IgG4-related hypothalamo-hypophysitis

Urushida, Yuki; Ishikawa, Daisuke; Yanaizumi, Mayuri; Nakamura, Takumi; Amari, Masakuni; Kawarabayashi, Tatsuhiko; Tosaka, Masahiko; Ikeda, Yoshio; Takatama, Masamitsu; Shoji, Mikio

doi:10.1016/j.ensci.2021.100362

Cited by 1 publication

(1 citation statement)

References 6 publications

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“…We used "hypophysitis," "IgG4-related hypophysitis," "diabetes insipidus" as keywords and searched on PubMed database. The review literature has been available for 115 cases of IgG4-RH before 2021, [1] so the search period was from January 1, 2021 to December 1, 2022, and 15 case reports were detected, [2][3][4][5][6][7][8][9][10][11][12][13][14][15][16] including 12 with complete clinical information and a total of 24 patients [2][3][4][5][6][7][9][10][11][12][13][14] (Table 1).…”

Section: Literature Reviewmentioning

confidence: 99%

Glucocorticoid impact therapy for recurrent IgG4-related disease with diabetes insipidus as the main manifestation: A case report and literature review

Yu,

Xu,

Wang

et al. 2023

Medicine

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Rationale: There is a relative wealth of experience in the initial treatment of IgG4-related disease (IgG4-RD), but little is known about therapeutic measures for recurrent cases combined with multiple organ and tissue involvement. Patient concerns: A 43-year-old man with a previous diagnosis of IgG4-RD due to recurrent right lacrimal gland enlargement with eyelid erythema presented with diabetes insipidus Diagnoses: We performed a pituitary Magnetic Resonance Imaging which revealed posterior pituitary rim changes with inhomogeneous enhancement and nodular-like thickening of the pituitary stalk, and performed a water-deprivation-vasopressin test confirmed central diabetes insipidus, and in combination with the patient’s elevated IgG4 levels and past medical conditions, we diagnosed central diabetes insipidus, IgG4-related hypophysitis, and IgG4-RD. Interventions: After the patient was admitted to the hospital we gave methylprednisolone 500 mg intravenously once daily for 4 days and again for 4 consecutive days after a 10-day interval. During this period combined with mycophenolate mofetil 250 mg twice daily and desmopressin acetate 0.1 mg 3 times daily. Outcomes: The patient was followed up for a sustained period of 6 months and no side effects of glucocorticoid therapy were noted, there were no signs of recurrence, and the daily urine output stabilized in the normal range. Lessons: We recognized that IgG4 levels do not reflect relapse or long-term control, and that glucocorticoid shock therapy is an optional and reliable treatment strategy for relapsed patients.

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