Immunoglobulin G4-related lacrimal sac dacryocystocele: A novel case report

Am J Ophthalmol Case Rep. 2020 Jun 20:19:100789. doi: 10.1016/j.ajoc.2020.100789. eCollection 2020 Sep.

Abstract

Purpose: Immunoglobulin G4-related disease is a systemic fibroinflammatory disease of unknown etiology. Immunoglobulin G4-related ophthalmic disease (IgG4-ROD) can manifest in multiple ways, but lacrimal sac involvement is rare. We present the first case in Chinese population of lacrimal sac IgG4-ROD.

Observations: Lacrimal sac IgG4-ROD is rare, and only 9 cases were reported in literature. Despite reported cases in Asian population, mainly in Japan, there was none from the Chinese population or South Asia. Our index case is a 67-year-old Chinese male, who presented with a left insidious nasolacrimal duct swelling mimicking dacryocystocele. Lacrimal sac IgG4-ROD was diagnosed with radiological, serological, pathological and immunohistochemical evidence. The under-reporting of this disease entity may suggest a benign course of such.

Conclusions and importance: This is the first reported case of biopsy proven lacrimal sac definite IgG4-ROD in Chinese patient on English literature. With the limited cases reported in literature, the pathology of Immunoglobulin G4 immune process in lacrimal sac demands further investigation.

Keywords: Chinese; Dacryocystocele; Immunoglobulin-G4 related disease; Lacrimal sac tumor.

Publication types

  • Case Reports