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Wegener granulomatosis causing compressive optic neuropathy in a child.

Vinay K Aakalu¹, Amjad Z Ahmad

¹Department of Ophthalmology and Visual Sciences, University of Illinois at Chicago, Chicago,IL 60612, USA. vaakalu@uic.edu

Ophthalmic Plastic and Reconstructive Surgery

|July 21, 2009

Summary

Wegener granulomatosis rarely causes compressive optic neuropathy in children. This case highlights successful treatment of unilateral vision loss and proptosis in a teen with this rare complication.

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Area of Science:

Ophthalmology
Rheumatology
Pediatrics

Background:

Wegener granulomatosis (WG), now known as granulomatosis with polyangiitis (GPA), is a rare autoimmune vasculitis.
Ophthalmic complications are common in WG/GPA, but compressive optic neuropathy is an infrequent manifestation, especially in pediatric patients.

Observation:

A 17-year-old male with a history of WG/GPA presented with acute unilateral vision loss, ocular pain, and proptosis in the left eye.
Ophthalmological examination revealed decreased visual acuity, pain on eye movement, and exophthalmos.
Computed tomography (CT) imaging demonstrated bilateral lacrimal gland enlargement causing compression of the left optic nerve.

Findings:

The patient received high-dose intravenous corticosteroids and daily oral cyclophosphamide.
Treatment led to a dramatic improvement in vision, resolution of ocular pain, and reduction of proptosis.
The patient was subsequently transitioned to maintenance therapy with mycophenolate mofetil and prednisone.

Implications:

This case underscores the importance of considering WG/GPA as a potential cause of compressive optic neuropathy in children and adolescents presenting with relevant ophthalmic symptoms.
Prompt diagnosis and aggressive immunosuppressive therapy are crucial for preserving vision and managing ocular complications in pediatric WG/GPA.
Ophthalmologists and rheumatologists should maintain a high index of suspicion for this rare but sight-threatening complication in affected children.