نتایج جستجو برای: dfnb59

تعداد نتایج: 25  

2011
Wang Jian Fan Ying-ying Wang Shu-juan Liang Peng-Fei Wang Jin-ling Qiu Jian-hua

BACKGROUND Mutations in OTOF and PJVK genes cause DFNB9 and DFNB59 types of hearing loss, respectively. The patients carrying pathogenic mutations in either of these genes may show the typical phenotype of auditory neuropathy spectrum disorder (ANSD). The aim of the present study was to identify OTOF and PJVK mutations in sporadic ANSD patients. METHODS AND FINDINGS A total of 76 unrelated Ch...

Journal: :The Journal of neuroscience : the official journal of the Society for Neuroscience 2007
Martin Schwander Anna Sczaniecka Nicolas Grillet Janice S Bailey Matthew Avenarius Hossein Najmabadi Brian M Steffy Glenn C Federe Erica A Lagler Raheleh Banan Rudy Hice Laura Grabowski-Boase Elisabeth M Keithley Allen F Ryan Gary D Housley Tim Wiltshire Richard J H Smith Lisa M Tarantino Ulrich Müller

Deafness is the most common form of sensory impairment in the human population and is frequently caused by recessive mutations. To obtain animal models for recessive forms of deafness and to identify genes that control the development and function of the auditory sense organs, we performed a forward genetics screen in mice. We identified 13 mouse lines with defects in auditory function and six ...

Journal: :Cell 2015
Sedigheh Delmaghani Jean Defourny Asadollah Aghaie Maryline Beurg Didier Dulon Nicolas Thelen Isabelle Perfettini Tibor Zelles Mate Aller Anaïs Meyer Alice Emptoz Fabrice Giraudet Michel Leibovici Sylvie Dartevelle Guillaume Soubigou Marc Thiry E. Sylvester Vizi Saaid Safieddine Jean-Pierre Hardelin Paul Avan Christine Petit

A deficiency in pejvakin, a protein of unknown function, causes a strikingly heterogeneous form of human deafness. Pejvakin-deficient (Pjvk(-/-)) mice also exhibit variable auditory phenotypes. Correlation between their hearing thresholds and the number of pups per cage suggest a possible harmful effect of pup vocalizations. Direct sound or electrical stimulation show that the cochlear sensory ...

پایان نامه :وزارت علوم، تحقیقات و فناوری - دانشگاه تربیت مدرس - دانشکده علوم پزشکی 1389

ناشنوایی شایع ترین نقص حسی در انسان است که در نیمی از موارد به علل ژنتیکی مربوط می شود. 75-80% موارد وراثتی به صورت اتوزومی مغلوب هستند. مشکل اصلی تشخیص در اختلالاتی مانند ناشنوایی، علل ناهمگن آنهاست؛ شایع ترین ژن های دخیل در ناشنوایی عبارتند از dfnb1 (gjb2&6)، dfnb3 (myo15a)، dfnb4 (slc26a4)، dfnb7/11 (tmc1)، dfnb8/10 (tmprss3)، dfnb9 (otof)، dfnb12 (cdh23)، dfnb59 (pjvk)، dfnb67 (tmhs). بنابر...

2016
Christine Petit Alice Emptoz Sedigheh Delmaghani Omar Akil Paul Avan Lawrence Lustig Saaid Safieddine M’hamed Grati Mariem Ben Said Bing Zou Imen Chak Qi Ma Qi Yao Bouthaina Hammami Denise Yan Rahul Mittal Abdelmonem Ghorbel Lingling Neng Mustafa Tekin Xiao Rui Shi Saber Masmoudi Zhongmin Lu Mounira Hmani Xuezhong Liu

Since the initial report on hearing restoration by cochlear gene transfer in a mouse mutant defective for vesicular gluta-mate transporter-3 (VGLUT3-/-), a growing number of studies tackle similar objectives in the perspective of developing inner ear gene therapy in humans. This presentation will focus on our main approaches to inner ear gene therapy, including the prevention of noise-induced h...

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