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    
Abstract/OtherAbstract:
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.
Authors:
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    
Affiliation:
Department of Dermatology, State University of New York-Health Science Center at Brooklyn 11203, USA.
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MeSH Terms
Descriptor/Qualifier:
Adult
Antibodies, Antinuclear / analysis
Diseases in Twins*
Female
HLA Antigens / analysis
Humans
Infant, Newborn
Lupus Erythematosus, Cutaneous / congenital*,  genetics*
Lupus Erythematosus, Systemic / genetics
Male
Pregnancy
Pregnancy Complications
Ribonucleoprotein, U1 Small Nuclear / analysis*
Twins, Dizygotic*
Chemical
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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