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Idiopathic granulomatous hypophysitis: a systematic review of 82 cases in the literature

Citation

Hunn, BHM and Martin, WG and Simpson Jr, S and McLean, CA, Idiopathic granulomatous hypophysitis: a systematic review of 82 cases in the literature, Pituitary pp. 1-9. ISSN 1573-7403 (2013) [Refereed Article]


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Licensed under the Creative Commons Attribution 4.0 International (CC BY 4.0)

DOI: doi:10.1007/s11102-013-0510-4

Abstract

Idiopathic granulomatous hypophysitis (IGH) is a rare inflammatory disease of the pituitary. There is debate in the scientific literature as to whether IGH represents a continuum of disease with lymphocytic hypophysitis or has a distinct pathogenesis. Due to the rare nature of the disease, previous descriptions have been limited to single case reports or small series. In the present study, a systematic review of the literature was performed for cases of IGH. 82 cases met inclusion criteria. Data was gathered on IGH clinical aspects, in order to elucidate any associations useful in determining pathogenesis, appropriate clinical treatment, or prognosis. Univariate and multivariate analysis was performed on available data. Female sex was significantly associated with IGH (p < 0.0001). Fever (p = 0.002), nausea or vomiting at presentation (p = 0.031), and histological evidence of necrosis (p = 0.022) correlated with reduced time to presentation. Panhypopituitarism at presentation predicted need for long term hormone replacement (p = 0.014). Hyperprolactinaemia (p = 0.032), normal gonadal (p = 0.037) and thyroid axes (p = 0.001) were associated with reduced likelihood of long-term hormone replacement. Anorexia (p = 0.017), cold intolerance (p = 0.046), and fatigue (p = 0.0033) were associated with death from IGH. Patients who had excisional surgery alone trended towards increased rates of symptom resolution, compared with patients who received corticosteroids as an adjunct to excisional surgery (p = 0.11). This article details the first systematic review of IGH, and presents evidence for a female predilection of the disease. Implications for pathogenesis, and a suggested clinical approach are discussed. An online disease registry has been established to facilitate further IGH research.

Item Details

Item Type:Refereed Article
Keywords:Hypophysitis, Granulomatous, Lymphocytic, Systematic Review
Research Division:Medical and Health Sciences
Research Group:Public Health and Health Services
Research Field:Epidemiology
Objective Division:Health
Objective Group:Clinical Health (Organs, Diseases and Abnormal Conditions)
Objective Field:Nervous System and Disorders
Author:Hunn, BHM (Dr Ben Hunn)
Author:Simpson Jr, S (Dr Steve Simpson JR)
ID Code:88841
Year Published:2013
Web of Science® Times Cited:13
Deposited By:Menzies Institute for Medical Research
Deposited On:2014-02-18
Last Modified:2017-11-01
Downloads:192 View Download Statistics

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