The Journal of rheumatology · 2010
Wegener's granulomatosis strictly and persistently localized to one organ is rare: assessment of 16 patients from the French Vasculitis Study Group database.
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Pagnoux Christian, Stubbe Muriel, Lifermann François, Decaux Olivier, Pavic Michel, Bérezné Alice, Delacroix-Szmania Isabelle, Méaux-Ruault Nadine, Bienvenu Boris, Cabane Jean, Guillevin Loïc, French Vasculitis Study Group
Аннотация
OBJECTIVE: To study the frequency and characteristics of patients with Wegener's granulomatosis (WG) strictly and persistently localized to one organ.
METHODS: Retrospective analysis of the French Vasculitis Study Group (FVSG) WG cohort.
RESULTS: Sixteen patients (3.2% of the cohort) were identified who had isolated lung nodules, ear-nose-throat, or ocular involvement that did not progress to systemic disease (median followup, 58 mo) over the period of observation. Ten received first-line therapy with cyclophosphamide, which was effective in 4. Cotrimoxazole alone achieved remission in one, combined with corticosteroids in 3. Eight required subsequent treatments because of first-line failure or relapse.
CONCLUSION: Strictly and persistently localized WG is uncommon. Optimal treatment remains to be determined.
METHODS: Retrospective analysis of the French Vasculitis Study Group (FVSG) WG cohort.
RESULTS: Sixteen patients (3.2% of the cohort) were identified who had isolated lung nodules, ear-nose-throat, or ocular involvement that did not progress to systemic disease (median followup, 58 mo) over the period of observation. Ten received first-line therapy with cyclophosphamide, which was effective in 4. Cotrimoxazole alone achieved remission in one, combined with corticosteroids in 3. Eight required subsequent treatments because of first-line failure or relapse.
CONCLUSION: Strictly and persistently localized WG is uncommon. Optimal treatment remains to be determined.