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Declaration of Funding NIH Training Grant T32 AI007469 (CG, LB) Conflict of Interest Dr. Hoffman is a consultant for Sobi Biovitrum -
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- Publisher
- Springer Journals
- Copyright
- Copyright © 2012 by Springer Science+Business Media, LLC
- Subject
- Biomedicine; Immunology; Infectious Diseases; Internal Medicine; Medical Microbiology
- ISSN
- 0271-9142
- eISSN
- 1573-2592
- DOI
- 10.1007/s10875-012-9767-z
- pmid
- 22926405
- Publisher site
- See Article on Publisher Site
Abstract
J Clin Immunol (2013) 33:162–171 DOI 10.1007/s10875-012-9767-z ORIGINAL RESEARCH Mutations of Complement Factor I and Potential Mechanisms of Neuroinflammation in Acute Hemorrhagic Leukoencephalitis Lori Broderick & Chhavi Gandhi & James L. Mueller & Christopher D. Putnam & Katayoon Shayan & Patricia C. Giclas & Karin S. Peterson & Seema S. Aceves & Robert M. Sheets & Bradley M. Peterson & Robert O. Newbury & Hal M. Hoffman & John F. Bastian Received: 3 July 2012 /Accepted: 9 August 2012 /Published online: 29 August 2012 Springer Science+Business Media, LLC 2012 Abstract Methods Extensive clinical workup of two patients pre- Purpose Acute Hemorrhagic Leukoencephalitis (AHLE) is senting with AHLE revealed complement abnormalities, a rare demyelinating disorder of acute onset, rapid deterio- specifically related to the alternative pathway and its ration and significant morbidity and mortality. Most often regulator, FI. Aggressive management with steroids, im- described as a post-infectious complication of an upper munoglobulin, and anakinra ultimately led to improve- respiratory illness, its precise pathophysiology remains un- ment of clinical status and near return to neurologic clear. We describe two pediatric patients with AHLE with baseline in both patients. Genetic sequencing of the FI partial complement factor I (FI) deficiency whose successful
Journal
Journal of Clinical Immunology – Springer Journals
Published: Aug 29, 2012