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April 10, 2001; 56 (7) Views & Reviews

Isolated angiitis of the CNS in children

S. Lanthier, A. Lortie, J. Michaud, R. Laxer, V. Jay, G. deVeber
First published April 10, 2001, DOI: https://doi.org/10.1212/WNL.56.7.837
S. Lanthier
From the Department of Pediatrics, Divisions of Neurology (Drs. Lanthier and deVeber) and Rheumatology (Dr. Laxer), and Department of Pathology (Dr. Jay), The Hospital for Sick Children and University of Toronto; the Département de Médecine, Service de Neurologie, Centre Hospitalier de l’Université de Montréal (Dr. Lanthier) and Département de Pédiatrie, Service de Neurologie, Hôpital Sainte-Justine (Dr. Lortie), and Université de Montréal; and the Department of Pathology (Dr. Michaud), Children’s Hospital of Eastern Ontario and University of Ottawa, Canada.
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A. Lortie
From the Department of Pediatrics, Divisions of Neurology (Drs. Lanthier and deVeber) and Rheumatology (Dr. Laxer), and Department of Pathology (Dr. Jay), The Hospital for Sick Children and University of Toronto; the Département de Médecine, Service de Neurologie, Centre Hospitalier de l’Université de Montréal (Dr. Lanthier) and Département de Pédiatrie, Service de Neurologie, Hôpital Sainte-Justine (Dr. Lortie), and Université de Montréal; and the Department of Pathology (Dr. Michaud), Children’s Hospital of Eastern Ontario and University of Ottawa, Canada.
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J. Michaud
From the Department of Pediatrics, Divisions of Neurology (Drs. Lanthier and deVeber) and Rheumatology (Dr. Laxer), and Department of Pathology (Dr. Jay), The Hospital for Sick Children and University of Toronto; the Département de Médecine, Service de Neurologie, Centre Hospitalier de l’Université de Montréal (Dr. Lanthier) and Département de Pédiatrie, Service de Neurologie, Hôpital Sainte-Justine (Dr. Lortie), and Université de Montréal; and the Department of Pathology (Dr. Michaud), Children’s Hospital of Eastern Ontario and University of Ottawa, Canada.
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R. Laxer
From the Department of Pediatrics, Divisions of Neurology (Drs. Lanthier and deVeber) and Rheumatology (Dr. Laxer), and Department of Pathology (Dr. Jay), The Hospital for Sick Children and University of Toronto; the Département de Médecine, Service de Neurologie, Centre Hospitalier de l’Université de Montréal (Dr. Lanthier) and Département de Pédiatrie, Service de Neurologie, Hôpital Sainte-Justine (Dr. Lortie), and Université de Montréal; and the Department of Pathology (Dr. Michaud), Children’s Hospital of Eastern Ontario and University of Ottawa, Canada.
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V. Jay
From the Department of Pediatrics, Divisions of Neurology (Drs. Lanthier and deVeber) and Rheumatology (Dr. Laxer), and Department of Pathology (Dr. Jay), The Hospital for Sick Children and University of Toronto; the Département de Médecine, Service de Neurologie, Centre Hospitalier de l’Université de Montréal (Dr. Lanthier) and Département de Pédiatrie, Service de Neurologie, Hôpital Sainte-Justine (Dr. Lortie), and Université de Montréal; and the Department of Pathology (Dr. Michaud), Children’s Hospital of Eastern Ontario and University of Ottawa, Canada.
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G. deVeber
From the Department of Pediatrics, Divisions of Neurology (Drs. Lanthier and deVeber) and Rheumatology (Dr. Laxer), and Department of Pathology (Dr. Jay), The Hospital for Sick Children and University of Toronto; the Département de Médecine, Service de Neurologie, Centre Hospitalier de l’Université de Montréal (Dr. Lanthier) and Département de Pédiatrie, Service de Neurologie, Hôpital Sainte-Justine (Dr. Lortie), and Université de Montréal; and the Department of Pathology (Dr. Michaud), Children’s Hospital of Eastern Ontario and University of Ottawa, Canada.
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Citation
Isolated angiitis of the CNS in children
S. Lanthier, A. Lortie, J. Michaud, R. Laxer, V. Jay, G. deVeber
Neurology Apr 2001, 56 (7) 837-842; DOI: 10.1212/WNL.56.7.837

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Abstract

Objective: To clarify the clinical features and pathologic manifestations of isolated angiitis of the CNS (IACNS) in children.

Methods: The authors report two new cases and summarize the literature of childhood IACNS confirmed by pathology.

Results: IACNS affecting small vessels (n = 5)—Neurologic manifestations included headaches, focal seizures, and progressive, behavioral, or multifocal neurologic impairment. MRI showed multifocal, T2-hyperintense, cerebral lesions without mass effect or tumor-like lesions. CSF, erythrocyte sedimentation rate, and cerebral angiograms were often normal. CNS biopsy disclosed a nongranulomatous vasculitis. Children were treated with prednisone alone or combined with cyclophosphamide. One child died. Four children had a favorable outcome. IACNS affecting large and medium arteries (n = 5)—Three children presented with acute ischemic stroke or TIA. Brain CT showed ischemic infarcts. Two children presented with subarachnoid hemorrhage. In this group, CSF, erythrocyte sedimentation rate, and angiograms were often abnormal. No patient received immunosuppressive therapy. Five children died. Autopsy showed granulomatous IACNS (n = 5)

Conclusions: Clinical and radiologic features correlate with the size of affected vessels. Prognosis differs between groups. Potential markers of poor outcome are acute stroke presentation secondary to large and medium-sized artery involvement, granulomatous angiitis, and delayed institution of immunosuppressive therapy.

  • Received August 22, 2000.
  • Accepted in final form December 23, 2000.
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