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J Clin Rheumatol ; 16(3): 125-8, 2010 Apr.
Article de Anglais | MEDLINE | ID: mdl-20375822

RÉSUMÉ

A 43-year-old Brazilian female presented in 2001 with nasal stuffiness and sinusitis. A biopsy was consistent with limited Wegener's granulomatosis although antineutrophil cytoplasmic antibodies were negative. Her nasal inflammation progressed despite trials of prednisone, methotrexate, and azathioprine. A septal perforation developed and a repeat biopsy showed granulomatous inflammation. In 2006 the patient was referred to Division of Rheumatology, University of California, Los Angeles. The nose was grossly erythematous and a magnetic resonance imaging revealed nasal destruction and sinusitis. Palatine biopsies showed chronic inflammation. Cyclophosphamide at 150 mg/d resulted in markedly improved mucocutaneous lesions. The patient developed a leg and arm rash in 2007. A skin biopsy was positive for Leishmania braziliensis. The cyclophosphamide was discontinued and amphotericin B was initiated with transient benefit. Remission was achieved with pentavalent antimony. Despite multiple nasopharyngeal biopsies, for a 6-year span, mucocutaneous leishmaniasis masqueraded as Wegener's granulomatosis. Cyclophosphamide not only resulted in clinical improvement, due to reduced inflammatory response, but also allowed widespread cutaneous dissemination.


Sujet(s)
Antiprotozoaires/usage thérapeutique , Granulomatose avec polyangéite/diagnostic , Leishmaniose cutanéomuqueuse/diagnostic , Leishmaniose cutanéomuqueuse/traitement médicamenteux , Méglumine/usage thérapeutique , Composés organométalliques/usage thérapeutique , Adulte , Amphotéricine B/administration et posologie , Brésil , Californie , Diagnostic différentiel , Femelle , Humains , Perfusions veineuses , Antimoniate de méglumine , Orientation vers un spécialiste
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