Martinez-Gil Mercedes, Yuen Kevin C J
Department of Internal Medicine, Creighton University School of Medicine, Phoenix, AZ 85004, USA.
Barrow Pituitary Center, Barrow Neurological Institute, University of Arizona College of Medicine and Creighton University School of Medicine, Phoenix, AZ 85013, USA.
JCEM Case Rep. 2025 Apr 22;3(6):luaf089. doi: 10.1210/jcemcr/luaf089. eCollection 2025 Jun.
Cushing disease (CD) is the most common form of adrenocorticotropin (ACTH)-dependent Cushing syndrome (CS), whereas unilateral adrenal adenoma is the most common cause of ACTH-independent CS. However, the occurrence of different subtypes of CS in a single individual is very rare. We present a case of a 44-year-old woman with distant histories of left adrenalectomy for an adrenal adenoma and total thyroidectomy following the diagnosis of papillary thyroid carcinoma. She was later diagnosed with CD, achieving disease remission after pituitary surgery, but subsequently developed adrenal CS from the remaining right adrenal gland. After discussing the potential advantages and drawbacks of another adrenalectomy to remove her right adrenal gland, the patient declined surgery and opted for medical management. After 7 years of imaging follow-up studies, her right adrenal adenoma has remained stable in size and she is biochemically controlled on low-dose osilodrostat therapy. Our case emphasizes the importance of recognizing the rare occurrence of successfully treated CD followed by the recurrence of CS from a different gland, and the adoption of management strategies tailored to each individual patient's preferences.
库欣病（CD）是促肾上腺皮质激素（ACTH）依赖性库欣综合征（CS）最常见的形式，而单侧肾上腺腺瘤是ACTH非依赖性CS最常见的病因。然而，同一个体出现不同亚型的CS非常罕见。我们报告一例44岁女性病例，该患者既往有因肾上腺腺瘤行左肾上腺切除术及诊断为甲状腺乳头状癌后行全甲状腺切除术的病史。她后来被诊断为库欣病，垂体手术后病情缓解，但随后剩余的右侧肾上腺又发生了肾上腺性CS。在讨论再次行肾上腺切除术切除右侧肾上腺的潜在利弊后，患者拒绝手术，选择药物治疗。经过7年的影像学随访研究，她的右侧肾上腺腺瘤大小保持稳定，且在低剂量奥西卓司他治疗下生化指标得到控制。我们的病例强调了认识到成功治疗的库欣病后罕见地出现来自不同腺体的库欣综合征复发的重要性，以及采用根据每个患者偏好量身定制的管理策略的重要性。
