Endocrine · 2026
Clinical management challenges of Cushing's disease associated with double pituitary adenomas: four cases and literature review.
СтатьяОбзорКлинический случайНазвание и аннотация: Перевод в очереди
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Zhang Di, Su Wan, Liu Jinghua, Liu Siti, Wei Yuchen, You Hui, Hou Bo, Mao Xinxin, Zhang Yi, Deng Kan, Liu Jifang, Yao Yong, Lu Lin
Аннотация
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.
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.