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; Michael L. Miller

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, Michael L. Miller^*

^*Corresponding author for this work

Research output: Contribution to journal › Article › peer-review

25 Scopus citations

Abstract

Primary angiitis of the central nervous system (PACNS) is a rare, idiopathic vusculitis diagnosed most frequently in adults. We deserihe 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.

Original language	English (US)
Pages (from-to)	2078-2080
Number of pages	3
Journal	Journal of Rheumatology
Volume	33
Issue number	10
State	Published - Oct 1 2006

Keywords

Children
Cyclophosphamide
Primary angiitis

ASJC Scopus subject areas

Rheumatology
Immunology and Allergy
Immunology

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title = "Cyclophosphamide treatment of primary angiitis of the central nervous system in children: Report of 2 cases",

abstract = "Primary angiitis of the central nervous system (PACNS) is a rare, idiopathic vusculitis diagnosed most frequently in adults. We deserihe 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.",

keywords = "Children, Cyclophosphamide, Primary angiitis",

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AU - Bitter, Kathryn J.

AU - Epstein, Leon G.

AU - Melin-Aldana, Hector

AU - Curran, John G.

AU - Miller, Michael L.

PY - 2006/10/1

Y1 - 2006/10/1

N2 - Primary angiitis of the central nervous system (PACNS) is a rare, idiopathic vusculitis diagnosed most frequently in adults. We deserihe 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.

AB - Primary angiitis of the central nervous system (PACNS) is a rare, idiopathic vusculitis diagnosed most frequently in adults. We deserihe 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.

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KW - Primary angiitis

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