Literature DB >> 21998865

Report of a further family with dominant deafness-onychodystrophy (DDOD) syndrome.

Susan M White1, Michael Fahey.   

Abstract

Deafness and onychodystrophy is a presumed autosomal dominant condition previously reported in five families. We report on a three-generation family with three affected members with absent finger and toenails, finger-like thumbs, and severe sensorineural deafness. In the hands, the first and fifth digits had absent nails and bulbous swelling of the distal phalanx. The second, third, and fourth digits were relatively spared. In the feet,there were hypoplastic great toenails and absent nails on second to fifth toes. Intelligence was normal. In addition to deafness and onychodystrophy, other features in this family included subtle facial dysmorphism in two individuals, cutis aplasia in one individual, epilepsy in one individual, and sudden infant death in two family members, one of whom had deafness and onychodystrophy and one who did not. A full autopsy of both infants did not reveal a cause. SNP microarray analysis of one family member showed no evidence of copy number change. This family's condition fits within the spectrum of dominant deafness-onychodystrophy syndrome (DDOD) and further characterises this rare condition.

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Year:  2011        PMID: 21998865     DOI: 10.1002/ajmg.a.34184

Source DB:  PubMed          Journal:  Am J Med Genet A        ISSN: 1552-4825            Impact factor:   2.802


  5 in total

1.  A Danish family with dominant deafness-onychodystrophy syndrome.

Authors:  Dina Vind-Kezunovic; Pernille M Torring
Journal:  J Dermatol Case Rep       Date:  2013-12-30

2.  Dominant deafness-onychodystrophy syndrome caused by an ATP6V1B2 mutation.

Authors:  Ibis Menendez; Claudia Carranza; Mariana Herrera; Nely Marroquin; Joseph Foster; Filiz Basak Cengiz; Guney Bademci; Mustafa Tekin
Journal:  Clin Case Rep       Date:  2017-02-08

3.  Case Report: Exome Sequencing Identified Variants in Three Candidate Genes From Two Families With Hearing Loss, Onychodystrophy, and Epilepsy.

Authors:  Yuan Li; Jianjun Xiong; Yi Zhang; Lin Xu; Jianyun Liu; Tao Cai
Journal:  Front Genet       Date:  2021-11-29       Impact factor: 4.599

Review 4.  Genetic architecture and phenotypic landscape of deafness and onychodystrophy syndromes.

Authors:  Xue Gao; Pu Dai; Yong-Yi Yuan
Journal:  Hum Genet       Date:  2021-07-07       Impact factor: 4.132

5.  De novo mutation in ATP6V1B2 impairs lysosome acidification and causes dominant deafness-onychodystrophy syndrome.

Authors:  Yongyi Yuan; Jianguo Zhang; Qing Chang; Jin Zeng; Feng Xin; Jianjun Wang; Qingyan Zhu; Jing Wu; Jingqiao Lu; Weiwei Guo; Xukun Yan; Hui Jiang; Binfei Zhou; Qi Li; Xue Gao; Huijun Yuan; Shiming Yang; Dongyi Han; Zixu Mao; Ping Chen; Xi Lin; Pu Dai
Journal:  Cell Res       Date:  2014-06-10       Impact factor: 25.617

  5 in total

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