Clinical management challenges of Cushing's disease associated with double pituitary adenomas: four cases and literature review.

IF 3.3 3区医学 Q2 ENDOCRINOLOGY & METABOLISM

Endocrine Pub Date : 2026-08-08 DOI:10.1007/s12020-026-04748-8

Di Zhang, Wan Su, Jinghua Liu, Siti Liu, Yuchen Wei, Hui You, Bo Hou, Xinxin Mao, Yi Zhang, Kan Deng, Jifang Liu, Yong Yao, Lin Lu

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引用次数: 0

Abstract

Purpose: To characterize clinical, radiological, and pathological features of double pituitary adenomas (DPAs) involving a corticotroph adenoma and evaluate challenges associated with preoperative localization and surgical management of Cushing's disease (CD).

Methods: We retrospectively reviewed four institutional cases and searched PubMed and Web of Science (1990-2025), identifying 30 publications comprising 68 additional cases. Demographic, clinical, radiological, immunohistochemical, surgical, and outcome data from the combined cohort were analyzed.

Results: In all four institutional cases, the lesion removed during initial transsphenoidal surgery (TSS) was a noncorticotroph adenoma, and hypercortisolism persisted. Follow-up pituitary magnetic resonance imaging (MRI) demonstrated a second suspected lesion in all four patients. In Cases 3 and 4, MRI abnormalities were subtle or equivocal, and ⁶⁸Ga-pentixafor positron emission tomography/computed tomography (PET/CT) revealed concordant focal uptake at the suspected sites. Revision TSS confirmed an adrenocorticotropic hormone (ACTH)- and T-PIT-positive corticotroph adenoma in all four patients, and all achieved early postoperative biochemical remission. In the combined cohort (72 patients), preoperative MRI identified both lesions in 12 patients (16.7%) and only one lesion in 39 (54.2%); MRI findings were unavailable or insufficiently described in 21 (29.2%). Sixteen patients (22.2%) underwent repeat surgery, whereas surgical-course information was unavailable in 21 (29.2%).

Conclusions: DPAs involving a corticotroph component should be considered as a possible explanation for persistent hypercortisolism after TSS when the resected lesion is negative for ACTH and T-PIT. Multidisciplinary reassessment and repeat expert pituitary imaging review may help identify an additional lesion. The role of adjunctive molecular imaging remains preliminary.

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库欣病合并双垂体腺瘤的临床治疗挑战：4例及文献回顾

目的：探讨双垂体腺瘤（dpa）伴促皮质性腺瘤的临床、影像学和病理特征，并评估库欣病（CD）术前定位和手术治疗的相关挑战。方法：我们回顾性地回顾了4个机构病例，并检索了PubMed和Web of Science(1990-2025)，确定了30篇出版物，其中包括68个额外的病例。对来自联合队列的人口统计学、临床、放射学、免疫组织化学、手术和结局数据进行分析。结果：在所有四个机构病例中，在最初的经蝶窦手术（TSS）中切除的病变是一个非促皮质性腺瘤，高皮质血症持续存在。随访垂体磁共振成像（MRI）显示所有4例患者均有第二个疑似病变。在病例3和4中，MRI异常是微妙的或模棱两可的，68Ga-pentixafor正电子发射断层扫描/计算机断层扫描（PET/CT）显示在可疑部位有一致的局灶摄取。修正TSS证实所有4例患者均为促肾上腺皮质激素（ACTH）和t - pit阳性促皮质腺瘤，所有患者术后早期生化缓解。在联合队列（72例患者）中，术前MRI发现12例（16.7%）患者有两个病变，39例（54.2%）患者只有一个病变；21例（29.2%）的MRI表现不可用或描述不充分。16例（22.2%）患者接受了重复手术，而21例（29.2%）患者无法获得手术过程信息。结论：当切除的病变ACTH和T-PIT呈阴性时，dpa涉及促皮质成分应被认为是TSS后持续高皮质醇的可能解释。多学科再评估和重复专家垂体影像学检查可能有助于确定额外的病变。辅助分子成像的作用仍处于初步阶段。

本文章由计算机程序翻译，如有差异，请以英文原文为准。

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来源期刊

Endocrine ENDOCRINOLOGY & METABOLISM-

CiteScore

6.50

自引率

5.40%

发文量

295

审稿时长

1.5 months

期刊介绍： Well-established as a major journal in today’s rapidly advancing experimental and clinical research areas, Endocrine publishes original articles devoted to basic (including molecular, cellular and physiological studies), translational and clinical research in all the different fields of endocrinology and metabolism. Articles will be accepted based on peer-reviews, priority, and editorial decision. Invited reviews, mini-reviews and viewpoints on relevant pathophysiological and clinical topics, as well as Editorials on articles appearing in the Journal, are published. Unsolicited Editorials will be evaluated by the editorial team. Outcomes of scientific meetings, as well as guidelines and position statements, may be submitted. The Journal also considers special feature articles in the field of endocrine genetics and epigenetics, as well as articles devoted to novel methods and techniques in endocrinology. Endocrine covers controversial, clinical endocrine issues. Meta-analyses on endocrine and metabolic topics are also accepted. Descriptions of single clinical cases and/or small patients studies are not published unless of exceptional interest. However, reports of novel imaging studies and endocrine side effects in single patients may be considered. Research letters and letters to the editor related or unrelated to recently published articles can be submitted. Endocrine covers leading topics in endocrinology such as neuroendocrinology, pituitary and hypothalamic peptides, thyroid physiological and clinical aspects, bone and mineral metabolism and osteoporosis, obesity, lipid and energy metabolism and food intake control, insulin, Type 1 and Type 2 diabetes, hormones of male and female reproduction, adrenal diseases pediatric and geriatric endocrinology, endocrine hypertension and endocrine oncology.