Document Detail


A new autosomal recessive congenital contractural syndrome in an Israeli Bedouin kindred.
MedLine Citation:
PMID:  12548738     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
We describe 23 cases with a syndrome of congenital contractures belonging to a large, inbred Israeli-Bedouin kindred. The phenotype described is similar to the Finnish type lethal congenital contracture syndrome yet differs in the following ways: by some additional craniofacial/ocular findings, by the lack of hydrops, multiple pterygia, and fractures, and by the normal duration of pregnancy. The major unique and previously undescribed clinical feature in our patients is a markedly distended urinary bladder as well as other urinary abnormalities. The vast majority of the cases died shortly after birth. Sonographic prenatal diagnosis was possible as early as 15 weeks gestation by demonstrating fetal akinesia, limb contractures, hydramnios, and distended urinary bladder. Linkage to 5q and 9q34 loci has been excluded.
Authors:
Daniella Landau; Anat Mishori-Dery; Reli Hershkovitz; Ginat Narkis; Khalil Elbedour; Rivka Carmi
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Publication Detail:
Type:  Journal Article    
Journal Detail:
Title:  American journal of medical genetics. Part A     Volume:  117A     ISSN:  1552-4825     ISO Abbreviation:  Am. J. Med. Genet. A     Publication Date:  2003 Feb 
Date Detail:
Created Date:  2003-01-27     Completed Date:  2003-05-20     Revised Date:  2008-05-21    
Medline Journal Info:
Nlm Unique ID:  101235741     Medline TA:  Am J Med Genet A     Country:  United States    
Other Details:
Languages:  eng     Pagination:  37-40     Citation Subset:  IM    
Copyright Information:
Copyright 2002 Wiley-Liss, Inc.
Affiliation:
Department of Neonatology, Soroka Medical Center and the Faculty of Health Sciences, Ben-Gurion University of the Negev, Beer-Sheva, Israel. landaud@bgumail.bgu.ac.il
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MeSH Terms
Descriptor/Qualifier:
Arabs / genetics*
Contracture / congenital,  genetics*
Family Health
Female
Genes, Recessive / genetics*
Humans
Infant, Newborn
Israel
Male
Pedigree
Pregnancy
Pregnancy Outcome
Prenatal Diagnosis
Syndrome

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


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