Bettale Chiara M, Allen Jason W, Mahdi Zaid K, Ioachimescu Adriana G
Emory University School of Medicine, Atlanta, GA 30322, USA.
Department of Radiology and Imaging Services, Emory University School of Medicine, Atlanta, GA 30322, USA.
JCEM Case Rep. 2023 Feb 1;1(1):luad007. doi: 10.1210/jcemcr/luad007. eCollection 2023 Jan.
A 55-year-old woman admitted for hypertensive emergency and myocardial infarction reported weight gain, muscle weakness, easy bruising, and recent-onset diabetes in the past 3 to 12 months. Urinary and salivary cortisol and adrenocorticotropin hormone (ACTH) levels were elevated. Pituitary imaging detected a macroadenoma. ACTH and cortisol did not increase after corticotropin-releasing hormone administration. Imaging revealed a large pancreatic mass. Pathology indicated a well-differentiated World Health Organization (WHO) grade 2 distal pancreatic neuroendocrine neoplasm which stained for ACTH by immunohistochemistry. Postoperatively, Cushing manifestations resolved, ACTH and cortisol levels became low, and patient required hydrocortisone replacement for 7 months. During the 3.5 years of follow-up, the pituitary macroadenoma size remained stable and pituitary hormone axes other than ACTH remained normal. This extremely rare case of ectopic ACTH-secreting pancreatic neuroendocrine tumor coexisting with a nonfunctioning pituitary macroadenoma illustrates the importance of dynamic endocrine testing in Cushing syndrome.
一名因高血压急症和心肌梗死入院的55岁女性报告称,在过去3至12个月内体重增加、肌肉无力、容易出现瘀伤,且近期患糖尿病。尿和唾液皮质醇以及促肾上腺皮质激素(ACTH)水平升高。垂体成像检测到一个大腺瘤。注射促肾上腺皮质激素释放激素后,ACTH和皮质醇未升高。成像显示胰腺有一个大肿块。病理检查表明是一个分化良好的世界卫生组织(WHO)2级远端胰腺神经内分泌肿瘤,免疫组化显示其ACTH染色阳性。术后,库欣综合征表现消失,ACTH和皮质醇水平降低,患者需要氢化可的松替代治疗7个月。在3.5年的随访期间,垂体大腺瘤大小保持稳定,除ACTH外的垂体激素轴仍保持正常。这种极为罕见的异位分泌ACTH的胰腺神经内分泌肿瘤与无功能垂体大腺瘤共存的病例,说明了动态内分泌检测在库欣综合征中的重要性。