Trimethoprim-sulfamethoxazole for scleritis associated with limited Wegener's granulomatosis: use of histopathology and anti-neutrophil cytoplasmic antibody (ANCA) test.
Ophthalmic involvement may be noted in < or = 58% of Wegener's granulomatosis cases, scleritis being one of the most frequent and potentially devastating manifestations. Cytotoxic immunosuppressive drug therapy is effective treatment for this disorder but potentially highly toxic. Recent uncontrolled and anecdotal reports have suggested a possible therapeutic role for a much less toxic agent, trimethoprim/sulfamethoxazole, in limited Wegener's granulomatosis. We report a patient who had a conjunctival nodule and scleritis. Biopsy of the nodule suggested Wegener's granulomatosis, confirmed serologically with serum anti-neutrophil cytoplasmic antibody (ANCA) testing. Treatment with oral trimethoprim/sulfamethoxazole was successful. Clinical response was paralleled by normalization of serial anti-neutrophil cytoplasmic antibody titers. This case is the first well-documented ophthalmologic report of limited Wegener's granulomatosis responding to trimethoprim/sulfamethoxazole and adds to the body of literature suggesting a potential role for this drug in selected cases of limited Wegener's granulomatosis.
Soukiasian
S H
SH
Department of Ophthalmology, Massachusetts Eye and Ear Infirmary, Boston.
Jakobiec
F A
FA
Niles
J L
JL
Pavan-Langston
D
D
D000284:Administration, Oral; D019268:Antibodies, Antineutrophil Cytoplasmic; D001323:Autoantibodies / Q000032:analysis*; D015415:Biomarkers; D003229:Conjunctival Diseases / Q000175:diagnosis / Q000188:drug therapy / Q000209:etiology; D005260:Female; D014890:Granulomatosis with Polyangiitis / Q000150:complications / Q000175:diagnosis / Q000188:drug therapy*; D006801:Humans; D007074:Immunoglobulin G / Q000032:analysis*; D008875:Middle Aged; D015423:Scleritis / Q000175:diagnosis / Q000188:drug therapy* / Q000209:etiology; D015662:Trimethoprim, Sulfamethoxazole Drug Combination / Q000627:therapeutic use*
D019268:Antibodies, Antineutrophil Cytoplasmic; D001323:Autoantibodies; D015415:Biomarkers; D007074:Immunoglobulin G; D015662:Trimethoprim, Sulfamethoxazole Drug Combination