Pituitary and Neuroendocrinology | 0070 | ECE2020 | 22nd European Congress of Endocrinology

ea0070aep560 | Pituitary and Neuroendocrinology | ECE2020

Proliferation index (Ki67) is a powerful predictor of recurrence in pituitary adenoma

Logan Ellis Hugh , Xekouki Evi , King Andy , Thomas Nick , Barazi Sinan , Maratos Eleni , Bullock Peter , Whitelaw Benjamin , Gilbert Jackie , Aylwin Simon

Introduction: In 2017 the term ‘Atypical adenoma’ was removed from the WHO classification, and ‘High-risk adenomas’ was introduced as a term to classify those with rapid growth, radiological invasion, and a high Ki-67 proliferation index. In this retrospective cohort study we have sought to identify the clinical and histological characteristics which are associated with worse outcomes.Methods: Data: We created a dataset of 1793 pa...

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ea0070aep561 | Pituitary and Neuroendocrinology | ECE2020

The expression level and binding affinity of glucocorticoid receptors in patients with Cushing’s syndrome

Zhang Qi , Liu Hui , Xiao Zhen , Yu Yerong

Summary: Objective: To evaluate the expression level and binding affinity of glucocorticoid receptors (GRs)in patients with endogenous Cushing’s syndrome(CS) and to determine whether there is partial glucocorticoid resistance.Methods: Fifty-eight patients with CS and forty-one age- and sex-matched healthy volunteers were studied. The expression level and binding affinity of GRs in peripheral blood mononuclear cells (PBMLs) were examined by flow cyt...

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ea0070aep562 | Pituitary and Neuroendocrinology | ECE2020

Treatment of acromegalic osteopathy in the real-life clinical practice: The baac (bone active drugs in acromegaly) multicenter study

Mazziotti Gherardo , Battista Claudia , Chiloiro Sabrina , Maffezzoni Filippo , Ferrante Emanuele , Grottoli Silvia , Grasso Ludovica , Gatto Federico , Olivetti Roberto , Arosio Maura , Bianchi Antonio , Carosi Giulia , Cellini Miriam , Chiodini Iacopo , De Marinis Laura , Di Somma Carolina , Ferlin Alberto , Ghigo Ezio , Lavezzi Elisabetta , Mantovani Giovanna , Pivonello Rosario , Porcelli Teresa , Prencipe Nunzia , Procopio Massimo , Scillitani Alfredo , Lania Andrea

Osteopathy is an emerging complication of acromegaly, characterized by increased bone turnover, profound abnormalities in trabecular bone structure and high risk of vertebral fractures (VFs). The therapeutic management of skeletal fragility in acromegaly is a challenge, since the risk of fractures may remain high after the control of acromegaly and the effectiveness of bone-active drugs in this clinical setting is unknown. In this retrospective-multicenter study, we aimed at e...

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ea0070aep563 | Pituitary and Neuroendocrinology | ECE2020

Macrophages involvement in neuroendocrine tumor behaviour and progression

Boemi Ilena , Vitali Eleonora , Trivellin Giampaolo , Smiroldo Valeria , Lavezzi Elisabetta , Zerbi Alessandro , Stratakis Constantine , Lania Andrea

Neuroendocrine neoplasms (NENs) are rare tumour showing a wide spectrum of clinical behaviours. Therapeutic options available for NET treatment are rarely curative and mostly palliative, as NETs frequently show resistance to pharmacological therapy. Cancers develop in complex tissue environments, which they depend on. Tumour-associated macrophages (TAMs) are a major cellular component of the tumour microenvironment. Two polarized state of macrophages are described in literatur...

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ea0070aep564 | Pituitary and Neuroendocrinology | ECE2020

Cushing’s syndrome & disease: Why does it take so long to diagnose; is the interdisciplinary medical team aware of the signs and symptoms; what are the consequences?

McBride Margaret

Background: CS and CD, a baffling portmanteau of symptoms, each often ascribed to other medical conditions, but together representing diagnostically, challenging medical conditions. Often coming, ‘disguised’, as other stand-alone medical conditions My diagnosis of CS and CD took me on a tortuous diagnostic journey taking almost 6 years to diagnose. My GP, during her 12 years of practice had never diagnosed CS. As part of my current PhD study of CS/CD, one of my obj...

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ea0070aep565 | Pituitary and Neuroendocrinology | ECE2020

Hypercortisolism-related molecular signature: Results from whole blood methylome analysis

Armignacco Roberta , Septier Amandine , Jouinot Anne , Neou Mario , Gaspar Cassandra , Perlemoine Karine , Bouys Lucas , Braun Leah , Riester Anna , Zennaro Maria-Christina , Reincke Martin , Bertherat Jérôme , Beuschlein Felix , Assié Guillaume

The effective treatment and optimal prognosis of hypercortisolism (Cushing’s syndrome – CS) depend on accurate and early diagnosis. However, hormonal assays can be complex, requiring multiple tests, and not predictive for any related complications, neither for their duration and severity. Identifying novel, specific and easily measurable biomarkers may improve CS diagnosis as well as the evaluation of complications. Since stress-associated epigenetic markers can be...

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ea0070aep566 | Pituitary and Neuroendocrinology | ECE2020

Plasma microRNA levels in patients with active acromegaly

Lutsenko Alexander , Belaya Zhanna , Nikitin Alexey , Solodovnikov Alexander , Lapshina Anastasia , Przhiyalkovskaya Elena , Melnichenko Galina

Background: Plasma microRNAs (miRs) (miR-4446-3p, miR-215-5p, miR-342-5p) were downregulated in patients with active acromegaly (AM) compared with healthy control subjects as assessed by next-generation sequencing.Aim: The aim of this study wasto validate the previously observed finding in the newly enrolled patients with AM using qRT-PCR.Materials and methods: Morning plasma samples were collected from fasting patients with AM and...

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ea0070aep567 | Pituitary and Neuroendocrinology | ECE2020

Multigene liquid biopsy (NETest) is diagnostic of pancreatic and small bowel neuroendocrine tumours and correlates with imaging

Malczewska Anna , Witkowska Magdalena , Makulik Karolina , Bocian Agnes , Walter Agata , Wojcik-Giertuga Monika , Pilch-Kowalczyk Joanna , Zajecki Wojciech , Bodei Lisa , Oberg Kjell , Kos-Kudla Beata

Introduction: There is a substantial unmet clinical need for an accurate and effective blood biomarker for gastroenteropancreatic neuroendocrine tumours (GEP-NETs). Current monoanalyte biomarkers are ineffective. The NETest, a novel multianalyte signature, provides molecular information relevant to disease biology. We therefore evaluated, under real-world conditions, the clinical utility of the NETest as a liquid biopsy in GEP-NETs.Aim(s): Independently ...

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ea0070aep568 | Pituitary and Neuroendocrinology | ECE2020

Modelling neuroendocrine tumours using 3 dimensional cell culturing

Krokker Lilla , Szabó Borbála , Németh Kinga , Sarkadi Balázs , Mészáros Katalin , Patócs Attila , Butz Henriett

Introduction: In vitro monolayer cell cultures are not able to faithfully model the biological features of the three dimensional (3D) structure of solid tumours. There are limited information about the applicability of 3D cell culturing methods for modeling of tumours of endocrine organs.Aim: The aim of our work was the establishment and characterization of three-dimensional in vitro models of various endocrine tumour cells in order to ...

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ea0070aep569 | Pituitary and Neuroendocrinology | ECE2020

Difference in plasma miRNA levels in inferior petrosal sinus samples from patients with ACTH-dependent Cushing’s syndrome as assessed by next-generation sequencing (NGS).

Belaya Zhanna , Malygina Anastasia , Nikitin Alexey , Koshkin Philipp , Sitkin Ivan , Rozhinskaya Liudmila , Melnichenko Galina , Dedov Ivan

Introduction: ACTH-secreting pituitary adenoma tissue differs from normal pituitary gland tissue in microRNA (miR) expression. MiRs are secreted into circulation and can be detected as potential biomarkersAim: To evaluate the difference in miR levels in plasma samples drained from inferior petrosal sinuses in patients with Cushing’s disease (CD) and ectopic-ACTH syndrome (EAS).Materials and methods: we enrolled 24 patients wi...

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ea0070aep570 | Pituitary and Neuroendocrinology | ECE2020

Challenges in the management of pituitary involvement in granulomatosis with Polyangitis (GPA): 2 cases managed at Imperial college healthcare trust

Behary Preeshila , Mendoza Nigel , Meeran Karim , Gontsarova Anastasia , Martin Niamh , Ellis Spencer , Tomlinson James , Levy Jeremy , Mc Adoo Stephen , Wernig Florian

Granulomatosis with Polyangitis (GPA) typically affects the lungs, kidneys and ENT system. Pituitary involvement is extremely rare, affecting less than 1% of all cases. The most common pituitary manifestations are secondary hypogonadism and Diabetes Insipidus (DI).Corticosteroids and immunosuppressive agents represent mainstay of medical management. Treatment needs to be initiated promptly and individualised according to clinical response.Case 1: A 54-ye...

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ea0070aep571 | Pituitary and Neuroendocrinology | ECE2020

Deletion of chromosome 1q24-1q32 and combined pituitary hormone deficiency type 4: Insight into the challenges of genotype-phenotype correlation

Giavoli Claudia , Ubertini Graziamaria , Giacchetti Federico , Rodari Giulia , Profka Eriselda , Cianfarani Stefano , Arosio Maura , Cappa Marco

Background: Interstitial deletions of the long arm of chromosome 1 are rare and classified as proximal or intermediate, the intermediate spanning bands 1q24–1q32. This region contains several genes, including LHX4, a LIM-homeodomain transcription factor essential in the early steps of pituitary ontogenesis. Indeed, mutations in the LHX4 gene are related to combined pituitary hormone deficiency type 4 (CPHD4, OMIM 602146).Aim: outline the impact of ...

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ea0070aep572 | Pituitary and Neuroendocrinology | ECE2020

Baseline IGF-I values influence the effect of rhGH therapy on fat mass: Short, medium and long term study on adults with GH deficiency

Profka Eriselda , Rodari Giulia , Giacchetti Federico , Draghi Alessandro , Arosio Maura , Giavoli Claudia

Background: One of the main effects of growth hormone therapy (rhGH) in the adult GH deficient patient (AGHD) is to positively modify body composition, with a reduction in fat mass (BF) and an increase in total lean mass (LM). However, the response to replacement therapy is highly variable and, contrary to what described in children, the potential predictive factors are not yet known.Aim of the study: To assess the impact of basal IGF-I levels on variati...

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ea0070aep573 | Pituitary and Neuroendocrinology | ECE2020

The SAGIT instrument: Potential utility in treatment decision-making in acromegaly

Sawicka-Gutaj Nadia , Ziółkowska Paulina , Ziemnicka Katarzyna , Szczepanek-Parulska Ewelina , Czarnywojtek Agata , Ruchała Marek

Introduction: The SAGIT instrument was developed for clinicians to assess the status and evolution of the disease in patients with acromegaly. Currently, it is investigated in the validation phase. The aim of our study was to evaluate the potential utility of SAGIT instrument for distinguishing acromegaly clinical stages and its usefulness in treatment decision-making at a single Polish endocrine center.Patients and Methods: Medical charts of adults with...

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ea0070aep574 | Pituitary and Neuroendocrinology | ECE2020

Cushing’s disease: Assessment of MGMT expression in pituitary corticotroph tumors and its relationship to clinical, pathological and ultrastructural parameters

Witek Przemyslaw , Maksymowicz Maria , Szamotulska Katarzyna , Stasiewicz Aleksandra , Wojciechowska-Luzniak Agnieszka , Zielinski Grzegorz

Background: O-6-Methylguanine-DNA-methyltransferase (MGMT) is a DNA repair enzyme. It has been demonstrated that its higher expression counteracts cytotoxicity of alkylating agents used in the treatment of glioblastoma or invasive pituitary tumors. Little is known on the relationships between MGMT expression and invasiveness of corticotroph adenomas in Cushing disease (CD).The Aim: To evaluate MGMT expression and its associations with clinical, neuropath...

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ea0070aep575 | Pituitary and Neuroendocrinology | ECE2020

Temporal trends in craniopharyngioma management and long term endocrine outcomes

HUSSEIN Ziad , Glynn Nigel , Martin Niamh , Alkrekshi Akram , Mendoza Nigel , Ramesh Nair , Mccullough Katherine , J Marcus Hani , Dorward Neil , Grieve Joan , Bouloux Peirre , Druce Maralyn , Baldeweg Stephanie

Background: The management of craniopharyngiomas remains controversial.Objective: This study sought to examine temporal trends in the management of craniopharyngioma and their impact on long-term patient outcomes, with focus on endocrine consequences.Methods: This was a cross sectional, multicentre study. Patients treated between 1951 and 2015 were identified and divided into four quartiles based on the date of initial surgery. Pat...

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ea0070aep576 | Pituitary and Neuroendocrinology | ECE2020

Sustained response to treatment with oral octreotide capsules: Results from the phase 3, randomized, double blind, placebo-controlled optimal study

Samson Susan , Nachtigall Lisa , Fleseriu Maria , Baldys Waligorska Agata , Jensterle Mojca , Ur Ehud , Molitch Mark E , Ludlam William , Patou Gary , Haviv Asi , Biermasz Nienke , Kennedy Laurence , Melmed Shlomo , Strasburger Christian J

Background: Patients with acromegaly responding to injectable somatostatin receptor ligands (SRL) are often treated for decades without deterioration of biochemical response (except for routine fluctuations in IGF-I control), unless there are changing clinical circumstances such as persistent or recurrent tumor growth. Oral octreotide capsules (OOC) have been formulated as a potential therapy for acromegaly and the safety and efficacy was evaluated in the CHIASMA OPTIMAL pivot...

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ea0070aep577 | Pituitary and Neuroendocrinology | ECE2020

Biochemical control of most patients reverting to injectable long-acting somatostatin receptor ligands is achieved after one dose: Results from the phase 3, randomized, double blind, placebo-controlled optimal study

Fleseriu Maria , Samson Susan , Nachtigall Lisa , Labadzhyan Artak , Elenkova Atanaska , Molitch Mark E , Ludlam William , Patou Gary , Haviv Asi , Biermasz Nienke , Strasburger Christian J , Kennedy Laurence , Melmed Shlomo

Background: Injectable somatostatin receptor ligands (SRLs) are currently the most widely used therapy for acromegaly. Oral octreotide capsules (OOC) are a potential therapy for acromegaly; the safety and efficacy were evaluated in the CHIASMA OPTIMAL pivotal study (Samson et al. ENDO 2020). As reported, mean IGF-I levels of the OOC group were maintained within normal range at end of treatment in all patients. However, some patients may not respond to OOC treatment (2...

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ea0070aep578 | Pituitary and Neuroendocrinology | ECE2020

Association between sagittal spine imbalance and radiological vertebral fractures in acromegaly: Does it reflect a pathophysiological link?

Cellini Miriam , Biamonte Emilia , Trenti Nicoletta , Mazza Massimiliano , Milani Davide , Ferrante Emanuele , Lavezzi Elisabetta , Mantovani Giovanna , Arosio Maura , Fornari Maurizio , Balzarini Luca , Lania Andrea , Mazziotti Gherardo

Chronic exposure to GH hypersecretion may alter the physiological balance of the spine through inducing degeneration of intervertebral discs and impaired trophism of facet joints. Moreover, GH in excess may also cause profound deterioration in bone microstructure with consequent increase in risk of fragility vertebral fractures (VFs). In this cross-sectional study,we evaluated for the first time in acromegaly the association between spine imbalance and VFs. Thirty-eight patien...

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ea0070aep579 | Pituitary and Neuroendocrinology | ECE2020

Once-weekly somapacitan in japanese adults with growth hormone deficiency was well tolerated, with similar efficacy to daily growth hormone: A randomised trial

Otsuka Fumio , Takahashi Yutaka , Tahara Shigeyuki , Ogawa Yoshihisa , Højby Rasmussen Michael , Takano Koji

Somapacitan is a long-acting, reversible albumin-binding growth hormone (GH) derivative. The objective of this trial was to evaluate the safety, efficacy and treatment satisfaction of onceweekly somapacitan versus daily GH (Norditropin) over 52 weeks in Japanese patients with adult GH deficiency (AGHD). This was a phase 3, multicentre, randomised, open-label, parallel-group, active-controlled trial (NCT03075644). Patients previously treated with GH were randomised 1:3 to daily...

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ea0070aep580 | Pituitary and Neuroendocrinology | ECE2020

Real life efficiency of pegvisomant therapy in acromegaly

Maria Lider Burciulescu Sofia , Livia Gheorghiu Monica , Caragheorgheopol Andra , Purice Mariana , Badiu Corin

Introduction: Pegvisomant (PEG) is an efficient treatment for acromegaly but the recommendation for this treatment is less active due to its high cost. In Romania it is reimbursed in doses up to 30 mg s.c/day or as 40 or 80 mg/week in combination with a somatostatin analog (SSA).Design: Retrospective analysis of 18 consecutive patients treated with PEG for acromegaly (either as monotherapy, or in combination with SSA and/or cabergoline (CAB...

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ea0070aep581 | Pituitary and Neuroendocrinology | ECE2020

Sheehan like syndrome in males: A case series of three patients developing pituitary infarction following hypotension

Qayum Ambreen , Zaman Shamaila , Mohammad Sanas , Todd Jeannie F , Meeran Karim

Pituitary apoplexy caused by pure infarction is rare. Here we present three cases of pituitary macroadenoma infarction following hypotension.1. An 84 year old gentleman had a 2 × 2 cm pituitary macroadenoma causing chiasmal compression and a bitemporal hemianopia. He had panhypopituitarism and was on prednisolone, thyroxine and testosterone replacement. Before surgical intervention of the macroadenoma, he fell and broke his humerus, which requi...

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ea0070aep582 | Pituitary and Neuroendocrinology | ECE2020

An asynchronous double growth hormone secreting pituitary adenoma ofa different proliferative potential – a case report

Trofimiuk-Muldner Malgorzata , Domagała Bartosz , Kluczynski Lukasz , Sokolowski Grzegorz , Zielinski Grzegorz , Maksymowicz Maria , Pekul Monika , Hubalewska-Dydejczyk Alicja

Background: Double pituitary adenomas are a rare entity, which requires clinical attention and a careful follow-up.Case report: A 37-year-old man presented with left-sided painful gynecomastia. He denied typical symptoms of excessive growth hormone (GH) secretion and did not show any acromegalic features. Due to low testosterone and LH levels with mild hyperprolactinaemia, the patient was referred to pituitary MR, which revealed an 11 × 13 mm right-...

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ea0070aep583 | Pituitary and Neuroendocrinology | ECE2020

Not just another headache – a rare case of recurrent pituitary collection

Tee Su , Andrew James Robert , Nicholson Claire , Mamoojee Yaasir

A 27 year old lady presented in 2016 with a 3 month history of intermittent headache and visual disturbance, 3.5 months post-partum. She had a past medical history of cluster headache; treated with verapamil and short courses of oral steroids (she had no recent steroid courses prior to presentation). She was found to have a partial right sixth nerve palsy but normal visual fields on Goldmann’s testing. She had biochemical evidence of secondary adrenal insufficiency (sodi...

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ea0070aep584 | Pituitary and Neuroendocrinology | ECE2020

A novel mechanism regulating dopamine receptor type 2 (DRD2) signal transduction in PRL- and ACTH-secreting pituitary tumoral cells: The role of cAMP/PKA-induced filamin A (FLNA) phosphorylation in the control of responsiveness to DRD2 agonist

Mangili Federica , Treppiedi Donatella , Catalano Rosa , Giardino Elena , Arosio Maura , Spada Anna , Mantovani Giovanna , Peverelli Erika

The actin binding protein filamin A (FLNA) is required for somatostatin receptor 2 (SST2) and dopamine receptor 2 (DRD2) expression, intracellular localization and signaling in GH- and PRL-secreting pituitary tumors, respectively, playing a role in tumor responsiveness to somatostatin analogs and dopaminergic drugs. FLNA functions are tightly regulated by several mechanisms, including FLNA phosphorylation. It has recently been shown that in GH-secreting pituitary tumors FLNA p...

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ea0070aep585 | Pituitary and Neuroendocrinology | ECE2020

Kisspeptin-54 accurately identifies hypothalamic dysfunction in men with congenital hypogonadotropic hypogonadism

Abbara Ali , Chia Eng Pei , Phylactou Maria , Clarke Sophie , Mills Edouard , Chia Germaine , Yang Lisa , Izzi-Engbeaya Chioma , Jayasena Channa , Comninos Alexander , Ivell Richard , Anand-Ivell Ravinder , Rademaker Jesse , Cheng Xu , Quinton Richard , Pittelout Nelly , Dhillo Waljit

Background: Hypogonadotrophic hypogonadism is characterised by hypogonadism in the context of low/inappropriately normal gonadotrophin levels. Congenital Hypogonadotrophic Hypogonadism (CHH) occurs due to disrupted GnRH neuronal migration, or impaired hypothalamic GnRH secretion or action. However, no direct test of hypothalamic GnRH neuronal function currently exists. Kisspeptin-54 is a neuropeptide that stimulates endogenous hypothalamic GnRH release. Thus, we investigated w...

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ea0070aep586 | Pituitary and Neuroendocrinology | ECE2020

Growth hormone-releasing hormone (GHRH) promotes survival and proliferation of neural stem cells and reduces amyloid-β-induced toxicity

Banfi Dana , Gesmundo Iacopo , Ghigo Ezio , Schally Andrew V , Granata Riccarda

Neurogenesis, a process by which new neurons are generated from precursors, still persists in discrete regions of the adult hippocampus. Impairment in neurogenesis is an important feature in the pathogenesis of neurodegenerative diseases, such asAlzheimer’s disease (AD). The hippocampus is critical for learning and memory and is a main target of AD, which causes massive neuronal death, reduction in neurogenesis and impairment in cognitive functions. Therefore, preventing...

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ea0070aep587 | Pituitary and Neuroendocrinology | ECE2020

Pancreatic neuroendocrine tumour with suspected extrarenal secretion of 1,25-dihydroxyvitamin D3

Yung Zhuang Choo Alwyn , Mohammed Kamrudeen

Background: Hypercalcaemia can occur in 20–30% of malignancy. Neuroendocrine malignancy-related hypercalcaemia is relatively rare with few reported cases, mostly in pancreatic neuroendocrine tumours. It could be related to humoral hypercalcaemia of malignancy (PTHrP secretion), production of 1,25-dihydroxyvitamin D3 (1,25(OH)2D3) by the tumour or production of PTH by the tumour1,2. Here we describe a case of metastatic neuroendocrine tumour with pos...

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ea0070aep588 | Pituitary and Neuroendocrinology | ECE2020

New advances in the diagnosis of gonadotroph tumours

García-Martínez Araceli , Morote-Guillén Cristina , Martínez-López Sebastián , Eugenia Torregrosa-Quesada María , Silva-Ortega Sandra , López-Muñoz Beatriz , Moreno Óscar , Aranda Ignacio , Picó Antonio

Introduction: Gonadotroph tumours (GT) are the most common subtype of silent pituitary neuroendocrine tumours. The diagnosis of this subtype of tumours is established after surgery. There are no reliable markers of aggressiveness to predict their clinical course and, moreover, immunohistochemistry (IHC) usually classifies GT as null cell tumours. The aim of the present study was to correlate molecular, immunohistochemical and biochemical gonadotropin expression, to quantify th...

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ea0070aep589 | Pituitary and Neuroendocrinology | ECE2020

The cushing’s collaborative patient survey results

Valassi Elena , Tabarin Antoine , Chiodini Iacopo , Feelders Richard , Andela Cornelie

Background: Early diagnosis of Cushing’s syndrome and initiation of effective treatment are essential to limit long-term morbidity and early mortality. However, diagnosis is often delayed due to the non-specificity of symptoms, or because symptoms are not recognised by physicians, leading to more complex treatment needs and a worsening of patient quality of life. A survey was conducted to better understand the patient experience of Cushing’s syndrome and the true b...

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ea0070aep590 | Pituitary and Neuroendocrinology | ECE2020

High-dose corticosteroid treatment of immunotherapy-induced hypophysitis: A multi-centric, restrospective analysis

Amereller Felix , Joshi Mamta , DEUTSCHBEIN Timo , Duffy Leo , Carroll Paul , Papa Sophie , Schilbach Katharina , Schopohl Jochen , Störmann Sylvère

Introduction: Hypophysitis is a frequent side effect of oncologic immunotherapy. The recommended standard treatment is systemic high-dose corticosteroids (HDS), however, recent studies have questioned the benefits of this treatment. We sought to examine the effect of HDS on immunotherapy-induced hypophysitis (IH) in a multi-centric cohort.Methods: Medical records of 41 patients with IH treated by specialist endocrinologists at three tertiary referral cen...

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ea0070aep591 | Pituitary and Neuroendocrinology | ECE2020

Assessment of hypercoagulability in patients with cushing syndrome before and after surgical cure

Liliana Serban Andreea , Tripodi Armando , Indirli Rita , Mantovani Beatrice , Sala Elisa , Padovan Lidia , Arosio Maura , Mantovani Giovanna , Ferrante Emanuele

Background: Patients with Cushing’s Syndrome (CS) have a high risk of venous thromboembolism (VTE) related to a hypercoagulable state. Previous studies showed increased levels of procoagulant factors but also an elevation of some of the anticoagulants factors and fibrinolytic enzymes. Once patients achieve disease remission, there is a significant decrease of some procoagulant factors but if these alterations are completely reversible is still unclear. Compared to tradit...

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ea0070aep592 | Pituitary and Neuroendocrinology | ECE2020

Morphometric vertebral fractures are highly prevalent in patients with non-functioning pituitary adenoma and related to older age and hypopituitarism

Frara Stefano , Di Filippo Luigi , Doga Mauro , Maria Formenti Anna , Losa Marco , Resmini Eugenia , Tecilazich Francesco , Mortini Pietro , Giustina Andrea

Introduction: Recent studies showed that morphometric vertebral fractures (VF) are an early and frequent complication in patients (pts) withhyperfunctioning pituitary adenomas and hypopituitarism (independently from the etiology). GH excess or severe deficiency play a negative key role on bone health in this clinical setting. Disease control or adequate replacement therapies still remain a cornerstone for the clinician to reduce the burden of osteo-metabolic complications. To ...

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ea0070aep593 | Pituitary and Neuroendocrinology | ECE2020

Course of serum potassium levels during the hypertonic saline infusion test for diagnosis of diabetes insipidus

Potasso Laura , Refardt Julie , Chifu Irina , Fassnacht Martin , Kristin Fenske Wiebke , Christ-Crain Mirjam

Introduction: Onset of hyperkalemia has previously been reported during the administration of hypertonic resuscitation solutions. Since the hypertonic saline infusion test has recently been validated for the diagnosis of diabetes insipidus, we aimed to investigate the course of plasma potassium levels during the test.Material and Methods: Data of 144 patients undergoing osmotic stimulation with hypertonic saline infusion at 11 tertiary medical centres in...

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ea0070aep594 | Pituitary and Neuroendocrinology | ECE2020

Concordant testing results in differential diagnosis in ACTH-dependent cushing’s syndrome: A retrospective study of high dose dexamethasone suppression test (HDDST) and DDAVP stimulation test

Qiao Jingtao , Zhang Weiwei , Yu Yerong

Background: Investigating the origin of ACTH determine disparate treatment and prognosis in ACTH-dependent Cushing’s Syndrome. We want to detect the value of combination of HDDST and DDAVP stimulation test for identifying Cushing’s disease (CD) and Ectopic ACTH-dependent Cushing’s Syndrome (EAS).Methods: We retrospectively recorded 102 ACTH-dependent Cushing’s Syndrome (87 CD and 15 EAS) in West China Hospital from January 1, 2010...

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ea0070aep595 | Pituitary and Neuroendocrinology | ECE2020

Gallium-68 -DOTATATE PET imaging in clinically non-functioning pituitary macroadenomas

M. Boertien Tessel , Booij Jan , Majoie Charles BLM , Drent ML , Pereira Alberto M , Biermasz Nienke , Simsek Suat , Groote Veldman Ronald , Stokkel Marcel PM , Bisschop Peter H , Fliers Eric

Background: Clinically non-functioning pituitary macroadenomas (NFMA) have been reported to express various somatostatin receptor (SSTR) subtypes, but results are inconsistent across different studies. This may be related to limited sensitivity and specificity of techniques used to date, i.e. immunohistochemistry in surgical specimens and 111In-DTPA-octreotide scintigraphy (Octreoscan) in vivo. The aim of this study was to assess SSTR expression in NFMA <em...

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ea0070aep596 | Pituitary and Neuroendocrinology | ECE2020

Efficacy and safety of long-acting pasireotide (LA-PAS) in patients with uncontrolled acromegaly: Results from the prospective cohort of european observational acronis study

Pivonello Rosario , Tabarin Antoine , Colao Annamaria , Kaltsas Gregory , Gonzalez-Molero Inmaculada , Neggers Sebastian , Venegas Moreno Eva , Kocjan Tomaz , Karges Wolfram , Krzyzanowska-Mittermayer Katarzyna , Heck Ansgar , Eden Engstrom Britt , Marques Ruben , O’Brien Eamonn , Manne Shobha , Vincenzi Beatrice , Schöfl Christof

Introduction: Results of the first European real-world evidence from retrospective cohort of ACRONIS study previously confirmed the efficacy and tolerability of LA-PAS in heavily pre-treated, uncontrolled acromegaly patients. Here, we report results of the second interim analysis reflecting the prospective cohort.Methods: Patients who were treated with LA-PAS for ≥6 months were included in this analysis. The primary objective was to document treatm...

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ea0070aep597 | Pituitary and Neuroendocrinology | ECE2020

High usage of drugs for neuropsychiatric morbidity in patients with Cushing´s disease before diagnosis and at 5–10 years follow-up – a nationwide study

Bengtsson Daniel , Ragnarsson Oskar , Berinder Katarina , Dahlqvist Per , Edén Engström Britt , Ekman Bertil , Höybye Charlotte , Wahlberg Jeanette , Burman Pia

Background: Neuropsychiatric symptoms and cognitive dysfunction are common in Cushing´s disease (CD), and seem only partly reversible after biochemical remission has been achieved.Aim: To investigate prescription of drugs associated with neuropsychiatric morbidity in a large national cohort of CD patients.Methods: Patients in the Swedish Pituitary Registry, diagnosed with CD between July 2006 and January 2018 were included, <e...

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ea0070aep598 | Pituitary and Neuroendocrinology | ECE2020

The importance of DHEA-S levels in cushing’s syndrome; is there a cut-off value in the differential diagnosis?

Ciftci Dogansen Sema , Soyluk Selcukbiricik Ozlem , Selek Alev , Erol Selvinaz , Hekimsoy Zeliha , Esen Pazir Ayse , Dursun Huseyin , Sahin Serdar , Oruk Gonca , Mert Meral , Soylu Hüseyin , Sarer Yurekli Banu , Eda Ertorer Melek , Omma Tülay , Evran Mehtap , Adaș Mine , Tanrikulu Seher , Aydin Kadriye , Pekkolay Zafer , Can Bulent , Karakilic Ersen , Karaca Zuleyha , Bilen Habib , Canturk Zeynep , Cetinarslan Berrin , Kadioglu Pinar , Yarman Sema

Purpose: To evaluate the relationship between basal dehydroepiandrosterone-sulfate (DHEA-S) levels and other tests used in the diagnosis and differential diagnosis of Cushing’s Syndrome (CS) among the patients with pathologically confirmed CS.Methods: In this multicenter study, the data of 623 patients with CS were evaluated retrospectively. The patients were classified as Group 1 (n = 353 Cushing’s disease;CD), Group 2 (n</e...

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ea0070aep599 | Pituitary and Neuroendocrinology | ECE2020

Midparental height is an important predictive parameter in a late diagnosis of acromegaly and gigantism in adults. Single centre, pilot study

Bogusławska Anna , Gilis-Januszewska Aleksandra , Godlewska Magdalena , Olszewska Marta , Hubalewska-Dydejczyk Alicja , Starzyk Jerzy

Introduction: Acromegaly is a rare disorder caused by excessive growth hormone production. Common clinical manifestations are visual changes as well as serious systemic complications. In childhood and adolescence, excessive growth hormone production leads to abnormal tall stature. To date, only a few studies have been published focusing on analysing the growth of patients with acromegaly, in particular regarding the growth of their parents and siblings. A&#...

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ea0070aep600 | Pituitary and Neuroendocrinology | ECE2020

Phenotypic differences between patients with familial pituitary neuroendocrine tumours due to MEN1 or AIP mutations

Marques Pedro , Magalhães Daniela , Caimari Francisca , Hernández Ramírez Laura , Collier David , Lim Chung , Stals Karen , Ellard Sian , Druce Maralyn , Akker Scott , Waterhouse Mona , Drake William , Grossman Ashley B. , Korbonits Marta

Introduction: Germline AIP and MEN1 mutations are the main known aetiologies of familial pituitary neuroendocrine tumours (PitNETs), which represent 5% of all PitNETs. We compared the clinical and tumour characteristics of AIP (AIP mut) and MEN1 mutation-positive (MEN1 mut) PitNET patients.Methods: We retrospectively analysed 70 MEN1 mut and 167 AIP mut patients with PitNETs. MEN...

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ea0070aep601 | Pituitary and Neuroendocrinology | ECE2020

A case of wolfram syndrome with primary hypogonadism

Calapkulu Murat , Erkam Sencar Muhammed , Ozturk Unsal İlknur , Duger Hakan , Kizilgul Muhammed , Bostan Hayri , Cakal Erman , Ozbek Mustafa

Introduction: Wolfram syndrome, is a rare autosomal recessive genetic disorder that is characterized by diabetes mellitus (DM), diabetes insipidus (DI), optic atrophy, and sensorineural deafness as well as various other possible disorders. DM is the first manifestation, and optic atrophy also onsets in the first decade of life. The onsets of DI and sensorineural deafness are in the second decade, urinary tract abnormalities are in the third decade, and neurologic abnormalities...

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ea0070aep602 | Pituitary and Neuroendocrinology | ECE2020

Effect of silibinin on acth synthesis and secretion in human adenomatous corticotropes in vitro

Francesca Cassarino Maria , Sesta Antonella , Libera Laura , Bardelli Donatella , Losa Marco , Lasio Giovanni , Pecori Giraldi Francesca

Silibinin, a milk thistle extract with known hepatoprotective effects, has recently been shown to act upon tumoral corticotropes and revert the Cushingoid phenotype in an allograft mouse model (Riebold et al. 2015). Silbinin is known to inhibit HSP90 -a chaperone to the glucocorticoid receptor-thereby restoring sensitivity to glucocorticoid negative feedback in tumoral corticotropes. Aim of the present study was to assess the effect of silibinin on ACTH synthesis and ...

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ea0070aep603 | Pituitary and Neuroendocrinology | ECE2020

TGF-β increases caspase 3 activation and migration in bronchial carcinoids

Borges de Souza Patricia , Tagliatti Valentina , Capobianco Simonaco , Rosaria Ambrosio Maria , Chiara Zatelli Maria

Introduction: Typical and atypical bronchial carcinoids (TBC and ABC) are well-differentiated neuroendocrine neoplasms (NEN). First-line treatment is radical removal of the primary tumour; however, management of metastatic BC is still challenging. Bronchial carcinoids (BC) may benefit from treatment with mTOR inhibitors, although response rates are moderate. TBC have shown reduced sensibility to mTOR inhibitors respect to ABC, notwithstanding its less aggressive behaviour. PI3...

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ea0070aep604 | Pituitary and Neuroendocrinology | ECE2020

Salivary cortisol and cortisone – Effects of liquorice and blood contamination

Bäcklund Nils , Imamovic Marcus , Brattsand Göran , Lundstedt Staffan , Aardal Elisabeth , Olsson Tommy , Dahlqvist Per

Context: Late-night salivary cortisol is a recommended screening method for Cushing’s syndrome. Liquorice intake may lead to falsely elevated salivary cortisol as glycyrrhizic acid inhibits the 11β-hydroxysteroid dehydrogenase type 2 induced conversion of cortisol to cortisone in the salivary glands. Furthermore, sample contamination by blood from the oral mucosa may also cause falsely elevated salivary cortisol levels. Salivary cortisone has been suggested to more ...

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ea0070aep605 | Pituitary and Neuroendocrinology | ECE2020

An education programme for patients with pituitary tumours and their relatives; preliminary results

Tiberg Persson Jenny , Jakobsson Sofie , Cid Käll Kristina , Kullin Lena , Olofsson Ann-Charlotte , Wittboldt Ia , Skoglund Thomas , Trimpou Penelope , Jakobsson Ung Eva , Johannsson Gudmundur

Background: Patients with pituitary tumour live with life-long consequences of their disease. The first year after pituitary surgery represents a time period of symptoms, fear of tumour recurrence, existential concerns as well as extensive medical evaluations and decisions about hormone replacement. To increase wellbeing, a person-centred care practice up to 1 year after pituitary surgery covering self-management support, accessibility and continuity has been implemented and a...

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ea0070aep606 | Pituitary and Neuroendocrinology | ECE2020

Clinical picture and MRI findings in patients with hypophysitis – a single-centre experience

Czajka-Oraniec Izabella , Stelmachowska-Banas Maria , Cylke Karolina , Zgliczynski Wojciech

Hypophysitis is a rare clinical condition manifesting as pituitary dysfunction: hypopituitarism or diabetes insipidus (DI) and pituitary enlargement causing mass effect (headaches, visual disturbances) due to the inflammation of the pituitary gland. We conducted a retrospective analysis of medical records of patients diagnosed with hypophysitis in the Department of Endocrinology of the Bielanski Hospital from 2014 to 2019. Twenty-five patients were identified, which represents...

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ea0070aep607 | Pituitary and Neuroendocrinology | ECE2020

Data mining analyses for precision medicine in acromegaly

Marques-Pamies Montserrat , Gil Joan , Jorda Mireia , Antonio Sampedro-Nunez Miguel , Webb Susan , Serra Guillermo , Salinas Vert Isabel , Blanco Alberto , Valassi Elena , Maria Martel Duguech Luciana , Carrato Cristina , Pico Antonio , García-Martínez Araceli , Simó-Servat Andreu , Biagetti Betina , Villabona Carles , Camara-Gómez Rosa , Fajardo-Montañana Carmen , Lamas Cristina , Alvarez Clara , Bernabeu Ignacio , Marazuela Monica , Puig-Domingo Manel

Since the first somatostatin receptor ligand (SRL) was used to treat acromegaly, predicting which patients could benefit from their use has become crucial to avoid months of ineffective treatment for non-responding patients. Although many biomarkers linked to SRL response have been identified, there is no consensus criterion on how to prescribe according to biomarker levels. In this study, we evaluate previously reported biomarkers using more exhaustive and accurate methods th...

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ea0070aep608 | Pituitary and Neuroendocrinology | ECE2020

Metabolism of glucose in patients with acromegaly treated with Pegvisomant and/or Pasireotide LAR after resistance to first generation somatostatin receptor ligands

Chiloiro Sabrina , Giampietro Antonella , Bianchi Antonio , Maria Formenti Anna , Maya Fleseriu Cara , Pontecorvi Alfredo , Giustina Andrea , De Marinis Laura

Introduction: Acromegaly (Acro) is characterized by insulin sensitivityreduction, glucose intolerance (IGT) and diabetes mellitus (DM2 in 15%–38% of patients). Studies that investigated action of medical therapies for acro on glucose metabolism, didn’t provide conclusive data. Association between blood glucose (BG) levels and serum IGF-I levels in patients with DM2 and acro has been suggested, however,IGF-I levels and hemoglobin A1c (HbA1c) correlation is still con...

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ea0070aep609 | Pituitary and Neuroendocrinology | ECE2020

Biliary stone disease in acromegaly under somatostatinanalogues: A longterm safety study

Prencipe Nunzia , Cuboni Daniela , Bona Chiara , Maria Berton Alessandro , Parasiliti Caprino Mirko , Fenoglio Luigi , Gasco Valentina , Ghigo Ezio , Grottoli Silvia

Introduction: Somatostatin analogues (SSA) are one of the main effective drug used in acromegaly. Sludge and/or cholelithiasis development represents oneof the more common side effect. The aim of the study was to analyze the frequency of biliary adverse events (BAE) and possible predictive factors, to propose a work-up strategy for their management.Method: This is a single centre, longitudinalretrospective study; we enrolled 91 acromegaly patients during...

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ea0070aep610 | Pituitary and Neuroendocrinology | ECE2020

Importance of sexual function assessment in multidimensional evaluation of AGHD patients: Baseline results from the management of adult growth hormone deficiency (MAGHD) study

Laura Monzani Maria , Pederzoli Simone , Volpi Laura , Magnani Elisa , Diazzi Chiara , Rochira Vincenzo

Background: Patients with adult growth hormone deficiency (AGHD) have impaired health-related quality of life (QoL). While the effects of reduced muscle mass and vitality-loss on QoL have been well characterized in AGHD, the impact of AGHD on sexual function, a recognized factor influencing well-being, has never been deepened.Aim: To investigate the prevalence of sexual dysfunction in AGHD patients referring to a single endocrinological center and groupe...

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ea0070aep611 | Pituitary and Neuroendocrinology | ECE2020

Effects of different therapeutic approaches on cardiovascular risk in patients with acromegaly: Results of a multicentric study

Reimondo Giuseppe , Spagnolo Federica , Puglisi Soraya , Lombardi Stocchetti Benedetta , Ragonese Marta , Ceccato Filippo , Ferraù Francesco , Berchialla Paola , Carla Scaroni , Cannavò Salvatore , Terzolo Massimo

The association between acromegaly and increased cardiovascular morbidity and mortality is widely recognised. The aim of this retrospective multicentric study is to evaluate the impact of different therapeutic approaches on the progression of cardiovascular risk (CVR) in acromegalic patients. At diagnosis and last follow up (follow up time of 13 ± 8 years),we assessed BMI, blood pressure, glycaemic and lipid profile, GH and IGF1 levels in 199 patients with acromegaly (116...

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ea0070aep612 | Pituitary and Neuroendocrinology | ECE2020

IGF-I variability and its association with demographic and clinical characteristics in patients with acromegaly treated with injectable somatostatin receptor ligands (SRLs); results from OPTIMAL, an international prospective phase 3 study

Samson Susan , Nachtigall Lisa , Fleseriu Maria , Ur Ehud , E Molitch Mark , Ludlam William , Patou Gary , Haviv Asi , Gilgun-Sherki Yossi , Biermasz Nienke , Strasburger Christian J , Kennedy Laurence , Melmed Shlomo

Background: Most patients responding to injectable somatostatin receptor ligands exhibit IGF-I variability around the upper limit of normal (ULN) during long-term follow up. These fluctuations are thought to result from assay variability, nutrition, comorbid conditions, concomitant medications and other unknown factors. The magnitude and factors affecting this variability arenot well understood in patients with acromegaly treated with injectable SRLs.Met...

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ea0070aep613 | Pituitary and Neuroendocrinology | ECE2020

Analysis of adverse events in adult patients with acromegaly receiving oral octreotide capsules: Results from the phase 3, randomized, double-blind, placebo-controlled optimal study

Labadzhyan Artak , Samson Susan , Nachtigall Lisa , Fleseriu Maria , Molitch Mark E , Ludlam William , Patou Gary , Haviv Asi , Baldys Waligorska Agata , Biermasz Nienke , Trainer Peter , Kennedy Laurence , Melmed Shlomo , Strasburger Christian J

Background: Distinguishing non-specific signs/symptoms of acromegaly from treatment-emergent adverse events (TEAEs) in patients treated with somatostatin receptor ligands has proven difficult given limited data from placebo-controlled studies. The CHIASMA OPTIMALstudy provides a novel data set to evaluate the incidence of adverse events (AEs) in patients randomized to oral octreotide capsules (OOC) or placebo.Methods: A multinational, randomized, placebo...

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ea0070aep614 | Pituitary and Neuroendocrinology | ECE2020

The effects of silent pituitary adenomas on pregnancy

Baș Aksu Özge , Demir Özgür , Gökçay Canpolat Asena , Çorapçıoğlu Demet

Silent pituitary adenomas are defined as a group of adenomas that are hormonally inactive and do not show clinical signs and symptoms of excess hormones. Data in the literature indicate that fertility is usually affected in women with silent pituitary adenomas.Aim: To investigate the possible effects of silent pituitary adenomas on pregnancy.Patients and method: Thirty patients followed by silent pituitary adenoma, without hormone ...

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ea0070aep615 | Pituitary and Neuroendocrinology | ECE2020

Cushing’s syndrome negatively affects socio-economic variables many years before the diagnosis: A nationwide registry-based cohort study

Ebbehoj Andreas , Søndergaard Esben , Jepsen Peter , Matilde Lundsgaard Svane Helene , Madsen Morten , Poulsen Per Løgstrup , Jorgensen Jens Otto

Introduction: Cushing’s syndrome (CS) results from prolonged glucocorticoid excess. Both iatrogenic and endogenous CS cause somatic and neuropsychiatric morbidity that do not fully reverse after biochemical disease control. However, the real-world socio-economic consequences for patients and their families are largely unknown.Aim: To determine the impact of CS on work status, income, education, marital status, parenthood, and depression before and ...

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ea0070aep616 | Pituitary and Neuroendocrinology | ECE2020

Prevalence, predictors and outcomes of acute, life-threatening and perioperative complications in cushing’s syndrome

Schernthaner-Reiter Marie Helene , Siess Christina , Micko Alexander , Wolfsberger Stefan , Scheuba Christian , Riss Philipp , Knosp Engelbert , Kautzky-Willer Alexandra , Luger Anton , Vila Greisa

Introduction: Cushing’s syndrome is associated with significant chronic and acute complications including acute thromboembolic and cardiovascular events. We aimed to study the prevalence and predictors of acute and perioperative complications in patients with active Cushing’s syndrome.Methods: The prevalence, predictors and outcomes of acute, life-threatening and perioperative complications were evaluated in a cohort of patients with active b...

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ea0070aep617 | Pituitary and Neuroendocrinology | ECE2020

SST5 expression and USP8 mutation in functioning and silent corticotroph pituitary tumors

Castellnou Solene , Vasiljevic Alexandre , Lapras Veronique , Raverot Véronique , Alix Eudeline , Borson-Chazot Francoise , Jouanneau Emmanuel , Raverot Gerald , Hélène Lasolle

Context: Somatostatin receptor type 5 (SST5) is inconsistently expressed by corticotroph tumors, with higher expression found in corticotropinomas having ubiquitin-specific protease 8 (USP8) mutations.Aims: To study the correlation between characteristics of corticotropinomasand SST5 expression/USP8 mutation status; to describe the response to pasireotide in 5 patients.Design: Retrospective cohort study.Metho...

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ea0070aep618 | Pituitary and Neuroendocrinology | ECE2020

Efficacy, safety and pitfalls of glucocorticoid therapy for patients with autoimmune hypophysitis

Tatsushima Keita , Takeshita Akira , Fukuhara Noriaki , Yamaguchi-Okada Mitsuo , Fukuhara Hirokazu , Nishioka Hiroshi , Takeuchi Yasuhiro

Background and aim: Autoimmune hypophysitis (AH) is a rare immune-mediated inflammatory disease. Glucocorticoid therapy represents a therapeutic choice for AH patients with symptoms from mass effect, such as headache and optic nerve compression. However, effective methods and adverse events of glucocorticoid therapy are unclear. We aimed to evaluate the efficacy and safety of glucocorticoid therapy in AH patients.Patients and Methods: The subjects were a...

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ea0070aep619 | Pituitary and Neuroendocrinology | ECE2020

Observational study of aetiology, efficacy of treatment and outcomes of hyponatraemia following pituitary surgery in a major neurosurgical centre

Hussein Ziad , Tzoulis Plutarchos , J Marcus Hani , Grieve Joan , Dorward Neil , Baldeweg Stephanie , Bouloux Pierre-Marc

Introduction: Hyponatraemia is a relatively common complication following pituitary surgery. However, there is sparse data about its optimal management and impact on clinical outcomes.Aims: To review the treatment and patient outcomes of hyponatraemia following pituitary surgery.Methods: A retrospective single-centre study included all inpatients who developed serum sodium (sNa) ≤ 132 mmol/l following pituitary surgery f...

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ea0070aep620 | Pituitary and Neuroendocrinology | ECE2020

Growth hormone deficiency in childhood acute lymphoblastic leukemia survivors – should systematic stimulation test be performed in all irradiated children?

Damasio Ines , Cavaco Daniela , Maciel Joana , Dias Daniela , Donato Sara , Pinheiro Sara , Figueiredo Ana , Simões-Pereira Joana , Pereira Conceiçao

Introduction: The combination of chemotherapy with prophylatic cranial radiotherapy (C-RT) allowed the improvement of survival rates of pediatric acute lymphoblastic leukemia (ALL) survivors while putting them at risk of develeping long-term endocrine deficiencies, like growth hormone deficiency (GHD).Current evidence suggests that the prevalence of GHD in children treated with radiation doses ≥ 30–50 Gy for non-pituitary brain tumors is...

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ea0070aep621 | Pituitary and Neuroendocrinology | ECE2020

9-CIS retinoic acid decreases pomc expression and cell viability in experimental model of ectopic cushing syndrome

Regazzo Daniela , Barbot Mattia , Ceccato Filippo , Nora Albiger , laura lizzul , Fiorella Calabrese , Federico Rea , Andrea Zuin , Boscaro Marco , Occhi Gianluca , Carla Scaroni

Ectopic Cushing syndrome (EAS) is a rare condition characterized by ACTH-dependent hypercortisolism resistant to normal physiologic suppression by glucocorticoids. EAS is due to an extra-pituitary tumor producing bioactive molecules generated by post-translational cleavage of the proopiomelanocortin gene (POMC). EAS is associated with significant morbidity and mortality and although surgical resection of the primary ACTH-producing tumor remains the mainstay of therapy, not all...

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ea0070aep622 | Pituitary and Neuroendocrinology | ECE2020

Sporadic neuroendocrine neoplasms in young-adult patients: Differences in natural history, prognosis and treatment compared to adult-elderly patients

Altieri Barbara , Modica Roberta , Bottiglieri Filomena , de Cicco Federica , Faggiano Antongiulio , Colao Annamaria

Introduction: Sporadic neuroendocrine neoplasms (NEN) occur rarely in young-adult (YA) patients, with an estimated incidence is about 2.8 cases per million, and data specific to their epidemiology are limited. The aim of our study was to better characterize the natural history, prognosis and management of NEN in YA patients (≤ 35 years old) compared to adult-elderly (AE; >35 years old).Methods: A retrospective observational study including 204 ...

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ea0070aep623 | Pituitary and Neuroendocrinology | ECE2020

Temporal trends in incidence, evaluation and management of neuroendocrine neoplasms of the appendix-15 years’ experience

Twito Orit , Haim Paran , Shmuel Avital , Vladimir Kravtsov , Pnina Rotman-Pikielny , Noa Klein

Introduction: Appendiceal neuroendocrine neoplasms (ANEN) account for approximately 50% of all primary tumors of the appendix and are between the first and third-most frequent gastrointestinal neuroendocrine neoplasms. Data regarding trends in incidence, management and prognosis of ANEN is limited.Aims: To evaluate temporal trends in ANEN incidence, evaluation and management over a 13-year period.Materials and methods: Appendectomy...

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ea0070aep624 | Pituitary and Neuroendocrinology | ECE2020

Contribution of NGS in the genetic analysis of neuroendocrine tumors associated with Multiple Endocrine neoplasia type 1 (MEN 1)

Hamny Illias , Françoise Borson-Chazot , Sophie Giraud

Objective: Next Generation Sequencing technique (NGS) allows the analysis of a whole set of genes in the same time. It is, now, used, in our laboratory for the genetic screening of neuroendocrine tumors associated with Multiple Endocrine neoplasia type 1(MEN1)The objective of the present study was to assess the efficiency of this approach and the relevance of the results regarding the current genetic screening recommendations.Methods: The data of all pat...

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ea0070aep625 | Pituitary and Neuroendocrinology | ECE2020

Malignant insulinoma treated with ¹⁷⁷Lu-DOTATATE: Results in the first year post-therapy

Concepción Terroba-Larumbe María , Maria Palacio-Mures Jose , Crespo-Soto Cristina , Perez-Saborido Baltasar , Crespo-Herrero Guillermo , Ventosa-Viña Marta , Acuña-Garcia Manuel , Alejo-Ramos Mirian , Anacabe-Goyogana Itziar , Cuellar-Olmedo Luis

Introduction: Neuroendocrine tumors (NETs) are rare and their clinical behavior and prognosis correlates with mitotic rate and Ki-67 index. Most patients with advanced NET have liver metastases unresectable and somatostatin analogues are the initial therapy of choice but when disease progresses despite treatment and there are positive somatostatin receptors, peptide receptor radioligand therapy (PRRT), using lutetium-177, is a therapeutic option.Clinical...

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ea0070aep626 | Pituitary and Neuroendocrinology | ECE2020

Surgical and survival outcomes of early peptide receptor radionuclide therapy for downstaging locally advanced or oligometastatic pancreatic neuroendocrine neoplasms

Minczeles Noémie , van Eijck CHJ , van Gils MJ , van Velthuysen MF , Nieveen van Dijkum EJM , Feelders Richard , de Herder Wouter W , Brabander* T , Hofland* Hans

Introduction: Pancreatic neuroendocrine neoplasm (pNEN) patients often present with locally advanced or metastatic disease. The objective response rate of peptide receptor radionuclide therapy (PRRT) in pNENs is 55%. Therefore, PRRT may be a possibility for patients who are not eligible for upfront curative surgery.Aims: To assess the potency of PRRT to render locally advanced or oligometastatic pNENs resectable and to evaluate the effect of surgery afte...

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ea0070aep627 | Pituitary and Neuroendocrinology | ECE2020

Absolute oral bioavailability and absorption, metabolism, excretion of [14C]-Labeled paltusotine (CRN00808), an orally bioavailable, nonpeptide, selective, somatostatin receptor 2 (sst2) biased agonist for the treatment of acromegaly

Madan Ajay , Luo Rosa , Ferrara-Cook Chris , Scott Struthers R. , van Marle Sjoerd , Krasner Alan

Injected depot formulations of somatostatin peptide analogs are routinely used to treat acromegaly and neuroendocrine tumors (NETs). Paltusotine (CRN00808), a small molecule nonpeptide selective somatostatin receptor 2 (sst2) agonist, is being evaluated for efficacy and safety in patients with acromegaly. The current Phase 1 study (NCT04246749) was conducted in two Parts: In Part A, the absorption, metabolism, excretion, and mass balance of a single oral dose of 20 mg [14C]-pa...

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ea0070aep628 | Pituitary and Neuroendocrinology | ECE2020

Safety and IGF-1 levels with once daily oral sst2 agonist paltusotine (CRN00808) in acromegaly patients previously treated with peptide long-acting somatostatin receptor ligands: Initial data from the open label ACROBAT Edge phase 2 study

Toth Miklos , Gordon Murray , Doknic Mirjana , Mezosi Emese , Randeva Harpal , Marmon Tonya , Fowler Kim , Luo Rosa , Monahan Michael , Madan Ajay , Ferrara-Cook Chris , Scott Struthers R. , Krasner Alan

Peptide long-acting somatostatin receptor ligands (SRLs) are a first line medical treatment for acromegaly but are not efficiently absorbed when delivered orally. Years of injections with SRLs are associated with variable dose delivery, injection site reactions, and excessive life burden. Paltusotine (CRN00808) is a nonpeptide small molecule somatostatin type 2 (sst2) receptor agonist with high oral bioavailability (70%) and a 42–50 hour terminal elimination half-life in...

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ea0070aep629 | Pituitary and Neuroendocrinology | ECE2020

The time, mode and markers of pituitary function deterioration in patients with PROP1 mutation. Single centre, longitudinal observation

Gilis-Januszewska Aleksandra , Rogoziński Damian , Kluczyński Łukasz , Godlewska Magdalena , Bogusławska Anna , Piwońska-Solska Beata , Zygmunt-Górska Agata , Wójcik Małgorzata , Starzyk Jerzy , Hubalewska-Dydejczyk Alicja

Introduction: The mode of pituitary function deterioration in patients with PROP1 mutation is not fully known and understood. The function of adrenal axis requires special attention.Aim: To investigate the time/mode/markers of pituitary function deterioration in families/sporadic patients with PROP1 mutation during longitudinal observation.Methods: We performed retrospective longitudinal (36.4 years, s.d. = 13.6) analy...

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ea0070aep630 | Pituitary and Neuroendocrinology | ECE2020

Clinical presentations of patients with MEN 1 syndrome and its phenocopies

Dimitrova Diana , Mamedova Elizaveta , Belaya Zhanna , Melnichenko Galina

Introduction: Multiple endocrine neoplasia type 1 (MEN 1) is a rare, autosomal dominant disease caused by mutations in the MEN1 gene. The syndrome predisposes an individual to the development of primary hyperparathyroidism (PHPT), gastroenteropancreatic neuroendocrine tumors (GEP-NETs), pituitary adenomas (PA), as well as other endocrine and non-endocrine tumors that usually manifest at a young age. If a patient with the MEN 1 phenotype does not carry m...

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ea0070aep631 | Pituitary and Neuroendocrinology | ECE2020

Functional panhypopituitarism of anorexia

Roque Catarina , Sousa Santos Francisco , Sequeira Duarte João

A 30-year-old male was admitted to the neurotraumatology ward after head trauma with cerebral concussion, subdural & subarachnoidal haemorrhage. The Endocrinologist was called to evaluate high urinary debits. The patient was healthy until 4 years before admission. He enrolled on variate patterns of alimentary restriction that resulted in the loss of 28 kg. Since then he had visited multiple doctors (Cardiology, Haemathology, Psychiatry, Gastroenterology, Urology, Endocrino...

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ea0070aep632 | Pituitary and Neuroendocrinology | ECE2020

A case of primary hypophysitis with cavernous sinus infiltration causing an unilateral internal carotid artery occlusion

Czajka-Oraniec Izabella , Stelmachowska-Banas Maria , Majos Agnieszka , Cylke Karolina , Zgliczynski Wojciech

Primary hypophysitis (PH) is a rare clinical entity usually presenting with headaches, pituitary deficiency, visual field defects and exceptionally with symptoms of cavernous sinus infiltration such as cranial nerve palsies. An internal carotid artery (ICA) occlusion is unusual complication of PH infiltrating the cavernous sinus. A 32-year old woman presented with a 4-year history of recurring headaches, right retro-orbital pain, periodic diplopia and secondary adren...

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ea0070aep633 | Pituitary and Neuroendocrinology | ECE2020

ACTH-secreting pituitary tumor with delayed aggressive clinical recurrence – management challenges

Gaita Isabela , Cristescu Alexandru , Enculescu Augustina , Poiana Catalina , Radian Serban

Background: Pituitary adenomas (PA) are slow-growing, benign intracranial tumors. Rarely, they can be histologically and clinically aggressive.Aim: To present a case of corticotroph PA with aggressive behavior following a long indolent interval after the initial surgery.Case presentation: A 47 years old female presented in Nov 2007 with right temporal hemianopsia and left eye blindness, associating type 2 DM and severe mixed dyslip...

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ea0070aep634 | Pituitary and Neuroendocrinology | ECE2020

Unusual case of women suffering from aggressive silent thyrotropinoma treated with somatostatin analogues during pregnancy

Zgliczynska Magdalena , Majos Agnieszka , Stelmachowska-Banas Maria , Zgliczynski Wojciech

Background: Thyroid-stimulating hormone (TSH) expressing tumors are one of the rarest pituitary adenomas and comprises 0.5–2% of all cases. To our knowledge, only four cases of thyrotropinoma in a pregnant women have been reported up to date.Case presentation: We present a case of 24-year old women who reported to gynecologist due to secondary amenorrhea and severe headaches. Commissioned laboratory test showed, among others, prolactin 31.0 ng/ml a...

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ea0070aep635 | Pituitary and Neuroendocrinology | ECE2020

Secondary amenorrhea, acute hydrocephalus and syndrome of inappropriate secretion of antidiuretic hormone as first manifestations of neurosarcoidosis

Zargni Asma , Oueslati Ibtissem , Mehrez Aymen Ben , Kamoun Elyes , Amor Zohra Ben , Nagi Sonia , Zouaghi Karim , Chihaoui Melika

Introduction: Sarcoidosis affects both the central and peripheral nervous system in 5–16% of patients. In most cases, such involvement occurs within a multi-systemic disease. Endocrine manifestations of neurosarcoidosis can be expressed by hypothalamic dysfunction, diabetes insipidus, hypopituitarism, hyperprolactinemia, in isolated fashion or variedly combined.Herein, we report a case of neurosarcoidosis revealed by an obstructive hydrocephalus, a...

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ea0070aep636 | Pituitary and Neuroendocrinology | ECE2020

Complicated endocrinopathy after liver transplantation for unusual diagnosis

Havrdova Tereza , Gottfriedova Halima , Brunová Jana

Introduction: Langerhans cell histiocytosis (LCH) is a very rare haematological disease, with difficult diagnosis for its very variable clinical picture. It is caused by monoclonal proliferation of cutaneous Langerhans cells. It affects most commonly the skin and bones, less often bone marrow, liver, pituitary and CNS.Case report: A 74-year-old female patient with progressive sclerosing cholangitis was referred to a transplant centre to consider liver tr...

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ea0070aep637 | Pituitary and Neuroendocrinology | ECE2020

A rare association of hypogonadotropic hypogonadism and GH deficiency in a patient with Addison’s disease

Chatti Hiba-Allah , Oueslati Ibtissem , Yazidi Meriem , Khessairi Nadia , Grira Wafa , Chaker Fatma , Chihaoui Melika

Introduction: Primary adrenal insufficiency is a classically rare but potentially serious disease due to the risk of acute adrenal crisis. Although autoimmune origin is the first etiology in adults, genetic causes are most common in children.Herein, we reporta case of coexisting hypogonadotropic hypogonadism and growth hormone (GH) deficiency in a patient with Addison’s disease.Observation: A 15-year-old boy was referred to o...

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ea0070aep638 | Pituitary and Neuroendocrinology | ECE2020

46XY DSD as initial clinical presentation in a patient with syndromic combined pituitary hormones deficiency

Lozovanu Vera , Emanuela Georgescu Carmen , Oana Irimia Ramona , Alina Silaghi Cristina

Background: Combined pituitary hormone deficiency (CPHD) is characterized by multiple pituitary hormone deficiencies, due to mutations of pituitary transcription factors involved in pituitary ontogenesis. The genetic basis for CPHD is complex, involving a variety of syndromic and non-syndromic presentations with variable degrees of phenotype-genotype correlations. In male infants with CPHD, gonadotropin deficiency is suggested by the presence of a microphallus and undescended ...

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ea0070aep639 | Pituitary and Neuroendocrinology | ECE2020

Clinical and hormonal features of congenital hypopituitarism

Kamoun Elyes , Oueslati Ibtissem , Chehida Anaam Ben , Khessairi Nadia , Yazidi Meriem , Grira Wafa , Chaker Fatma , Chihaoui Melika

Introduction: Congenital hypopituitarism is a rare disease induced by mutations in transcription factors involved in the pituitary development or ubiquitous transcription factors. It can be associated with a malformative syndrome. We herein describe clinical and hormonal features of congenital hypopituitarism in two Tunisian families.Observation 1: We report the case of three sisters, born at term to healthy consanguineous parents. The index case was ref...

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ea0070aep640 | Pituitary and Neuroendocrinology | ECE2020

Looking for cyclic cushing’s syndrome

Elena Lopez Alaminos Maria , Gonzalez Fernandez Mikel , Almendra Alvarado Rosas Karol , Serrano Urzaiz Leticia , Martínez García María , Trincado Pablo , de Diego Garcia Patricia

Introduction: Cyclic Cushing´s syndrome (CCS) is a rare disorder with a wide clinical variability, characterized by repeated episodes of cortisol excess and periods of normal or even low cortisol secretion. Different periodical diagnostic tests are required to confirm hypercortisolism and thereafter imaging techniques and laboratory studies can be performed to localize the cause.Case report: A 17-year-old female patient with a personal history of ad...

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ea0070aep641 | Pituitary and Neuroendocrinology | ECE2020

Medical treatment leading to remission of ectopic cushing’s syndrome

Seguna Desiree , Shiafkou Marianna , Sahdev Anju , Al-Mrayat Ma’en , Akker Scott

A 35 year-old lady presented to her local hospital with a 3 year history of fatigue, weight gain, recurrent tonsillitis and oligomenorrhoea. Examination revealed facial plethora, round facies and thin skin on the dorsum of the hand. She appeared tanned, with evidence of spontaneous bruising and difficulty standing up from a chair unaided. No striae were present. Blood pressure was 152/91 mmHg. Investigations revealed a 0900 h cortisol of 632 nmol/l (ACTH 59 ng/l) and an elevat...

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ea0070aep642 | Pituitary and Neuroendocrinology | ECE2020

An uncommon cause of atrial fibrillation

Llaro Mary , Martinez Ana , Quiroga Ivan , Parra Ramírez Paola , Moya Elisa , Blanco Benito

Thyrotropin-secreting pituitary adenomas (TSHomas) are rare functioning pituitary adenomas, accounting for less than 1%. They commonly present as macroadenomas with symptoms of hyperthyroidism. Though surgery is the first line therapy, cure is achieved in only 30–40%, with high rate of post-surgical hypopituitarism. We propose somatostatin analogs having a new role in the management. We report a case of 73 years-old man with a 6-month history of palpitations and progress...

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ea0070aep643 | Pituitary and Neuroendocrinology | ECE2020

Osmotic demyelination syndrome in a patient with noonan syndrome and anterior hypopituitarism

Harn Chua Tzy , Jia Loh Wann

Introduction: Management of severe hyponatremia is challenging because of the need to balance the risk of overcorrection leading to osmotic demyelination syndrome (ODS) as well as under-correction causing complications, particularly in a patient with hypocortisolism and hypothyroidism. Case reports of patients with Noonan syndrome with hypopituitarism are rarely reported in literature. We report a case of a patient with Noonan syndrome and untreated anterior hypopituitarism wh...

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ea0070aep644 | Pituitary and Neuroendocrinology | ECE2020

A case of developmental delay by 18q23 deletion syndrome

Chaves Catarina , Martinho Mariana , Brandão Carla , Rodrigues Catarina , Cunha Filipe , Garrido Susana , Vieira Margarida , Almeida Margarida

Introduction: Monosomy 18q represents a partial deletion of the long arm of chromosome 18, with an estimated prevalence of 1:100 000. This syndrome is characterized by a highly variable phenotype. The symptoms and their severity depend on which part of the chromosome is missing. Most common manifestations are hypotonia, developmental delay, short stature, growth hormone deficiency, hearing loss and external ear anomalies, intellectual disability, palatal defects, dysmorphic fa...

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ea0070aep645 | Pituitary and Neuroendocrinology | ECE2020

Sodium copeptin ratio differentiates patients with and without acute diabetes insipidus post neurosurgery

chiang Cherie , Boehm Emma , Sherfan Julie , Perera Nimalie , Wentworth John , King James

Background: The diagnosis of central diabetes insipidus (DI) relies on indirect measurement of serum/urine sodium and osmolality. Since the diagnosis can only be made when an inappropriately dilute urine is paired with a significantly concentrated serum, the process is tedious for the clinician and uncomfortable for the patient. Copeptin is the C-terminal portion of the anti-diuretic hormone (ADH) prohormone which correlates with the less stable ADH, therefore providing a dire...

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ea0070aep646 | Pituitary and Neuroendocrinology | ECE2020

Differences in acquired and idiopathic adult-onset growth hormone deficiency

Yee Cheng Boey , Gill Lizy , Brown Sarah , Palau Isabel , Allie Nicole

Introduction: Growth hormone deficiency (GHD) is a medical condition affecting adults and children who lack sufficient growth hormone. The objective of this study was to compare diagnoses in acquired and idiopathic adult-onset GHD patients.Methodology: Results are from Ipsos’ EU5 GHD Therapy Monitor Study 2018, which is an online study conducted among physicians treating both paediatric and adult GHD patients. 156 sampled physicians provided d...

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ea0070aep647 | Pituitary and Neuroendocrinology | ECE2020

Evaluation of the of alpha2-adrenergic receptors stimulation effect on copeptin secretion, based on the result of the test with clonidine used in the diagnosis of children with short stature

Stawerska Renata , Kolasa-Kicińska Marzena , Krygier Adrian , Hilczer Maciej , Lewinski Andrzej

Introduction: Copeptin can be used as a tool to directly assess serum antidiuretic hormone (ADH) level. A certain pool of ADH are synthesized in hypothalamus, together with corticoliberin (CRH). Then they are released into the pituitary portal circulation, where through the V1bR receptor ADH stimulates adrenocorticotropic cells and ACTH secretion, followed by cortisol and catecholamines production. The stimulus for ADH secretion is, among others, stress. In turn, presynaptic s...

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ea0070aep648 | Pituitary and Neuroendocrinology | ECE2020

Typical carcinoid of the nose: The role of somatostatin agonist following surgery

Pazos Fernando , Obeso Sergio

Carcinoid tumors (CT) seldom develop in the head and neck area. Several authors doubt the existence of authentic neuroendocrine tumors in the nose. Most of the CT arise in the larynx.Clinical case: A 54 years old woman, who had a right-sided nasal obstruction, rhinorrhea, and sometimes purulent discharge in 2016. In 2002 she had hyperthyroidism by Graves disease and treated for two years with remission after that. The examination of the nose revealed a d...

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ea0070aep649 | Pituitary and Neuroendocrinology | ECE2020

Multi-hormonal secretion in a young male with a large cystic pancreatic neuroendocrine neoplasm

Boutzios Georgios , Koukoulioti Eleni , Garmpis Nikolaos , Giovannopoulos Ilias , Bourgalis-Michalopoulos Konstantinos , Mantas Dimitrios

Introduction: The molecular pathogenesis of pancreatic neuroendocrineneoplasms (PNEN) is increasingly yet incompletely understood. PNEN typically occur sporadically or may occur within the context of hereditary tumor predisposition syndromes. PNEN can also be associated with endocrinopathy of hormonal hypersecretion. We present a young male with a multi-hormonal neuroendocrine neoplasm of pancreas.Case presentation: A 24-year-old male, non-smok...

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ea0070aep650 | Pituitary and Neuroendocrinology | ECE2020

Differentiation of Insulinoma from accessory spleen by 99mTc-labelled heat-denaturated red blood cell scintigraphy

Gezer Emre , Çetinarslan Berrin , Karakaya Dilek , Cantürk Zeynep , Selek Alev , Sözen Mehmet , İșgören Serkan

Introduction: Neuroendocrine tumors (NETs) are rare tumors harboring overexpression of somatostatin receptors (SSTRs) on their cell membrane. Gallium 68-tetraazacyclododecane tetraacetic acid-octreotate (68Ga-DOTATATE) positron emission tomography/computed tomography (PET/CT) is an important imaging modality in diagnosis and staging of NETs. Because some organs such as spleen, adrenal glands and liver physiologically express SSTR, it might be challenging to distinguish some pa...

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ea0070aep651 | Pituitary and Neuroendocrinology | ECE2020

Predictors of Cushing’s disease remission after transsphenoidal endoscopic surgery

Kuritsyna Natalia , Tsoy Uliana , Cherebillo Vladislav , Paltsev Artem , Tsiberkin Alexandr , Vasilieva Elena , Grineva Elena

Purpose: The postoperative levels of cortisol and ACTH are the most commonly used markers for the prediction of Cushing’s disease (CD) remission after transsphenoidal surgery (TSS). However, there is still no consensus on the collection time and threshold values of basal cortisol and ACTH levels after surgery to predict remission of CD.Aim: To study the significance of early postoperative serum cortisol and plasma ACTH testing in the prognosis of C...

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ea0070aep652 | Pituitary and Neuroendocrinology | ECE2020

Acrodat – a useful tool to manage acromegaly patients

Alves Helena , Almeida Ferreira Marta , Tavares Patrícia , Ramalho Diogo , Machado Catarina , Almeida Lucia , Correia Sara , João Oliveira Maria

Introduction: Acromegaly is a rare condition associated with metabolic abnormalities, risk of cardiovascular complications and increased mortality. Even when biochemical control is achieved, following surgery or medical treatment, patients still complain about disease-related symptoms.Material and Methods: Retrospective study of acromegaly patients, followed in an Endocrine outpatient clinic. Clinical and biochemical data was recorded. ACRODAT was used t...

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ea0070aep653 | Pituitary and Neuroendocrinology | ECE2020

Characterization of pituitary adenomas by immunohistochemistry of pituitary-specific transcription factors and their correlation with hormonal subtypes

García-Martínez Araceli , Silva-Ortega Sandra , López-Muñoz Beatriz , Moreno-Pérez Óscar , Monjas Irene , Abarca Javier , Picó Antonio , Aranda Ignacio

Introduction: The immunohistochemical characterization of Pit-1, Tpit and SF-1 transcription factors allows the identification of the three adenohypophyseal cell lines and has been incorporated into the latest WHO classification of pituitary adenomas (PA). The aim of the present study was to quantify the protein expression of pituitary-specific transcription factors (TF) by immunohistochemistry (IHC) and to correlate these results with the identification based on hormonal prot...

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ea0070aep654 | Pituitary and Neuroendocrinology | ECE2020

Our experience with lu-dotatate in patients with neuroendocrine tumors in a tertiary hospital

Munoz Diego , Carlos Percovich Hualpa Juan , Weber Bettina , Atencia Goñi José , González Fernández Laura , Miguélez González María , Brox Torrecilla Noemí , José Añez Ramos Roberto , Maricel Rivas Montenegro Alejandra , López Guerra Aurelio , Lucía Olmedilla Isishi Yoko , Ángeles Vélez Romero María , Ágreda García Javier , Picallo Pérez María , Monereo Megías Susana , García Centeno Rogelio

Introduction: Lutetium-177-DOTATATE (Lu177) is approved in patients with well-differentiated (G1 and G2) neuroendocrine tumors (NETs) positive to somatostatin receptors, progressive and unresectable or metastatic. Lu177 has been shown to increase progression-free survival and the quality of life of these patients.Material and Methods: Retrospective descriptive study of a series of 15 cases with NETs treated with Lu177. Demographic data, tumor characteris...

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ea0070aep656 | Pituitary and Neuroendocrinology | ECE2020

Characteristics of severe forms of Cushing’s disease compared to milder cases

Mourelatos Panagiotis , Mitravela Vasiliki – Ioanna , Balomenaki Maria , Margaritopoulos Dimitris , Diamantopoulos Aristeidis , Kanellopoulou Sofia , Vassiliadi Dimitra , Tsagarakis Stylianos

Introduction: Cushing’s disease (CD) is associated with significant morbidity and mortality due to cerebro-cardiovascular, thrombotic or infectious complications. However, there is significant variation in the clinical presentation and consequences of hypercortisolism, resulting in a wide clinical spectrum, ranging from mild to severe or even life-threatening disease requiring immediate treatment.Aim: The description of patients with severe CD in c...

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ea0070aep657 | Pituitary and Neuroendocrinology | ECE2020

The impact of variations in laboratory measurements of IGF-1 and random growth hormone on the classification of acromegaly disease activity status: Lessons from the UK Acromegaly Register Reference Laboratory

Bashir Bilal , Adam Safwaan , Monaghan Philip , Plummer Zoe , Archer Natasha , Ayuk John , Trainer Peter

Background: The UK Acromegaly Register contains data for 2700 patients. IGF-1 and random growth hormone (GH) measurements are used for disease monitoring. The registry reference laboratory (RRL) uses the Immunodiagnostic systems (IDS)-iSYS immunoassay platform for GH and IGF-1. The RRL uses age- and sex-specific reference ranges for IGF-1. We compared IGF-1 and GH results fromlocal laboratories to those of the RRL (pre-defined as the Gold standard) to determine the extent of d...

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ea0070aep658 | Pituitary and Neuroendocrinology | ECE2020

Prevalence of non-alcoholic fatty liver disease in patients with Cushing disease (CD)

Remon Pablo , Venegas-Moreno Eva , Deniz-Garcia Alejandro , Millan Raquel , Dios-Fuentes Elena , Martin Natalia , Rico Maria C , Madrazo Ainara , Romero-Gómez Manuel , Soto-Moreno Alfonso

Introduction: The prevalence and comorbidities of NAFLD in CD are unknown. There is only one study wich set the prevalence of NAFLD by CT images in 20%1.Objectives: Describe the prevalence of NAFLD and fibrosis in CD and analize predictive biomarkers of NAFLDMethods: Transversal descriptive study. 31 patients with CD; 11 active, 20 cured. Women 24 (77%), men 7 (23%). 54 years mean age. NAFLD was valorated by Hepatic Stea...

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ea0070aep659 | Pituitary and Neuroendocrinology | ECE2020

Tumour progression in clinically non-functioning pituitary adenomas- does somatostatin analogue therapy decrease the risk?

Zawada Natalia

Introduction: The management of clinically non-functioning pituitary adenomas (NFPA) remains a debated issue. Surgery, which is indisputably indicated in invasive NFPA, is rarely curative. Moreover, it is not always feasible due to potential complications or contraindications. Expression of somatostatin receptors (SSTR) form the rationale for the use of somatostatin analogues (SSA) in NFPA.Aim: To compare the risk of NFPA progression in patients treated ...

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ea0070aep660 | Pituitary and Neuroendocrinology | ECE2020

Predictors for remission after transsphenoidal surgery in acromegaly: A dutch multicenter study

Coopmans Eva , Postma Mark , Wolters Thalijn , van Meyel Sebastiaan , Teodora Netea-Maier Romana , Neggers Sebastian , van Beek Andre

Context: Transsphenoidal surgery (TSS) is the primary treatment of choice in acromegaly. It is important to identify patients in whom surgical cure is not attainable at an early stage, both to inform patients on expected treatment outcome and to select those who are more likely to need additional therapy.Objective: To identify predictors for remission and relapse after TSS in acromegaly.Design: Retrospective study in three large te...

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ea0070aep661 | Pituitary and Neuroendocrinology | ECE2020

Metyrapone treatment in endogenous Cushing’s syndrome. Results from a prospective multicenter, open-label, phase III/IV study: Prompt

Nieman Lynnette , Boscaro Marco , Carla Scaroni , Deutschbein Timo , Mezosi Emese , Driessens Natacha , Emanuela Georgescu Carmen , Hubalewska-Dydejczyk Alicja , Berker Dilek , Jarzab Barbara , Maiter Dominique , Reincke Martin , Loli Paola , Zampetti Benedetta , Atmaca Aysegul , Badiu Corin , Beckers Albert , Bolanowski Marek , Cavagnini Francesco , Unger Nicole , Giordano Roberta , Hanzu Felicia , Terzolo Massimo , Bostnavaron Martine , Marsault Pauline , Toth Miklos

Metyrapone treatment in endogenous Cushing’s syndrome. Results from a prospective multicenter, open-label, phase III/IV study: PROMPTBackground: Metyrapone is a steroidogenesis inhibitor approved in Europe for the treatment of endogenous Cushing’s syndrome (CS) based on observational retrospective studies published over more than 50 years. We present data from the first prospective study designed to confirm metyrapone efficacy and good tolera...

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ea0070aep662 | Pituitary and Neuroendocrinology | ECE2020

Predictors of response to medical therapy with pegvisomant and pasireotide lar in SRLs-resistant acromegaly

Chiloiro Sabrina , Giampietro Antonella , Bianchi Antonio , Mirra Federica , Donfrancesco Federico , Maya Fleseriu Cara , Lauretti Liveraba , Anile Carmelo , Gessi Marco , Rindi Guido , Pontecorvi Alfredo , Fleseriu Maria , Giustina Andrea , De Marinis Laura

Background: Pegvisomant(PegV) and Pasireotide LAR(Pasi) are commonly used in acromegaly patients resistant to first-generation SRLs. Predictors of response to therapy with PegV and Pasi in this subset of patients are still unclear. These findings may be useful in choosing the most appropriate therapeutic option for the personalized treatment of patients affected by more aggressive disease. We aimed to identify individual predictors of responses.Methods: ...

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ea0070aep663 | Pituitary and Neuroendocrinology | ECE2020

Efficacy of monotherapy versus combined GH receptor antagonist therapy in patients with somatostatin receptor analogues resistant acromegaly

Simona Andreea Găloiu , Margarit Emma , Lavinia Popa Maria , Baciu Ionela , Niculescu Dan , Trifanescu Raluca , Serban Radian , Dumitrașcu Anda , Caragheorgheopol Andra , Poiana Catalina

Background and Aims: GH receptor antagonist (Pegvisomant –PEG) has been shown to obtain IGF1 normalization in 60–90% of patients with acromegaly in clinical trials. The aim of the study is to evaluate real life response and side effects of PEG treatment in monotherapy versus combined with somatostatin analogues and/or cabergoline in patients with somatostatin analogues resistant acromegaly.Methods: We included 40 patients (22F/18 M) cons...

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ea0070aep664 | Pituitary and Neuroendocrinology | ECE2020

The role of repeated prolactin samples in patients with hyperprolactinaemia

Rodrigues Catarina , Cunha Filipe , Chaves Catarina , Silva Vieira Margarida , Silva André , Garrido Susana , Martinho Mariana , Almeida Margarida

Introduction: Current guidelines recommend a single prolactin sampling for the diagnosis of hyperprolactinaemia. Nonetheless, in some patients, prolactin levels may normalize in a subsequent sampling or if prolactin is collected through a venous catheter some time after puncture. We aimed to assess the percentage of patients in which prolactin remained elevated in repeated sampling and to determine the best prolactin cutoff associated with persistent hyperprolactinaemia.<p...

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ea0070aep665 | Pituitary and Neuroendocrinology | ECE2020

Assessment of disease control in patients with acromegaly treated with long-acting somatostatin analogs (SMSa) varies according to the time when IGF-I levels are measured during the month following the injection

Cristina Albrici , Lecoq Anne-Lise , Vialon Magaly , Solange Grunenwald , Cocco Aldo , Hamdi Safouane , Vincenzo Cimino , Maione Luigi , Caron Philippe , Chanson Philippe

Introduction: Acromegaly is associated with multiple comorbidities and excess mortality. However, disease burden is reduced by maintaining IGF-1 (and/or GH) levels under safe levels. First generation long-acting SMSa, administered at monthly intervals, represent the first line medical treatment. According to guidelines, its efficacy is evaluated through IGF-I measurements. However, there are no data indicating the optimal time for measuring IGF-I levels after the SMSa injectio...

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ea0070aep666 | Pituitary and Neuroendocrinology | ECE2020

Surgical and clinical outcome in elderly patients affected by non-functional pituitary adenoma treated by endoscopic transnasal transsphenoidal surgery: A monocentric study

Biamonte Emilia , Milani Davide , Lavezzi Elisabetta , Betella Nazarena , Cellini Miriam , Battista Lasio Giovanni , Mazziotti Gherardo , Lania Andrea

The rate of diagnosis of pituitary adenomas in the elderly is increasing, as a consequence of the improved life expectancy. The management of elderly patients affected by pituitary adenomas are still a subject of debate in literature. In particular, data on the safety of endoscopic transnasal transsphenoidal surgery (TNS) are still controversial. The primary endpoint of the present study is to assess the surgical outcome of a cohort of patients who underwent TNS for non-functi...

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ea0070aep667 | Pituitary and Neuroendocrinology | ECE2020

Gender differences in airway obstruction in men and women with acromegaly: A longitudinal study

Störmann Sylvère , Schilbach Katharina , Amereller Felix , W Angstwurm Matthias , Schopohl Jochen

Introduction: Acromegaly is a slowly progressing systemic disease that leads to numerous changes in metabolism and organ function. A large cross-sectional study showed signs of subclinical airway obstruction in female patients. We performed a longitudinal study of lung function testing to compare parameters of airway obstruction in male and female patients with acromegaly over time.Methods: 40 patients with acromegaly (24 male, 16 female) underwent repea...

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ea0070aep668 | Pituitary and Neuroendocrinology | ECE2020

Withdrawal from long-acting somatostatin receptor ligand injections in adult patients with acromegaly: Results from the phase 3, randomized, double-blind, placebo-controlled optimal study

Nachtigall Lisa , Samson Susan , Fleseriu Maria , Bolanowski Marek , E Molitch Mark , Ludlam William , Patou Gary , Haviv Asi , Manning Patrick , Biermasz Nienke , Giustina Andrea , Strasburger Christian J , Melmed Shlomo , Kennedy Laurence

Background: Data on the impact of withdrawal from long-acting somatostatin receptor ligand (SRL) injections on disease activity in patients with acromegaly are limited. The CHIASMA OPTIMAL study assessed the efficacy and safety of oral octreotide capsules (OOC) in adult patients with acromegaly responding to SRL injections. The placebo arm of this study allowed for assessment of acromegaly activity in patients after withdrawal from SRL treatment.Methods:...

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ea0070aep669 | Pituitary and Neuroendocrinology | ECE2020

Impact of imputation method on efficacy results from the phase 3 optimal study of oral octreotide capsules in adult patients with acromegaly

Samson Susan , Nachtigall Lisa , Fleseriu Maria , Gordon Murray , Jensterle Mojca , Elenkova Atanaska , Molitch Mark E , Ludlam William , Patou Gary , Haviv Asi , Biermasz Nienke , Trainer Peter , Strasburger Christian J , Kennedy Laurence , Melmed Shlomo

Objective: The phase 3 CHIASMA OPTIMAL study assessed efficacy and safety of oral octreotide capsules (OOC) in patients with acromegaly controlled on injectable somatostatin receptor ligands (SRLs). Sensitivity analyses were conducted for efficacy endpoints using two methods of imputation (i.e., the process of replacing clinical data with substitution values) to address missing data points due to some subjects reverting back to their prior injectable SRL treatment.<p class...

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ea0070aep670 | Pituitary and Neuroendocrinology | ECE2020

Eucaloric very low-carbohydrate ketogenic diet as a new supportive treatment modality for acromegaly?

Coopmans Eva , Berk Kirsten AC , El-Sayed Nour , Neggers Sebastian , Van der Lely Aart Jan

Objective: Acromegaly is caused by a growth hormone (GH)-secreting pituitary tumor and its signs are linked to increasedGH-dependent hepatic insulin-like growth factor I (IGF-I) synthesis. A eucaloric very low-carbohydrate ketogenic diet (euVLCK; < 50 g/day) induces ketosis and reduces portal insulin concentrations, which down-regulate hepatic GH receptors and reduce IGF-I synthesis. Somatostatin receptor ligands (SRLs) reduce GH secretion by the pituitary tumor,resul...

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ea0070aep671 | Pituitary and Neuroendocrinology | ECE2020

To score or not to score? Is Ki-67 analysis worthwhile in pituitary neuroendocrine tumours?

Benjamin Loughrey Paul , Craig Stephanie , Herron Brian , Abdullahi Sidi Fatima , McQuaid Stephen , Kelly Paul , Humphries Matt , Parkes Eileen , McArt Darragh , Hunter Steven , Korbonits Marta , James Jacqueline

Pituitary neuroendocrine tumours (PitNETs) are heterogeneous and have limited biomarkers to predict their behaviour, thus making their prognostication difficult. Ki-67 is a protein expressed in active phases of the cell cycle and is one of the biomarkers utilized in routine assessment of PitNET tissue. Current European Society of Endocrinology recommendations advise that histopathological analysis of PitNETs should as a minimum include Ki-67 proliferation index and anterior pi...

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ea0070aep672 | Pituitary and Neuroendocrinology | ECE2020

Giant prolactinomas: Is it possible to stop treatment?

Mallea-Gil Susana , Diez Sabrina , Tubert Gloria , Cuccia Mariela , Rodriguez Noelia , Stalldecker Graciela , de los Angeles Sosa Maria , Peressotti Bruno , Palazzo Adriana , Ballarino Carolina

Giant prolactinomas are tumors with a large size >4 cm and/or prolactin levels higher than 3000 ng/ml and/or highly invasive growth. Today dopamine agonists (DA), mainly cabergoline (CAB), are the first-line choice in the treatment of these tumorsTo assess the efficacy, safety and long-term follow-up of patients with giant prolactinomas treated with DA. We retrospectively reviewed the clinical records of 33 patients with giant prolactinomas. Mea...

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ea0070aep673 | Pituitary and Neuroendocrinology | ECE2020

Development of a local reference range for hypertonic saline-stimulated copeptin

Brooks Emily , Bachmeier Caroline , Vorster Juanita , Sorbello Jane , Peer Faseeha , Chikani Viral , Dimeski Goce , Ungerer Jacobus , Pretorius Carel , Inder Warrick J

Hypertonic saline stimulated copeptin measurements have recently been described for the diagnosis of central DI. A copeptin cut-off of > 4.9 pmol/l has a diagnostic accuracy of 96.5% for distinguishing primary polydipsia from central DI1. A copeptin assay has recently been established in our laboratory. Validation of hypertonic saline-stimulated copeptin concentrations in our local population is needed before this test can be used with confidence in patients pre...

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ea0070aep674 | Pituitary and Neuroendocrinology | ECE2020

A key role for conservative treatment in the management of pituitary apoplexy

Marx Claire , Rabilloud Muriel , Borson-Chazot Francoise , Tilikete Caroline , Jouanneau Emmanuel , Raverot Gerald

Objective: The management of pituitary apoplexy, a rare emergency neuroendocrine condition, is controversial. The aim of the present study was to compare the outcome of patients with pituitary apoplexy managed either by a conservative or surgical approach.Methods: A retrospective cohort study including patients diagnosed between 2007 and 2018 in a tertiary French university hospital. Pituitary apoplexy score was retrospectively applied in a perspective o...

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ea0070aep675 | Pituitary and Neuroendocrinology | ECE2020

Gender dimorphism of intramuscular fatty infiltration and related muscle dysfunction in patients with long-term control of acromegaly

Martel-Duguech Luciana , Alonso-Pérez Jorge , Bascuñana Helena , Diaz-Manera Jordi , Alonso-Jimenez Alicia , Llauger Jaume , Nuñez-Peralta Claudia , Montesinos Paula , Webb Susan M. , Valassi Elena

Background: Muscle weakness persists in patients with acromegaly (ACRO) even long-term after disease control. Mechanisms determining this sustained impairment are not known. We hypothesized that alteration of muscle architecture, due to intramuscular fatty infiltration, is associated with muscle dysfunction in these patients.Patients and Methods: Thirty-seven acromegalic patients [21 females and 16 males, mean (± s.d.)age, 53 ± 9 years...

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ea0070aep676 | Pituitary and Neuroendocrinology | ECE2020

European Society of Endocrinology audit and multi-country comparison of Adult Growth Hormone Deficiency (AGHD) treatment in clinical practice in Europe and Australia; –how closely are protocols and best practice recommendations followed

Martel-Duguech Luciana , Screen Nik , Jorgensen Jens Otto L , Korbonits Marta , Johannsson Gudmundur , Webb Susan M

Background: Current guidelines recommend that susceptible patients should be tested and treated for AGHD. Still, it is not universally recognised as a distinct entity and reimbursement of GH replacement therapy is not available in some countries.Aim: 1) to record current practice of AGHD management throughout Europe and benchmark it against existing guidelines, 2) to evaluate educational status of health care professionals.Patients...

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ea0070aep677 | Pituitary and Neuroendocrinology | ECE2020

Carpal tunnel syndrome is common in patients with undiagnosed acromegaly – a swedish nationwide study

Vouzouneraki Konstantina , Esposito Daniela , Mukka Sebastian , Granfeldt Daniel , Ragnarsson Oskar , Dahlqvist Per , Olsson Daniel S

Background: Carpal tunnel syndrome (CTS) is common in patients with acromegaly, with a reported prevalence of 19–64%. We have studied CTS in a large national cohort of patients with acromegaly, and the temporal relationship between the diagnosis of the two diseases.Methods: Retrospective nationwide cohort study including patients diagnosed with acromegaly in Sweden between 2005 and 2017. Patients with acromegaly were identified in the Swedish Natio...

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ea0070aep678 | Pituitary and Neuroendocrinology | ECE2020

Levoketoconazole in the treatment of endogenous Cushing’s syndrome: Extended evaluation phase results of the SONICS study

Fleseriu Maria , Auchus Richard J , Greenman Yona , Zacharieva Sabina , Geer Eliza B , Salvatori Roberto , Pivonello Rosario , Feldt-Rasmussen Ulla , Kennedy Laurence , Buchfelder Michael , Biller Beverly M K , Cohen Fredric , Heaney Anthony P

Background: The phase 3, open-label SONICS study of levoketoconazole demonstrated sustained reduction in mean urinary free cortisol (mUFC) in adults with endogenous Cushing’s syndrome (CS). The extended evaluation phase (Ext) of SONICS reported here further assessed long-term safety, tolerability, and benefit/risk of this treatment.Methods: SONICS consisted of dose-titration (150 – 600 mg BID to attain maximally tolerated dose for mUFC normal...

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ea0070aep679 | Pituitary and Neuroendocrinology | ECE2020

Role of NGS in the diagnostic work-up of pituitary tumors and ‘incidental findings’

Del Sindaco Giulia , Sala Elisa , Carosi Giulia , Cremaschi Arianna , Mungari Roberta , Liliana Serban Andreea , Mantovani Beatrice , Indirli Rita , Ferrante Emanuele , Persani Luca , Arosio Maura , Mantovani Giovanna

Background: Pituitary tumors are mostly sporadic, but in less than 5% of cases they can be associated to genetic syndromes, so harbouring germline mutations. Familial pituitary tumors are often more aggressive, so it’s important to detect them, for both a better early diagnosis and genetic counselling. Before the development of Next-Generation Sequencing (NGS), Sanger sequencing was the most widely used method of DNA sequencing. Therefore, DNA samples were analysed follo...

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ea0070aep680 | Pituitary and Neuroendocrinology | ECE2020

fMRI for cognitive evaluation of adult survivors of childhood craniopharyngioma

Svärd Daniel , Marie Erfurth Eva , Fjalldal Sigridur , Hellerstedt Robin , Mannfolk Peter , Mårtensson Johan , Follin Cecilia

Introduction: The morbidity of craniopharyngioma (CP) includes cognitive dysfunction with attention deficits, impaired episodic memory and processing speed (Fjalldal et al., 2013) and CP patients with hypothalamic damage are more affected. fMRI is a technique used to investigate cortical activity by detecting alterations in blood flow in response to stimuli or actions. Previously pre- and post-meal fMRI responses to visual food cues were investigated showing higher ac...

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ea0070aep681 | Pituitary and Neuroendocrinology | ECE2020

Effects of long-term growth hormone replacemente therapy on body composition in adult patients with growth hormone deficiency

María García Moreno Rosa , Parra Ramírez Paola , Baonza Saiz Gonzalo , Martin Rojas-Marcos Patricia , Lecumberri Beatriz , Álvarez Escolá Cristina

Introduction: Growth hormone deficiency (GHD) is associated with worsening body composition and growth hormone replacement therapy (GHRT) has shown to improve it in several studies. The objectives of this study were to evaluate the effects of long-term GHRT on body composition in adult patients with GHD andassess, in this population, the agreement between two different methods to measure fat mass that are total body fat mass (FM) and relative fat mass (RFM).<p class="abste...

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ea0070aep682 | Pituitary and Neuroendocrinology | ECE2020

Perception of stature and quality of life in young adult men referred for tall stature during adolescence. Perception of stature and quality of life in young adult men referred for tall stature during adolescence.

De Schepper Jean , Wynand Justine , De Waele Kathleen , Van der Straaten Saskia , Cools Martine , Den Brinker Marieke , Dotrement Hilde , Vanbesien Jesse , Gies Inge

Background and aims: Little is known about the long-term psychosocial effect of tallness and its treatment in male adolescents.1 Therefore, psychosocial outcome, including adult coping with tall stature were investigated in young adult men, who had consulted for tall stature during adolescence.Methods: All adult men (age >18 years) who had been referred for tall stature or rapid growth during adolescence (ages 11 to 16 years) between 2010 ...

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ea0070aep683 | Pituitary and Neuroendocrinology | ECE2020

Antitumor effects of growth hormone-releasing hormone (GHRH) antagonists in ACTH- and GH-secreting pituitary neuroendocrine tumor cell lines

Fanciulli Alessandro , Gesmundo Iacopo , Gamba Giacomo , Maria Clara Alvarez Villamarin , Dieguez Carlos , Zatelli Chiara , Cannavò Salvatore , Grottoli Silvia , Arvat Emanuela , Castaño Justo P. , Luque Raùl M. , Cai Renzhi , Sha Wei , Victor Schally Andrew , Granata Riccarda

Pituitary neuroendocrine tumors (PitNETs) are mostly benign lesions originating from the anterior pituitary and represent 10–15% of all the intracranial neoplasms. PitNETscan be classified in non-secretory, clinically non-functioning pituitary adenomas (NFPAs), and secretory, comprising prolactin (PRL), growth hormone (GH) and adrenocoticotropic hormone (ACTH) PitNETs. Surgical resectionis the first line treatment for PitNETs, whereas chemotherapy and radiotherapy are pr...

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ea0070aep684 | Pituitary and Neuroendocrinology | ECE2020

Evaluation of somatostatin and dopamine receptor expression in nonfunctioning pituitary adenomas

Cano Gonzales David , Flores-Martinez Alvaro , Venegas Moreno Eva , Dios Elena , Remon Pablo , Deniz-Ruiz Alejandro , Madrazo-Atutxa Ainara , Luque Raul M. , Castaño Justo P. , Soto-Moreno Alfonso

Introduction: Non-functioningpituitary adenomas (NFPAs) are of benign nature but a sizeable number of NFPAs show aggressive features. Surgery is not always effective in treating NFPAs and thus other therapeutic options are needed. Previous studies have reported that NFPAs express somastostatin receptors (SSTRs) and dopamine receptors (DRDs).Aim: To systematically analyze the expression of SSTRs and DRDs in a large cohort of clinically well-characterized ...

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ea0070aep685 | Pituitary and Neuroendocrinology | ECE2020

High prevalence of pituitary deficiency after cranial radiation therapy for skull base meningioma: The importance of an annual screening.

Raymond Perrine , Cuny Thomas , Klein Marc , Bernier-Chastagner Valerie

Purpose: To determine the risk of developing hypopituitarism after cranial irradiation for skull base meningiomas. Cranial irradiation is often chosen as first treatment in skull base meningiomas to avoid risk to proximally-located critical structures. While cranial irradiation has been associated with high risk of inducing hypopituitarism, few studies concern meningiomas.Methods and materials: Fifty-two adult patients receiving photon-beam therapyfor sk...

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ea0070aep686 | Pituitary and Neuroendocrinology | ECE2020

Role of 68ga-dotatoc pet/ct in the management of neuroendocrine tumors

Hernández-Montoliu Laura , Peiró Inmaculada , Luis Vercher Jose , Suils Judith , Teulé Alexandre , Sánchez Cristina , Vilarrasa N , Puig de la Bellacasa Jordi , Guerrero Fernando , Villabona Carles

Introduction: Primary tumours in some patients with metastatic neuroendocrine tumors (NET) cannot be found by conventional imaging as CT, MRI and scintigraphy. 68Ga-DOTATOC PET/CT (68Ga-PET) appears to have superior sensitivity, specifity, and better resolution than 99mTC-octreotide SPECT/TC (SSTR scintigraphy) and can improve decision-making process, however its cost is higher, and its availability is limited.Aims: Evalu...

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ea0070aep687 | Pituitary and Neuroendocrinology | ECE2020

Endoscopic transsphenoidal pituitary surgery (ETPS) for pituitary tumors: Outcomes and complications

Venegas-Moreno Eva , Remon Pablo , Dios-Fuentes Elena , Deniz-Garcia Alejandro , Cardenas Eugenio , Kaen Ariel , Martin-Schrader Ignacio , Gonzalez-Rivera Natividad , Roldan Florinda , Cano-Gonzalez David , Soto-Moreno Alfonso

Methods: Restrospective descriptive study of patients who underwent ETPS for pituitary adenoma removal performed by the same surgical team in Virgen del Rocio Hospital of Seville from January 2014 to January 2019.Results: 174 patients had a pituitary adenoma (44 GH, 36 ACTH, 94 Non functioning). 54.02% were female. In acromegaly, 39 (88.63%) patients were operated by ETPS as initial approach. 20 (51.28%) patients had cavernous sinus invation. At outcome ...

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ea0070aep688 | Pituitary and Neuroendocrinology | ECE2020

Do cardiovascular risk factors predispose to pituitary apoplexy?

Gargouri Imen , Faten Hadjkacem , Othman Wafa Ben , Mouna Mnif , Mohamed Abid

Introduction: Pituitary apoplexy (PA) is a rare incident defined by the occurrence of necrosis and/or haemorrhage of the pituitary gland. The low incidence of PA makes it difficult to bring out from the studies its precipitating factors and especially its relationship with cardiovascular risk factors (CVR). The objectives of this studies were to characterize a number of patients with PA and to establish the link between PA and their CVRs.Methods: It is a...

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ea0070aep689 | Pituitary and Neuroendocrinology | ECE2020

Trabecular bone score as a useful tool for assessment of fracture risk in acromegaly

Jawiarczyk-Przybyłowska Aleksandra , Halupczok-Żyła Jowita , Syrycka Joanna , Rolla Małgorzata , Zembska Agnieszka , Kubicka Eliza , Bolanowski Marek

Acromegaly is a rare disease, caused by an excessive secretion of growth hormone (GH), and consequently the insulin like growth factor 1 (IGF-1). Patients with acromegaly have an increased risk of fractures, which might be correlated with insufficient quality of bone. It is known that use of FRAX in acromegaly is not validated, however we do not have other useful tools to assess real risk for fractures. The new valuable tool in the assessment of bone structure is the trabecula...

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ea0070aep690 | Pituitary and Neuroendocrinology | ECE2020

Metabolic abnormalities in patients with childhood onset of hypopituitarism – single center,long term observation

Kluczyński Łukasz , Gilis-Januszewska Aleksandra , Rogoziński Damian , Godlewska Magdalena , Pantofliński Jacek , Zygmunt-Górska Agata , Wójcik Małgorzata , Starzyk Jerzy , Hubalewska-Dydejczyk Alicja

Background: Hypopituitarism is a condition caused by deficiency in one or multiple pituitary hormones. The disease is associated with various metabolic disorders and decreased quality of life that are particularly marked in patients with childhood onset of the disorder. There are manyfactors influencing the metabolic status of patients such as different treatment modalities (surgery, radiotherapy, chemotherapy) and current supplementation (e.g. steroid or testosterone/estradio...

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ea0070aep691 | Pituitary and Neuroendocrinology | ECE2020

Early basal growth hormone level and nadir growth hormone level in oral glucose tolerance test as predictors for surgical cure in acromegaly

Tsiberkin Alexandr , Tsoy Uliana , Cherebillo Vladislav , Kuritsyna Natalya , Dalmatova Anna , Polezhaev Andrej , Belousova Lidia , Grineva Elena

Introduction: Growth hormone (GH) level is a direct marker of active residual adenoma tissue after acromegaly surgery. However, the value of early postoperative GH for prediction of surgical cure in acromegaly has not been established and therefore is not used in everyday clinical practice.Aim: The aim of our work was to study the 24-hour basal postoperative GH level and the nadir of GH level after the oral glucose tolerance test (OGTT) two weeks after s...

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ea0070aep692 | Pituitary and Neuroendocrinology | ECE2020

Quantitative bone assessment by radiofrequency echographic multi-spectrometry (REMS) in patients with acromegaly – a preliminary study

Halupczok-Żyła Jowita , Rolla Małgorzata , Jawiarczyk-Przybyłowska Aleksandra , Kolackov Katarzyna , Bolanowski Marek

Introduction: Acromegaly is a rare diseaseresulting most commonly from pituitary adenoma. A chronic increase of growth hormone (GH) and insulin-like factor 1 (IGF-1) leads to many systemic complications including osteoporosis and fractures. Radiofrequency echographic multi-spectrometry (REMS) is a new non-ionizing technique to diagnose osteoporosis.Purpose: The objective of the study was to evaluate bone mineral density (BMD) by REMS in patient...

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ea0070aep693 | Pituitary and Neuroendocrinology | ECE2020

Analysis of the value of bilateral inferior petrosal sinus sampling in the diagnosis and treatment of Cushing’s disease in patients with a negative or inconclusive result of magnetic resonance imaging

Styk Andrzej , Zięcina Piotr , Piasecki Piotr , Brzozowski Krzysztof , Witek Przemysław , Narloch Jerzy , Wierzbicki Marek , Zieliński Grzegorz

Diagnosis and treatment of Cushing’s disease is one of the greatest challenges of modern endocrinology. This is related to the fact that pituitary corticotropinomas are small tumours that are difficult to visualize in magnetic resonance imaging (MR). In about 40% of patients with Cushing’s disease, MR scans of the pituitary gland do not reveal any changes. The method that allows to localize the source of adrenocorticotropin (ACTH) secretion is bilateral inferior pe...

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ea0070aep694 | Pituitary and Neuroendocrinology | ECE2020

Micromegaly or acromegaly? A retrospective longitudinal study on clinical aspects and comorbidities in a large cohort of patients referred to a single tertiary center

Carosi Giulia , Sala Elisa , Mangone Alessandra , Del Sindaco Giulia , Mungari Roberta , Cremaschi Arianna , Ferrante Emanuele , Arosio Maura , Mantovani Giovanna

Introduction: The diagnosis of acromegaly is confirmed in the presence of high IGF-1 levels and inadequate suppression of growth hormone (GH) after glucose load. According to guidelines, a GH nadir (GHn)>0.4 ng/ml is considered diagnostic with ultrasensitive assays. However, some acromegalic patients with lower GHn, also called ‘micromegalic’, are reported, but a systematic collection of their clinical features is unavailable.Aim of the s...

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ea0070aep695 | Pituitary and Neuroendocrinology | ECE2020

Markers of recurrence in cushing’s disease: The role of post-operative serum cortisol and desmopressin test

Barbot Mattia , Ceccato Filippo , Lizzul Laura , Mondin Alessandro , Regazzo Daniela , ZIlio Marialuisa , Scaroni Carla

Introduction: Transsphenoidal surgery (TSS) is the first-choice treatment in Cushing’s disease (CD). When performed by experienced neurosurgeon, post-operative remission can be achieved in 70–80% of cases; however, recurrences during follow-up are quite frequent, involving around 20–30% of patients initially in remission. Early detection of relapses is crucial to avoid cortisol-related comorbidities. The aim of the study was to find reliable predictors of rec...

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ea0070aep696 | Pituitary and Neuroendocrinology | ECE2020

Long-term response to cabergoline treatment in men with macroprolactinoma is independent of tumor size

Rudman Yaron , Duskin-Bitan Hadar , Pertzov Barak , Manisterski Yossi , Masri-Iraqi Hiba , Shimon Ilan

Objective: To study the outcomeof menwith macroprolactinoma followingcabergoline treatment based on tumor size.Design: A retrospective cohort study using a single tertiary referral center registry.Methods: The study included 96 men with macroprolctinoma, aged 16–84 years (mean 47.3 years) treated from 1993 to 2019,for a mean follow-up of 7.5 years. The cohort was subdivided into 3 groups according to baseline adenoma diameter...

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ea0070aep697 | Pituitary and Neuroendocrinology | ECE2020

Abstract unavailable...

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ea0070aep698 | Pituitary and Neuroendocrinology | ECE2020

Inferior petrosal sinus sampling – 16 years of experience from a single tertiary center in Israel

Fischer Matan , Cahn Avivit , Glaser Benjamin , Leibowitz Gil , Stokar Joshua , Dresner-Pollak Rivka , Cohen José E. , Moshe Gomori J.

Introduction: IPSS (Inferior petrosal sinus sampling) is a reliable test for differentiating Cushing’s disease from ectopic ACTH secretion. In the past 30 years, Hadassah Hebrew University Hospital has been the sole national referral center for IPSS in Israel, carrying out the test for all patients country-wide.Methods: In this retrospective study we reviewed the records of all patients who underwent IPSS in our institution, for whom electronic dat...

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ea0070aep699 | Pituitary and Neuroendocrinology | ECE2020

Determinants of epicardial fat in patients with acromegaly

Biagetti Betina , Herance Raúl , Casteràs Anna , Ferrer Roser , Mesa Jordi , Simo Canonge Rafael

Background: Epicardial adipose tissue (EAT), the visceral fat of de myocardium, has been positively related to insulin resistance (IR), cardiovascular risk and the left ventricular myocardial mass. Whether EAT thickness in acromegaly is mediated by GH/IGF-1, IR or other factors remains to be elucidated.Objective: The aims of this study were: 1) To investigate whether EAT in patients with ACRO is linked to IR and pro-inflammatory and anti-inflammatory rel...

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ea0070aep700 | Pituitary and Neuroendocrinology | ECE2020

Disconnection hyperprolactinaemia and its correlation with tumour size: A study of 210 patients with histologically proven non-functioning pituitary macroadenomas

Das Arpan , Elalfy Omar , Hayhurst Caroline , Goetz Pablo , Mohamed Amr , Steve Davies J , Aled Rees D , Lansdown Andrew

Background: Serum prolactin levels at presentation can be useful in distinguishing between non-functioning pituitary macroadenomas and prolactinomas in order to guide appropriate therapy. Although thresholds have been suggested to distinguish between the two tumour-types, there remains some debate regarding discriminatory levels.Objective: To assess the baseline serum prolactin levels in a series of patients with histologically-proven non-functioning pit...

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ea0070aep701 | Pituitary and Neuroendocrinology | ECE2020

Role of 1 mg ACTH Stimulation Test to predict adrenocortical reserve and need of glucocorticoid therapy in non-functioning pituitary adenomas undergoing surgery

Garg Arpit , Mishra Sunil , Mithal Ambrish , Dubey Sudhir , Singh VP

Background: Peri-operative glucocorticoids are routinely administered to patients undergoing trans-sphenoidal surgery for non-functional pituitary adenomas (NFPA) irrespective of the integrity of hypothalamic-pituitary-adrenal (HPA) axis.Aim: To evaluate HPA axis before and three months after endoscopic trans-sphenoidal adenomectomy (E-TSA) in patients with clinically NFPA utilizing 1 mg ACTH stimulation test and determine the need for glucocorticoid adm...

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ea0070aep702 | Pituitary and Neuroendocrinology | ECE2020

Gigantism associated with hyperprolactinemia due to a pituitary macroadenoma in an adolescent girl

Nikitas Skarakis Spyridon , Chroussos Georgios , Kanaka-Gantenbein Christina , Charmandari Evangelia , Karachaliou Feneli

Aim: To describe the clinical course of an adolescent girl with gigantism.Background and methodsIn young persons with open epiphyses, overproduction of GH results in gigantism, with consequent longitudinal growth acceleration1. Pituitary gigantism is extremely rare, and its cause in the majority of patients is a pituitary adenoma1. Here, we present a 12, 5 years-old girl investigated for tall stature [183.5&#1...

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ea0070aep703 | Pituitary and Neuroendocrinology | ECE2020

Cabergoline resistance in a patient with prolactinoma with debut before puberty

Fedorova Natalia , Dzeranova Larisa , Pigarova Ekaterina , Vorotnikova Svetlana

Prolactinomas are the most common hormone-active pituitary tumors, most common in young women, whose main reason for going to the doctor is menstrual irregularity. At the onset of the disease before puberty in children, there is a delay in sexual development, in girls primary amenorrhea.A 27-year old woman has been observed since 15 years, when hyperprolactinemia was first detected, level of PRL was 10 900 IU/l, and according to MRI pituitary endose...

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ea0070aep704 | Pituitary and Neuroendocrinology | ECE2020

A rare etiology of hyperprolactinemia: Factitious hypoglycemia

Kalthoum Mehdi , Gharbi Radhouan , Kandara Hajer , Adel Meriem , Jemel Manel , Kammoun Ines

Introduction: While prolactin is most well known for its role in lactation and suppression of reproduction, its physiological functions are quite diverse. There are many etiologies of hyperprolactinemia, including physiologic as well as pathologic causes. Factitious hypoglycemia a rare case of induced hyperprolactinemiaCase report: We report a case of 17-years-old girl presented to with sever hypoglycemia mesured at 30 mg/dl. The patient had no significa...

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ea0070aep705 | Pituitary and Neuroendocrinology | ECE2020

Pituitary gland metastasis of endometrial cancer: A case report

Farzaliyeva Aydan , Turhan İyidir Özlem , Pınar Altay Feride , Bașçıl Tütüncü Neslihan

Introduction: Tumour metastasis to pituitary gland is very rare and constitutes only 1% of all pituitary lesions. Breast and lung cancer are the most common neoplasms reported to metastasize to pituitary gland. Most of the pituitary metastasis are asymptomatic, and thus can be easily overlooked in imaging studies. Polyuria and polydipsia due to diabetes insipidus are the most common sypmtoms in these patients. Patients may also present with visual impairment and symptoms of pa...

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ea0070aep706 | Pituitary and Neuroendocrinology | ECE2020

Microprolactinoma, PCOS Or stress induced hyperprolactinemia? – case report

Oprea Luciana , banica andreea , Ilie Iuliana , Iordachescu Carmen , Musat Madalina

Hyperprolactinemia is one of the most common problems in clinical endocrinology. It relates with various etiologies (physiological, pharmacological, pathological), the clarification of which requires careful history taking and clinical assessment.We present the case of a 15-year-old girl, with history of secondary amenorrhea and headache in the presence of mildly elevated prolactin level, diagnosed as a microprolactinoma on MRI scan (4–5 mm) and tr...

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ea0070aep707 | Pituitary and Neuroendocrinology | ECE2020

A prolactinoma of rare localization as part of familial isolated pituitary adenomas

Asbar Hind , Rafi Sana , El Mghari Ghizlane , El Ansari Nawal

Introduction: The syndrome of familial isolated pituitary adenomas (FIPA) or predisposition to pituitary adenomas (PAP) is characterized by the presence within the same family of at least 2 isolated pituitary adenomas without any other type of associated endocrine tumor. We report the case of a prolactinoma attached to the pituitary stalk as part of FIPA, revealed by erectile dysfunction.Case report: A 40 year old man presented with eretile dysfunction s...

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ea0070aep708 | Pituitary and Neuroendocrinology | ECE2020

A rare coexistence of acromegaly, myasthenia gravis, sicca syndrome, and elevated serum IgG4 levels: A single case report

Wu Wan-Chen , Po-Hao Huang Abel

62-year-old woman was an employee of catering service. She had progressive limb weakness, easpecially proximal part since May, 2015, with diurnal change. She had difficulties in lifting arms, holding plates steady, and knee buckling while walking, which made her quit the job. Generalized myasthenia gravis (MG) was confirmed by positive acetylcholine receptor antibody (12.8 nmol/l) and marked decremental changes in repetitive stimulation test in November, 2015. Her limb weaknes...

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ea0070aep709 | Pituitary and Neuroendocrinology | ECE2020

A case of cyclic cushing’s disease – cortisol surfing; catching the cortisol wave!

Sutton-Smith Mark , Yasin Zeeshan , Boharoon Hessa , Idowu Oluwagbemiga , Alegre-Abarrategui Javier , Nair Ramesh , Meeran Karim , Todd Jeannie F.

A 28-year-old woman presents with a one-year history of marked weight gain of 18 kg, increasing hirsutism and night sweats. Clinically, hyperandrogenism and Cushing’s syndrome were suspected. A diagnosis of Polycystic ovarian syndrome was made based on Rotterdam criteria and Metformin was initiated. Initial testing for Cushing’s syndrome demonstrated a high 24 h urinary free cortisol (306 nmol/l) with incomplete cortisol suppression on an overnight dexamethaso...

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ea0070aep710 | Pituitary and Neuroendocrinology | ECE2020

Successful pregnancy in a woman with acromegaly treated with lanreotide

Orłowska-Florek Renata , Sztembis Joanna , Gala-Błądzińska Agnieszka

Introduction: Pregnancy in an acromegalic woman is rare and generally safe, but tumor expansion may occur. As this is a rare occurrence, little is known about the optimum management of such patients during pregnancy. We present the case of a woman with acromegaly treated with Lanreotide during pregnancyCase report: A 29-year-old woman with acromegaly underwent transsphenoidal resection of invasive pituitary macroadenoma in August 2014, without cure. In N...

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ea0070aep711 | Pituitary and Neuroendocrinology | ECE2020

Isolated adrenocorticotropic hormone deficiency in a female with ectopic posterior hypophysis

Boutzios Georgios , Koukoulioti Eleni , Bourgalis-Michalopoulos Konstantinos , Giovannopoulos Ilias , Miliou Myrto , Papaoiconomou Eleni , Tsourouflis Gerasimos

Introduction: Some endocrine disorders, including hypophysitis and isolated adrenocorticotropic hormone (ACTH) deficiency, are caused by an autoimmune response to endocrine organs. Previous data have also shown that isolated ACTH deficiency may be associated at least in part with a paraneoplastic syndrome. We present a rare case with isolated ACTH deficiency along with ectopic posterior hypophysis.Case presentation: A 53-yea...

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ea0070aep712 | Pituitary and Neuroendocrinology | ECE2020

Thickening of the pituitary stalk: About 4 cases

Bouzid Aicha , Meskine Djamila

Introduction: Pathologies of the pituitary stem are very often revealed during diabetes insipidus. The introduction of MRI constituted, in this context, a major diagnostic contribution. We report 4 cases of thickening of the pituitary stem observations. These are 4 patients (3 women and one man) whose age varies from 30 to 47 years, the circumstances of discovery are represented by amenorrhea in women and polyuropolydipsic syndrome in men. Clinically, diabetes insipidus was fo...

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ea0070aep713 | Pituitary and Neuroendocrinology | ECE2020

Pregnancy and pituitary adenomas: A case series

Dumitriu Roxana , Dusceac Roxana , Florentina Burcea Iulia , Baciu Ionela , Capatina Cristina , Dumitrascu Anda , Poiana Catalina

Introduction: Pituitary tumours may interfere with fertility and pregnancy may be uncommon in these cases, but some patients can conceive spontaneous. Also in pregnancy, due to the physiologic changes of the pituitary gland, gradual volume increase and cellular hyperplasia that target hormonal secretion, the evaluation of the pituitary function is very complex.Clinical cases: We present 5 cases of female patients (24 – 35 years old) that were diagn...

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ea0070aep714 | Pituitary and Neuroendocrinology | ECE2020

Short stature in turner syndrome: Should we assess growth hormone secretion?

Kamoun Elyes , Oueslati Ibtissem , Chiboub Marwa , Yazidi Meriem , Grira Wafa , Chaker Fatma , Chihaoui Melika

Introduction: Short stature is a common feature in Turner syndrome. It is caused by haplo-insufficiency of the SHOX gene. Growth hormone deficiency does not occur in this disorder as confirmed by the normal GH response to stimulation tests. However, few cases of coexisting GH deficiency and Turner syndrome have been reported. We herein describe two cases of GH deficiency in patient with Turner syndrome.Observation 1: A 20-year-old patient was referred fo...

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ea0070aep715 | Pituitary and Neuroendocrinology | ECE2020

Case of differential diagnosis in a patient presenting with paroxysmal arterial hypertension

Vaychulis Irina

Arterial hypertension (AH) may be the initial clinical presentation of various endocrine disorders. An accurate diagnosis provides an opportunity to achieve a cure of AH in some cases. Female patient, born in 1971, was referred to endocrinologist in 2018, at the age of 47 y, presented with AH and adrenal adenoma. Since the age of 35 y (2006), after the third delivery she had been suffering from AH with paroxysmal BP surges up to 180/120 mmHg, accompanied by panic, anxiety, swe...

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ea0070aep716 | Pituitary and Neuroendocrinology | ECE2020

A rare case of panhypopituitarism caused by TNF-α inhibitors

Koutroukas Vaios , Dimitropoulos Ioannis

Aim: To present an unusual case of hypophysitis caused by TNF-α inhibitors and demonstrate the importance of astuteness with regards to endocrine axes testing when clinical circumstances indicate.Case: A 78-year-old gentleman with known fistulating Crohn’s disease (treated with adalimumab and infliximab in the past) presented with vomiting, dizziness, hyponatraemia and hypotension in early December 2018. SST showed he was cortisol-deficient, i...

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ea0070aep717 | Pituitary and Neuroendocrinology | ECE2020

Natural course of incidentally found nonfunctioning pituitary macroadenoma

Dusek Tina , Kasnar Ana-Marija , Kraljevic Ivana , Skoric Polovina Tanja , Solak Mirsala , Balasko Annemarie , Zibar Tomšić Karin , Popovac Hrvoje , Ozretic David , Jovanovic Ivan , Kastelan Darko

Introduction: The widespread use of neuroimaging leads to the increased detection of asymptomatic pituitary adenomasnot causing significant mass effect. Their optimal management remains unclear.Aim of the study: To explore the natural course of incidentally found nonfunctioning pituitary macroadenomas.Patients and Methods: This was a longitudinal study that enrolled patients with clinically nonfunctioning pituitary macroadenomas wh...

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ea0070aep718 | Pituitary and Neuroendocrinology | ECE2020

Evaluation of early predictors of metabolic syndrome in patients with gastroenteropancreatic neuroendocrine tumors (GEP-NET)

Bottiglieri Filomena , Modica Roberta , Barrea Luigi , de Cicco Federica , Minotta Roberto , Muscogiuri Giovanna , Savastano Silvia , Faggiano Antongiulio , Colao Annamaria

Metabolic syndrome and obesity (MetS) are supposed to have a role in cancer but data analysing their association with GEP-NET are lacking. Aim of this study was to explore the association of early predictors of MetS with GEP-NET, usingas tools Fatty Liver Index (FLI), a predictor of non-alcoholic fatty liver disease (NAFLD)and Visceral Adiposity Index (VAI), a gender-specific indicator of adipose dysfunction. A cross-sectional case-control observational study was conducted. VA...

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ea0070aep719 | Pituitary and Neuroendocrinology | ECE2020

Experience in complex therapy of selective and non-selective dopamine agonists in women with resistant prolactinomas for recovering of ovulation function

Vorotnikova Svetlana , Dzeranova Larisa , Pigarova Ekaterina , Fedorova Natalia

Experience in complex therapy of selective and non-selective dopamine agonists in women with resistant prolactinomas for recovering of ovulation function.Introduction: Prolactinomas are the most common pituitary adenomas and dopamine agonists (DA) still remain the first choice of treatment. Nevertheless, it does not always exert an adequate effect and endocrinologists face the challenge of resistant prolactinomas more frequently. This problem is very imp...

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ea0070aep720 | Pituitary and Neuroendocrinology | ECE2020

An analysis of craniopharyngioma patients in malta: Epidemiology, patient characterisation and long-term sequelae

Craus Sarah , Gruppetta Mark

Background: Despite being benign, craniopharyngiomas are challenging tumours to manage and can cause significant morbidity and mortality in both the paediatric and adult population.Method: Our aim was to analyse epidemiology, patient characteristics and long-term sequalae through a population-based study in Malta. A thorough research was carried out to identify patients who were diagnosed with craniopharyngioma in our local population. Subjects were iden...

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ea0070aep721 | Pituitary and Neuroendocrinology | ECE2020

Septo-optic dysplasia with late-onset diagnosis: An uncommon presentation of a rare disease

Duarte Vitória , Ivo Catarina , Veríssimo David , Franco Sara , Bastos Filipa , Marcelino Mafalda , Vara Luiz Henrique , Portugal Jorge

Background: Septo-optic dysplasia (SOD), also known as de-Morsier syndrome, is a rare congenital disorder characterized by any combination of optic nerve hypoplasia, pituitary dysfunction and midline abnormalities of the brain. Clinical diagnosis requires the presence of at least two of the features. This disorder is equally prevalent in males and females, with a reported incidence of 1/10,000 live births. There is wide variation in the severity of the clinical features found....

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ea0070aep722 | Pituitary and Neuroendocrinology | ECE2020

Doses of tolvaptan needed in chronic therapy of SIADH-induced euvolemic hyponatremia vary according to the etiology of SIADH

Pérez Candel Xavier , Lopez Nevado Celia , Cuesta Martin , Angel Rodriguez Cabezas Miguel , Puebla Virginina , de Miguel Paz , Gomez Hoyos Emilia , Ruiz Teresa , Calle Pascual Alfonso , Runkle Isabel

Introduction: Clinical trials indicate that Tolvaptan is safe and effective in the treatment of patients with sustained mild/moderate SIADH-induced euvolemic hyponatremia. Tolvaptan doses are often modified during chronic use. We present the weekly doses of a series of patients on chronic therapy, followed up in outpatient clinic.Methods: Retrospective, cross-sectional. 114 patients receiving tolvaptan for chronic SIADH were followed up in a Hyponatremia...

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ea0070aep723 | Pituitary and Neuroendocrinology | ECE2020

Soluble alpha klotho measurement: Comparison of measurements by different commercially available assays in healthy subjects and active acromegaly

Ribeiro de Oliveira Longo Schweizer Junia , Schilbach Katharina , Haenelt Michael , Störmann Sylvère , Schopohl Jochen , Bidlingmaier Martin

Background: Recent studies reported high soluble α-klotho concentrations (α-klotho) in active acromegaly, with normalization after successful therapy. We showed that α-klotho correlates to GH and even better to IGF-I. So far, most studies employed an immunoassay from Immuno-Biological Laboratories (IBL) for measurement of α-klotho, but other assays are available. Agreement between assays remains questionable since α-klotho exists in several isoforms: K...

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ea0070aep724 | Pituitary and Neuroendocrinology | ECE2020

Metformin and everolimus: A promising combination for neuroendocrine tumors treatment

Vitali Eleonora , Boemi Ilena , Piccini Sara , Tarantola Giulia , Zerbi Alessandro , Smiroldo Valeria , Lavezzi Elisabetta , Spada Anna , Mantovani Giovanna , Lania Andrea

Treatment options for neuroendocrine tumors (NET), including everolimus, are rarely curative, as NETs frequently show resistance to medical therapy. In fact, the use of everolimus, an mTOR inhibitor, is limited by the development of resistance, probably due to the activation of Akt signalling. Metformin seems to have anticancer effects in neuroendocrine tumors (NETs). A recent retrospective study found longer progression-free-survival in diabetic pancreatic neuroendocrine tumo...

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ea0070aep725 | Pituitary and Neuroendocrinology | ECE2020

One year of growth hormone therapy in spanish adults with prader-willi syndrome (PWS) improves body composition without changes in bone mineral density

Casamitjana Laia , Giménez-Palop Olga , Pareja Rocío , Corripio Raquel , León Josep , Caixàs Assumpta

Introduction: PWS is the most common cause of genetic obesity. These patients have an abnormal body composition with increased amounts of fat mass (FM), reduced lean body mass (LBM) and diminished bone mineral density (BMD), all similar to patients with growth hormone deficiency (GHD).The abnormal body composition has been described due to impairment of the activity of GH-IGF system and to hypogonadism. Studies ongrowth hormone (GH) treatment in PWS adults from other European ...

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ea0070aep726 | Pituitary and Neuroendocrinology | ECE2020

Efficacy and safety of urea in syndrome of inappropriate secretion of antidiuretic hormone on a secondary hospital

Moreno Tirado Antonio , González Lázaro Paloma , Contreras Pascual Cristina , Montalban Mendez Cristina , Garcia Manzanares Vazquez De Agredos Alvaro , Del Val Zaballos Florentino , Silva Fernandez Julia , Javier Gomez Alfonso Francisco , Rosa Gomez Garcia Ines , Lomas Meneses Amparo

Introduction: The most common cause of hyponatraemia is the syndrome of inappropriate antidiuretic hormone (SIADH). The diagnosis typically requires hyponatraemia in the setting of reduced serum osmolality, inappropriately concentrated urine with normal sodium excretion levels, and the absence of interfering medications, hypothyroidism and adrenal insufficiency. Unless hypertonic saline is indicated for acute onset profound hyponatraemia and/or with severe symptoms, the mainst...

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ea0070aep727 | Pituitary and Neuroendocrinology | ECE2020

Multiple metastatic NET of unknown primary site in a young patient with carcinoid syndrome: Can we treat it as a gastrointestinal NET?

Lambrinoc Diana , Morea Alexandru , Dușceac Roxana , Poiană Cătălina

Introduction: Neuroendocrine tumours (NETs) of unknown primary site are relatively uncommon, representing about 10% of all NETs. Of these, particularly the well-differentiated NETs often present initially with liver metastases, and most of these represent gastroenteropancreatic NETs. The presence of carcinoid syndrome is also common.Case presentation: We describe the case of a 40 years-old patient presented in 2013 with flushing, diarrhea, and back pain....

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ea0070aep728 | Pituitary and Neuroendocrinology | ECE2020

A relationship between motilin and ghrelin in human motilin receptor transgenic mice

Matsuura Bunzo , Kawamura Tomoe , Nakaguchi Hironobu , Kanzaki Sayaka , Senba Hidenori , Miyake Teruki , Furukawa Shinya , Hiasa Yoichi

Motilin (M), erythromycin (EM) and ghrelin (G) are recognized as important regulators of gastrointestinal motor function in humans that are mediated by class I guanine nucleotide-binding protein (G protein)-coupled motilin receptor (MR) and growth hormone secretagogue receptor (GHSR). These receptors have also been demonstrated as clinically useful pharmacological targets. However, a molecular relationship between MR and GHSR activation has been unclear. We generated human MR ...

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ea0070aep729 | Pituitary and Neuroendocrinology | ECE2020

Characterization of epithelial-mesenchymal transition in growth hormone-secreting adenomas

Gil Joan , Marques-Pamies Montserrat , García-Martínez Araceli , Serra Guillermo , Webb Susan , Antonio Sampedro-Nunez Miguel , Pico Antonio , Marazuela Monica , Jorda Mireia , Puig-Domingo Manel

First generation somatostatin receptor ligands (SRL) are the first-line drugs in primary acromegaly treatment or after surgical failure in patients with active acromegaly. In previous studies we confirmed the association of the expression of SSTR2, Ki67 and E-cadherin (CDH1) in responsive GH-secreting adenomas response to SRL. Moreover, E-cadherin showed a greater predictive capacity than most of the markers described. Loss of E-cadherin is a typical mark of ...

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ea0070aep730 | Pituitary and Neuroendocrinology | ECE2020

Global methylation-demethylation status in pituitary neuroendocrine tumors as potential therapeutic target

Szabó Borbála , Németh Kinga , Mészáros Katalin , Szücs Nikolette , Czirják Sándor , Reiniger Lilla , Patócs Attila , Butz Henriett

Background: The altered DNA methylation of certain genes in Pituitary Neuroendocrine Tumors (PitNETs) are well known. However little information is available regarding global methylation changes and the process of demethylation in these tumors. In addition, influencing global methylation-demethylation could be a potential new therapeutic option especially in clinically non-functional PitNETs.Material and Methods: Overall, 44 fresh frozen pituitary adenom...

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ea0070aep731 | Pituitary and Neuroendocrinology | ECE2020

The expression of oxytocin receptor (OXTR) in metastatic pancreatic neuroendocrine tumors (PNETs)

Katalinic Darko , Bildat Stephan , Kattner Elke , Soerensen Lilly , Aleric Ivan , Vcev Aleksandar

Introduction: Pancreatic neuroendocrine tumors (PNETs) are rare malignant neoplasms which incidence is continually increasing. They are characterized by diverse biological behaviour and impact on the patients’ prognosis, ranging from clinicaly indolent to very aggressive. Oxytocin receptor (OXTR) is a member of the family of G-protein receptors and is present on the cell-surface of the gastrointestinal organs. Unfortunately, theimpact of OXTR signaling on the development...

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ea0070aep732 | Pituitary and Neuroendocrinology | ECE2020

Metastatic insulinoma managed with lutetium (177LU) and somatostatin analog

Sözen Mehmet , Canturk Zeynep , Cetinarslan Berrin , Selek Alev , Gezer Emre

Background: Insulinoma is a rare tumour representing 1–2% of all pancreatic neoplasms and it is malignant in only 10% of cases. Locoregional invasion or metastases define malignancy, whereas dimension (> 2 cm), CK19 status, tumor staging and grading (Ki67 > 2%), and age of onset (> 50 years) can be considered elements of suspect.Case presentation: We report a case of malignant insulinoma in a 80 year old woman presenting symptoms c...

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ea0070aep733 | Pituitary and Neuroendocrinology | ECE2020

Tobacco smoke induces aberrant pulmonary neuroendocrine differentiation via different MAPK pathways

Liang Zhaofeng , Xie Wei , Wu Rui , Xie Chunfeng , Li Xiaoting , Zhong Caiyun

Tobacco smoke (TS) is the major cause of lung cancer, including pulmonary neuroendocrine tumors such as small cell lung cancer and large cell neuroendocrine carcinoma. Unlike squamous cell carcinomas and adenocarcinomas, the precursor lesions for subtypes of pulmonary neuroendocrine tumors have not been historically defined, and theexpression of neuroendocrine markers has been served as critical indices for the identification of these tumors. Mitogen-activated protein kinases ...

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ea0070aep734 | Pituitary and Neuroendocrinology | ECE2020

Use of tolvaptan in acute post-surgical hyponatremia in patients with pituitary diseases

Ferrante Emanuele , Indirli Rita , Mantovani Beatrice , Cremaschi Arianna , Liliana Serban Andreea , Sala Elisa , Locatelli Marco , Bertani Giulio , Arosio Maura , Mantovani Giovanna

Introduction: Hypotonic hyponatremia is frequently observed after pituitary surgery. In this context, use of vasopressin V2-receptor antagonists is not standardized. The aim of this retrospective study is to explore the role of Tolvaptan in the management of acute hyponatraemia after pituitary surgery.Methods: We collected clinical, safety and efficacy data of patients treated with Tolvaptan after pituitary surgeryin our Centre between April 2011 and Feb...

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ea0070aep735 | Pituitary and Neuroendocrinology | ECE2020

Metabolic status of patients with combined pituitary hormone deficiency due to PROP1 mutation

Rogoziński Damian , Gilis-Januszewska Aleksandra , Kluczyński Łukasz , Godlewska Magdalena , Hubalewska-Dydejczyk Alicja

Introduction: Combined pituitary hormone deficiency (CPHD) due to PROP1 mutation is a rare cause of childhood-onset pituitary disfunction. Treatment with growth hormone is crucial for the proper development during childhood and adolescence, and to maintain adequate metabolic processes in the adulthood. There are other factors influencing patients metabolism e.g. steroid and estradiol/testosterone treatment.Aim: To assess metabolic status of patients with...

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ea0070aep736 | Pituitary and Neuroendocrinology | ECE2020

Kisspeptin levels in children and adolescents

Gumeniuk Olga

Kisspeptin (metastin, HH13, KiSS-1, KiSS-1 metastasis-suppressor, KiSS-1 metastasis suppressor) is a neuropeptid that is encoded by the KISS1 gene (Messager et al., 2005). Kisspeptin-GPR54 signaling has an significant role in initiating secretion of gonadotropin-releasing hormone at puberty (K. Skorupskaite et al., 2014). Is known that kisspeptin to play a role in tumor suppression (especially in breast tissue), (E.J. M...

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ea0070aep737 | Pituitary and Neuroendocrinology | ECE2020

Body composition and bone mineral density in 107 patients with childhood onset growth hormone deficiency (CO-GHD) at the time of transition from pediatric to adulthood endocrine care

Doknic Mirjana , Stojanovic Marko , Milenkovic Tatjana , Zdravkovic Vera , Maja Jesic , Todorovic Sladjana , Katarina Mitrovic , Vukovic Rade , Miljic Dragana , Pekic Djurdjevic Sandra , Soldatovic Ivan , MilanPetako

Transition from childhood to adulthood is a vulnerable period in all adolescents and particularly inCO-GHD patients. Low bone mineral density (BMD) and body composition alterations are frequently reported in young adults with CO-GHD, but relevant large monocentric studies are lacking.Patients and Methods: In a monocentric, observational, retrospective cross-sectional study conducted from 2005–2019, 107 CO-GHD patients were analyzed (17–26 yea...

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ea0070aep738 | Pituitary and Neuroendocrinology | ECE2020

Management of pregnant women with prolactinomas and analysis of their pregnancy outcomes

Nasirova Khurshida , Muminova Sitorakhon

The purpose of the study was to study the characteristics of the course of pregnancy and childbirth in women with prolactinomas, as well as the effect of pregnancy on the size and functional activity of prolactin-secreting pituitary adenomas.Materials and methods: 70 women with prolactinomas during pregnancy were examined. The age of the examined patients from 20 to 38 years (28.9 ± 0.5 years). The duration of the disease ranged from 3 months to 20 ...

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ea0070aep739 | Pituitary and Neuroendocrinology | ECE2020

Interleukin-6 response to insulin-induced hypoglycemia is associated with hypothalamic-pituitary-adrenal axis activation

Drummond Juliana , Vieira Erica , Rocha Beatriz , Pedrosa William , Ribeiro-Oliveira Jr Antonio , Teixeira Antonio

Introduction: Increased plasma levels of interleukin-6 (IL-6) in response to acute hypoglycemia have been well documented. Counter-regulatory hormones are likely to play a role in this inflammatory response.Objective: To study the interaction between IL-6 and counter-regulatory hormones during hypoglycemic stress.Methods: We conducted an exploratory single center study involving 23 patients (mean age = 34.15 ± 8.87 years, ...

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ea0070aep740 | Pituitary and Neuroendocrinology | ECE2020

The effect of dual-release versus conventional hydrocortisone on fatigue, measured by ecological momentary assessments

Boesen Victor , Borresen Stine , Christoffersen Thea , Klose Marianne , Watt Torquil , Feldt-Rasmussen Ulla

Background: Replicating the physiological cortisol secretion is key in treatment of glucocorticoid insufficient individuals. A sub-optimal replication may explain the impaired quality of life experienced by these individuals. The study investigates fatigue measured by ecological momentary assessments in patients treated with conventional hydrocortisone compared with a once-daily dual-release formula of hydrocortisone (Plenadren) that follows the cortisol physiology more closel...

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ea0070aep741 | Pituitary and Neuroendocrinology | ECE2020

Cushing’s syndrome in the 21st century, not what it used to be: A single institution experience

Rosset Arza , Greenman Yona , Shefer Gabi , Osher Etty , Stern Naftali , Tordjman Karen

Background: True Cushing’s syndrome (CS) is an exceedingly rare condition, thus to avoid unnecessary and costly testing, a diagnostic investigation should only start based on solid clinical suspicion. The current obesity epidemic has become a hurdle in discriminating subjects at real risk for CS from those with the metabolic syndrome. Moreover, many CS patientsrecently diagnosed do not fit the typical clinical profile of ‘at risk subjects’.<p class="abste...

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ea0070aep742 | Pituitary and Neuroendocrinology | ECE2020

Visual impairment in patients with pituitary apoplexy

Gargouri Imen , Faten Hadjkacem , Wafa Belabed , Salah Dhouha Ben , Mouna Mnif , Nadia Charfi , Nabila Rekik , Mohamed Abid

Introduction: Pituitary apoplexy (PA) is a rare incident defined by the occurrence of necrosis and/or haemorrhage of the pituitary gland. PA is a clinical syndrome characterized by the sudden onset of headache, vomiting, visual impairment and decreased consciousness in some cases. The objectives of our study are to describe its clinical features and characterize the visual impairments in a cohort of PA in the region of Sfax.Methods: It is a retrospective...

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ea0070aep743 | Pituitary and Neuroendocrinology | ECE2020

Central hypothyroidism as an adverse effect of bexarotene treatment for cutaneous T-cell lymphoma

Felder Susan , Zechmann Stefan , Baur Katharina , Seelig Eleonora , Christ Emanuel , Holbro Andreas , Potlukova Eliska

Introduction: Central hypothyroidism is rare, estimated to occur in 1:20,000 to 1:80,000 in the general population. Pituitary mass lesions and adverse effects of their treatment are the most common causes of central hypothyroidism. In recent years, the incidence of pituitary hormonal deficiencies as adverse effects of novel molecular targeting anticancer drugs has risen sharply. Especially checkpoint inhibitors are associated with partial or complete hypopituitarism. In this r...

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ea0070aep744 | Pituitary and Neuroendocrinology | ECE2020

Pasireotide-induced hyperglycemia and efficacy of antidiabetic treatments in patients with acromegaly: Evaluation of two referral centers

F.S. Grasso Ludovica , Chiloiro Sabrina , Pirchio Rosa , Giampietro Antonella , Auriemma Renata S. , Bianchi Antonio , De Martino Maria Cristina , Negri Mariarosaria , Colao Annamaria , Pontecorvi Alfredo , De Marinis Laura , Pivonello Rosario

Pasireotide (PAS) has a safety profile similar to first-generationsomatostatin analogues (SSA), except for a higher frequency of hyperglycaemia-related adverse events (AEs). However, consensus on the best management of PAS-induced hyperglycaemia in acromegalic patients has still to be defined. The current study aims at investigating the effects of long-term PAS treatment on glucose metabolism, by evaluating the clinical management of hyperglycemia-relatedAEs in acromegalic pat...

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ea0070aep745 | Pituitary and Neuroendocrinology | ECE2020

The characteristic of patients with pituitary stalk lesions – single center,long term observation

Kluczyński Łukasz , Gilis-Januszewska Aleksandra , Rogoziński Damian , Godlewska Magdalena , Pantofliński Jacek , Zygmunt-Górska Agata , Wójcik Małgorzata , Starzyk Jerzy , Hubalewska-Dydejczyk Alicja

Background: Pituitary stalk lesions (PSL) is a general term used to describe changes located in the pituitary infundibulum. Special anatomical locus makes the diagnosis difficult to establish. Most of the patients with PSL characterizes with hypopituitarism and multiple metabolic abnormalities.Aim: To present the characteristic of patients with PSL.Methods: We analyzed data of 35 patients (21 M/14 W) with pituitary stalk ...

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ea0070aep746 | Pituitary and Neuroendocrinology | ECE2020

Preoperative pegvisomant as a potential therapeutic option to improve cardiac function in Acromegaly-induced cardiomyopathy: Two cases

Ernst Matthias E. , Anand Gurpreet , Beuschlein Felix

Introduction: Acromegaly is a rare chronic disorder caused by growth hormone hypersecretion due to GH-producing pituitary adenoma. Surgery is the first-line treatment modality. However, patients with severe cardiac involvement are high-risk candidates for pituitary surgery. These patients may benefit from rapid preoperative biochemical control of acromegaly. There is emerging evidence of efficacy of pegvisomant, a GH-receptor antagonist, (either alone or in combination with so...

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ea0070aep747 | Pituitary and Neuroendocrinology | ECE2020

A prospective, non-interventional, observational, multi-center study of quality of life in people with acromegaly receiving lanreotide autogel

Bolanowski Marek , Hubalewska-Dydejczyk Alicja , Kos Kudla Beata , Ruchala Marek , Witek Przemyslaw , Zgliczynski Wojciech , Houchard Aude , Bartmanska Marta

Background: Patients with acromegaly have substantially reduced quality of life (QoL). Delayed diagnosis is common and a long duration of untreated acromegaly leads to decreased QoL and increased morbidity and mortality.Aim: This longitudinal study examined QoL in patients with acromegaly who received lanreotide autogel.Material and Methods: This was a 2-year prospective, observational, multicenter study conducted in Poland (NCT023...

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ea0070aep748 | Pituitary and Neuroendocrinology | ECE2020

Soluble klotho: A possible predictor of quality of life in acromegaly patients

Coopmans Eva , El-Sayed Nour , Frystyk Jan , Erik Magnusson Nils , Otto Jorgensen Jens , Jan Van der Lely Aart , Janssen Joseph AM , Muhammad Ammar , Neggers Sebastian

Purpose: Although quality of life (QoL) is improved in patients with acromegaly after disease control, QoL correlates only weakly with traditional biomarkers. Our objective is to investigate a potential relation between the new serum biomarker soluble Klotho (sKlotho), GH and insulin-like growth factor 1 (IGF-1) levels and QoL.Methods: In this prospective cohort study, we investigated 54 acromegaly patients biochemically well-controlled on combination tr...

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ea0070aep749 | Pituitary and Neuroendocrinology | ECE2020

Pregnancy outcomes in women receiving growth hormone therapy enrolled in NordiNet international outcome study (IOS) and American Norditropin Studies: Web Enabled Research (ANSWER Program)

Biller Beverly , Höybye Charlotte , Camilla Birkegård Anna , Pietropoli Alberto , Weber Matthias

Recombinant human growth hormone (GH) is not indicated for use during pregnancy and in women of childbearing potential not using contraception. Nonetheless, in clinical practice, some women taking GH replacement conceive during treatment and many continue GH during their pregnancy. Here we report data on GH-treated women enrolled in two complementary, international, non-interventional registry studies, NordiNet IOS (NCT00960128; 2006–2016) and ANSWER (NCT01009905; 2002&#...

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ea0070aep750 | Pituitary and Neuroendocrinology | ECE2020

Country-specific differences in adult growth hormone deficiency diagnosis and treatment rates

Nedjatian Navid , den Hollander Bjørnar , Amirouche Yacine

Background: Adult growth hormone deficiency (AGHD) is a rare disease associated with adverse metabolic abnormalities and possibly increased cardiovascular morbidity and mortality. Although basic prevalence data is available, little is known about the rates of diagnosed and treated patients.Aim: To understand the rates of AGHD diagnosis/treatment and details about patients’ growth hormone (GH) dose, age and treatment duration....

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ea0070aep751 | Pituitary and Neuroendocrinology | ECE2020

Evaluation of haemolitic complement activity (ch50) and igg subclasses in adult growth hormone deficiency: Preliminary data

Vergani Edoardo , Bruno Carmine , Basile Umberto , Napodano Cecilia , Piunno Gaia , Stefanile Annunziata , Gulli Francesca , Mancini Antonio

Adult Growth Hormone Deficiency (GHD) is a condition characterized by low grade chronic inflammation; increased levels of free light chains of immunoglobulins have been described; moreover, it shares some features with metabolic syndrome (MetS), including insulin resistance and oxidative stress, but with a different pattern of antioxidant response. Finally, it is known that GH, directly or via IGF-1 activity, influences many aspects of immune response; both humoral and cellula...

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ea0070aep752 | Pituitary and Neuroendocrinology | ECE2020

FDG PET contribution to ectopic prolactinoma diagnosis - case report

Krcma Michal , Fuchsova Radka

Pituitary adenomas are in the majority of cases diagnosed by magnetic resonance imaging (MRI). In some cases, especially in functional tumors, there are too small adenomas for MRI resolution and we need other diagnostic modality (including petrosal sinus catheterization for laterality). Usefulness of positron emission tomography (PET) with fluorodeoxyglucose was demonstrated in some case report. In this case, we present 36 y/o women with no other comorbidities, which came at f...

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ea0070aep753 | Pituitary and Neuroendocrinology | ECE2020

SF-12 or SF-36 in pituitary disease? towards concise patient-reported outcomes measurements

Van der Meulen Merel , Biermasz Nienke , Zamanipoor Najafabadi Amir , Lobatto Daniel , van Furth Wouter , Andela Cornelie , Pereira Alberto M , Vliet-Vlieland Thea P.M.

Objective: Pituitary diseases cause a wide range of local and systemic symptoms and severely affect patients’ health-related quality of life (HRQoL), which can be monitored using both disease-specific and generic questionnaires. The most frequently used generic HRQoL questionnaire is the Short Form-36 (SF-36), generating a mental (MCS) and physical component score (PCS). The shorter 12-item version (SF-12) can improve efficiency of patient monitoring and outcome measurem...

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ea0070aep754 | Pituitary and Neuroendocrinology | ECE2020

The role of surgery in the treatment of craniopharyngioma: A comparison of the transsphenoidal approach and the interbasal hemisphere approach based on surgical outcomes

Matsuo Takayuki , Kamada Kensaku

The transsphenoidal approach has been utilized in intrasellar craniopharyngioma surgeries. However, the advent of endoscopic extended transsphenoidal approach (EETSA) has expanded its indication to suprasellar craniopharyngiomas. We compared the indication and limitations of EETSA to those of unilateral basal interhemispheric approach (UBIHA), which presents similar indications for surgery. We analyzed 60 patients with tumors located below the foramen of Monro and the lateral ...

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ea0070aep755 | Pituitary and Neuroendocrinology | ECE2020

An analysis of incidence and characteristics of cushing’s syndrome in malta: A population based study

Giordano Imbroll Miriam , Giovanna Abela Alexia , Vassallo Josanne , Gruppetta Mark

Aim: There are few reports discussing incidence and characteristics of the whole group of Cushing’s syndrome patients in the literature. The aim was to establish the incidence of endogenous Cushing’s syndrome with in-depth analysis of their various subtypes in a well-defined population.Methods: Retrospective cross-sectional analysis of Cushing’s syndrome patients diagnosed between 2008 and 2017. A thorough search for patients was carrie...

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ea0070aep756 | Pituitary and Neuroendocrinology | ECE2020

Patients receiving a range of doses of prior injectable somatostatin receptor ligands respond to oral octreotide in the treatment of acromegaly: Results from the phase 3 optimal study

Nachtigall Lisa , Samson Susan , Fleseriu Maria , Bolanowski Marek , Molitch Mark E , Ludlam William , Patou Gary , Haviv Asi , Manning Patrick , Biermasz Nienke , Giustina Andrea , Strasburger Christian J , Melmed Shlomo , Kennedy Laurence

Background: Injectable somatostatin receptor ligands (SRLs) are the most widely used therapy to control acromegaly. Oral octreotide capsules (OOC) have been formulated as a potential therapy for this disorder and the efficacy and safety was evaluated in the CHIASMA OPTIMAL prospective phase 3 study in patients with acromegaly who were controlled on injectable SRL treatment of varying doses (Samson et al. ENDO 2020).Methods: Patients with confirm...

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ea0070aep757 | Pituitary and Neuroendocrinology | ECE2020

AIP gene germline mutations in non-selected patients with sporadic pituitary macrodenomas

Trofimiuk-Muldner Malgorzata , Domagała Bartosz , Sokolowski Grzegorz , Skalniak Anna , Pach Dorota , Hubalewska-Dydejczyk Alicja

Up to 5% of all pituitary tumors are hereditary (e.g. due to menin or AIP genes mutations). AIP gene mutations are more common in subjects with acromegaly, less than 30 years old at the onset of disease, and with FIPA family history. The study was aimed at the assessment of the frequency and characteristics of AIP-mutation related tumors in non-selected patients with pituitary macroadenomas.Material and Methods: The study included subsequent 131 patients...

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ea0070aep758 | Pituitary and Neuroendocrinology | ECE2020

Silent corticotroph adenoma: Experience with six cases

Serra Soler Guillermo , Antich Barceló Carlos , Bodoque Cubas Javier , García Fernández Honorato , Mas Bonet Antonio , Antón Valentí Ester , Mena Ribas Elena , Tofé Povedano Santiago , Argüelles Jiménez Iñaki , Pereg Macazaga Vicente

Introduction: Silent corticotroph adenomas (SCA) represent a subtype of pituitary adenomas whose prevalence is 3% of pituitary adenomas. They are characterized by having a positive immunohistochemistry to corticotropin (ACTH) without clinical or biochemical data of hypercortisolism. The diagnosis is a challenge since they are considered as ANF, and it is with the pathology report after surgery when they are classified as SCA. They have more aggressive behavior and recur more t...

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ea0070aep759 | Pituitary and Neuroendocrinology | ECE2020

MRI follow-up of patients with acromegaly treated with first-generation somatostatin analogues (SMSa): Is there a difference during primary or post-operative treatment?

Naia Grandgeorge , Giovanni Barchetti , Solange Grunenwald , Fabrice Bonneville , Caron Philippe

Objectives: First-generation SMSa are the medical treatment of choice in the management of acromegaly, mainly as adjuvant treatment of pituitary surgery when normal IGF-1 is not obtained or as primary treatment in selected patients. The main objective of this study is to evaluate regular pituitary MRI follow-up of acromegalic patients treated with SMSa as adjuvant treatment of pituitary surgery. The secondary objective is to compare the anti-tumoral effect of SMSa in post-oper...

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ea0070aep760 | Pituitary and Neuroendocrinology | ECE2020

Associations of endocrine and metabolic complications of childhood- and adult-onset craniopharingeoma with the histological subtype

Jazbinsek Soncka , Jensterle Mojca , Kotnik Primoz

Objective: Studies comparing endocrine and metabolic outcomes of childhood- and adult-onset craniopharyngioma (CP) are generally based on the age-of-disease onset only. The aim of this study was to analyze the role of histological tumor subtype (adamantinomatous (aCP) or papillary (pCP)) on the development of long-term endocrine and metabolic CP comorbidities.Methods: 47 patients with CP treated from 1979 onwards (19 patients with childhood-onset disease...

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ea0070aep761 | Pituitary and Neuroendocrinology | ECE2020

The united russian registry on patients with acromegaly

Belaya Zhanna , Golounina Olga , Rozhinskaya Liudmila , Isakov Mikhail , Lutsenko Alexander , Przhiyalkovskaya Elena , Pigarova Ekaterina , Panyushkina Galina , Dreval Alexander , Melnichenko Galina

Background: The registry is the main source of information about patients with acromegaly for assessing the quality of medical care, effectiveness of treatment, determining the compliance of real clinical practice with existing standards and patient management protocols.Aims: To analyze the epidemiological, demographic and clinical characteristics of acromegaly in the Russian Federation and the effectiveness of various treatment methods.<p class="abs...

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ea0070aep762 | Pituitary and Neuroendocrinology | ECE2020

Immunohistochemical subtypes of growth hormone-secreted pituitary adenoma and relationship with the clinical course and seconder malignancy

Akkus Gamze , Cetinalp Ralp , Bagir Emine , Evran Mehtap , Sengoz Sinem , Zorludemir Suzan , Sert Murat , Tetiker Tamer

Purpose: Most of the acromegaly cases are due to agrowth hormone-secreting pituitary adenomas of which immunohistochemical subtype seperated as sparsely granulated adenoma (SGA) and densely granulated adenoma (DGA). SGA’s were reported to have more aggressive clinical course and therapy resistance. We investigated our patients for these aspects.Method: Forty (F21, M19) patients with acromegaly who diagnosed and operated for pituitary adenoma at our...

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ea0070aep763 | Pituitary and Neuroendocrinology | ECE2020

Prolactinoma In childhood and adolescence – a systematic review and meta-analysis

Bhushan Arya Ved , Aylwin Simon , Kapoor Ritika R , Buchanan Charles R

Background and ObjectiveData on the epidemiology, effects of dopamine agonists and long term outcome of prolactinoma in children and adolescents have been gradually accumulating but are still scarce. We conducted a systematic review and meta-analysis of published literature (1994 – 2019) to study the epidemiology of prolactinoma in patients < 20 years old, and determine the management strategies adopted.Methods: Relevant ...

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ea0070aep764 | Pituitary and Neuroendocrinology | ECE2020

Combined pitutitary hormone deficiency in a patient with charge syndrome

Nikitas Skarakis Spyridon , Karachaliou Feneli , Mitrogiorgou Marina , Simatou Aristofania , Bountouvi Evangelia , Papaevangelou Vassiliki

Background: CHARGE is an autosomal-dominant syndrome which includes a variable combination of ocular coloboma, heart defects, atresia of the choanae, retardation of growth and development, and genitourinary and ear abnormalities CHARGE syndrome has rarely been associated with anterior pituitary dysfunction and with structural abnormalities of the pituitary gland only twice. We report the case of a child with CHARGE association and congenital hypopituitarism due to structural a...

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ea0070aep765 | Pituitary and Neuroendocrinology | ECE2020

Controversies in the spectrum of GH-IGF-I axis disorders requiring replacement therapy

Rodari Giulia , Cavenaghi Ivan , Profka Eriselda , Giacchetti Federico , Arosio Maura , Giavoli Claudia

Background: Growth hormone deficiency (GHD) is the most frequent endocrinological disorder inchildren with short stature, but there are significant controversies in the diagnosis due to lack of reliablediagnostic criteria. Moreover, at final height (FH) attainment, many subjects diagnosed withisolated GHD re-test normal. It is not clear whether this represents a form of transient GHD or a false positive diagnosis during childhood.Aim: To evaluate differe...

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ea0070aep766 | Pituitary and Neuroendocrinology | ECE2020

Severe heart failure in a young male patient with unrecognized hypopituitarism consequent to hemorrhagic fever with renal syndrome and undiagnosed hemochromatosis

Caric Bojana , Stojanović Marko , Kos Ljiljana , Grbić Aleksandra , Đekić Danijel , Malešević Gabrijela , Miljić Dragana

Background: The partial or complete hypopituitarism is described as late complication of hemorrhagic fever with renal syndrome (HFRS). Imaging methods of pituitary gland examination in the chronic phase showed pituitary atrophy, but a precise pathogenic mechanism that causes pituitary damage in HRFS remains unclear. While hypopituitarism in HFRS is rarely described, cardiac failure as a known complication of hypopituitarism is even more rarely described. We present a case of s...

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ea0070aep767 | Pituitary and Neuroendocrinology | ECE2020

Severe hyponatremia revealing hypopituitarism of undetermined etiology

Zargni Asma , Oueslati Ibtissem , Skouri Wafa , Abidi Sahar , Yazidi Meriem , Khessairi Nadia , Chaker Fatma , Chihaoui Melika

Introduction: Hyponatremia represents a frequent electrolyte disorder. It is defined as a serum sodium level below 135 mmol/L and it is severe when serum level is below 125 mmol/l. Endocrine disorders including adrenal deficiency and hypothyroidism are uncommon causes of hyponatremia. Secondary adrenal insufficiency is related with hyponatremia through increased ADH secretion.Herein, we report a case of severe hyponatremia revealing hypopituitarism.<...

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ea0070aep768 | Pituitary and Neuroendocrinology | ECE2020

A case of isolated ACTH deficiency and autoimmune pancreatitis associated with pembrolizumab treatment

Unluturk Uğur , Hanife Oguz Seda , Aksoy Sercan , Erbas Tomris

Background: Endocrine-related adverse effects of immune check-point inhibitors (ICIs) have been identified in growing number of cancer patients.Case presentation: A 49-year-old male with laryngeal cancer applied to ouroutpatient clinic with weakness, loss of appetite, weight loss, nausea and vomiting for the last three weeks. He was diagnosed with T3N1M0 laryngeal squamous cell cancer seven months before this admission and was given concurrent cisplatin ...

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ea0070aep769 | Pituitary and Neuroendocrinology | ECE2020

Empty sella: Who thinks of it while studying hyponatremia?

Ramalho Ana , Costa Joana , Carvalho João , Maria Amador Susana , Resendes Sousa André , Marques Santos Sofia , Cantante Helena

Hyponatremia is a common disorder, especially among the elderly, and an important morbidity and mortality cause. Empty sella syndrome, on the other hand, is a rare entity, more common in women between their fourth and sixth decade of life, and endocrine dysfunction has been reported in 10–60% of the cases. A 68-year-old man, with a past medical history of essential hypertension and glaucoma, presented at the emergency room due to malaise, asthenia, somnolence and anorexi...

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ea0070aep770 | Pituitary and Neuroendocrinology | ECE2020

Massive pulmonary embolism in a newly diagnosed acromegaly patient

Hepsen Sema , Ucan Bekir , Akhanli Pinar , Ozturk Unsal Ilknur , Cakal Erman

Introduction: Acromegaly is caused by growth hormone (GH) and insulin-like growth factor (IGF-1) excess and leads to various comorbidities. However, the association between acromegaly and thromboembolic diseases is unclear. Herein, we present a newly diagnosed acromegaly patient with massive pulmonary embolism.Case report: A 61-year-old female was hospitalized with acromegaly pre-diagnosis. She complained about extensive arthralgia and snoring. She had h...

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ea0070aep771 | Pituitary and Neuroendocrinology | ECE2020

Persistent hypercortisolism due to cushing disease

Volkova Natalya , Degtyareva Yuliya , Ganenko Lilia , Davidenko Ilya , Dzherieva Irina , Reshetnikov Igor , Zibarev Alexander , Sorokina Julia

Background: Cushing’s disease (CD) develops as a result of ACTH-secreting pituitary adenoma. Transsphenoidal selective surgery (TSS) is the first-line therapy, and success is largely determined by the experience of the surgeon. There are several reasons for the persistence of the disease, but it is almost impossible to predict such an outcome in advance, which leads to multiple interventions and has a pronounced effect on the quality of life of patients.<p class="abs...

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ea0070aep772 | Pituitary and Neuroendocrinology | ECE2020

The effect of chronic glucocorticoid exposure on brown adipose tissue in patients with cushing’s disease

Selek Alev , Furkan Çayir Berat , Tarkun Ilhan , Sözen Mehmet , Cetinarslan Berrin , Canturk Zeynep , Gezer Emre

Objective: Various effects of glucocorticoids occur in brown adipose tissue depending on their duration of action. As a result of exposure to glucocorticoids, thermogenesis and oxygen use in brown fat tissue are reduced in animal studies. In this study, we aimed to compare the levels of UCP1 (uncoupling protein 1), Irisin, BMP 7 (bone morpogenic protein), PRDM16, which are brown addipose tissue markers, between patients with Cushing’s disease and healthy controls<p c...

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ea0070aep773 | Pituitary and Neuroendocrinology | ECE2020

A rare outcome after surgical removal of insulinoma- case presentation

Ghenoiu Sandra , Verdes Andreea , Dumitrașcu Anda , Capatina Cristina , Poiana Catalina

Introduction: Insulinoma is a rare tumor that causes spontaneous fasting hypoglycemia in a supposedly healthy adult. Mostly these tumors are single and benign, few are malignant and a minority is represented by multiple adenomas. The treatment of choice is surgical removal of the insulinoma, which cures the condition in the majority of cases. However, after performing the surgery, less than 10% of patients either develop diabetes mellitus or continue to have persistent hypogly...

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ea0070aep774 | Pituitary and Neuroendocrinology | ECE2020

Evaluation of factors associated with presence and growth of thyroid nodules in patients with acromegaly

Pascual-Corrales Eider , Valderrabano Herrero Pablo , Quiñones de Silva Jonathan , Baonza Saiz Gonzalo , Martínez Vaello Victoria , Araujo Castro Marta

Objective: To investigate factors associated with the presence and growth rate of thyroid nodules(TN) in patients with acromegaly.Methods: We retrospectively evaluated the medical records of 52 patients with acromegaly followed in our institution (University Hospital Ramón y Cajal). The frequencies of TN and papillary thyroid cancer (PTC) and potential factors associated with nodular thyroid disease (NTD) were investigated. In patients with NTD (<em...

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ea0070aep775 | Pituitary and Neuroendocrinology | ECE2020

FSH levels are related to E-cadherin expression and subcellular location in non-functioning pituitary neuroendocrine tumours

Jensen Kolnes Anders , Astrid Berland Øystese Kristin , Olarescu Cristina , Casar-Borota Olivera , Bollerslev Jens , Jørgensen Anders

Objectives: To study the effect of epithelial-to-mesenchymal transition (EMT) on hormone expression in gonadotroph non-functioning pituitary neuroendocrine tumours (NF-PitNET).Background: Gonadotroph PitNETs can express FSH and LH or be hormone negative, however they rarely secrete hormones. During tumour development, epithelial cells develop a mesenchymal phenotype. This process is characterised by decreased membranous E-cadherin and translocation of E-...

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ea0070aep776 | Pituitary and Neuroendocrinology | ECE2020

To optimize the acute octreotide suppression test in predicting the efficacy of long acting somatostatin analogues in acromegaly

Yuting Gao , Liao Zhihong , Du Qin , Cai Ke

A 6-hour octreotide suppression test (OST) is useful in the selection of patients with acromegaly for chronic somatostatin depot analogues treatment. However, it is time consuming and that brings inconvenience to patients. We aim to simplify the blood sampling of OST, and determine the reliability of a short version of the classic 6-hour OST. The data provided in the supplementary tables of the primary manuscript were used to re-analyze the efficacies of the simplified OST tes...

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ea0070aep777 | Pituitary and Neuroendocrinology | ECE2020

Acromegaly is associated with reduced socioeconomic status and more so in female patients: A nationwide population-based study

Dal Jakob , Nielsen Eigil H , Feldt-Rasmussen Ulla , Andersen Marianne , Feltoft Claus , Vestergaard Peter , Stochholm Kirstine , Otto Jorgensen Jens

Context: Acromegaly is a rare and insidious disease associated with severe somatic morbidity but data on socioeconomic status are scarce.Objective: To study the socioeconomic status in acromegaly in a population based, nationwide follow-up study.Patients and Methods: All incident cases of acromegaly (n = 576) during the period 1997–2010 were included, representing 10.116 years at risk. For every patient, 100 pers...

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