Double pituitary adenomas: report of two cases and systematic review of the literature

Front Endocrinol (Lausanne). 2024 Apr 2:15:1373869. doi: 10.3389/fendo.2024.1373869. eCollection 2024.

Abstract

Objective: Double pituitary adenomas (DPA) are a rare clinical condition, and our knowledge of them is limited. Missing the second lesion leading to incomplete biochemical remission after surgery is an important challenge in DPA management. This study aims to analyze independent prognostic factors in DPA patients and summarize clinical experiences to prevent surgical failure.

Methods: Two cases of DPA patients with Cushing's disease diagnosed and surgically treated at Peking Union Medical College Hospital are reported. A literature review was performed on the online database Pubmed, and 57 DPA patients from 22 retrieved articles were included. Demographic characteristics, endocrine manifestations, diagnostic methods, tumor size, and immunohistochemical features of 59 patients were analyzed. Binary logistic regression models were used to identify independent prognostic factors affecting postoperative biochemical remission.

Results: Among 59 DPA patients, the mean ± SD age was 43.64 ± 14.42 years, with 61.02% being female (n = 36). The most common endocrine manifestations were Cushing's syndrome (23/59, 38.98%) and acromegaly (20/59, 33.90%). The most prevalent immunohistochemical types were ACTH-immunopositive (31/118, 26.27%) and GH-immunopositive (31/118, 26.27%) tumors. Microadenomas (<1cm) were the most frequent in terms of tumor size (62/92, 67.39%). The detection rate for double lesions on 3.0T MRI was 50.00% (14/28), which significantly higher than 1.5T MRI (P = 0.034). Univariate analysis revealed that female, Cushing's syndrome and only single lesion detected by surgical exploration were associated with significantly worse prognosis (P<0.05). Multivariate analysis identified double lesion detected by surgical exploration (OR = 0.08, P = 0.003) and contiguous type tumor (OR = 0.06, P = 0.017) as independent protective factors for DPA patients.

Conclusions: The double lesion detected by surgical exploration is independently associated with a better prognosis for DPA patients. Comprehensive intraoperative exploration are crucial measures to avoid missing causative lesions.

Keywords: acromegaly; biochemical remission; cushing disease; double pituitary adenomas (DPA); prognostic analysis.

Publication types

  • Case Reports
  • Systematic Review

MeSH terms

  • Acromegaly* / complications
  • Adenoma* / diagnosis
  • Adult
  • Cushing Syndrome* / diagnosis
  • Female
  • Humans
  • Male
  • Middle Aged
  • Pituitary ACTH Hypersecretion* / complications
  • Pituitary Neoplasms* / complications
  • Pituitary Neoplasms* / diagnosis
  • Pituitary Neoplasms* / surgery

Grants and funding

The author(s) declare financial support was received for the research, authorship, and/or publication of this article. This study was supported by National High Level Hospital Clinical Research Funding (No. 2022-PUMCH-B-114) from YY, and Youth Science Foundation of Peking Union Medical College Hospital (No. pumch201911867) from YZ.