ECEESPE2025 ePoster Presentations Adrenal and Cardiovascular Endocrinology (170 abstracts)
Significant metabolic syndrome improvement in a patient with adrenal Cushing’s syndrome treated preoperatively with osilodrostat
Vasiliki Daraki 1 , Evangelia Pissadaki 1 , Eleni-Konstantina Syntzanaki 1 , Irini Lambiri 2 , Vasiliki Venetsanaki 1 , Panagiotis- Nikolaos Tsakalomatis 1 , Fotis Mermigkas 1 , Rodanthi Vamvoukaki 1 , Grigoria Betsi 1 , Maria Sfakiotaki 1 , Chrysanthi Aggeli 3 , George Zografos 3 & Paraskevi Xekouki 1
1University General Hospital of Heraklion, Department of Endocrinology Diabetes and Metabolic Diseases, Crete, Greece; 2University General Hospital of Heraklion, Department of Thoracic Medicine, Crete, Greece; 3Athens General Hospital, 1st Department of Surgery, Athens, Greece
JOINT2402
Background: Cushing syndrome (CS) is associated with high morbidity and mortality, due to its metabolic consequences. The treatment of choice is surgical resection of the causative tumor. Data on the preoperative use of osilodrostat, an inhibitor of 11β-hydroxylase, in patients with adrenal CS (ACS) are scarce. We present a rare case of ACS treated preoperatively with osilodrostat with significant metabolic syndrome improvement.
Case presentation: A 47-year-old woman, with a previous history of cervical cancer, presented to the outpatient clinic with a 3,6 cm left adrenal mass discovered incidentally, 4 years prior to presentation. She suffered from poorly-controlled diabetes mellitus (DM) (HbA1c 9,5%), dyslipidemia, chronic obstructive pulmonary disease (COPD) and significant weight gain (25 kgr, BMI 46) over the last 8 years. Clinical examination revealed facial plethora, posterior cervicothoracic fat pad and severe truncal obesity. Laboratory tests were indicative of adrenal Cushing syndrome (morning serum cortisol 20,7 μg/dl after low dose oral dexamethasone suppression, adrenocorticotropin (ACTH)5,32 pg/ml and urinary free cortisol 384 μg/24hr (normal values 20,9-292 μg/24hr). Adrenalectomy was postponed twice due to her severe obesity and poorly-controlled DM and COPD. The patient was lost to follow-up. She returned a year later with deterioration of her clinical symptoms. Her urinary free cortisol levels were further increased (>1000 μg/24hr). Treatment with osilodrostat was decided at a dose of 4 mg daily. However, a week later she presented with symptoms suggestive of adrenal insufficiency (nausea, fatigue and low blood pressure). Osilodrostat was reduced to 1 mg daily, while oral hydrocortisone 20 mg daily was added with symptom resolution. Two weeks later, hydrocortisone was stopped and osilodrostat was continued with a slow titration initially to 2 mg for 6 months and subsequently to 3 mg. Ten months after her 1st osilodrostat dose, the patient achieved a significant weight loss (20 kg, BMI:38), while her DM and dyslipidemia were well controlled (HbA1c 6,1%) without treatment modification. Her last urinary free cortisol measurement was within normal range (147 μg/24hr). She has been scheduled for adrenalectomy next month.
Discussion: Treatment of ACS with osilodrostat, has been rarely reported in the literature. In our patient, a 10-month treatment with low osilodrostat dose resulted in significantly reduced cortisol levels concomitantly with important weight loss and improvement of all metabolic parameters prior to curative adrenalectomy. Further studies are warranted to evaluate preoperatively osilodrostat use on surgery-related morbidities and outcomes in such patients.
Volume 110
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Endocrine Abstracts
ISSN 1470-3947 (print) | ISSN 1479-6848 (online)
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