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Case Report
Cyclophosphamide Treatment of Primary Angiitis
of the Central Nervous System in Children:
Report of 2 Cases
KATHRYN J. BITTER, LEON G. EPSTEIN, HECTOR MELIN-ALDANA, JOHN G. CURRAN, and MICHAEL L. MILLER ABSTRACT. Primary angiitis of the central nervous system (PACNS) is a rare, idiopathic vasculitis diagnosed most frequently in adults. We describe 2 children presenting with hemiplegia from PACNS treated with cyclophosphamide. Diagnosis in one child was based on abnormal angiography. Oral, but not intravenous (IV), cyclophosphamide was effective in preventing progressive weakness. The second child had unremarkable angiography, but brain biopsy revealed vasculitis; IV cyclophosphamide prevented further weakness. Both cases highlight the importance of early diagnosis and treatment. (J Rheumatol 2006;33:2078-80) Key Indexing Terms:
CYCLOPHOSPHAMIDE From the Department of Pediatrics, Feinberg School of Medicine, Northwestern University, Chicago, Illinois, USA. K.J. Bitter, MD; L.G. Epstein, MD; H. Melin-Aldana, MD; J.G. Curran, MD; M.L. Miller, MD, Division of Immunology and Rheumatology, Children's Memorial Hospital. Address reprint requests to Dr. M.L. Miller, Division of Immunology and Rheumatology, Children's Memorial Hospital, 2300 Children's Plaza, Chicago, IL 60614. E-mail: millermd@northwestern.edu Accepted for publication April 4, 2006.
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