trimethoprim--sulfamethoxazole-drug-combination and Conjunctival-Diseases

We report a case of toxoplasmosis with bilateral maculopathy in a 7-year-old boy diagnosed with ataxia telangiectasia (AT) at age 6. AT manifests as ataxia, apraxia, telangiectasia, and dysarthria. Common ophthalmologic findings in AT include fine conjunctival telangiectasia. Patients also suffer from recurrent sinopulmonary infections; however, serious opportunistic infection is rarely diagnosed. At 8 years of age he developed disseminated Toxoplasma gondii (toxoplasmosis) infection and meningoencephalitis. This ophthalmologic finding and the subsequent toxoplasmosis meningoencephalitis have not been previously reported in AT.

Topics: Anti-Infective Agents; Ataxia Telangiectasia; Cerebrospinal Fluid; Child; Conjunctival Diseases; Eye Infections, Parasitic; Humans; Magnetic Resonance Imaging; Male; Polymerase Chain Reaction; Retinal Diseases; Tomography, Optical Coherence; Toxoplasma; Toxoplasmosis, Ocular; Trimethoprim, Sulfamethoxazole Drug Combination

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.

Topics: Administration, Oral; Antibodies, Antineutrophil Cytoplasmic; Autoantibodies; Biomarkers; Conjunctival Diseases; Female; Granulomatosis with Polyangiitis; Humans; Immunoglobulin G; Middle Aged; Scleritis; Trimethoprim, Sulfamethoxazole Drug Combination

Topics: Conjunctival Diseases; Drug Combinations; Drug Hypersensitivity; Humans; Male; Middle Aged; Sulfamethoxazole; Trimethoprim; Trimethoprim, Sulfamethoxazole Drug Combination