Document Detail

Association of arthrogryposis multiplex congenita with maternal antibodies inhibiting fetal acetylcholine receptor function.
MedLine Citation:
PMID:  8941654     Owner:  NLM     Status:  MEDLINE    
Arthrogryposis multiplex congenita (AMC), characterized by multiple joint contractures developing in utero, results from lack of fetal movement. Some cases are genetically determined, but AMC occasionally complicates pregnancy in patients with myasthenia gravis (MG) suggesting involvement of circulating maternal antibodies. We previously demonstrated antibodies that inhibited the function of fetal acetylcholine receptor (AChR) in one healthy woman with an obstetric history of recurrent AMC. Here we study sera from this woman, from one other with a similar history, and from three (one asymptomatic) whose babies had neonatal MG and AMC. All five maternal sera had high titers of antibodies that inhibited alpha-Bungarotoxin (alpha-BuTx) binding to fetal AChR, and their sera markedly inhibited fetal AChR function with little effect on adult AChR function. Moreover, in a further survey, 3 of 20 sera from anti-AChR negative AMC mothers inhibited fetal AChR function significantly at 1:100 dilution. These results demonstrate the role of antibodies to fetal AChR and perhaps other muscle antigens in some cases of AMC. More generally, they suggest that placental transfer of antibodies directed at fetal antigens should be considered as a cause of other recurrent fetal or perinatal disorders.
S Riemersma; A Vincent; D Beeson; C Newland; S Hawke; B Vernet-der Garabedian; B Eymard; J Newsom-Davis
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Publication Detail:
Type:  Case Reports; Journal Article; Research Support, Non-U.S. Gov't    
Journal Detail:
Title:  The Journal of clinical investigation     Volume:  98     ISSN:  0021-9738     ISO Abbreviation:  J. Clin. Invest.     Publication Date:  1996 Nov 
Date Detail:
Created Date:  1997-01-23     Completed Date:  1997-01-23     Revised Date:  2009-11-18    
Medline Journal Info:
Nlm Unique ID:  7802877     Medline TA:  J Clin Invest     Country:  UNITED STATES    
Other Details:
Languages:  eng     Pagination:  2358-63     Citation Subset:  AIM; IM    
Neurosciences Group, Institute of Molecular Medicine, John Radcliffe Hospital, Oxford, United Kingdom.
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MeSH Terms
Antibodies, Blocking / immunology*
Antibodies, Monoclonal / immunology
Arthrogryposis / complications,  etiology,  immunology*
Bungarotoxins / immunology
Fetal Diseases / immunology*
Immunoglobulin G / immunology
Myasthenia Gravis / complications,  immunology*
Precipitin Tests
Receptors, Cholinergic / immunology*,  physiology
Seroepidemiologic Studies
Reg. No./Substance:
0/Antibodies, Blocking; 0/Antibodies, Monoclonal; 0/Bungarotoxins; 0/Immunoglobulin G; 0/Receptors, Cholinergic

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