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Lymphocytic panhypophysitis and anti-rabphilin-3A antibody with pulmonary sarcoidosis

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Title: Lymphocytic panhypophysitis and anti-rabphilin-3A antibody with pulmonary sarcoidosis
Authors: Takahashi, Yuka Browse this author
Kameda, Hiraku Browse this author →KAKEN DB
Miya, Aika Browse this author
Nomoto, Hiroshi Browse this author →KAKEN DB
Cho, Kyu Yong Browse this author
Nakamura, Akinobu Browse this author →KAKEN DB
Nishimura, Hiroki Browse this author
Kimura, Hirokazu Browse this author →KAKEN DB
Suzuki, Masaru Browse this author →KAKEN DB
Konno, Satoshi Browse this author →KAKEN DB
Shimizu, Ai Browse this author
Matsuno, Yoshihiro Browse this author →KAKEN DB
Okamoto, Michinari Browse this author
Motegi, Hiroaki Browse this author →KAKEN DB
Iwata, Naoko Browse this author
Fujisawa, Haruki Browse this author
Suzuki, Atsushi Browse this author
Sugimura, Yoshihisa Browse this author
Miyoshi, Hideaki Browse this author →KAKEN DB
Atsumi, Tatsuya Browse this author →KAKEN DB
Keywords: Lymphocytic panhypophysitis
Anti-rabphilin-3a antibody
Issue Date: 6-Apr-2023
Publisher: Springer
Journal Title: Pituitary
Volume: 25
Issue: 2
Start Page: 321
End Page: 327
Publisher DOI: 10.1007/s11102-021-01200-0
Abstract: Purpose To explore the clinical significance of anti-rabphillin-3A antibody for the differential diagnosis of lymphocytic panhypophysitis. Methods and results A 58-year-old Japanese man developed uveitis of unknown cause in 2017. In 2019, he became aware of polyuria. In August 2020, he noticed transient diplopia and was diagnosed with right abducens nerve palsy. At the same time, he complained of fatigue and loss of appetite. Head magnetic resonance imaging demonstrated enlargement of the pituitary stalk and pituitary gland, corresponding to hypophysitis. Hormone stimulation tests showed blunted responses with respect to all anterior pituitary hormones. Central diabetes insipidus was diagnosed on the basis of a hypertonic saline loading test. Taking these findings together, a diagnosis of panhypopituitarism was made. Computed tomography showed enlargement of hilar lymph nodes. Biopsies of the hilar lymph nodes revealed non-caseating epithelioid cell granulomas that were consistent with sarcoidosis. Biopsy of the anterior pituitary revealed mild lymphocyte infiltration in the absence of IgG4-positive cells, non-caseating granulomas, or neoplasia. Western blotting revealed the presence of anti-rabphilin-3A antibody, supporting a diagnosis of lymphocytic panhypophysitis. Because the patient had no visual impairment or severe uveitis, we continued physiological hormone replacement therapy and topical steroid therapy for the uveitis. Conclusion To the best of our knowledge, this is the first case of anti-rabphilin 3A antibody positive lymphocytic panhypophysitis comorbid with sarcoidosis, diagnosed by both pituitary and hilar lymph node biopsy. The utility of anti-rabphilin-3A antibody for the differential diagnosis of hypophysitis like this case should be clarified with further case studies.
Rights: This is a post-peer-review, pre-copyedit version of an article published in Pituitary. The final authenticated version is available online at:
Type: article (author version)
Appears in Collections:北海道大学病院 (Hokkaido University Hospital) > 雑誌発表論文等 (Peer-reviewed Journal Articles, etc)

Submitter: 亀田 啓

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