Searchable abstracts of presentations at key conferences in endocrinology
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25th European Congress of Endocrinology

Istanbul, Turkey
13 May 2023 - 16 May 2023

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ECE 2023 will be held in Istanbul, Turkey from 13-16 May 2023. Istanbul is known for its rich culture, historical sites and dynamic nightlife, and we are excited to come together in this city, and look forward to meeting old friends, making new ones, and sharing science, collaborations and ideas.

Poster Presentations

Pituitary and Neuroendocrinology

ea0090p129 | Pituitary and Neuroendocrinology | ECE2023

Asymptomatic Familial Hyperprolactinemia Caused by a Unique bi-Allelic Variant in the Prolactin-Receptor Gene

Natif Yoav , Jean Matan M. , Birk Ohad S. , Eskin-Schwartz Marina , Fraenkel Merav , Yoel Uri

Introduction: Hyperprolactinemia is usually an acquired condition. Typical clinical manifestations include hypogonadism, infertility, and galactorrhea. Rarely, hyperprolactinemia has been attributed to a variant in the prolactin receptor gene (PRLR), presenting as agalactia, without hypogonadism, infertility, or galactorrhea.Aim: In the current study we aimed to delineate the clinical phenotype and the genetic basis of marked hyperprolactinemia ...

ea0090p130 | Pituitary and Neuroendocrinology | ECE2023

Increased Plasma Leptin and Agouti-Related Peptide May Be Related To Sleep Alterations In Patients With Active Cushing’s Syndrome

Sulu Cem , Mefkure Ozkaya Hande , Kadioglu Pinar

Aim: To evaluate sleep architecture of patients with Cushing’s disease (CD) and to explore whether agouti-related peptide (AgRP) leptin, and interleukin-6 (IL-6) play a permissive role in sleep alterations.Methods: We performed polysomnography on 26 patients with active CD and age 26 age- and sex-matched control subjects. Blood samples were obtained from all participants for the analyses of AgRP, leptin, and IL-6. The laboratory and sleep-related pa...

ea0090p131 | Pituitary and Neuroendocrinology | ECE2023

Somatostatin receptor expression in pituitary tumours according to the 2022 WHO classification: An insight into the rarer morphological subtypes

Fookeerah Prishila , Varikatt Winny , McLean Mark

Introduction: Pituitary tumours may express somatostatin receptors (SSTR), which are a potential drug target to achieve tumour reduction and control of hormone secretion. There is a well-established role for somatostatin analogues in the management of acromegaly and Cushing’s disease. The recent 2022 WHO classification of pituitary tumours provides a comprehensive approach to the diagnosis of pituitary tumours, allowing identification of additional morphological subtypes....

ea0090p132 | Pituitary and Neuroendocrinology | ECE2023

Comparative Efficacy and Safety of Osilodrostat Vs Metyrapone for the Treatment of Cushing’s Syndrome – a Matching-Adjusted Indirect Treatment Comparison

Tyas Emma , Hickey Conor , Hemstock Matthew , Binowski Grzegorz , Rios Carl , Schmidt Fabian

Objectives: Endogenous Cushing’s Syndrome (CS) is a rare, chronic condition that results in high morbidity, caused by prolonged exposure to elevated levels of circulating free cortisol levels. Osilodrostat, a newly approved steroidogenesis inhibitor, has been shown to achieve fast and high rates of cortisol normalization, improving manifestations of hypercortisolism in patients with Cushing’s Disease (CD), a form of CS. We evaluated relative complete response (CR) ou...

ea0090p133 | Pituitary and Neuroendocrinology | ECE2023

Rare adverse event of treatment with somapacitan after switch from somatotropine

Rakusa Matej , Janez Andrej , Jensterle Mojca

Introduction: Somapacitan is 23.3-kDa human GH derivate linked to small noncovalent albumin binding moiety that facilitates reversible endogenous albumin binding to delay somapacitan elimination. The longer half-life and improved pharmacodynamics allow injections once per week. Some patients find a daily regimen with established treatment difficult, especially because treatment may be lifelong or at least for several years. Poor adherence with medication can lead to poorer cli...

ea0090p134 | Pituitary and Neuroendocrinology | ECE2023

Efficacy and Safety Analysis of Setmelanotide As a Novel Treatment for Hypothalamic Obesity

Roth Christian L. , Shoemaker Ashley H. , Gottschalk Michael , Miller Jennifer L. , Yuan Guojun , Chen Evan , Scimia Cecilia , Jennifer Abuzzahab M.

Background: Hypothalamic injury and impaired melanocortin-4 receptor (MC4R) pathway signaling, often a result of surgery or radiation for a benign tumor, may lead to hypothalamic obesity (HO). After injury, sudden weight gain and appetite changes unresponsive to existing therapies develop. Setmelanotide, an MC4R agonist, is approved for chronic weight management in patients with certain MC4R pathway–associated diseases. We report interim results of a Phase 2 study of setm...

ea0090p135 | Pituitary and Neuroendocrinology | ECE2023

Prolactinomas under high doses cabergoline in portuguese patients: The Resistant Prolactinoma Study

Manique Ines , Benido Silva Vania , Carvalho Davide , Marques Olinda , Nobre Ema , Joao Bugalho Maria , Palha Ana , Ribeiro Isabel , Pereira Josue , Almeida Rui , Sagarribay Amets , Cerqueira Luis , Amaral Claudia

Background: In 15% of prolactinomas there is dopamine agonist(DA) resistance. The clinical characteristics, the best diagnostic and therapeutic management of these cases have not been established so far.Aim: To characterize patients taking a high dose cabergoline(≥ 3mg/week) and then describe clinical, biochemical and imaging features of cabergoline-resistant cases.Methods: Retrospective study with 5 centers representing the ...

ea0090p136 | Pituitary and Neuroendocrinology | ECE2023

Characteristics of follicular-stellate cells in newborn pituitary gland

Nicoleta Nastase Valeria , Barb Alina , Cosma Andrei , Raluca Ceausu Amalia , Raica Marius

Introduction: The development of pituitary gland is orchestrated by genetic signals, transcription factors and signaling molecules with a crucial role in cell proliferation and differentiation. Anterior pituitary gland is formed by both granular (secretory) and agranular cells represented mainly by follicular-stellate cells. It is known that these cells form a supporting network around secretory cells and produce many kind of growth factors, but their major function remains un...

ea0090p137 | Pituitary and Neuroendocrinology | ECE2023

The Effect of Dual-release vs Conventional Hydrocortisone on the Metabolic Profile in Secondary Adrenal Insufficiency

Thurmann Jorgensen Nanna , Brun Boesen Victor , Willemoes Borresen Stina , Christoffersen Thea , Rye Jorgensen Niklas , Watt Torquil , Feldt-Rasmussen Ulla , Klose Marianne

Background and aim: The dual-release hydrocortisone Plenadren® has shown promising results of improved cardiovascular and metabolic profiles after treatment of adrenal insufficiency, possibly due to an improved circadian profile of cortisol, but results are ambiguous. Aim: To further investigate the potential effect of dual-release hydrocortisone on the metabolic profile as compared to conventional hydrocortisone in patients with secondary adrenal i...

ea0090p138 | Pituitary and Neuroendocrinology | ECE2023

Post operative copeptin does not predict occurrence of the syndrome of inappropriate antidiuresis (SIAD) after pituitary transsphenoidal surgery

Efthymiadis Agathoklis , Rostom Hussam , James Tim , Shine Brian , Guha Nishan , Cudlip Simon , Christ-Crain Mirjam , Pal Aparna

Introduction: The incidence of the syndrome of inappropriate antidiuresis (SIAD) post pituitary surgery is estimated at 3-20% with the condition potentially leading to significant morbidity and prolonged admission. Copeptin, a surrogate marker for vasopressin activity, has been shown to be a diagnostic tool in the diagnosis of AVP deficiency (formerly known as diabetes insipidus) post pituitary surgery. There is limited data regarding the utility of copeptin in predicting SIAD...

ea0090p139 | Pituitary and Neuroendocrinology | ECE2023

Patients with acromegaly continue to derive clinical benefit from pasireotide long-acting release (LAR) for up to 12 years of treatment: Follow-up analysis of patients who received pasireotide during the clinical trial programme

Gadelha Monica , Grineva Elena , Miguel Escalante Pulido Jesus , Rollin Guilherme , Leelawattana Rattana , De Block Christophe , Piacentini Andrea , Schmidt Fabian , Pedroncelli Alberto M , Kapoor Nitin

Introduction: Acromegaly is associated with significant morbidity and reduced quality of life. A robust clinical programme (14 trials) established pasireotide as an effective second-generation somatostatin receptor ligand for the treatment of various endocrine conditions, including acromegaly. Patients who completed a previous pasireotide trial and continued to receive clinical benefit, according to the parent study investigator, could continue treatment in the B2412 rollover ...

ea0090p140 | Pituitary and Neuroendocrinology | ECE2023

Molecular and clinical relevance of the somatostatin system in brain tumors

De la Rosa-Herencia Ana S. , G-Garcia Miguel E. , Vazquez-Borrego Mari C. , Blanco Acevedo Cristobal , Solivera Juan , Gahete Manuel D. , Fuentes-Fayos Antonio C. , M Luque Raul

High-grade astrocytomas (HGAs) remain the most prevalent and malignant brain tumor based on its locally aggressive behavior, being glioblastoma (grade-IV astrocytoma) the most lethal. The current standard treatment for glioblastomas consists of surgery followed by radiotherapy and/or chemotherapy. However, the average period of survival is about 14 months after the first intervention. Therefore, there is a clear clinical need for the identification of novel diagnostic, prognos...

ea0090p141 | Pituitary and Neuroendocrinology | ECE2023

177Lu DOTATATE Peptide Receptor Radionuclide Therapy (PRRT) in advanced Phaeochromocytomas and paragangliomas (PPGL) – a single centre experience at a ENETS Centre of Excellence

Mansukhbhai Shekhda Kalyan , Armeni Eleni , Hayes Aimee , Caplin Martyn , Toumpanakis Christos , Mandair Dalvinder , Quigley Ann-Marie , Navalkissoor Shaunak , Grossman Ashley , Khoo Bernard

Introduction: Phaeochromocytomas and paragangliomas (PPGL) are rare neuroendocrine tumours with no standardised protocol for treatment. 177Lu-DOTATATE peptide receptor radionuclide therapy (PRRT) is an emerging treatment option for patients with metastatic and/or inoperable PPGL, and with low toxicity. We present our experience with PRRT in advanced PPGL, treated in a ENETS Centre of Excellence.Materials and Methods: 165 patients with...

ea0090p142 | Pituitary and Neuroendocrinology | ECE2023

Characterization of a large cohort of patients with “micromegaly”: long-term follow-up and preliminary data on treatment response

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

Background: In a previous study, we retrospectively analysed a group of patients with high insulin growth factor 1 (IGF1) but normal growth hormone (GH) suppression, identifying among them a subgroup of “micromegalic” patients presenting with clinical features of acromegaly and high rate of comorbidities. We therefore expanded our cohort of patients, extended the follow-up time and collected preliminary data on treatment response aiming to better characterize this co...

ea0090p143 | Pituitary and Neuroendocrinology | ECE2023

Role of fT4 and TSH-index fluctuations as early diagnostic tools in milder forms of central hypothyroidism: data from 221 patients with pituitary lesions from a single tertiary center

Carrara Silvia , Galazzi Elena , Nicoli Federico , Moro Mirella , Fatti Letizia , Cangiano Biagio , Persani Luca

Diagnosis of Central Hypothyroidism (CeH) is commonly given when FT4 concentrations are below the lower limit of normal range. A reduction in FT4 concentrations greater than 20% was proposed in ETA 2018 Guidelines as an unphysiological fluctuation indicating the onset of milder forms of CeH with FT4 still within the normal range. Similarly, TSH-index was proposed to quantify thyrotrope reserve, hence as a tool to detect patients at risk of CeH. Here we verified the performance...

ea0090p144 | Pituitary and Neuroendocrinology | ECE2023

Glucocorticoid Resistance Syndrome: a Rare Disorder that we Must not Forget. A Clinical Case

Martin Timon Iciar , Moreno-Ruiz Inmaculada , Meizoso-Pita Olalla , Huguet Isabel , Llavero Valero Maria , Trivino Yannuzzi Vanessa , Sevillano-Collantes Cristina

Primary generalized glucocorticoid resistance or Chrousos syndrome is a rare disorder, which affects all tissues expressing the human glucocorticoid receptor. It has been defined by an absence of overt Cushing’s syndrome (CS) signs (no skin weakness, muscle atrophy, or osteoporosis) associated with biological hypercortisolism consisting of high urinary free cortisol (UFC) and an absence of negative feedback loop of cortisol on HPA, defined as 8-AM cortisol level >50 n...

ea0090p145 | Pituitary and Neuroendocrinology | ECE2023

Somatostatin receptor type 2 (SSTR2) expression regulation by miR-375 in a murine pituitary corticotroph tumor cell model

Pivonello Claudia , Patalano Roberta , Monto Tatiana , Negri Mariarosaria , Amatrudo Feliciana , Paola Provvisiero Donatella , Simeoli Chiara , Paola Nicola , Larocca Angelica , Massimo Crescenzo Erminio , Colao Annamaria , Pivonello Rosario

Cushing’s disease (CD), caused by an adrenocorticotropic hormone (ACTH)-secreting pituitary tumor, is the most common form of hypercortisolism. Pituitary surgery is the first-line treatment of CD and medical treatment is an alternative second-line approach to control cortisol excess. Long-term exposure to glucocorticoids (GC) downregulates the expression of somatostatin receptor type 2 (SSTR2) but not type 5 (SSTR5) in human and mouse ACTH-secreting tumor cell cultures; t...

ea0090p146 | Pituitary and Neuroendocrinology | ECE2023

Dopamine agonist-resistance in patients with macroprolactinoma: prevalence and predictive factors

Terzi Ameni , Oueslati Ibtissem , Hadj Sliman Chayma Bel , Abid Arige , Yazidi Meriem , Chihaoui Melika

Introduction: Dopamine agonists, mainly cabergoline and bromocriptine, represent the first-line treatment option in the management of prolactinomas. They allow prolactin normalization and tumor shrinkage. However, a subset of individuals with macroprolactinoma could exhibit a varying degree of resistance to dopamine agonists. The aim of this study was to determine the prevalence of dopamine agonist-resistance in patients with macroprolactinoma and to assess its predictive fact...

ea0090p147 | Pituitary and Neuroendocrinology | ECE2023

Baseline clinical and treatment characteristics of dopamine agonist treated patients in a Dutch national cohort of patients with prolactinoma

Zandbergen Ingrid , Pelsma Iris , van Furth Wouter , Biermasz Nienke

Introduction: Dopamine agonists (DA) are first-line treatment for prolactinomas and effectively control hyperprolactinemia in most patients. However, side effects may negatively impact health-related quality of life; and post-withdrawal remission rates are low, resulting in prolonged DA treatment.Methods: Dutch multicenter prospective observational cohort study mapping standard care for 316 prolactinoma patients. Baseline clinical characteristics and tre...

ea0090p148 | Pituitary and Neuroendocrinology | ECE2023

Uncontrolled Acromegaly, Diabetes Mellitus and Metabolic Syndrome are the Main Risk Factors of Colon Polyposis Recurrence in Patients with Acromegaly

Pirchio Rosa , Simona Auriemma Renata , Vergura Alice , Pivonello Rosario , Colao Annamaria

Colon polyps represent a frequent complication of acromegaly. Knowledge about prevalence and risk factors of polyps’ recurrence in acromegalic patients is still limited. The current retrospective study aimed at investigating polyps’ prevalence and recurrence risk factors in acromegalic patients. One hundred and forty-three patients (73 males, 70 females) referring to “Federico II” university diagnosed with acromegaly from 2000 to 2022 who underwent at least...

ea0090p149 | Pituitary and Neuroendocrinology | ECE2023

Is a 20% decrease in free T4 (fT4) levels a reliable marker of secondary hypothyroidism in patients with non-functioning pituitary macroadenoma (NFPA)?

McLaren David , Mitchell Ellen , Z Safdar Nawaz , Gibbons Stephen , D Murray Robert

Background: Central hypothyroidism can be difficult to diagnose as TSH values often reside within the normal range. The Endocrine Society clinical guideline for hormone replacement in hypopituitarism suggests levothyroxine is indicated for treatment of secondary hypothyroidism where fT4 levels decrease by ≥20%. To determine the reliability of this biochemical marker of secondary hypothyroidism we evaluated evolution of TFTs over time within a cohort of NFPA.<p class=...

ea0090p150 | Pituitary and Neuroendocrinology | ECE2023

Epidemiology, clinical presentation, treatment and outcome of acromegaly in the island of Crete, Greece. 27 years of follow up study

Chrysoulaki Maria , Vergou Stavrina , Maria Makri Artemis , Mpouhlaki Elpida , Michailidis Stefanos , DARAKI Vasiliki , Sfakiotaki Maria , betsi Grigoria , Kontolaimaki Kalliopi , Vamvoukaki Rodanthi , Sifaki-Pistola Dimitra , Lionis Christos , Goulielmos Georgios , Xekouki Paraskevi

Background: Acromegaly is a rare endocrine disorder due to growth hormone excess attributed to a pituitary tumor, in most cases. Epidemiological data of acromegaly are derived from national tumor registries or population studies while there are no published data in the Greek population except for a few small studies regarding treatment outcome.Methods: We carried out a retrospective ongoing observational study to determine the epidemiology, presentation ...

ea0090p151 | Pituitary and Neuroendocrinology | ECE2023

Novel provocation test with MDMA (“ecstasy’) reveals oxytocin deficiency as a new pituitary entity in patients with central diabetes insipidus – a randomized placebo-controlled trial

Atila Cihan , Holze Friederike , Murugesu Rakithan , Rommers Nikki , Hutter Nina , Varghese Nimmy , Clara Odilia Sailer , Eckert Anne , Heinrichs Markus , Liechti Matthias , Christ-Crain Mirjam

Introduction: Patients with arginine vasopressin deficiency (AVP-D), known as central diabetes insipidus (cDI), often report psychological symptoms such as heightened anxiety levels, difficulties describing emotions, and depressed mood despite adequate treatment with desmopressin. Given the anatomical proximity, disruptions of the hypothalamic-pituitary axis causing an AVP-D could also disturb the oxytocin (OXT) system. OXT regulates socio-emotional functioning, including fear...

ea0090p152 | Pituitary and Neuroendocrinology | ECE2023

Protein tyrosine phosphatase receptors N and N2 regulate gonadotropin-releasing hormone neuron function

Sokanovic Srdjan , Constantin Stephanie , Lamarca Dams Aloa , Mochimaru Yuta , Smiljanic Kosara , Bjelobaba Ivana , Maso Previde Rafael , Stojilkovic Stanko

Simultaneous knockout of the neuroendocrine marker genes Ptprn and Ptprn2, which encode the protein tyrosine phosphatase receptors N and N2, respectively, causes infertility of female mice while males are fertile. To clarify the mechanism of sex-specific roles of Ptprn and Ptprn2 in mice reproduction, we analyzed the effects of their double knockout (DKO) on the hypothalamic-pituitary-gonadal axis. In DKO females, a delay in puberty and lack...

ea0090p153 | Pituitary and Neuroendocrinology | ECE2023

Comparison of Clinical, Hormonal, Pathological and Treatment Outcomes of Ectopic ACTH Syndrome by Gender: Results of a Multicenter Study

Ciftci Sema , Yilmaz Nusret , Soyluk Selcukbiricik Ozlem , Hekimsoy Zeliha , Gokcay Canpolat Asena , Topsakal Senay , Yaylali Guzin , Misiroglu Fuat , Gul Nurdan , Kubat Uzum Ayse , Hacioglu Aysa , Yorulmaz Goknur , Ozisik Hatice , Sarer Yurekli Banu , Pekkolay Zafer , Karaca Zuleyha , Duran Cevdet , Nur Kebapci Medine , Yarman Sema

Objective: Data on gender comparisons of diagnosis, management and prognostic factors in Cushing’s disease are very limited, however, there is no such a comparison in ectopic ACTH syndrome (EAS) in the literature. The aim of this study is to compare clinical and hormonal data, neuroendocrine tumor (NET) localization, treatment and survival outcomes in Cushing’s syndrome due to EAS by gender.Material-Methods: Eleven experienced centers from our ...

ea0090p154 | Pituitary and Neuroendocrinology | ECE2023

Clinical utility of the Octreotide Challenge Test in Acromegaly

Khan Irfan , Guma Muna , Mamoojee Yaasir , James Andy

Background: Acromegaly is a rare condition characterised primarily by tumourous production of excess Growth Hormone (GH) from a pituitary adenoma. The Octreotide Challenge Test (OCT) has been used in the RVI to predict short term efficacy of long-acting Somatostatin Analogue (SSA) therapy.Aims: We retrospectively reviewed the OCT results of all patients with acromegaly treated at RVI from 2005 to 2021 to evaluate its clinical utility.<p class="abstex...

ea0090p155 | Pituitary and Neuroendocrinology | ECE2023

Development of a new score to predict dopamine agonist resistance in patients with macroprolactinoma

Terzi Ameni , Oueslati Ibtissem , Hadj Sliman Chayma Bel , Ayari Sabrina , Yazidi Meriem , Chihaoui Melika

Introduction: Surgical treatment is indicated in patients with dopamine agonist-resistant macroprolactinoma. However, the resection of the tumor is usually partial because of fibrotic changes under long-term dopamine agonist therapy. Thus, early identification and management of patients at high risk of dopamine agonist resistance is crucial to a complete resection outcome and a better remission. The aim of this study was to develop a score predicting dopamine agonist resistanc...

ea0090p156 | Pituitary and Neuroendocrinology | ECE2023

Incidence of complications of acromegaly 10 years after achieving biochemical control of GH – a single centre retrospective observational study

Mulholland Sarah , Clayton Richard , Jose Biju , Varadhan Lakshminarayanan

Aim: Acromegaly is associated with multiple co-morbidities including diabetes mellitus (DM), obstructive sleep apnoea (OSA), arthropathy, cardiovascular events and malignancies. Early diagnosis and treatment, with an intention to cure or control the growth hormone (GH) exposure can reduce risk of these complications. The aim of this study is to assess the development of acromegaly-related complications amongst patients who have completed at least 10 years of follow up after ac...

ea0090p157 | Pituitary and Neuroendocrinology | ECE2023

Various cut-off points for paradoxical increase of growth hormone after glucose load in patients with acromegaly and their clinical implications

Godlewska Magdalena , Bogusławska Anna , Grochowska Anna , Hubalewska-Dydejczyk Alicja , Gilis-Januszewska Aleksandra

Introduction: Paradoxical increase of growth hormone after glucose load (PR) and its clinical significance remain the focus of researchers. Different cut off points for diagnosis of PR are used in the literature. Aim : Our aim was to investigate 3 different definitions of PR and their clinical implications.Material and Methods: We analyzed 116 consecutive patients diagnosed with acromegaly in 2012-2022. We included 89 patients with...

ea0090p158 | Pituitary and Neuroendocrinology | ECE2023

Expression of omentin in the porcine anterior pituitary and action on cell proliferation, cell cycle, and secretion of FSH, LH, PRL, GH, TSH and ACTH

Respekta Natalia , Pich Karolina , Mlyczyńska Ewa , Dobrzyń Kamil , Opydo Małgorzata , Kamiński Tadeusz , Smolińska Nina , Dupont Joelle , Rak Agnieszka

Background : Omentin, also known as intelectin-1, is a member of the family of adipose tissue-related hormones known as adipokines, that exert anti-inflammatory, anti-atherosclerotic, and cardiovascular protective effects. Omentin expression has been detected in adipocytes as well as in a variety of extra-adipose tissues, including the ovary. Interestingly, omentin expression in the pituitary has been shown to be regulated by pituitary hormones. These observations led us to in...

ea0090p159 | Pituitary and Neuroendocrinology | ECE2023

Mild Autonomous Cortisol Secretion May Be Related To Decreased Thalamic Volume

Sulu Cem , Kadioglu Pinar , Mefkure Ozkaya Hande

Aim: To investigate the volumetric changes in cortical and subcortical deep gray matter (GM) structures and cognitive alterations in patients with mild autonomous cortisol secretion (MACS). We also sought to assess whether the anticipated changes in brain volume and cognitive functions of the patients with MACS differ from the changes in patients with overt Cushing’s Syndrome (CS). Methods: In this cross-sectional study, volumes of the cortex, hippo...

ea0090p160 | Pituitary and Neuroendocrinology | ECE2023

Multimodal therapy for the management of an aggressive corticotroph tumour after bilateral adrenalectomy

Fookeerah Prishila , Varikatt Winny , McLean Mark

Background: Rapid progression of corticotroph tumours can occur after bilateral adrenalectomy for treatment of Cushing’s disease. There are currently no guidelines to assist with management of aggressive cases of Nelson’s syndrome, particularly after surgery and radiotherapy.Clinical case: A 53 year old woman presented with classic clinical features of hypercortisolism. Initial investigations showed a markedly elevated 24 hour urine cortisol le...

ea0090p161 | Pituitary and Neuroendocrinology | ECE2023

Neurosarcoidosis Presenting with Panhypopituitarism

Al-Qaysi Amina , Chandran Shanthi

Introduction: Sarcoidosis is a multisystem inflammatory disorder that affects the lungs and the lymph nodes commonly. The involvement of the nervous system in this disease is infrequent with prevalence lower than 10%. The diagnosis could be challenging when the neurologic disease presents in a patient without previously known sarcoidosis.Case Report: A 39-year-old Caucasian gentleman was brought to the Emergency department with a one week’s history ...

ea0090p162 | Pituitary and Neuroendocrinology | ECE2023

An unusual sellar dermoid cyst

Akbar Shahzad , Taqi Muhammad , Shah Najeeb , Mongolu Shiva , Aye Mo

Introduction: Dermoid cysts are rare (0.5% of intracranial tumours). They are best considered a spectrum: from epidermoid cysts containing desquamated squamous epithelium to teratomas containing all three embryonic tissues. Sellar dermoid cysts are even rarer [ ID: ORPHA:91351]. Case: A 47-year old white British male was referred following incidental discovery of bitemporal field defect on routine eye test. He was otherwise completely asymptomat...

ea0090p163 | Pituitary and Neuroendocrinology | ECE2023

Pituitary apoplexy as the first manifestation of non-functioning pituitary neuroendocrine tumor

Dzialach Lukasz , Sobolewska Joanna , Respondek Wioleta , Wojciechowska-Luzniak Agnieszka , Witek Przemysław

Introduction: Pituitary apoplexy (PA) is a rare but potentially life-threatening complication that may occur in pituitary neuroendocrine tumors (PitNETs). Non-functioning PitNETs (NF-PitNETs), specifically macroadenomas, seem to have a higher risk of apoplexy. Case description: A 45-year-old male presented to the emergency unit with a one-week history of binocular visual deterioration (blurred vision, diplopia, visual field defect) with left-side predomi...

ea0090p164 | Pituitary and Neuroendocrinology | ECE2023

FTO gene polymorphisms and their role in acromegaly

Jawiarczyk-Przybyłowska Aleksandra , Kuliczkowska-Płaksej Justyna , Halupczok-Żyła Jowita , Kolačkov Katarzyna , Zembska Agnieszka , Zdrojowy-Wełna Aleksandra , Bolanowski Marek

Acromegaly is characterized by two-fold higher mortality than in the general population. The leading causes of morbidity and mortality in patients with acromegaly are cardiovascular diseases (CVD). Many population-based studies suggest a role of genetic background in CVD and associated metabolic disturbances. In 2007, FTO (fat mass and obesity associated) gene polymorphisms were discovered. According to a few studies, the polymorphisms of FTO gene are associated with ...

ea0090p165 | Pituitary and Neuroendocrinology | ECE2023

Longer surveillance period needed for Ectopic ACTH secretion and Cushing’s syndrome – two unusual cases

Htut Zin , Bahowairath Fatima , Todd Jeannie

Ectopic Cushing’s syndrome is a rare condition accounting for 10–20% of all cases of ACTH-dependent Cushing. Neuroendocrine tumours (NETs), mainly bronchial carcinoids and small cell lung cancer, are the most frequent causes of ectopic ACTH secretion(1). Here we report two challenging cases with long duration of surveillance and monitoring.Case 1: A 42-year-old lady with a florid Cushing syndrome underwent bilateral adrenalectomy in 2002. An IP...

ea0090p166 | Pituitary and Neuroendocrinology | ECE2023

Doege-Potter Syndrome, a case report

Guerrero Alba , Romero Fabiola , Lopez Helen , Hun Lee Chong , Muller Alejandra , Rodriguez Roque , Valinotti Elizabeth

Introduction: 2 to 3% of the causes of hypoglycemia in the population without diabetes mellitus correspond to tumors of cells other than islets. Below is a rare case of hypoglycemia related to a pleural tumor. Clinical case: A 65-year-old woman with arterial hypertension, psychiatric treatment for confusion and behavioral changes of 6 months of evolution, palpitations and night sweats, loss of 20 kg of weight, several admissions to the emergency room, un...

ea0090p167 | Pituitary and Neuroendocrinology | ECE2023

Somatostatin analogue treatment is associated with lack of progression of pNETs <20mm in size in patients with MEN1

Frizelle Isolda , Velusamy Anand , McGowan Barbara , Breen Louise , Duncan Emma , Izatt Louise , Christodoulou Dimitra , Joshi Mamta , Paul Carroll

Introduction: Pancreatic neuroendocrine tumours (pNETs) are the main cause of mortality in patients with multiple endocrine neoplasia type 1 (MEN1). The CLARINET study demonstrated that somatostatin analogue treatment improved progression free survival in patients with enteropancreatic NETs but little is known about the role of SSA in preventing progression of pNETs in MEN1 (1). Many centres have started using SSA treatment in MEN1 patients with enlarging pNETs or tumours &#62...

ea0090p168 | Pituitary and Neuroendocrinology | ECE2023

Quantitative assessment of the signal intensity of somatotropin pituitary tumors and its clinical implications in consecutive newly-diagnosed patients with acromegaly

Godlewska Magdalena , Bogusławska Anna , Grochowska Anna , Hubalewska-Dydejczyk Alicja , Gilis-Januszewska Aleksandra

Introduction: Somatotroph Pituitary Neuroendocrine Tumor’s (sPitNET) signal intensity in T2-weighted MR images (SI) was reported as prognostic marker for tumor’s aggressiveness and poor response to 1st generation somatostatin analogs (SSA). Tumors are arbitrary divided into 3 groups: hyperintensive (HYPER), isointensive (ISO) and hypointensive (HYPO). Aim: Our aim was to quantitatively assess SI of sPitNETs and investigate its clinic...

ea0090p169 | Pituitary and Neuroendocrinology | ECE2023

Lymphocytic Hypophysitis – many faces of the same disesase. Watchful waiting could be suggested therapeutic approach in majority of patients

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

Introduction: Clinical manifestation of hypophysitis depends on presence of hormonal abnormalities and/or enlargement of pituitary structures. Both the diagnosis and treatment still remain challenging. Hormonal replacement is the basic therapy, while steroids are the first-line treatment in case of mass related symptoms. The course of the disease varies from spontaneous remission to atrophy of the pituitary gland.Aim: To characterize and sum up clinical/...

ea0090p401 | Pituitary and Neuroendocrinology | ECE2023

Mortality in acromegaly diagnosed in the elderly in Spain is higher in women compared to the general Spanish population

Biagetti Betina , Iglesias Pedro , Villar Taibo Rocio , Dolores Moure Maria , Paja Fano Miguel , Araujo Castro Marta , Ares Blanco Jessica , Cristina Alvarez Escola Maria , Vicente Almudena , Alvarez Guivernau Elia , Novoa-Testa Iria , Cordido Carballido Fernando , Camara-Gomez Rosa , Lecumberri Beatriz , Garcia Gomez Carlos , Bernabeu Ignacio , Manjon Miguelez Laura , Gaztambide Sonia , Webb Susan , Luis Menendez Torre Edelmiro , Jose Diez Juan , Simo Canonge Rafael , Puig-Domingo Manel

Context: There is no data on mortality of acromegaly diagnosed in the elderly. Objective: To compare clinical characteristics, GH-related comorbidities, therapeutic approaches and mortality of patients diagnosed before or after 2010 and to assess overall mortality compared with the general Spanish population. Setting: Spanish tertiary care centers.Design, Patients, and Methods: Retrospective evaluation of 118...

ea0090p402 | Pituitary and Neuroendocrinology | ECE2023

Treatment Patterns in Acromegaly: Analysis of Real-World US Insurance Claims from the MarketScan® Database

Brue Thierry , Fleseriu Maria , Barkan Ariel , Duquesne Edouard , Houchard Aude , del Pilar Schneider Maria , Ribeiro-Oliveira Antonio , Melmed Shlomo K.

Background: Medical treatment for acromegaly (characterized by excess growth hormone [GH] production) includes somatostatin receptor ligands (SRLs), dopamine agonists (DAs), and GH receptor antagonists (GHRAs). However, recent real-world United States (US) treatment evaluations are few. We present treatment patterns for patients receiving medications for acromegaly (1/1/2010–31/7/2022).Methods: De-identified data were extracted from MarketScan&...

ea0090p403 | Pituitary and Neuroendocrinology | ECE2023

Bilateral Inferior Petrosal Sinus Sampling and Utility of Maximum Stimulated Petrosal Sinus ACTH to Baseline ACTH ratio in Adrenocorticotropin-dependent Cushing’s Syndrome

Maldar Aasim , Chauhan Phulrenu , Kulkarni Aniruddha , Misra B. K. , Shah N. F. , Chadha Manoj

Methods: The diagnosis of Cushing’s Syndrome (CS) was made as per the prevalent endocrine society guidelines. CS patients with serum ACTH>15 pg/ml were considered as ACTH-dependent CS. Baseline ACTH (both inferior petrosal sinuses and peripheral) values were obtained, and since 2014, stimulated ACTH values (5 and 10 minutes post one unit iv vasopressin) were obtained as standard protocol. Prolactin corrected ACTH ratios were additionally being calculated since 2016. The h...

ea0090p404 | Pituitary and Neuroendocrinology | ECE2023

Prevalence of transient and permanent diabetes insipidus after transsphenoidal pituitary surgery: A systematic review and meta-analysis

Fountas Athanasios , Coulden Amy , Allotey John , Karavitaki Niki

Introduction: Patients undergoing transsphenoidal pituitary surgery (TPS) may develop water balance disorders post-operatively, with diabetes insipidus (DI) being the most common. Nonetheless, data on the prevalence of post-operative DI are not consistent necessitating a systematic review of the literature.Aim: To estimate the prevalence of DI following TPS in patients with pituitary adenomas (PAs), craniopharyngiomas and Rathke’s cleft cysts (RCCs)...

ea0090p405 | Pituitary and Neuroendocrinology | ECE2023

The TRH test is a valuable diagnostic test in central hypothyroidism with low T4

Norma Smith* Liv , Ellegaard Christensen* Sara , Rosendal Christian , Angela Gulisano Helga , Ettrup Kare , H Nielsen Eigil , Karmisholt Jesper , Dal Jakob

Objective: To evaluate the value of the thyrotropin-releasing hormone (TRH) test in the diagnosis of central hypothyroidism (CH) in patients with pituitary disease.Methods: Systematic evaluation of 368 TRH tests including measurements of TBG corrected thyroxine (T4corr), baseline TSH (TSH0) and fold and absolute TSH increase (TSHfold, TSHabsolute) in patients with pituitary disease.Result...

ea0090p406 | Pituitary and Neuroendocrinology | ECE2023

Somatotroph Pituitary Neuroendocrine Tumors (Pitnets)/Adenomas Expressing Steroidogenic Factor (SF1)

Guaraldi Federica , Roncaroli Federico , Villa Chiara , Zoli Matteo , Waqar Mueez , Higham Claire , Lally Imogen , Baussart Bertrand , Assie Guillaume , Morandi Luca , Mazzatenta Diego , Asioli Sofia

Rationale and aim: A recent pangenomic study documented a subset of somatotroph mostly GNAS-wildtype PitNET/adenomas co-express steroidogenic factor 1 (SF1). Aim of our study was to investigate clinical and molecular features of a cohort of these tumour subtype. Study sample: We identified 20 cases of SF1-expressing somatotropinomas out of 173 (11.6%) operated via transsphenoidal endoscopic approach in 3 referral Pituitary Centers patients. Pre- and post...

ea0090p407 | Pituitary and Neuroendocrinology | ECE2023

TGFβ1 and activins as new therapeutic targets for the treatment of dopamine-resistant prolactinomas

Pena-Zanoni Milagros , Ines Abeledo-Machado Alejandra , Bornancini Dana , Diaz-Torga Graciela

Background: Pituitary tumours are commonly benign adenomas, accounting for 10-15% of all intracranial neoplasms. Among functioning pituitary tumours, prolactinomas are the most frequently observed in the clinic (about 40%). Dopamine, acting through the dopamine type 2 receptor (Drd2), is the main inhibitor of lactotroph proliferation and prolactin secretion, therefore, these tumours are usually treated with dopaminergic agonists with a high efficiency. However, there is a subs...

ea0090p408 | Pituitary and Neuroendocrinology | ECE2023

Pooled analysis from two osilodrostat Phase III studies in Cushing’s disease (LINC 3 and LINC 4): Clinical improvements according to urinary and late-night salivary cortisol levels

Newell-Price John , Fleseriu Maria , Pivonello Rosario , Feelders Richard , Gadelha Monica , Lacroix Andre , Witek Przemysław , Heaney Anthony , Piacentini Andrea , Pedroncelli Alberto , Biller Beverly M.K.

Introduction: In two Phase III studies (LINC3, NCT02180217; LINC4, NCT02697734), osilodrostat, (potent oral 11β-hydroxylase inhibitor), provided rapid, sustained reductions in mean urinary free cortisol (mUFC) and late-night salivary cortisol (LNSC), alongside improvements in clinical signs of hypercortisolism and health-related quality of life (HRQoL), in Cushing’s disease (CD) patients. mUFC and LNSC are recommended for monitoring treatment response. We assessed wh...

ea0090p409 | Pituitary and Neuroendocrinology | ECE2023

Inflammatory infiltrate in wild-type and non-wild-type GNAS somatotropinomas

Chiloiro Sabrina , Cicia Martina , Giampietro Antonella , Gessi Marco , Lauretti Liverana , Paolo Mattogno Pier , Carlino Angela , Olivi Alessandro , Rindi Guido , Pontecorvi Alfredo , De Marinis Laura , Doglietto Francesco , Bianchi Antonio

Introduction: Somatotropinomas are benign pituitary tumors with heterogeneous biological and clinical behavior. The tumor microenvironment (TME) is an environment generated by the interaction between tumor cells and the host immune system. It affects both the behavior of the tumor and the outcome of the therapy. The GNAS gene encodes the alpha subunit of G proteins associated with hormone receptors such as TSH, PTH, GHRH, FSH/lH, ACTH. Mutation of this gene has been associated...

ea0090p410 | Pituitary and Neuroendocrinology | ECE2023

Impact of Surgery or Medical Treatment with the Selective Glucocorticoid Receptor Modulator Relacorilant on Hypercoagulopathy in Patients with Cushing Syndrome

Simeoli Chiara , Di Paola Nicola , Stigliano Antonio , Lardo Pina , Kearney Tara , Mezosi Emese , Ghigo Ezio , Giordano Roberta , Mariash Cary N. , Donegan Diane , Feelders Richard , Hand Austin , Moraitis Andreas G. , Pivonello Rosario

In patients with Cushing syndrome (CS), hypercoagulability represents a significant concern, leading to elevated risk for thrombotic events. After curative surgery, hypercoagulability persists for several months; CS treatment guidelines recommend anticoagulation therapy for ≤ 3 months. In patients with Cushing disease, hemostatic parameters may even worsen after surgery, independent of surgical outcome; improvements begin ~3 months after successful surgery (Casonato ...

ea0090p412 | Pituitary and Neuroendocrinology | ECE2023

Copeptin levels increase in response to both insulin-induced hypoglycemia and arginine but not to clonidine – data from GH-stimulation tests

Stankovic Jelena , Kristensen Kurt , Birkebaek Niels , Otto Jorgensen Jens , Sondergaard Esben

Background: The differential diagnosis of the polyuria-polydipsia syndrome is challenging. The water-deprivation test is the current gold standard, but the test is cumbersome, and the diagnostic performance is poor. Copeptin, which is a split product of the vasopressin pre-propeptide, appears to be a robust biomarker in the circulation and a promising tool for the diagnosis of patients with polyuria and polydipsia, especially when measured in conjunction with intravenous infus...

ea0090p413 | Pituitary and Neuroendocrinology | ECE2023

Sleep Disorders And Osas Risk in Patients with Prolactinoma: A Single Center Experience

Del Vecchio Guendalina , Sorice Federica , Scairati Roberta , Pirchio Rosa , Di Meglio Sara , Pivonello Rosario , Colao Annamaria , Simona Auriemma Renata

Purpose: Dopaminergic system is implicated in the development of sleep disorders, and dopaminergic agents are used in clinical practice to treat daytime sleepiness and sleep disorders. The present study aimed at investigating the effects of hyperprolactinemia and its control with dopamine-agonists (DA) on sleep quality, excessive daytime sleepiness (ESS) and risk of obstructive sleep apnea syndrome (OSAS) in prolactinomas.Methods: A single-centre study w...

ea0090p414 | Pituitary and Neuroendocrinology | ECE2023

Assessment of adrenal axis function in patients with PROP1 mutation – longitudinal observation

Kluczyński Łukasz , Rogoziński Damian , Zygmunt-Gorska Agata , Wojcik Małgorzata , Piwońska-Solska Beata , Starzyk Jerzy , Hubalewska-Dydejczyk Alicja , Gilis-Januszewska Aleksandra

Introduction: Mutation in PROP1 gene is the cause of different forms of pituitary dysfunction. The assessment of the functioning of the adrenal axis still raises the most doubts.Aim& methods: A retrospective longitudinal (mean 35 years, SD 16.24) analysis of 32 patients (19W/13M, including 5 families and sporadic cases) with PROP1 mutation was performed to define the optimal diagnostic approach in evaluation of adrenal axis. In the study morning cort...

ea0090p415 | Pituitary and Neuroendocrinology | ECE2023

Synchronous multiple pituitary neuroendocrine tumors (PitNETs) consisting of a null cell and PIT-1 lineage adenomas: a peculiar acromegalic case report

Bruco Alessia , Zoli Matteo , Melotti Sofia , Ribichini Danilo , Pizza Gianmarco , Mao Federico , Mazzatenta Diego , Asioli Sofia , Pagotto Uberto

Pituitary adenomas constitute the third most common intracranial neoplasm. Adenoma cells either express no hormone (null cell adenoma) or hormones of different cell lineage. About 1% of pituitary adenomas shows a plurihormonal expression pattern, which may either be attributed to one cell lineage or rarely to different cell lineages. In addition, the development of separate pituitary adenomas may be observed in 0.5–1.5% of surgical specimens and in up to 10% of autopsy ca...

ea0090p416 | Pituitary and Neuroendocrinology | ECE2023

Treatment, complications and mortality of Cushing’s Disease: twenty-year report from a referral Centre

Mondin Alessandro , Ceccato Filippo , Voltan Giacomo , Mazzeo Pierluigi , Tizianel Irene , Scaroni Carla , Barbot Mattia

Context: Cushing’s disease (CD) is rare condition burdened by several systemic complications that in turn increase mortality. The main goal of CD treatment is to promptly reduce cortisol excess, but whether remission can revert cortisol-related complications and guarantee a normal life expectancy remains debated. Aim: To assess the prevalence of cortisol-related complications and mortality in a large monocentric cohort of CD patients followed at our...

ea0090p417 | Pituitary and Neuroendocrinology | ECE2023

A negative association between growth hormone secretion and inflammation in adult patients with non-functioning pituitary mass

Seki Yasufumi , Bokuda Kanako , Morimoto Satoshi , Watanabe Satoshi , Yamashita Kaoru , Takano Noriyoshi , Watanabe Daisuke , Ichihara Atsuhiro

Background: Growth hormone (GH) deficiency, which causes visceral obesity and non-alcohol fatty liver disease, increases cardiovascular event risks. High-sensitivity C-reactive protein (hs-CRP), an inflammatory marker associated with increased cardiovascular risk, reportedly decreased after GH supplementation in GH-deficient patients, however, the association between GH secretion and inflammation remains unclear.Patients and Methods: We retrospectively i...

ea0090p418 | Pituitary and Neuroendocrinology | ECE2023

Search for new biomarkers of adult growth hormone deficiency metabolic syndrome: a comprehensive overview of a four peptides analysis

Mancini Antonio , Vergani Edoardo , Bruno Carmine , Oliva Alessandro , Curro Diego

Adult growth hormone deficiency (aGHD) is characterized by an altered metabolic profile and increased cardiovascular risk. Neudesin is a newly discovered protein mainly secreted from adipose tissue and brain, under evaluation for its possible activity as negative regulator of energy expenditure. Liver expressed antimicrobial peptide (LEAP)-2 is a competitive antagonist of ghrelin on its receptor. Spexin is a polypeptide related to the galanin-kisspeptin family, secreted along ...

ea0090p419 | Pituitary and Neuroendocrinology | ECE2023

Reference intervals for soluble alpha klotho and the impact of biological variables

Ribeiro de Oliveira Longo Schweizer Junia , Schilbach Katharina , Haenelt Michael , Pauline Gagliardo Anica , Peters Anette , Thorand Barbara , Stormann Sylvere , Schopohl Jochen , Bidlingmaier Martin

Background: Alpha klotho is a transmembrane protein that regulates calcium metabolism. Its soluble portion has been linked to endocrine functions. We have recently shown high concentrations of serum soluble alpha klotho (sαKL) in active acromegaly. There is limited information about regulation of sαKL secretion. We aimed to gain insight about sαKL as a potential biomarker for H-related diseases.Methods: SαKL was measured by ELISA (IBL...

ea0090p420 | Pituitary and Neuroendocrinology | ECE2023

Italian guidelines for the management of prolactinomas

Cozzi Renato , Simona Auriemma Renata , Pasquale De Menis Ernesto , Esposito Felice , Ferrante Emanuele , Iati Giuseppe , Mazzatenta Diego , Poggi Maurizio , Ruda Roberta , Tortora Fabio , Cruciani Fabio , Mitrova Zuzana , Saulle Rosella , Vecchi Simona , Basile Michele , Cappabianca Paolo , Paoletta Agostino , Papini Enrico , Persichetti Agnese , Samperi Irene , Scoppola Alessandro , Bozzao Alessandro , Caputo Marco , Doglietto Francesco , Ferrau Francesco , Lania Andrea , Laureti Stefano , Lello Stefano , Locatelli Davide , Maffei Pietro , Minniti Giuseppe , Peri Alessandro , Ruini Chiara , Settanni Fabio , Silvani Antonio , Veronese Nadia , Grimaldi Franco , Attanasio Roberto

Aim: This guideline (GL) is aimed at providing a reference for the management of prolactin (PRL)-secreting pituitary adenoma in non-pregnant adults.Methods: For each question, the panel identified potentially relevant outcomes, which were then rated for their impact on therapeutic choices.Results: The present GL provides recommendations about the roles of pharmacological and neurosurgical treatment for the management of prolactinom...

ea0090p421 | Pituitary and Neuroendocrinology | ECE2023

Factors Affecting Quality of Life in Patients with Acromegaly: Single Center Experience

Merve Ok Ayşe , Bektaş Fatih , Bilik Oyman Gamze , Mutlu Ummu , Hacişahinoğulları Hulya , Yenidunya Yalin Gulsah , Gul Nurdan , Kubat Uzum Ayse , Aral Ferihan , Yarman Sema , Soyluk Selcukbiricik Ozlem

Objective: Despite treatment, the quality of life in patients with acromegaly remains lower than in the healthy population due to the classical findings and complications related to the disease. Therefore, in the follow-up of acromegaly, evaluation of quality of life along with symptoms and biochemical parameters is recommended. The aim of our study was to evaluate the quality of life, anxiety and depression status of our acromegaly patients and to assess the factors affecting...

ea0090p422 | Pituitary and Neuroendocrinology | ECE2023

Effects of a GLP-1 agonist on consumption of alcohol in patients treated for smoking cessation

Sofia Probst Leila , Monnerat Sophie , Lengsfeld Sophia , Burkard Thilo , Meienberg Andrea , Bathelt Cemile , Christ-Crain Mirjam , Winzeler Bettina

Introduction: Alcohol use disorder AUD causes high socio-economic costs and has a detrimental impact on health globally, being considered a key risk factor for non-communicable diseases. In Switzerland, four substances are currently approved for treatment of AUD, with varying effects. Taking into consideration the complex nature of the disease and the heterogeneity of affect patients, it becomes evident that there is need for new treatment targets. A body of preclinical studie...

ea0090p423 | Pituitary and Neuroendocrinology | ECE2023

Immune profile in acromegalic patients and the impact of the current medical treatment. Resultsfrom the PROMISE study

Feola Tiziana , De Alcubierre Dario , Sada Valentina , Puliani Giulia , Sciarra Francesca , Cozzolino Alessia , Appetecchia Marialuisa , Sbardella Emilia , Jaffrain-Rea Marie-Lise , Anna Venneri Mary , Isidori Andrea

Context: Acromegaly is associated with several metabolic and cardio-respiratory comorbidities, with a relative increase in the last years of cancer mortality. Although the GH/ IGF-1 axis has long been supposed to play a role on immune modulation, immune function in acromegaly is poorly known.Objective: This observational, prospective, single site, pilot study (NCT05069324) aims to analyse peripheral blood mononuclear cells (PBMCs) subpopulations in acrom...

ea0090p424 | Pituitary and Neuroendocrinology | ECE2023

The acro-TIME score: a new clinical, pathological and immune integrative approach to early identify acromegaly patients resistant to treatment with first generation somatostatin ligands

Chiloiro Sabrina , Giampietro Antonella , Gessi Marco , Lauretti Liverana , Mattogno Pierpaolo , Costanza Flavia , Carlino Angela , Olivi Alessandro , Rindi Guido , Pontecorvi Alfredo , De Marinis Laura , Doglietto Francesco , Bianchi Antonio

Somatotropinomas are benign pituitary tumors, with a heterogenous biological and clinical behavior. Tumor microenvironment reflects the interaction between tumor cells and the host immune system and may regulate tumor behavior and therapy outcome. We develop a scoring system that includes clinical, pathological and immune markers to early identify fg-SRLs resistant acromegaly pts. 43 acromegaly pts were included according the following criteria (1) first line treatment with su...

ea0090p425 | Pituitary and Neuroendocrinology | ECE2023

Longitudinal multiomics characterization of paired primary and recurrent aggressive pituitary tumors from the same patient reveals genomic stability and transcriptomic and epigenetic heterogeneity with metabolic pathways alterations

Taniguchi Keiko , Chavez-Santoscoy Alejandra , Hinojosa-Alvarez Silvia , Hernandez-Perez Jesus , Valenzuela-Perez Alejandra , Vela Patino Sandra , Andonegui-Elguera Sergio , Cano-Zaragoza Amayrani , Martinez Florencia , Kerbel Jacobo , Zhang Zidong , Smith Gergory , Rubenstein Aliza , Sze Cheng Wan , Mendelev Natalia , Anne Amper Mary , Zamojski Michel , Zaslavsky Elena , Ruf-Zamojski Frederique , Sealfon Stuart , Mercado Moises , Marrero-Rodriguez Daniel

Pituitary tumors (PT) represent 20% of all intracranial neoplasms. Some behave aggressively, growing rapidly and invading surrounding tissues, with high recurrence rates and are frequently resistant to conventional therapies. We performed exome and transcriptome sequencing, as well as methylation profiling of primary and recurrent PT of the same patient. The cohort consisted of patients with GH- and gonadotropin-producing PT and a patient with an ACTH carcinom...

ea0090p426 | Pituitary and Neuroendocrinology | ECE2023

Acromegaly in the elderly: additional challenges in timely recognizing

Pekic Djurdjevic Sandra , Stojanovic Marko , Doknic Mirjana , Miljic Dragana , Milojevic Toplica , Milicevic Mihailo , Stanimirovic Aleksandar , Scepanovic Vuk , Manojlovic Gacic Emilija , Banjalic Sandra , Jovanovic Marija , Jemuovic Zvezdana , Nikolic Djurovic Marina , Grujicic Danica , Popovic Vera , Petakov Milan

Background: Increased life expectancy and wider availability of neuroimaging lead to increase in diagnosis of acromegaly in elderly patients, after the age of 70. Delay in diagnosis is attributable to milder symptomatology, higher prevalence of comorbidities and symptoms overlapping with normal aging process.Objective: To investigate clinical characteristics, presenting symptoms, comorbidities and treatment modalities of acromegaly in patients diagnosed ...

ea0090p427 | Pituitary and Neuroendocrinology | ECE2023

Long-term Surgical Remission in Patients with Acromegaly in South-Eastern Norway

Maria Falch Camilla , Kirstine Dupont Anne , Olarescu Cristina , Bollerslev Jens , Berg-Johnsen Jon , Heck Ansgar

Context: Immediate and sustained cure of acromegaly can only be achieved by surgery, however, most growth hormone (GH) producing adenomas are macroadenomas (≥ 10 mm) at diagnosis, with reported surgical cure rates of approximately 50% compared to microadenomas (<10 mm) with an expected higher cure rate (around 80%). Further, long-term data on remission rates after surgery are sparse. Aim: Estimate short- and long-term surgical remission rates o...

ea0090p428 | Pituitary and Neuroendocrinology | ECE2023

Effects of acromegaly treatment on left ventricular systolic function assessed by speckle tracking echocardiography: results from a prospective single-center study

Popielarz-Grygalewicz Agata , Stelmachowska-Banaś Maria , Raczkiewicz Dorota , Kochman Wacław , Zgliczyński Wojciech

Background: Despite the preserved left ventricular(LV) ejection fraction, patients with acromegaly are characterized by subclinical systolic dysfunction, i.e. abnormal global longitudinal strain(GLS) assessed by speckle-tracking echocardiography(STE). The effect of acromegaly treatment on LV systolic function assesed by STE has not been evaluated so far.Patients and methods: Thirty two naïve acromegaly patients with no detectable heart disease were ...

ea0090p429 | Pituitary and Neuroendocrinology | ECE2023

Clinicopathological aspects of growth-hormone and prolactin-producing pituitary neuroendocrine tumors

Dumitriu Roxana , Florentina Burcea Iulia , Nicoleta Nastase Valeria , Raluca Ceausu Amalia , Raica Marius , Maria Cimpean Anca , Poiana Catalina

Introduction: Pituitary neuroendocrine tumors (PitNET) are characterized by a heterogenous behavior. The immunohistochemical (IHC) staining of anterior pituitary hormones, along with that of pituitary transcription factors and with the clinical and biochemical characteristics of the patients can be integrated in a risk stratification system, which helps the therapeutic management.Material and Methods: We analyzed 37 patients with confirmed diagnosis of a...

ea0090p430 | Pituitary and Neuroendocrinology | ECE2023

Menin, p27 and pAKT role in prolactinoma development

Pena-Zanoni Milagros , Ines Abeledo-Machado Alejandra , Bornancini Dana , Marcial-Lopez Agustina , Rulli Susana , Diaz-Torga Graciela

Molecular mechanisms involved in lactotrophs cell proliferation that led to prolactinoma development are not fully understood. Cyclin-dependant kinase (CDK) p27Kip1 is a key cell cycle regulator, and it is regulated by Menin. The transgenic mice model MEN1+/-, as the p27 -/- knock-out mice, develop pituitary adenomas and other neuroendocrine tumours. Menin can also interfere with phosphorylated AKT (pAKT) function, known for i...

ea0090p431 | Pituitary and Neuroendocrinology | ECE2023

Morphological and functional parameters of the cardiovascular system in acromegalic patients: impact of body composition

Sagova Ivana , Dragula Milan , Mokaň Marian , Vanuga Peter

Objective: Cardiovascular diseases are the most common comorbidities in acromegaly. Potential parameters in pathology of cardiovascular comorbidities are changes in levels of growth hormone (GH) and insulin-like growth factor 1 (IGF-1) as well as body composition parameters.Purpose: The aim of this study was to examine morphological and functional parameters of the cardiovascular system by echocardiography and to assess its relationship with disease acti...

ea0090p432 | Pituitary and Neuroendocrinology | ECE2023

Design of the foresiGHt Trial: A Multicenter, Randomized, Placebo- and Active-Controlled Trial to Compare Once-Weekly Lonapegsomatropin to Placebo and Daily Somatropin in Adults with Growth Hormone Deficiency (GHD)

Gilis-Januszewska Aleksandra , Fleseriu Maria , Otto Jorgensen Jens , CJ Yuen Kevin , Hoybye Charlotte , Mao Meng , Kang Jennifer , Song Wenjie , Komirenko Allison , Shu Aimee , Beckert Michael

Background: Adult GHD results from insufficient growth hormone (GH) secretion from the anterior pituitary gland and may represent either a continuation of childhood-onset GHD or GHD acquired during adulthood. Clinically, adult GHD is associated with central adiposity, decreased lean muscle mass, increased fat mass, decreased bone mineral density, and reduced quality of life. Current standard of care consists of GH replacement via daily injections. Lonapegsomatropin (SKYTROFA; ...

ea0090p433 | Pituitary and Neuroendocrinology | ECE2023

Lanreotide-Induced Acalculous Acute Pancreatitis in a Person with Acromegaly: an Unexpected Severe Side Effect

Ramalho Diogo , Alves Helena , Henrique Carmona Alexandrino Jorge , Jesus Nuno , Melo Gustavo , Joao Oliveira Maria

Introduction: Somatostatin analogues (SA) are the first-line treatment for Acromegaly in patients whose surgery failed to achieve cure or it was contraindicated. The main reported adverse events of SA are mild and transient, and include nausea, abdominal cramps, diarrhea and flatulence. Other side effects are of rare occurrence.Case report: The authors describe a case of a 44-year-old previously asymptomatic male patient with a growth hormone-secreting p...

ea0090p434 | Pituitary and Neuroendocrinology | ECE2023

Awaken a sleeping giant: Incidentally discovered pituitary gigantism

Rusu Eva , Peica Andreea , Gliga Tudor , Milea Gheorghe Ana , Adrian Radu Mircea , Stanoiu- Pinzariu Oana , Emanuela Georgescu Carmen

Introduction: Pituitary gigantism is a rare disorder characterized by growth hormone (GH) excess that occurs before epiphyseal growth plates fusion leading to rapid and excessive linear growth in childhood and very tall adult stature. It can be sporadic or coexist with genetic disorders such as FIPA, X-LAG, McCune-Albright, Carney complex, MEN 1 or 4, and Neurofibromatosis type 1.Case report: We present a case of a 12 years old boy with no medical histor...

ea0090p435 | Pituitary and Neuroendocrinology | ECE2023

Osilodrostat as an effective and safe treatment for ectopic Cushing Syndrome- case report

Minasyan Mari , Boguslawska Anna , Rzepka Ewelina , Kluczyński Łukasz , Gilis-Januszewska Aleksandra , Hubalewska-Dydejczyk Alicja

Osilodrostat as an inhibitor of adrenal 11B-hydroxylas, is an effective medication used in the management of endogenous hypercortisolism. There are limited data regarding usage of osilodrostat in ectopic Cushing Syndrome (CS). Surgery is usually the first line treatment in ectopic CS, however the source of ectopic hormonal production often remains undiscovered for a long period of time. Meanwhile dangerous state of severe hypercortisolism needs to be managed pharmacologically ...

ea0090p436 | Pituitary and Neuroendocrinology | ECE2023

The Indian Male Aging Study Shows Differential Relationships Between Age and Modifiable Risk Factors for Hypothalamic-Pituitary-Testicular Axis Disruptions in Older Men

Chandra Abhishek , Kumar Singh Vijay

Introduction: It is unknown what causes ageing men to have lower testosterone levels and how those levels relate to risk factors. Less research has been done on the health of ageing males than on postmenopausal women.Objective: The aim was to look into how men-aging lifestyles and health related to their levels of reproductive hormones.Method: A prospective cohort study in Indian men included 320 community-dwelling men between the ...

ea0090p437 | Pituitary and Neuroendocrinology | ECE2023

Acromegaly management in the Scandinavian countries – a DELPHI consensus survey

Arlien-Soborg Mai C. , Dal Jakob , Heck Ansgar , Stochholm Kirstine , H Nielsen Eigil , Feltoft Claus , Krogh Rasmussen AEse , Feldt-Rasmussen Ulla , Andreassen Mikkel , Klose Marianne , Leo Nielsen Torben , Andersen Marianne , Lehmann Christensen Louise , Krogh Jesper , Jarlov Anne , Lyngvi Fougner Stine , Nermoen Ingrid , Bollerslev Jens , Oksnes Marianne , Dahlqvist Per , Olsson Tommy , Berinder Katarina , Hoybye Charlotte , Petersson Maria , AEkerman Anna-Karin , Wahlberg Jeanette , Ekman Bertil , Eden Engstrom Britt , Johannsson Gudmundur , Ragnarsson Oskar , Agusta Sigurjonsdottir Helga , Burman Pia , Otto Jorgensen Jens

Background: Acromegaly is associated with increased morbidity and mortality if left untreated. The therapeutic options include surgery, medical treatment, and radiotherapy. Guidelines regarding treatment algorithms and follow-up vary considerably.Aim: To evaluate consensus on the treatment and follow-up of acromegaly in the Scandinavian countries.Methods: To map the landscape of acromegaly management in the Scandinavian countries, ...

ea0090p438 | Pituitary and Neuroendocrinology | ECE2023

The Role of the GH Receptor Polymorphism as Prognostic Factor of Vertebral Fractures in Acromegaly Patients Resistant to First Generation SSAs and Treated with GH Receptor Antagonist or Second-Generation Somatostatin Ligand

Costanza Flavia , Chiloiro Sabrina , Giampietro Antonella , Mattogno Pierpaolo , Amato Infante , Angelini Flavia , Lauretti Liverana , Olivi Alessandro , Pontecorvi Alfredo , Doglietto Francesco , De Marinis Laura , Bianchi Antonio

Acromegaly is associated with skeletal fragility and an increased prevalence of vertebral fractures (VF). In recent years several authors have tried to investigate the markers that can predict the risk of bone fragility in this endocrine disorder. Two different isoforms of the GH receptor (GHR) have been described so far, which differ in the presence or absence of a transcript of exon 3 of the GHR gene. Both isoforms produce a functional receptor, but the exon 3-deleted isofor...

ea0090p439 | Pituitary and Neuroendocrinology | ECE2023

Serial sampling of dried blood spots collected by a novel automated body-worn system, Fluispotter®

Krogh Jesper , Plomgaard Peter , Frikke-Schmidt Ruth , Velschow Sten , Johannesen Jesper , hilsted linda , Schroeder Malene , Feldt-Rasmussen Ulla

Repeated blood sampling is required in certain clinical and research settings, which is currently performed by drawing blood from venous catheters requiring manual handling of each sample at time of collection. A novel body-worn device for repeated serial samples, Fluispotter®, with automated extraction, collection and storage of up to 20 venous dried blood spot samples (DBS) over the course of 20 h may overcome problems with current methods for serial sampling. The purpo...

ea0090p440 | Pituitary and Neuroendocrinology | ECE2023

DUONEN multicenter study - personalized PRRT treatment with 177Lu- or 177Lu/90Y-DOTA-TATE in patients with neuroendocrine tumors based on individual dosimetry

Opalinska Marta , Kamiński Grzegorz , Dedecjus Marek , Kowalska Aldona , Kolodziej Maciej , Saracyn Marek , Garnuszek Piotr , Lenda-Tracz Wioletta , Borkowska Anna , Gąsior-Perczak Danuta , Budzyńska Anna , Kubik Agata , Kacperski Krzysztof , Szubstarska Patrycja , Chalewska Wioletta , Długosińska Joanna , Januszkiewicz-Caulier Joanna , Hubalewska-Dydejczyk Alicja , Mikolajczak Renata

Introduction: The good expression of somatostatin receptors in neuroendocrine tumor tissue enables effective treatment with peptide receptor radionuclide therapy (PRRT). To date, there is no clear consensus on the optimal PRRT arrangement due to the possibility of the use of different radiopharmaceutical regimens. In theory, the simultaneous use of two radionuclides (90Y and 177Lu) with different energy and radiation ranges should be more effective than monotherapy. The main o...

ea0090p441 | Pituitary and Neuroendocrinology | ECE2023

Prevalence and related risk factors of simple renal cysts and nephrolithiasis in acromegaly

Bostan Hayri , Kizilgul Muhammed , Calapkulu Murat , Kozan Kalkisim Hatice , Betul Gulden Topcu Fatma , Gul Umran , Duger Hakan , Hepsen Sema , Ucan Bekir , Cakal Erman

Background: The growth hormone (GH)/insulin-like growth factor-1 (IGF-1) axis has effects on renal growth and electrolyte regulation. However, data on adverse renal comorbidities such as renal cysts and stones in acromegaly is scarce. The current study aimed to investigate the prevalence of radiologically detectable renal comorbidities and possible related factors in the acromegaly population.Methods: A total of 125 patients with acromegaly (46.4&pm;11.6...

ea0090p672 | Pituitary and Neuroendocrinology | ECE2023

Phosphoproteomics analysis of aryl hydrocarbon receptor interacting protein (AIP) knockout cells reveals AIP-mediated kinase signalling cascades

Barry Sayka , Haworth Oliver , Rajeeve Vinothini , Cutillas Pedro , Korbonits Marta

Introduction: Aryl hydrocarbon receptor interacting protein (AIP) is a multifunctional co-chaperone protein: it behaves as a tumour suppressor in the pituitary, but may have other roles including oncogenic function in other tissues. Protein phosphorylation is an important posttranslational modification that regulates protein activity, which is crucial for understanding protein function. To understand the molecular pathways altered in AIP deficient cells, we have performed glob...

ea0090p673 | Pituitary and Neuroendocrinology | ECE2023

Pooled analysis of osilodrostat dosing across LINC 2, LINC 3 and LINC 4 in Cushing’s disease

Fleseriu Maria , Pivonello Rosario , Lacroix Andre , Biller Beverly M.K. , Feelders Richard , Gadelha Monica , Bertherat Jerome , Belaya Zhanna , Piacentini Andrea , Pedroncelli Alberto , Newell-Price John

Introduction: Phase II (LINC2, NCT01331239) and Phase III (LINC3, NCT02180217; LINC4, NCT02697734) studies showed that osilodrostat, a potent oral 11β-hydroxylase inhibitor, was an effective long-term treatment for Cushing’s disease patients. In this LINC programme pooled analysis, we examined how dose uptitration and adjustments during long-term maintenance can provide rapid, sustained mean urinary free cortisol (mUFC) control, and minimise AEs.<p class="abstext...

ea0090p674 | Pituitary and Neuroendocrinology | ECE2023

Sleep Quality and Associated Factors in Patients with Non-functioning Pituitary Adenoma

Barlas Tugba , Muhittin Yalcin Mehmet , Ecem Avci Doga , Kaplan Yigit , Akturk Mujde , Toruner Fusun , Karakoc Ayhan , Alev Altinova

Aim: Sleep disturbances are widespread and closely associated with pituitary diseases, even those under long-term therapeutic management. The aim of this study was to investigate sleep quality, depression, anxiety, and physical activity in patients with non-functioning pituitary adenoma (NFPA) and determine the factors that might be associated with decreased quality of sleep.Method: Eighty-two patients with NFPAs and 82 age- and gender-matched control su...

ea0090p675 | Pituitary and Neuroendocrinology | ECE2023

Psychopathological characteristics in patients with arginine vasopressin deficiency (central diabetes insipidus) and primary polydipsia

Beck Julia , Atila Cihan , Refardt Julie , Zoran Erlic , Drummond Juliana , Santana Soares Beatriz , Beuschlein Felix , Winzeler Bettina , Christ-Crain Mirjam

Introduction: The differential diagnosis between arginine vasopressin deficiency (AVP-D), formally known as central diabetes insipidus, and primary polydipsia (PP) is challenging. In clinical routine, psychopathologic findings are often used as a hallmark for diagnosing PP; thus, it is often referred to as psychogenic polydipsia. Yet, psychopathologic characteristics are barely assessed in patients with AVP-D, and to date, no data exist comparing AVP-D and PP with regard to th...

ea0090p676 | Pituitary and Neuroendocrinology | ECE2023

Hyperosmolar Hyperglycaemic State (HHS) and severe decompensated heart failure as presenting features of ectopic ACTH Syndrome

McDonnell David , Cussen Leanne , Martin-Grace Julie , Cotter Paul , Grogan Liam , Agha Amar

Ectopic ACTH syndrome is a rare condition occurring in five to ten percent of ACTH dependent hypercortisolism. We present the case of a fifty eight year old gentleman who presented with severe hyperosmolar hyperglycaemic state (blood glucose fifty seven mmol/l) and acute decompensated heart failure associated with elevated liver enzymes. Liver ultrasound followed by computed tomography of thorax abdomen and pelvis showed a lung tumour with liver metastases. Clinical suspicion ...

ea0090p677 | Pituitary and Neuroendocrinology | ECE2023

The diagnostic accuracy of dex-CRH test in differentiating between Cushing’s disease and pseudo-Cushing syndrome. A single-center experience

De Herdt Carlien , Dirinck Eveline , Eva Philipse , De Block Christophe

Background: Distinguishing pseudo-Cushing syndrome (PCS) from mild forms of Cushing’s disease (CD) is a challenge. The dexamethasone-Corticotropin Releasing Hormone test (dex-CRH) assumes that cortisol response to CRH is preserved in CD while suppressed in PCS after the low dose dexamethasone suppression test (LDDST). Methods: Persons with a suspicion of CD based on mild clinical features and a positive first-line screening test were included. They ...

ea0090p678 | Pituitary and Neuroendocrinology | ECE2023

Genetic Analyses Of 20 Turkish FIPA Families Utilizing Whole Exome Sequencing: Preliminary Results Of A Multicenter Collaborative Study

Eda Ertorer Melek , Tuncer Feyza , Ciftci Sema , Tanrikulu Seher , Soyluk Selcukbiricik Ozlem , Topaloğlu Omercan , Evran Mehtap , Kadioglu Pinar , Aydin Sevcan , Can Bulent , Sehit Canan , Pekkolay Zafer , Oruk Gonca , Cetinarslan Berrin , Yarman Sema

Objective: Hereditary pituitary adenomas (PAs) are rare and occur either isolated or as part of a syndrome. Familial isolated pituitary adenoma (FIPA) is the presence of only PA in at least two members of a family, where “Aryl hydrocarbon receptor interacting protein-AIP” gene mutations have been identified in 10-20% of cases. However, the cause of tumorigenesis in the majority is unknown. We aimed to identify novel genetic variants in a cohort of FIPA patie...

ea0090p679 | Pituitary and Neuroendocrinology | ECE2023

Prevalence of steatosis and organ-specific distribution of adipose tissue in patients affected by Cushing’s syndrome

Ferrante Emanuele , Oberti Giovanna , Mantovani Beatrice , Indirli Rita , Cremaschi Arianna , Sala Elisa , Carosi Giulia , Ludovica Fracanzani Anna , Mantovani Giovanna , Lombardi Rosa , Arosio Maura

Introduction: Endogenous hypercortisolism is associated with cardio-metabolic complications and promotes the deposition of lipids in different tissues, particularly in the liver. However, the prevalence of hepatic steatosis in patients with Cushing’s syndrome (CS) has been little investigated so far and only one previous study reported a prevalence of 20% using computed tomography. The aim of the study is to evaluate the prevalence of hepatic steatosis and the organ-speci...

ea0090p680 | Pituitary and Neuroendocrinology | ECE2023

Impact of Setmelanotide Treatment on Reducing Hyperphagia in Pediatric and Adult Patients With Hypothalamic Obesity

Roth Christian L. , Shoemaker Ashley H. , Gottschalk Michael , Miller Jennifer , Yuan Guojun , Malhotra Sonali , Scimia Cecilia , Jennifer Abuzzahab M.

Objective: To report hunger-related results from a Phase 2 trial of setmelanotide in patients with hypothalamic obesity (HO).Methods: A Phase 2, open-label, 16-week trial of setmelanotide in patients aged ≥6 to ≤ 40 years with body mass index (BMI) ≥95th percentile (aged <18 years) or ≥35 kg/m2 (aged ≥18 years) and HO caused by hypothalamic damage secondary to brain tumor, surgical resection, and/or chemothera...

ea0090p681 | Pituitary and Neuroendocrinology | ECE2023

Erythropoiesis in endogenous Cushing syndrome: sex-related and subtype-specific differences

Detomas Mario , Deutschbein Timo , Tamburello Mariangela , Chifu Irina , Kimpel Otilia , Sbiera Silviu , Kroisz Matthias , Fassnacht Martin , Altieri Barbara

Context: Endogenous Cushing syndrome (CS) is associated with hematological abnormalities. Nevertheless, conflicting data have been reported on erythropoiesis. Some studies reported an association between CS and anemia in men, whereas others described erythrocytosis in affected patients. Furthermore, it is unclear whether there are CS subtype-specific changes in red blood cells (RBC) parameters.Objective: To investigate sex and subtype-specific changes in...

ea0090p682 | Pituitary and Neuroendocrinology | ECE2023

A rollover study for patients who continued to receive benefit from pasireotide at completion of an earlier trial (B2412): An 8-year interim analysis

Gadelha Monica , Bronstein Marcello , Grineva Elena , Kapoor Nitin , De Block Christophe , Miguel Escalante Pulido Jesus , Rollin Guilherme , Baggenstoss Rejane , Piacentini Andrea , Pedroncelli Alberto , Gallardo Wilson

Introduction: A robust clinical programme of 14 trials demonstrated pasireotide as an effective treatment for patients with rare endocrine disorders, including acromegaly and Cushing’s disease (CD). Patients with acromegaly or CD have significant morbidity, reduced quality of life and, if inadequately treated, higher mortality risk than the general population. This 8-year interim analysis evaluated long-term safety of pasireotide treatment in patients with acromegaly, CD ...

ea0090p683 | Pituitary and Neuroendocrinology | ECE2023

Association of Pituitary Adenoma (Pa)/Pituitary Neuroendocrine Tumor (Pit-Net) and Cerebral Aneurysm: risk factors, peculiar features and management

Guaraldi Federica , Zoli Matteo , Nawabi Noah , Kilgallon John L. , Gori Davide , Asioli Sofia , Mazzatenta Diego , Richard Smith Tim

Rationale and aim: The incidence of intracranial aneurysms (IA) appears increased in patients with PAs/PitNETs. Changes in hemodynamics and vascular structure secondary to mass effect, inflammation, and hormonal changes (primarily, GH hypersecretion) have been suggested as risk factors. Study aim was to define typical features, timing of occurrence and treatment, and identify risk factors in patients with PA/PitNET and IA.Patients and methods: Data of 57...

ea0090p684 | Pituitary and Neuroendocrinology | ECE2023

Fibroblast Activation Protein in Patients with Growth Hormone Deficiency and Acromegaly: Before and After Treatment

Baek Amanda , Arlien-Soborg Mai C. , Kathrine Nissen Pedersen Anne , Jessen Niels , Bjerre Mette , Otto Lunde Jorgensen Jens

Background: It is well-known that growth hormone (GH) potently stimulates collagen turnover and it is associated with fibrosis in several tissues, but the mechanisms involved in GH-stimulated fibrosis are poorly elucidated. Fibroblast activation protein-α (FAPα) is an enzyme that can cleave collagens and is expressed almost only under pathological conditions such as fibrosis. FAPα also cleaves and inactivates fibroblast growth factor 21 (FGF21), which is a circu...

ea0090p685 | Pituitary and Neuroendocrinology | ECE2023

Panhypopituitarism in an Adult Patient with William-Beuren Region Duplication Syndrome: A Rare Case Report

Sema Yaman kalender Derya , Gasımlı Roya , Subaşıoğlu Aslı , Sercan Erturk Mehmet , Cavdar Umit , Gonca Oruk Guzide , Onder Pamuk Barış

Background: William-Beuren region duplication syndrome (WBDS) is a rare multisystem disease caused by the gain on chromosome 7q and transmitted autosomal dominant, with approximately a population frequency of 1 in 13,000-20,000. The age of diagnosis is variable, but generally, it is diagnosed during childhood. It include endocrine (growth hormone deficiency) and non-endocrine (facial dysmorphology, cardiovascular problems, gastrointestinal and genitourinary problems, neurologi...

ea0090p686 | Pituitary and Neuroendocrinology | ECE2023

Diagnostic criteria for the hypothalamic syndrome in childhood

van Santen Hanneke , van Schaik Jiska , van Roessel Ichelle , Boekhoff Svenja , Beckhaus Julia , Muller Hermann

Objective: Hypothalamic syndrome (HS) in childhood is a rare condition. Its epidemiology is not well known because incidence and prevalence are related to very rare underlying diseases. In addition, different criteria for the syndrome are used across studies. Recognizing HS may be difficult, due to its rareness and variety of symptoms. Having diagnostic criteria for signs and symptoms of hypothalamic dysfunction may aid in early recognition and diagnosis, in the reporting and ...

ea0090p687 | Pituitary and Neuroendocrinology | ECE2023

Smartphone App on Follow-up of Adult Growth Hormone Deficiency (AGHD) Patients: Results From the Management of AGHD (MAGHD) Study

Bellelli Alessio , Golinelli Caterina , Laura Monzani Maria , Pederzoli Simone , Magnani Elisa , Beltrami Elisa , Pacchioni Marco , Orsini Mirko , Diazzi Chiara , De Vincentis Sara , Rochira Vincenzo

Background: AGHD significantly impacts on metabolism, body composition and quality of life (QoL), including sexuality.AIM: To investigate the impact of a smartphone App on AGHD monitoring, especially QoL and comorbidities as well as on both body composition and biochemical parameters.Methodology: 83 AGHD patients were enrolled in this prospective, open-label, monocentric study (MAGHD Study) of 24-month duration. Patients were evalu...

ea0090p688 | Pituitary and Neuroendocrinology | ECE2023

Incidental pituitary macroadenoma: natural history and surgical results

Matei Constantinescu Stefan , Alexopoulou Orsalia , Duprez Thierry , Furnica Raluca , Fomekong Edward , Raftopoulos Christian , Maiter Dominique

Introduction: The Incidental diagnosis of non-functioning pituitary macroadenomas (NPFMA) is becoming more prevalent with the spread of modern imaging techniques. More clinical data about their natural history and surgical results are needed. Methods: We retrospectively analyzed medical files of patients referred to our clinic for an incidental NFPMA between 2010 and 2019. In particular, we compared patients that experienced tumor growth or not during su...

ea0090p690 | Pituitary and Neuroendocrinology | ECE2023

Evaluation of Serum Omentin-1 Levels and Atherosclerotic Risk Factors in Patients with Prolactinoma

Can Erdi Zuleyha , Ozkan Cigdem , Ozgur Ceylan Mustafa , Eren Guzel Hamza , Ercan Gizem , Bozkaya Giray , Hilal Adibelli Zehra

Background: Prolactinoma is the most common functioning pituitary adenoma. Subclinical atherosclerosis and cardiovascular risk in patients with prolactinoma were reported but have not been clarified yet.Aim: Our study aims to compare prolactinoma patients with a control group in terms of lipid profile, body fat distribution, and subclinical atherosclerosis markers.Methods: This single-center, prospective study included 32 patients ...

ea0090p691 | Pituitary and Neuroendocrinology | ECE2023

Diabetes Mellitus Secondary to Cushing’S Disease or Acromegaly: a Single-Centre Experience

Scairati Roberta , Di Meglio Sara , Pirchio Rosa , Vergura Alice , Di Paola Nicola , Larocca Angelica , Del Vecchio Guendalina , Simeoli Chiara , Simona Auriemma Renata , Colao Annamaria , Pivonello Rosario

Rationale: Type 2 diabetes mellitus (DM) is common in patients with Cushing’s disease (CD) and Acromegaly (AC), and contributes to increased mortality. This study aimed at investigating the prevalence and characteristics of DM in CD and AC patients at diagnosis and after disease control (DC).Patients and Methods: 34 CD patients (8M,26F, 24 microadenomas(mA),6 macroadenomas (MMA) and 4 empty sella) and 35 AC patients (16M,19F, 7 mA,28 MMA) were inclu...

ea0090p692 | Pituitary and Neuroendocrinology | ECE2023

Characterisation of the clinical and biologic behaviour of acromegaly caused by pluri-hormonal tumours in a cohort of 75 patients

Martins Ferreira Mafalda , Carreira Ana , Cardoso Luis , Oliveira Patricia , Melo Miguel , Paiva Isabel

Introduction: The clinical and biochemical spectrum of pituitary acromegaly is wide and variable. There are pure somatotroph tumors(ST), immunohistochemically positive only for GH, and pluri-hormonal(PL), also positive for other hormones, and they appear to have different presentations and responses to treatment.Aims: To compare the clinical presentation, imaging characteristics and response to treatment of acromegalic patients according to the pituitary...

ea0090p693 | Pituitary and Neuroendocrinology | ECE2023

Characterizing the expression of thyroid hormone transporter MCT8 for the treatment of Allan-Herndon-Dudley syndrome

Molenaar Anna , Schriever Sonja , Cebrian Serrano Alberto , Pathak Ekta , Maity-Kumar Gandhari , Stander Lisa , Englmaier Felix , Muller Timo , Pfluger Paul

Allan-Herndon-Dudley syndrome (AHDS) is a genetic disorder characterized by intellectual disability and movement disorders. AHDS patients have mutations in the monocarboxylate transporter 8 (MCT8) that impedes thyroid hormone (TH) transport to the brain during crucial phases of brain development. In mice, the resulting structural and functional pathologies of human AHDS patients can be mimicked by knocking out murine MCT8 and the Solute Carrier Organic Anion Transporter Family...

ea0090p694 | Pituitary and Neuroendocrinology | ECE2023

Management challenges of gestational pituitary adenoma

Fatma Mnif , Trimeche Oumeyma , Mouhaymen Missaoui Abdel , Ben Salah Dhoha , Elleuch Mouna , Haj Kacem Akid Faten , Charfi Nadia , Mnif Mouna , Rekik Majdoub Nabila , Abid Mohamed

Pituitary apoplexy is a rare condition and gestational pituitary apoplexy (GPA) is even more exceptional. Clinical presentation of GPA is often non-specific overlapping with other conditions, making its diagnosis challenging. Additionally, therapeutic management is often limited since it depends on both the mother’s and the fetus’s prognosis. Herein we report of a woman who was diagnosed with GPA and we discuss the management of her case. A 34-year-old woman with no ...

ea0090p695 | Pituitary and Neuroendocrinology | ECE2023

Sporadic neuroendocrine neoplasm: a survival analysis in a monocentric cohort

Liccardi Alessia , Minotta Roberto , Cannavale Giuseppe , Benevento Elio , Modica Roberta , Colao Annamaria

Introduction: Neuroendocrine neoplasms (NEN) are heterogeneous tumors, usually sporadic and non-functioning, with a favorable long-term prognosis if localized. Metastases are common and survival outcomes of metastatic patients (pts) is worse than in localized disease.Aim: To investigate survival outcomes in a monocentric cohort of sporadic NEN.Materials and Methods: Clinical data of pts with histologically confirmed sporadic NEN, r...

ea0090p696 | Pituitary and Neuroendocrinology | ECE2023

In the Phase III studies LINC 3 and LINC 4, osilodrostat was effective and well tolerated in patients of Asian and non-Asian origin with Cushing’s disease

Akira Shimatsu , Biller Beverly M.K. , Fleseriu Maria , Pivonello Rosario , Jig Lee Eun , Rattana Leelawattana , Hee Kim Jung , Walia Rama , Yu Yerong , Liao Zhihong , Piacentini Andrea , Pedroncelli Alberto , Snyder Peter

Introduction: Osilodrostat (oral 11β-hydroxylase inhibitor) demonstrated rapid, sustained cortisol normalisation in Phase III studies (LINC 3, NCT02180217; LINC 4, NCT02697734) in patients with Cushing’s disease (CD). Relative osilodrostat bioavailability is ~20% higher in Asian patients than other ethnicities; body weight is not a major determinant of this difference. This analysis of LINC 3 and LINC 4 evaluated osilodrostat efficacy and safety in Asian and non-Asia...

ea0090p697 | Pituitary and Neuroendocrinology | ECE2023

Transphenoidal surgery for sellar and suprasellar lesions: a prospective analysis of factors affecting inpatient length of stay

Ali Mudassir , Olaremi Japhet , Ali Rashid Razan , Mamoojee Yaasir

Background: The recent Get It Right First Time (GIRT) report suggests short stay pathways should be aimed for in pituitary surgery in England, with 1–2 days being realistic. We prospectively audited the length of stay (LOS) for patients undergoing transsphenoidal surgery (TSS) for sellar and suprasellar lesions at the Royal Victoria infirmary hospital (RVI) over a 2-year period, from October 2019.Methods: Data was prospectively collected for each pa...

ea0090p698 | Pituitary and Neuroendocrinology | ECE2023

Evaluation of the Association of Primary Empty Sella Syndrome in Neurobehçet Syndrome

Yilmaz Taner , Nur Kebapci Medine , Korkmaz Cengiz , Kaşifoğlu Timucin , Uskudar Cansu Dondu , Şule Yaşar Bilge Nazife

Central nervous system involvement is observed in 5-14% of Behçet’s disease and this form is called neurobehçet syndrome (NBS). There are two main forms of CNS involvement in Neurobehçet syndrome, in which parenchymal involvement and vascular involvement are prominent. Cerebral venous thrombosis (CVT), the most prominent form of vascular involvement, has been reported in 8% in Behçet’s disease and in approximately 18% in NBS. Although CVT is the m...

ea0090p699 | Pituitary and Neuroendocrinology | ECE2023

Non-functioning PitNETs: positive or differential diagnosis?

Florentina Burcea Iulia , Dobre Ramona , Nicoleta Nastase Valeria , Raluca Ceausu Amalia , Maria Cimpean Anca , Raica Marius , Poiana Catalina

Introduction: Clinically non-functioning pituitary neuroendocrine tumours (NFPitNETs) include all anterior pituitary tumours that are not hormonally active. In histopathology (HP) and immunohistochemistry (IHC), most of NFPitNETs are gonadotroph (1). Immunolabeling is negative in only 10% of cases (‘null-cell’). Occasionally, NFPitNETs are positive for GH, PRL, TSH or ACTH despite absent or only minimal secretion of these hormones in vivo, which are known as...

ea0090p700 | Pituitary and Neuroendocrinology | ECE2023

Grading of pituitary adenomas (PitNets): clinical and prognostic implications in a monocentric series

Giuffrida Giuseppe , Casablanca Rosalinda , Ragonese Marta , Giovinazzo Salvatore , Ruxandra Cotta Oana , Certo Rosaria , Alessi Ylenia , Scordo Marianna , Pizzimenti Cristina , Tuccari Giovanni , Flavio Angileri Filippo , Ferrau Francesco , Cannavo Salvatore

Rationale: Pituitary neuroendocrine tumors (PitNets) are aggressive in 20% of cases, with local invasion, relapse/scarce response to conventional treatment, in the absence of reliable predictive parameters. In 2018, Trouillas et al. proposed a 5-tier prognostic classification, not widely validated yet. In our study we evaluated the outcomes of a PitNets monocentric series in the context of this classification.Materials & Methods: We retrospe...

ea0090p701 | Pituitary and Neuroendocrinology | ECE2023

Emergence of De-novo Steroid-Responsive Conditions Following Remission of Cushing’s Syndrome: A Case Report and Scoping Review

Desgagnes Noemie , Senior Laura , Lithgow Kirstie

Background: Endogenous Cushing’s syndrome is caused by chronically elevated glucocorticoid levels. The most common etiology is ACTH hypersecretion from a pituitary adenoma; other causes include hypersecretion of cortisol from an adrenal source or ectopic ACTH secretion. Following successful treatment, a period of adrenal insufficiency is expected due to chronic suppression of the HPA axis. Onset and exacerbation of steroid-responsive conditions have been reported followin...

ea0090p702 | Pituitary and Neuroendocrinology | ECE2023

A Low Incidence of Transient Anti-Drug Antibodies Is Observed Upon Long-Term Exposure to Lonapegsomatropin in Children With Growth Hormone Deficiency

Holse Mygind Per , Komirenko Allison , Mao Meng , Shu Aimee , Breinholt Vibeke M.

Objectives: Lonapegsomatropin (SKYTROFA; TransCon hGH), a prodrug of somatropin, is approved by the EMA and FDA for once-weekly treatment of pediatric growth hormone deficiency (GHD). Lonapegsomatropin uses TransCon® technology, which combines a parent drug transiently linked to an inert carrier to achieve sustained release. The characterization of anti-drug antibodies (ADAs) was assessed in 3 phase 3 trials of lonapegsomatropin in pediatric GHD.Met...

ea0090p703 | Pituitary and Neuroendocrinology | ECE2023

Diabetes insipidus in children secondary to a germinoma diagnosed after 3 years of follow-up

Mezoued Mouna , Khadidja Bessaid , Azzouz Malha

Central diabetes insipidus (CDI) is rare in children and has a wide variety of causes. In children, the majority of central DI is acquired, but 30 to 50% is considered idiopathic. However, the natural history of isolated idiopathic central ID is unpredictable and germinoma should always be considered during the first 3 years of follow-up.Case report: We report the observation of a 16 year old patient, in whom the diagnosis of idiopathic central diabetes ...

ea0090p704 | Pituitary and Neuroendocrinology | ECE2023

Solitary pulmonary nodule developing ectopic Cushing’s Syndrome over the years: Awakening of a Sleeping Giant

Bostan Hayri , Kizilgul Muhammed , Ozcelik Ozgur , Gul Umran , Taskin Turkmenoglu Tugba , Ozbek Mustafa , Cakal Erman

Background: A rare cause of ectopic Cushing’s Syndrome (ECS) is pulmonary neuroendocrine neoplasm (NEN) with a broad spectrum of clinical behavior. At the time of diagnosis, pulmonary NENs may present as occult tumors or well-defined round-to-oval lesions in close relationship to the bronchus. Herein, we describe a NEN that developed ECS three years later in a patient followed up for a solitary pulmonary nodule (SPN).Case Presentation: A 56-year-old...

ea0090p705 | Pituitary and Neuroendocrinology | ECE2023

Isolated IgG4 Hypophysitis

Ni Aung Nwe , Butt Nouman

Hypophysitis is an inflammatory disorder of the pituitary gland and among them, IgG-4 hypophysitis is rare and presents in less than 5 % of cases.Case: 59 years old lady with a past medical history of asthma presented Rheumatology with? giant cell arthritis due to constant frontal headache, pain behind her eyes, and raised inflammatory markers (ESR 15, CRP 17) three weeks after COVID infection. After review, rheumatology team concluded unlikely giant cel...

ea0090p706 | Pituitary and Neuroendocrinology | ECE2023

Rare causes of adrenal tumours and the possible role of adrenal arterial embolization - two case reports

Manique Ines , Cortez Luisa , Abreu Silvestre , Silva-Nunes Jose

Introduction: Adrenal oncocytoma (AO) and ganglioneuroma (GN) are rare causes of adrenal masses, corresponding usually to nonfunctional and benign tumors. However, 20% of the AO shows some elements of malignancy and 10–20% of them affect adrenal hormone production. GNs account for 0.3%-2% of adrenal incidentalomas, with punctate calcifications in CT imaging being highly suggestive. However, preoperative differential diagnosis remains challenging. Adrenal arterial emboliza...

ea0090p707 | Pituitary and Neuroendocrinology | ECE2023

Acromegaly and its ophthalmic, neurological and psychiatric complications

Cristiana Istrate Andra , Christina Ungureanu Maria , Leustean Letitia , Juliette Mogos Simona , Bilha Stefana , Florescu Alexandru , Preda Cristina

Introduction: Acromegaly is a rare, complex condition that throughout its evolution affects the body on numerous levels, especially if inappropriately managed. Some of the complications it develops over time are ophthalmic, neurological and psychiatric.Aim: The aim is to asses the impact of long-term acromegaly on the eye, nervous system and mental health of previously diagnosed patients.Patients and method: 90 patients’ recor...

ea0090p708 | Pituitary and Neuroendocrinology | ECE2023

Congenital, isolated, and lifetime growth hormone deficiency provides superior cognitive performance in senescence

Batista Vanderlan O. , Kellner Michael , Salvatori Roberto , Oliveira Walter , Faro Andre , Santos Lucas B. , Melo Enaldo V. , Oliveira-Santos Alecia A. , Oliveira Carla R. P. , Almeida Viviane , Barros-Oliveira Cynthia S. , Santos Elenilde G. , Santana Nathalie O. , Villar-Gouy Keila R. , Leal Angela , Amorim Rivia S. , H. Aguiar-Oliveira Manuel

Mice with isolated GH deficiency (IGHD) due to GHRH receptor mutations live longer than their normal siblings with an extended healthspan, i.e., the period of life free from disabilities. Human IGHD individuals due to a mutation in the GHRH receptor gene from Itabaianinha, Brazil, has a normal lifespan with an extended healthspan (1). Our hypothesis is that their aging is accompanied by a delayed cognitive decline. Accordingly, we used the Literacy Independent Cognitive Assess...

ea0090p709 | Pituitary and Neuroendocrinology | ECE2023

Adrenocortical, somatotropic and antidiuretic response to nasal glucagon in healthy subjects

Varaldo Emanuele , Bioletto Fabio , Cuboni Daniela , Prencipe Nunzia , Bona Chiara , Barale Marco , Ghigo Ezio , Grottoli Silvia , Maria Berton Alessandro , Gasco Valentina

Rationale: In the diagnosis of hypopituitarism, the glucagon stimulation test allows for the simultaneous and safe evaluation of the somatotropic and corticotropic axes; recent data have highlighted a stimulating action also on the neurohypophyseal secretion of arginine-vasopressin. This procedure involves the intramuscular or subcutaneous administration of 1-1.5 mg of glucagon based on the patient’s weight (respectively less or more than 90 kg). Nowadays no data are avai...

ea0090p710 | Pituitary and Neuroendocrinology | ECE2023

Chordomas of the parasellar region: description of 4 cases of a rare disease

Cosme Ines , Nobre Ema , Joao Bugalho Maria

Background: Chordomas are rare, invasive and slow growing bone tumours with different locations. Parasellar chordomas are even more rare and the initial manifestations include visual and/or neurological symptoms. In addition, endocrine dysfunction may be present. We describe 4 cases of parasellar chordomas.Clinical cases: 1) Female, 55-years-old, reported headache associated with restriction of left eye movement for the past 3 months. Brain magnetic reso...

ea0090p711 | Pituitary and Neuroendocrinology | ECE2023

Prevalence and associated factors related to combined hypopituitarism in patients with congenital Growth Hormone deficiency

Haj Kacem Akid Faten , Belabed Wafa , Mouhaymen Missaoui Abdel , Safi Wajdi , Ben Salah Dhoha , Mnif Fatma , Mejdoub Nabila , Mnif Mouna , Abid Mohamed

Background and aim: Congenital Growth hormone deficiency (CGHD) may result from genetic or congenital disorders of pituitary development. It could be isolated or combined with other types of hypopituitarism. This study aims to evaluate the prevalence of combined hypopituitarism and its associated factors in patients with CGHD.Patients and Methods: We conducted a retrospective study (1991-2019) at the Endocrinology department of Hedi Chaker University Hos...

ea0090p712 | Pituitary and Neuroendocrinology | ECE2023

Successful management of severe hypoglycemia by octreotide LAR in an insulinoma patient

Hocaoglu Erhan , Aydemir Ensar , Ates Coskun , Mercan Saridas Filiz , Yasar Muge , Oz Gul Ozen , Cander Soner , Ersoy Canan , Erturk Erdinc

Introduction: Surgical removal of the tumor is the primary treatment modality for insulinomas. However, in some patients, surgery is not possible. Octreotide, a somatostatin analogue, is known to suppress insulin secretion. However, there are little data on the use of octreotide in the treatment of insulinoma. Here, we present an insulinoma patient with severe clinical findings and successful control of hypoglycemia after long-acting octreotide.Case: A 7...