Literature DB >> 19375527

Radial aplasia in CHARGE syndrome: a new association.

E M M Burkitt Wright1, R O'Connor, B A Kerr.   

Abstract

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.

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Year:  2009        PMID: 19375527     DOI: 10.1016/j.ejmg.2009.03.017

Source DB:  PubMed          Journal:  Eur J Med Genet        ISSN: 1769-7212            Impact factor:   2.708


  6 in total

Review 1.  Chromodomain proteins in development: lessons from CHARGE syndrome.

Authors:  W S Layman; E A Hurd; D M Martin
Journal:  Clin Genet       Date:  2010-04-08       Impact factor: 4.438

2.  Atypical phenotypes associated with pathogenic CHD7 variants and a proposal for broadening CHARGE syndrome clinical diagnostic criteria.

Authors:  Caitlin L Hale; Adrienne N Niederriter; Glenn E Green; Donna M Martin
Journal:  Am J Med Genet A       Date:  2015-11-21       Impact factor: 2.802

Review 3.  Molecular and phenotypic aspects of CHD7 mutation in CHARGE syndrome.

Authors:  Gabriel E Zentner; Wanda S Layman; Donna M Martin; Peter C Scacheri
Journal:  Am J Med Genet A       Date:  2010-03       Impact factor: 2.802

4.  Mature middle and inner ears express Chd7 and exhibit distinctive pathologies in a mouse model of CHARGE syndrome.

Authors:  Elizabeth A Hurd; Meredith E Adams; Wanda S Layman; Donald L Swiderski; Lisa A Beyer; Karin E Halsey; Jennifer M Benson; Tzy-Wen Gong; David F Dolan; Yehoash Raphael; Donna M Martin
Journal:  Hear Res       Date:  2011-08-23       Impact factor: 3.208

5.  The chromatin remodeling protein CHD7, mutated in CHARGE syndrome, is necessary for proper craniofacial and tracheal development.

Authors:  Ethan D Sperry; Elizabeth A Hurd; Mark A Durham; Elyse N Reamer; Adam B Stein; Donna M Martin
Journal:  Dev Dyn       Date:  2014-07-10       Impact factor: 3.780

6.  Three novel mutations of CHD7 gene in two turkish patients with charge syndrome; A double point mutation and an insertion.

Authors:  O Giray Bozkaya; E Ataman; C Randa; D Onur Cura; S Gürsoy; O Aksel; A Ulgenalp
Journal:  Balkan J Med Genet       Date:  2015-12-30       Impact factor: 0.519

  6 in total

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