Document Detail


A novel nonsense B3GALTL mutation confirms Peters plus syndrome in a patient with multiple malformations and Peters anomaly.
MedLine Citation:
PMID:  21067481     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
Peters plus syndrome is an autosomal recessive rare congenital disorder defined by corneal Peters anomaly with short disproportionate stature, development delay and dysmorphic facial features. In addition, cardiac, genito-urinary and/or central nervous system malformations can be present. Mutations in the beta-1,3-galactosyltransferase-like glycosyltransferase gene (B3GALTL) have been reported in patients with Peters plus syndrome prompting phenotype-genotype studies because of the variable clinical spectrum related to the syndrome. A 20 month old boy presenting with bilateral Peters anomaly in association with multiple developmental anomalies including cerebral malformations was found to carry a novel homozygous B3GALTL nonsense mutation [p.Tyr366X]. This is the first stop mutation described in association with this gene. The present report confirms the wide clinical spectrum of Peters plus syndrome, underlines the major clinical criteria of the syndrome and the major implication of B3GALTL gene in this condition. Ophthalmologic examination in multiple developmental anomalies remains an important clinical issue that may lead to specific gene screening. In Peters plus syndrome B3GALTL molecular test provides diagnosis confirmation and improves dramatically genetic counselling for the families.
Authors:
K Aliferis; C Marsal; V Pelletier; B Doray; M M Weiss; C M J Tops; C Speeg-Schatz; S A J Lesnik; H Dollfus
Publication Detail:
Type:  Case Reports; Journal Article    
Journal Detail:
Title:  Ophthalmic genetics     Volume:  31     ISSN:  1744-5094     ISO Abbreviation:  Ophthalmic Genet.     Publication Date:  2010 Dec 
Date Detail:
Created Date:  2010-11-11     Completed Date:  2011-01-21     Revised Date:  -    
Medline Journal Info:
Nlm Unique ID:  9436057     Medline TA:  Ophthalmic Genet     Country:  England    
Other Details:
Languages:  eng     Pagination:  205-8     Citation Subset:  IM    
Affiliation:
Centre de Référence pour les Affections Rares en Génétique Ophtalmologique, Hôpitaux Universitaires de Strasbourg, Strasbourg, France. k.aliferis@gmailcom
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MeSH Terms
Descriptor/Qualifier:
Abnormalities, Multiple / genetics*
Anterior Eye Segment / abnormalities
Codon, Nonsense*
Corneal Opacity / genetics
Developmental Disabilities / genetics
Eye Abnormalities / genetics
Galactosyltransferases / genetics*
Glucosyltransferases / genetics*
Humans
Infant
Male
Nervous System Malformations / genetics
Polymerase Chain Reaction
Syndrome
Chemical
Reg. No./Substance:
0/Codon, Nonsense; EC 2.4.1.-/B3GALTL protein, human; EC 2.4.1.-/Galactosyltransferases; EC 2.4.1.-/Glucosyltransferases

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


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