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A Rare Unilateral Case of Ocular Mucous Membrane Pemphigoid

Roufas, Athena MBBS MMed(Ophth Sci)*; Kim, Peter MBBS(Hons), FRANZCO*; Reeves, Dianne BMed FRCPA†; Sutton, Gerard MBBS, MD, FRANZCO*‡§

Cornea:
doi: 10.1097/ICO.0b013e3181da580a
Case Report
Abstract

Purpose: Ocular mucous membrane pemphigoid (MMP) is known to be rapidly progressive in younger patients and affects both eyes. We are aware of only one other reported case of unilateral ocular MMP. The case presented in this study is unique in that it demonstrates genuinely unilateral ocular MMP in a younger patient.

Methods: We report a case of a 50-year-old man who presented with a 3-month history of left eye redness, irritation, and mild discharge. He also suffered from mouth ulcers, skin lesions, and recurrent nose bleeds secondary to nasal mucosal lesions.

Results: Examination revealed unilateral lid granulomas and cicatricial conjunctivitis in his left eye. There were no abnormal findings in his right eye. Biopsy of the lesions showed nonspecific inflammation with positive immunofluorescence for immunoglobulin G and C3 on epithelial basement membranes of conjunctiva, buccal mucosa, and skin, which were consistent with MMP. He was treated with immunosuppression and had no disease progression at 12-month follow-up.

Conclusions: This represents a rare case of unilateral nonprogressive ocular MMP in a younger patient. Histological analysis and immunofluorescence testing excluded a neoplastic process and confirmed the diagnosis. This case demonstrates that the presentation of unilateral eyelid pyogenic granulomas should include ocular MMP in the differential diagnosis once a neoplastic process has been excluded.

Author Information

From the *The Save Sight Institute, The University of Sydney and The Sydney Eye Hospital, Sydney, Australia; †Department of Anatomical Pathology, Prince of Wales Hospital, Sydney, Australia; ‡Department of Ophthalmology, Auckland University, Auckland, New Zealand; and §Vision Eye Institute, Sydney, Australia.

Received for publication October 10, 2009; revision received January 23, 2010; accepted February 18, 2010.

Reprints: Associate Professor Gerard L. Sutton, Level 3, 270 Victoria Avenue, Chatswood 2069, Sydney, Australia (e-mail: gsutton@theeyeinstitute.com.au) or Dr. Athena Roufas, PO Box 233, Enmore NSW 2042, Sydney, Australia (e-mail: athena.roufas@gmail.com).

© 2010 Lippincott Williams & Wilkins, Inc.