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Title Recurrent and Refractory Dacryocystitis as a Manifestation of IgG4-Related Disease: Two Cases with Different Presentations
Number 327
Author Lucy Li
Principal Unit where research was undertaken University of Cambridge

Purpose

IgG4-related disease (IgG4-RD) is a systemic fibro-inflammatory condition with recognised orbital involvement; however, nasolacrimal duct obstruction (NLDO) is uncommon. We present two cases of NLDO associated with IgG4-RD to highlight diagnostic and management challenges.

Methods

Retrospective review of two patients with different presentations of NLDO. Clinical features, surgical management, histopathology, and serological findings were analysed.

Results

Case 1 presented with bilateral epiphora and recurrent episodes of right acute dacryocystitis and orbital cellulitis on a background of chronic dacryocystitis and sinonasal disease. Despite multiple ENT and lacrimal procedures, symptoms persisted, ultimately requiring bilateral sequential dacryocystectomy leading to a histopathological diagnosis of IgG4-RD nearly four years after initial presentation. Case 2 presented with left chronic dacryocystitis and an irreducible lacrimal sac mucocoele with a contralateral dry eye. Following left dacryocystectomy, histopathological analysis of the lacrimal sac demonstrated dense lymphoplasmacytic infiltration consistent with IgG4-RD. Elevated serum IgG4 levels supported the diagnosis.

Conclusion

IgG4-RD should be considered in cases of atypical, recurrent, or treatment-resistant dacryocystitis. Histopathological evaluation plays a critical role in diagnosis. Early recognition may influence management strategies and prompt appropriate systemic assessment for IgG4-RD.


Additional Authors

First name Last name Hospital / Institution
Cornelius Rene
Twishaa Sheth


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