Literature DB >> 11891690

ABCD syndrome is caused by a homozygous mutation in the EDNRB gene.

Joke B G M Verheij1, Jürgen Kunze, Jan Osinga, Anthonie J van Essen, Robert M W Hofstra.   

Abstract

ABCD syndrome is an autosomal recessive syndrome characterized by albinism, black lock, cell migration disorder of the neurocytes of the gut (Hirschsprung disease [HSCR]), and deafness. This phenotype clearly overlaps with the features of the Shah-Waardenburg syndrome, comprising sensorineural deafness; hypopigmentation of skin, hair, and irides; and HSCR. Therefore, we screened DNA of the index patient of the ABCD syndrome family for mutations in the endothelin B receptor (EDNRB) gene, a gene known to be involved in Shah-Waardenburg syndrome. A homozygous nonsense mutation in exon 3 (R201X) of the EDNRB gene was found. We therefore suggest that ABCD syndrome is not a separate entity, but an expression of Shah-Waardenburg syndrome.

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Year:  2002        PMID: 11891690     DOI: 10.1002/ajmg.10172

Source DB:  PubMed          Journal:  Am J Med Genet        ISSN: 0148-7299


  11 in total

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Journal:  Pediatr Surg Int       Date:  2009-06-12       Impact factor: 1.827

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Authors:  Yassamine Doubaj; Véronique Pingault; Siham C Elalaoui; Ilham Ratbi; Mohamed Azouz; Hicham Zerhouni; Fouad Ettayebi; Abdelaziz Sefiani
Journal:  Mol Syndromol       Date:  2015-01-28

3.  Novel mutations of endothelin-B receptor gene in Pakistani patients with Waardenburg syndrome.

Authors:  Raheela Jabeen; Masroor Ellahi Babar; Jamil Ahmad; Ali Raza Awan
Journal:  Mol Biol Rep       Date:  2011-05-06       Impact factor: 2.316

Review 4.  Chromosomal and related Mendelian syndromes associated with Hirschsprung's disease.

Authors:  S W Moore
Journal:  Pediatr Surg Int       Date:  2012-09-23       Impact factor: 1.827

Review 5.  The contribution of associated congenital anomalies in understanding Hirschsprung's disease.

Authors:  S W Moore
Journal:  Pediatr Surg Int       Date:  2006-03-04       Impact factor: 1.827

Review 6.  Genetic insights, disease mechanisms, and biological therapeutics for Waardenburg syndrome.

Authors:  Sida Huang; Jian Song; Chufeng He; Xinzhang Cai; Kai Yuan; Lingyun Mei; Yong Feng
Journal:  Gene Ther       Date:  2021-02-25       Impact factor: 4.184

7.  Genomic amplification of the caprine EDNRA locus might lead to a dose dependent loss of pigmentation.

Authors:  Fiona Menzi; Irene Keller; Irene Reber; Julia Beck; Bertram Brenig; Ekkehard Schütz; Tosso Leeb; Cord Drögemüller
Journal:  Sci Rep       Date:  2016-06-22       Impact factor: 4.379

8.  Chromosome 13q deletion syndrome involving 13q31‑qter: A case report.

Authors:  Yue-Ping Wang; Da-Jia Wang; Zhi-Bin Niu; Wan-Ting Cui
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Journal:  PLoS One       Date:  2017-02-07       Impact factor: 3.240

Review 10.  Hereditary Hearing Impairment with Cutaneous Abnormalities.

Authors:  Tung-Lin Lee; Pei-Hsuan Lin; Pei-Lung Chen; Jin-Bon Hong; Chen-Chi Wu
Journal:  Genes (Basel)       Date:  2020-12-30       Impact factor: 4.096

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