Document Detail

Neonatal lupus erythematosus: discordant disease expression of U1RNP-positive antibodies in fraternal twins--is this a subset of neonatal lupus erythematosus or a new distinct syndrome?
MedLine Citation:
PMID:  7722044     Owner:  NLM     Status:  MEDLINE    
Neonatal lupus erythematosus (NLE) is an uncommon disease that is manifested by cutaneous lesions, cardiac conduction defects, or both, that appear in utero or shortly after birth. In approximately 95% of patients, anti-Ro antibody (Ro[SS-A]) has been identified and has become the serologic marker for NLE. Since 1987 there have been four reported cases of Ro- and anti-La antibody (La[SS-B])-negative, U1RNP antibody-positive, NLE. Our affected twin, as well as all other infants with U1RNP-positive NLE, had cutaneous lesions similar to those in Ro-positive NLE, although they lacked systemic abnormalities, including cardiac conduction defects. HLA typing of mothers with infants with U1RNP-positive NLE revealed the presence of HLA-DR4, DQw1, or DQw3 phenotypes. Our typing confirms these findings. As with Ro-positive NLE, no distinct HLA associations were demonstrated in the infants. Unlike Ro-positive mothers, all mothers with a U1RNP-positive infant with NLE had connective tissue disease at the time of the diagnosis and had a different spectrum of disease. We describe the clinical, serologic, and immunogenetic findings in the first reported case of U1RNP-positive NLE in dizygotic twins in whom the NLE disease expression was discordant.
B A Solomon; T A Laude; A R Shalita
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Publication Detail:
Type:  Case Reports; Journal Article    
Journal Detail:
Title:  Journal of the American Academy of Dermatology     Volume:  32     ISSN:  0190-9622     ISO Abbreviation:  J. Am. Acad. Dermatol.     Publication Date:  1995 May 
Date Detail:
Created Date:  1995-05-24     Completed Date:  1995-05-24     Revised Date:  2004-11-17    
Medline Journal Info:
Nlm Unique ID:  7907132     Medline TA:  J Am Acad Dermatol     Country:  UNITED STATES    
Other Details:
Languages:  eng     Pagination:  858-62     Citation Subset:  IM    
Department of Dermatology, State University of New York-Health Science Center at Brooklyn 11203, USA.
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MeSH Terms
Antibodies, Antinuclear / analysis
Diseases in Twins*
HLA Antigens / analysis
Infant, Newborn
Lupus Erythematosus, Cutaneous / congenital*,  genetics*
Lupus Erythematosus, Systemic / genetics
Pregnancy Complications
Ribonucleoprotein, U1 Small Nuclear / analysis*
Twins, Dizygotic*
Reg. No./Substance:
0/Antibodies, Antinuclear; 0/HLA Antigens; 0/Ribonucleoprotein, U1 Small Nuclear; 0/SS-A antibodies; 0/SS-B antibodies

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