Isolated pituitary sarcoidosis stained with a specific MAB against Propionibacterium acnes: a case report

Kaoru Yamashita; Michita Niiyama; Yasufumi Seki; Daisuke Watanabe; Osamu Isozaki; Kosaku Amano; Takashi Komori; Yoshikazu Okada; Yoshinobu Eishi; Atsuhiro Ichihara

doi:10.1530/endoabs.37.EP717

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Endocrine Abstracts (2015) 37 EP717 | DOI: 10.1530/endoabs.37.EP717

ECE2015 Eposter Presentations Pituitary: clinical (121 abstracts)

Isolated pituitary sarcoidosis stained with a specific MAB against Propionibacterium acnes: a case report

Kaoru Yamashita ¹ , Michita Niiyama ¹ , Yasufumi Seki ¹ , Daisuke Watanabe ¹ , Osamu Isozaki ¹ , Kosaku Amano ² , Takashi Komori ³ , Yoshikazu Okada ² , Yoshinobu Eishi ³ & Atsuhiro Ichihara ¹

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¹Department of Endocrinology and Hypertension, Tokyo Women’s Medical University, Tokyo, Japan; ²Department of Neurosurgery, Tokyo Women’s Medical University, Tokyo, Japan; ³Department of Human Pathology, Tokyo Medical and Dental University Graduate School, Tokyo, Japan.

Introduction: Sarcoidosis is a systemic granulomatous disorder usually affects multiple organs. Approximately 10% of sarcoidosis occurs as isolated neurosarcoidosis in CNS and <10% of neurosarcoidosis develops in a restricted hypothalamic–pituitary area. Recent study has identified Propionibacterium acnes in lungs and lymph nodes of many patients with sarcoidosis using a MAB specific for this bacterium (PAB), providing strong evidence for P. acne as a pathogen of sarcoidosis (Modern Pathology, 2012). We report a case of isolated pituitary sarcoidosis, manifested as an acute onset of secondary adrenocortical insufficiency.

Case: A 29-year-old man suddenly developed severe headache, malaise, and appetite loss 1 year after an extensive skin peeling therapy for pimples. MRI revealed a diffusely and symmetrically enlarged pituitary, protruded upward with a thickened stalk and inhomogeneously enhanced with gadolinium, suggesting anterior hypophysitis. Endocrinologically, baseline ACTH and cortisol was 4.8 pg/ml and 0.4 μg/dl respectively. FT₄ was 0.72 ng/dl, testosterone was 10.3 ng/dl, PRL was 36.1 μg/l, and peak GH response to GHRP2 was 2.3 μg/l. Administration of hydrocortisone improved clinical symptoms and recovered hypogonadism and hypothyroidism by 2 weeks. Blood levels of ACE, lysozyme, and IgG4 were normal. Whole body gallium scanning did not detect any organ involvement other than the pituitary. Pituitary biopsy revealed many non-caseous granulomas accompanied with calcification. Immunostaining with PAB was positive for giant cells and infiltrated mononuclear cells. This patient fulfilled a diagnostic criterion of neurosarcoidosis by clinical manifestation of hypopituitarism, exclusion of other systemic inflammatory disorders, and histological identification of typical sarcoid granulomas.

Conclusion: Isolated pituitary sarcoidosis is an extremely rare inflammatory disorder. Currently six cases were reported and only three of them were biopsy-proven. This is the seventh case of isolated pituitary sarcoidosis and the first case in which P. acnes were identified definitely in sarcoid granulomas of the pituitary.