• Mayari Ito, Aric Vaidya, Hirohiko Kakizaki, and Yasuhiro Takahashi.
• Department of Oculoplastic, Orbital & Lacrimal Surgery, Aichi Medical University Hospital, Nagakute, Aichi, Japan.
• Medicine (Baltimore). 2023 Jan 6; 102 (1): e32618e32618.

PurposeImmunoglobulin G4 (IgG4)-related dacryoadenitis is rarely resolved spontaneously without steroids. Here, we report a case of IgG4-related dacryoadenitis and extra-ophthalmic lesions with spontaneous regression.MethodsThis is a clinical case report. A 56-years-old man had a 1-year and 7-month-old histories of neck and eyelid swelling, respectively. On the first examination, the lacrimal and submandibular glands were palpable bilaterally. Computed tomographic images showed enlargement of the lacrimal gland on both sides, right pulmonary hilar lymph node, and pancreas, and thickening of the abdominal aortic wall. Blood tests demonstrated elevated serum IgG4 level and positive hepatitis B surface antibody. Pathological examination of the biopsied lacrimal gland specimens revealed marked IgG4-positive plasma cell infiltration.ResultsThe patient was monitored carefully without steroid administration. Serum IgG4 level had gradually decreased during follow-up period and reached the normal range 3 years after the biopsy. At 4-year follow-up, the lacrimal and submandibular glands were not palpable on either side. Computed tomographic images demonstrated no enlargement of the lacrimal gland, submandibular gland, or lymph nodes, and improvement of the enlarged pancreas and thickened abdominal aortic wall.ConclusionOur case indicates that careful observation can be an option in selected cases with risks of steroid treatment or silent clinical course.Copyright © 2022 the Author(s). Published by Wolters Kluwer Health, Inc.

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