Literature DB >> 16160809

[Unilateral sensineural deafness associated with mutations in the PAX3-gene in Waardenburg syndrome type I].

M Ptok1, S Morlot.   

Abstract

Waardenburg syndrome (WS) type 1 occurs due to a mutation in the PAX3-gene on the long arm of chromosome 2. It is an autosomal dominant mutation with highly variable expression and high penetrance. Symptoms include the absence of melanocytes in the skin, hair, eyes and cochlea due to an early developmental disturbance in melancoytes from the neural crest. An inner ear disturbance is characteristic. Here we present a 4 year old girl with unilateral hearing loss, dystopia canthorum and partial albinism. Screening the entire PAX 3 gene revealed C64A und T164A mutations in exon I und II, both being missense mutations. Neither mutation has not been reported previously.

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Year:  2006        PMID: 16160809     DOI: 10.1007/s00106-005-1315-1

Source DB:  PubMed          Journal:  HNO        ISSN: 0017-6192            Impact factor:   1.284


  13 in total

1.  A cascade of genes related to Waardenburg syndrome.

Authors:  M Tachibana
Journal:  J Investig Dermatol Symp Proc       Date:  1999-09

2.  Transcription factor hierarchy in Waardenburg syndrome: regulation of MITF expression by SOX10 and PAX3.

Authors:  S B Potterf; M Furumura; K J Dunn; H Arnheiter; W J Pavan
Journal:  Hum Genet       Date:  2000-07       Impact factor: 4.132

3.  A new syndrome combining developmental anomalies of the eyelids, eyebrows and nose root with pigmentary defects of the iris and head hair and with congenital deafness.

Authors:  P J WAARDENBURG
Journal:  Am J Hum Genet       Date:  1951-09       Impact factor: 11.025

Review 4.  Molecular basis of congenital hypopigmentary disorders in humans: a review.

Authors:  R E Boissy; J J Nordlund
Journal:  Pigment Cell Res       Date:  1997 Feb-Apr

5.  Mutations of PAX3 unlikely in Waardenburg syndrome type 2.

Authors:  S Arias
Journal:  Nat Genet       Date:  1993-09       Impact factor: 38.330

Review 6.  Cyclic AMP a key messenger in the regulation of skin pigmentation.

Authors:  R Buscà; R Ballotti
Journal:  Pigment Cell Res       Date:  2000-04

7.  PAX3 gene structure, alternative splicing and evolution.

Authors:  T D Barber; M C Barber; T E Cloutier; T B Friedman
Journal:  Gene       Date:  1999-09-17       Impact factor: 3.688

8.  Homozygous and heterozygous inheritance of PAX3 mutations causes different types of Waardenburg syndrome.

Authors:  Bernd Wollnik; Turgut Tukel; Oya Uyguner; Asadollah Ghanbari; Hulya Kayserili; Melike Emiroglu; Memnune Yuksel-Apak
Journal:  Am J Med Genet A       Date:  2003-09-15       Impact factor: 2.802

9.  [Waardenburg syndrome. A heterogenic disorder with variable penetrance].

Authors:  F Apaydin; M Bereketoglu; O Turan; K Hribar; M M Maassen; O Günhan; H-P Zenner; M Pfister
Journal:  HNO       Date:  2004-06       Impact factor: 1.284

10.  Analysis of SOX10 mutations identified in Waardenburg-Hirschsprung patients: Differential effects on target gene regulation.

Authors:  Kwok Keung Chan; Corinne Kung Yen Wong; Vincent Chi Hang Lui; Paul Kwong Hang Tam; Mai Har Sham
Journal:  J Cell Biochem       Date:  2003-10-15       Impact factor: 4.429

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  1 in total

1.  [Propionic acidemia and sensorineural hearing loss: is there a connection at the molecular genetics level?].

Authors:  S Brosch; A Rauffeisen; M Baur; L Michels; F K Trefz; M Pfister
Journal:  HNO       Date:  2008-01       Impact factor: 1.284

  1 in total

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