{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,3,31]],"date-time":"2026-03-31T03:57:02Z","timestamp":1774929422099,"version":"3.50.1"},"reference-count":18,"publisher":"The Endocrine Society","issue":"4","license":[{"start":{"date-parts":[[2026,3,31]],"date-time":"2026-03-31T00:00:00Z","timestamp":1774915200000},"content-version":"vor","delay-in-days":12,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2026,3,19]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:p>Multiple endocrine neoplasia type 1 (MEN1) syndrome is a rare autosomal dominant disorder characterized by predisposition to a multitude of endocrine neoplasms. Cushing syndrome (CS) within MEN1 presents complex diagnostic and therapeutic challenges. We report a case of a 36-year-old male with adrenocorticotropic hormone (ACTH)-dependent Cushing disease and MEN1 syndrome with thymic and pancreatic neuroendocrine tumors. The patient presented with weight gain, weakness and proximal myopathy. Biochemical testing confirmed ACTH-dependent hypercortisolism. Imaging revealed a pituitary microadenoma, a large anterior mediastinal mass, and pancreatic lesions. Genetic analysis confirmed a pathogenic heterozygous MEN1 frameshift variant. After thymectomy, he declined further surgery. Treatment with thymectomy and octreotide long-acting release (LAR) was ineffective. Initiation of osilodrostat (titrated to 5\u2005mg twice daily) resulted in a 76% reduction in urinary cortisol levels and a 47\u2005kg weight loss, with significant functional improvement. Pancreatic and pituitary lesions remained stable. This is the first reported case of successful symptom management of MEN1-associated Cushing disease with osilodrostat, establishing it as an effective therapeutic option for medically complex MEN1 cases where surgery is not feasible or is declined.<\/jats:p>","DOI":"10.1210\/jcemcr\/luag039","type":"journal-article","created":{"date-parts":[[2026,3,31]],"date-time":"2026-03-31T02:40:57Z","timestamp":1774924857000},"source":"Crossref","is-referenced-by-count":0,"title":["Symptom management with osilodrostat in multiple endocrine neoplasia type 1 with a Cushing syndrome presentation"],"prefix":"10.1210","volume":"4","author":[{"ORCID":"https:\/\/orcid.org\/0009-0007-8327-7038","authenticated-orcid":false,"given":"Hasan Frookh","family":"Jamal","sequence":"first","affiliation":[{"name":"Department of Endocrinology, Government Hospital, Salmaniya Medical Complex , Manama 00973 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