Document Detail


Radial aplasia in CHARGE syndrome: a new association.
MedLine Citation:
PMID:  19375527     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
CHARGE syndrome affects up to 1 in 8500 births, and is most commonly due to de novo truncating mutations in the CHD7 gene. In addition to the 4 major (choanal atresia, coloboma, cranial nerve dysfunction and characteristic ear abnormalities) and 7 minor features (genital hypoplasia, developmental delay, cardiac anomalies, growth retardation, orofacial clefting, tracheo-oesophageal fistula and characteristic facies) proposed by Blake et al. [K.D. Blake, S.L.H. Davenport, B.D. Hall, M.A. Hefner, R.A. Pagon, M.S. Williams, A.E. Lin, J.M. Graham Jr., CHARGE association: an update and review for the primary pediatrician, Clin. Pediatr. (Phila) 37 (1998) 159-173.], many different features have been described in affected patients. Limb defects do not feature in the original designation of the condition, but occasional reports have noted tibial aplasia and other less severe limb anomalies. Presented here is the first case of radial aplasia in a patient with CHARGE syndrome due to a novel frameshift mutation of CHD7.
Authors:
E M M Burkitt Wright; R O'Connor; B A Kerr
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Publication Detail:
Type:  Case Reports; Journal Article     Date:  2009-04-16
Journal Detail:
Title:  European journal of medical genetics     Volume:  52     ISSN:  1878-0849     ISO Abbreviation:  Eur J Med Genet     Publication Date:    2009 Jul-Aug
Date Detail:
Created Date:  2009-07-27     Completed Date:  2009-11-02     Revised Date:  -    
Medline Journal Info:
Nlm Unique ID:  101247089     Medline TA:  Eur J Med Genet     Country:  Netherlands    
Other Details:
Languages:  eng     Pagination:  239-41     Citation Subset:  IM    
Affiliation:
Medical Genetics Research Group and Regional Genetics Service, University of Manchester and Central Manchester University Hospitals Foundation Trust, St Mary's Hospital, Hathersage Road, Manchester M13 0JH, UK.
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MeSH Terms
Descriptor/Qualifier:
Abnormalities, Multiple / genetics
Adolescent
Choanal Atresia / genetics*
Coloboma / genetics*
DNA Helicases / genetics
DNA-Binding Proteins / genetics
Ear / abnormalities*
Exons
Female
Frameshift Mutation
Growth Disorders / genetics*
Heart Defects, Congenital / genetics*
Humans
Pregnancy
Radius / abnormalities*
Sequence Analysis, DNA
Syndrome
Upper Extremity Deformities, Congenital / genetics*
Chemical
Reg. No./Substance:
0/DNA-Binding Proteins; EC 3.6.1.-/DNA Helicases; EC 5.99.-/CHD7 protein, human

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