Document Detail


Clinical features of anti-factor H autoantibody-associated hemolytic uremic syndrome.
MedLine Citation:
PMID:  21051740     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
Atypical hemolytic uremic syndrome (aHUS) is a rare form of thrombotic microangiopathy that associates, in 70% of cases, with genetic or acquired disorders leading to dysregulation of the alternative pathway of complement. Autoantibody directed against Factor H causes at least 6% to 10% of aHUS cases, but only a few clinical reports are available. Here, we describe the clinical, biologic, genetic features, treatment, and outcome of 45 patients who presented with aHUS associated with anti-FH autoantibody. We found that this form of aHUS primarily affects children between 9 and 13 years old but it also affects adults. It presents with a high frequency of gastrointestinal symptoms and with extrarenal complications and has a relapsing course. Activation of the alternative pathway of complement at the onset of disease portends a poor prognosis. Early specific treatment may lead to favorable outcomes. These data should improve the recognition and diagnosis of this form of aHUS and help identify patients at high risk of a poor outcome.
Authors:
Marie-Agnès Dragon-Durey; Sidharth Kumar Sethi; Arvind Bagga; Caroline Blanc; Jacques Blouin; Bruno Ranchin; Jean-Luc André; Nobuaki Takagi; Hae Il Cheong; Pankaj Hari; Moglie Le Quintrec; Patrick Niaudet; Chantal Loirat; Wolf Herman Fridman; Véronique Frémeaux-Bacchi
Publication Detail:
Type:  Journal Article; Research Support, Non-U.S. Gov't     Date:  2010-11-04
Journal Detail:
Title:  Journal of the American Society of Nephrology : JASN     Volume:  21     ISSN:  1533-3450     ISO Abbreviation:  J. Am. Soc. Nephrol.     Publication Date:  2010 Dec 
Date Detail:
Created Date:  2010-12-01     Completed Date:  2011-01-07     Revised Date:  2011-12-21    
Medline Journal Info:
Nlm Unique ID:  9013836     Medline TA:  J Am Soc Nephrol     Country:  United States    
Other Details:
Languages:  eng     Pagination:  2180-7     Citation Subset:  IM    
Affiliation:
Service d'Immunologie Biologique, Hôpital Européen Georges Pompidou, Assistance Publique-Hôpitaux de Paris, Paris, France. marie-agnes.durey@egp.aphp.fr
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MeSH Terms
Descriptor/Qualifier:
Adult
Age Factors
Autoantibodies / immunology*
Child
Child, Preschool
Cohort Studies
Complement Activation / immunology
Complement Factor H / immunology*
Female
Follow-Up Studies
Hemolytic-Uremic Syndrome / diagnosis,  epidemiology,  immunology*,  therapy*
Humans
Immunoglobulins, Intravenous / administration & dosage
Infant
Kidney Transplantation
Male
Middle Aged
Plasma Exchange / methods
Risk Assessment
Severity of Illness Index
Sex Factors
Stem Cell Transplantation / methods
Time Factors
Treatment Outcome
Chemical
Reg. No./Substance:
0/Autoantibodies; 0/Immunoglobulins, Intravenous; 80295-65-4/Complement Factor H

From MEDLINE®/PubMed®, a database of the U.S. National Library of Medicine


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