ECEESPE2025 ePoster Presentations Endocrine Related Cancer (100 abstracts)
A rare case report of prolonged adrenal insufficiency following discontinuation of osilodrostat treatment for severe hypercortisolism due to ectopic ACTH syndrome
Hamza Benderradji 1,2 , François Walasiak 1 , Antoine Danais 1 , Arnaud Jannin 1,3 , Nicolas Venissac 4 , Samuel Boury 5 , Amandine Beron 6 , Bryan Ducrocq 7 , Robert Caiazzo 2 , Marie-Christine Vantyghem 1,8 & Christine Do Cao 1
1Department of Endocrinology, Diabetology, and Metabolism, Lille University Hospital, Lille, France; 2Department of Endocrine and Metabolic Surgery, Lille University Hospital, Lille, France; 3CANTHER-Cancer-Heterogeneity Plasticity and Resistance to Therapies, University of Lille, UMR9020-U1277-CNRS, INSERM, CHU Lille, Lille, France; 4Department of Thoracic Surgery, Calmette Hospital, University Hospital of Lille, Lille, France; 5Department of Radiology, Lille University Hospital, Lille, France; 6Department of Nuclear Medicine, Lille University Hospital, Lille, France; 7Department of Pathology, Lille University Hospital, Lille, France; 8INSERM U1190 Translational Research in Diabetes, Firendo Rare Diseases Competence Center PRISIS, (Pathologies of insulin Resistance and Insulin Sensitivity), Lille University Hospital, Lille, France
JOINT2543
Osilodrostat is an 11β-hydroxylase inhibitor indicated for treatment of Cushing’s syndrome in adult patients. Transient use of osilodrostat may lead to prolonged adrenal insufficiency, a rare adverse event worth noting. We report here the case of an 81-year-old woman who was treated with osilodrostat (20 mg/day) for five months for severe hypercortisolism secondary to ectopic ACTH secretion [midnight cortisol at 81.7 µg/dL (2–7), with ACTH at 137 pg/mL, and urinary free cortisol at 5972.1 µg/24h (20–65)]. CT scan revealed a bronchial tumour located at the lower right pulmonary lobe and enlarged adrenals. Following tumour resection (well differentiated neuroendocrine histology), osilodrostat was discontinued. One month postoperatively, basal cortisol at 8h00 was low at 2.2 µg/dL (9–22) with inadequate ACTH secretion at 40 pg/mL (<46.0) supposedly linked to previously suppressed corticotropic cells by excessive cortisol production. The patient was given accordingly a physiological replacement dose of hydrocortisone. 68Ga-DOTATOC PET/CT performed six months post-surgery ruled out residual or recurrent neuroendocrine tumor and an abdominal CT scan demonstrated normal appearance of adrenals. Ten months after discontinuation of osilodrostat, the patient exhibited persistent adrenal insufficiency. Basal hormone analysis revealed markedly elevated ACTH levels (175 pg/mL) and undetectable cortisol levels this time in favor of a residual iatrogenic effect of osilodrostat. Prolonged primary adrenal insufficiency after transient use of osilodrostat is a rare but serious side effect. Only few cases have been documented in the past two years. The underlying pathophysiological mechanism remains unclear and quite unexpected, considering the short half-life of the drug. Clinical follow up and continued adrenal function monitoring are advisable following osilodrostat discontinuation to prevent adrenal crisis in those rare at-risk patients.
Volume 110
Article tools
My recent searches
My recently viewed abstracts
Authors
Endocrine Abstracts
ISSN 1470-3947 (print) | ISSN 1479-6848 (online)
© Bioscientifica 2025 |
Privacy policy |
Cookie settings
BiosciAbstracts
Bioscientifica Abstracts is the gateway to a series of products that provide a permanent, citable record of abstracts for biomedical and life science conferences.
Find out more