Rapid exacerbation of lymphocytic infundibuloneurohypophysitis

Kimitaka Shibue, Toshihito Fujii, Hisanori Goto, Yui Yamashita, Yoshihisa Sugimura, Masahiro Tanji, Akihiro Yasoda, Nobuya Inagaki

Research output: Contribution to journalArticle

Abstract

Rationale: Lymphocytic hypophysitis is a relatively rare autoimmune disease defined by lymphocytic infiltration to the pituitary. Its rarity and wide spectrum of clinical manifestations make clarification of the pathology difficult. Here, we describe a case we examined from the primary diagnosis to final discharge, showing the serial progression of lymphocytic infundibuloneurohypophysitis (LINH) to panhypopituitarism with extrapituitary inflammatory invasion in a short period, and responding favorably to high-dose glucocorticoid treatment. Patient concerns: Polyuria, General fatigue and Nausea/Vomiting. Diagnoses: Central diabetes insipidus (CDI), Lymphocytic infundibuloneurohypophysitis (LINH). Interventions: Desmopressin acetate, High-dose glucocorticoid (GC) treatment. Outcomes: He was prescribed desmopressin acetate and subsequently discharged. A month later, he revisited our hospital with general fatigue and nausea/vomiting. A screening test disclosed hypopituitarism with adrenal insufficiency. MRI revealed expanded contrast enhancement to the peripheral extrapituitary lesion. He received high-dose GC treatment and the affected lesion exhibited marked improvement on MRI, along with the recovery of the anterior pituitary function. Lessons: This case demonstrates the potential for classical LINH to develop into panhypopituitarsim. We consider this is the first documentation of approaching the cause of atypical LINH with progressive clinical course from the pathological viewpoint.

Original languageEnglish
Article numbere6034
JournalMedicine (United States)
Volume96
Issue number9
DOIs
Publication statusPublished - 01-03-2017

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Glucocorticoids
Deamino Arginine Vasopressin
Nausea
Vomiting
Fatigue
Neurogenic Diabetes Insipidus
Polyuria
Adrenal Insufficiency
Hypopituitarism
Rare Diseases
General Hospitals
Documentation
Autoimmune Diseases
Therapeutics
Autoimmune Hypophysitis
Pathology
Combined Pituitary Hormone Deficiency

All Science Journal Classification (ASJC) codes

  • Medicine(all)

Cite this

Shibue, K., Fujii, T., Goto, H., Yamashita, Y., Sugimura, Y., Tanji, M., ... Inagaki, N. (2017). Rapid exacerbation of lymphocytic infundibuloneurohypophysitis. Medicine (United States), 96(9), [e6034]. https://doi.org/10.1097/MD.0000000000006034
Shibue, Kimitaka ; Fujii, Toshihito ; Goto, Hisanori ; Yamashita, Yui ; Sugimura, Yoshihisa ; Tanji, Masahiro ; Yasoda, Akihiro ; Inagaki, Nobuya. / Rapid exacerbation of lymphocytic infundibuloneurohypophysitis. In: Medicine (United States). 2017 ; Vol. 96, No. 9.
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Shibue, K, Fujii, T, Goto, H, Yamashita, Y, Sugimura, Y, Tanji, M, Yasoda, A & Inagaki, N 2017, 'Rapid exacerbation of lymphocytic infundibuloneurohypophysitis', Medicine (United States), vol. 96, no. 9, e6034. https://doi.org/10.1097/MD.0000000000006034

Rapid exacerbation of lymphocytic infundibuloneurohypophysitis. / Shibue, Kimitaka; Fujii, Toshihito; Goto, Hisanori; Yamashita, Yui; Sugimura, Yoshihisa; Tanji, Masahiro; Yasoda, Akihiro; Inagaki, Nobuya.

In: Medicine (United States), Vol. 96, No. 9, e6034, 01.03.2017.

Research output: Contribution to journalArticle

TY - JOUR

T1 - Rapid exacerbation of lymphocytic infundibuloneurohypophysitis

AU - Shibue, Kimitaka

AU - Fujii, Toshihito

AU - Goto, Hisanori

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AU - Sugimura, Yoshihisa

AU - Tanji, Masahiro

AU - Yasoda, Akihiro

AU - Inagaki, Nobuya

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Y1 - 2017/3/1

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