نتایج جستجو برای: nonsyndromic deafness

تعداد نتایج: 9132  

2016
Takehiko Ueyama Yuzuru Ninoyu Shin-ya Nishio Takushi Miyoshi Hiroko Torii Koji Nishimura Kazuma Sugahara Hideaki Sakata Dean Thumkeo Hirofumi Sakaguchi Naoki Watanabe Shin-ichi Usami Naoaki Saito Shin-ichiro Kitajiri

DIAPH1 encodes human DIA1, a formin protein that elongates unbranched actin. The c.3634+1G>T DIAPH1 mutation causes autosomal dominant nonsyndromic sensorineural hearing loss, DFNA1, characterized by progressive deafness starting in childhood. The mutation occurs near the C-terminus of the diaphanous autoregulatory domain (DAD) of DIA1, which interacts with its N-terminal diaphanous inhibitory ...

2016
Takehiko Ueyama Yuzuru Ninoyu Shin-Ya Nishio Takushi Miyoshi Hiroko Torii Koji Nishimura Kazuma Sugahara Hideaki Sakata Dean Thumkeo Hirofumi Sakaguchi Naoki Watanabe Shin-Ichi Usami Naoaki Saito Shin-Ichiro Kitajiri

DIAPH1 encodes human DIA1, a formin protein that elongates unbranched actin. The c.3634+1G>T DIAPH1 mutation causes autosomal dominant nonsyndromic sensorineural hearing loss, DFNA1, characterized by progressive deafness starting in childhood. The mutation occurs near the C-terminus of the diaphanous autoregulatory domain (DAD) of DIA1, which interacts with its N-terminal diaphanous inhibitory ...

2012
Yongyi Yuan Xun Zhang Shasha Huang Lujie Zuo Guozheng Zhang Yueshuai Song Guojian Wang Hongtian Wang Deliang Huang Dongyi Han Pu Dai

BACKGROUND Thirty thousand infants are born every year with congenital hearing impairment in mainland China. Racial and regional factors are important in clinical diagnosis of genetic deafness. However, molecular etiology of hearing impairment in the Tibetan Chinese population living in the Tibetan Plateau has not been investigated. To provide appropriate genetic testing and counseling to Tibet...

2013
Cynthia J. Schoen Margit Burmeister Marci M. Lesperance

We previously demonstrated that a mutation in the 5' untranslated region of Diaphanous homolog 3 (DIAPH3) results in 2 to 3-fold overexpression of the gene, leading to a form of delayed onset, progressive human deafness known as AUNA1 (auditory neuropathy, nonsyndromic, autosomal dominant, 1). To investigate the mechanism of deafness, we generated two lines of transgenic mice overexpressing Dia...

Journal: :American journal of human genetics 2008
Jérôme Ruel Sarah Emery Régis Nouvian Tiphaine Bersot Bénédicte Amilhon Jana M Van Rybroek Guy Rebillard Marc Lenoir Michel Eybalin Benjamin Delprat Theru A Sivakumaran Bruno Giros Salah El Mestikawy Tobias Moser Richard J H Smith Marci M Lesperance Jean-Luc Puel

Autosomal-dominant sensorineural hearing loss is genetically heterogeneous, with a phenotype closely resembling presbycusis, the most common sensory defect associated with aging in humans. We have identified SLC17A8, which encodes the vesicular glutamate transporter-3 (VGLUT3), as the gene responsible for DFNA25, an autosomal-dominant form of progressive, high-frequency nonsyndromic deafness. I...

2015
So Young Kim Ah Reum Kim Kyu Hee Han Min Young Kim Eun-Hee Jeon Ja-Won Koo Seung Ha Oh Byung Yoon Choi Berta Alsina

INTRODUCTION The contribution of Gap junction beta-2 protein (GJB2) to the genetic load of deafness and its mutation spectra vary among different ethnic groups. OBJECTIVE In this study, the mutation spectrum and audiologic features of patients with GJB2 mutations were evaluated with a specific focus on residual hearing. METHODS An initial cohort of 588 subjects from 304 families with varyin...

Journal: :The Laryngoscope 2008
Simon I Angeli

OBJECTIVES/HYPOTHESIS The aim of this study was to 1) determine the prevalence of DFNB1 in a cohort of children with prelingual nonsyndromic sensorineural hearing loss (HL), 2) study phenotype/genotype correlations, and 3) establish guidelines for genetic counseling of DFNB1. STUDY DESIGN Prospective cohort study. METHODS A total of 119 unrelated children (107 sporadic and 12 familial cases...

2012
Oscar Diaz-Horta Duygu Duman Joseph Foster Aslı Sırmacı Michael Gonzalez Nejat Mahdieh Nikou Fotouhi Mortaza Bonyadi Filiz Başak Cengiz Ibis Menendez Rick H. Ulloa Yvonne J. K. Edwards Stephan Züchner Susan Blanton Mustafa Tekin

Identification of the pathogenic mutations underlying autosomal recessive nonsyndromic hearing loss (ARNSHL) is difficult, since causative mutations in 39 different genes have so far been reported. After excluding mutations in the most common ARNSHL gene, GJB2, via Sanger sequencing, we performed whole-exome sequencing (WES) in 30 individuals from 20 unrelated multiplex consanguineous families ...

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