نتایج جستجو برای: syndromic deafness

تعداد نتایج: 13200  

Journal: :Biomedical Research and Therapy 2023

Introduction: Non-syndromic hearing loss (NSHL) in children, which has numerous causes, can impede or even postpone the acquisition of spoken language. In Viet Nam, screening programs and genetic testing for NSHL are rarely applied. this study, 31 pediatric patients had their medical histories collected alongside sequencing results GJB2 TECTA genes to determine prevalence these mutations commun...

2016
Somayeh Reiisi Mohammad Amin Tabatabaiefar Mohammad Hosein Sanati Morteza Hashemzadeh Chaleshtori

OBJECTIVES Non-syndromic sensorineural hearing loss (NSHL) is a common disorder affecting approximately 1 in 500 newborns. This type of hearing loss is extremely heterogeneous and includes over 100 loci. Mutations in the GJB2 gene have been implicated in about half of autosomal recessive non-syndromic hearing loss (ARNSHL) cases, making this the most common cause of ARNSHL. For the latter form ...

Journal: :Brain : a journal of neurology 2008
Patrizia Amati-Bonneau Maria Lucia Valentino Pascal Reynier Maria Esther Gallardo Belén Bornstein Anne Boissière Yolanda Campos Henry Rivera Jesús González de la Aleja Rosanna Carroccia Luisa Iommarini Pierre Labauge Dominique Figarella-Branger Pascale Marcorelles Alain Furby Katell Beauvais Franck Letournel Rocco Liguori Chiara La Morgia Pasquale Montagna Maria Liguori Claudia Zanna Michela Rugolo Andrea Cossarizza Bernd Wissinger Christophe Verny Robert Schwarzenbacher Miguel Angel Martín Joaquín Arenas Carmen Ayuso Rafael Garesse Guy Lenaers Dominique Bonneau Valerio Carelli

Mutations in OPA1, a dynamin-related GTPase involved in mitochondrial fusion, cristae organization and control of apoptosis, have been linked to non-syndromic optic neuropathy transmitted as an autosomal-dominant trait (DOA). We here report on eight patients from six independent families showing that mutations in the OPA1 gene can also be responsible for a syndromic form of DOA associated with ...

2015
Fei Liu Jiongjiong Hu Wenjun Xia Lili Hao Jing Ma Duan Ma Zhaoxin Ma

Autosomal dominant non-syndromic hearing loss is highly heterogeneous, and eyes absent 4 (EYA4) is a disease-causing gene. Most EYA4 mutations founded in the Eya-homologous region, however, no deafness causative missense mutation in variable region of EYA4 have previously been found. In this study, we identified a pathogenic missense mutation located in the variable region of the EYA4 gene for ...

2016
Alok K. Sharma Tobias Krieger Alan C. Rigby Israel Zelikovic Seth L. Alper

Mutations in the human SLC26A4/Pendrin polypeptide (hPDS) cause Pendred Syndrome /DFNB4, syndromic deafness with enlargement of the vestibular aqueduct and low-penetrance goiter. Here we present data on cloning, protein overexpression and purification, refolding, and biophysical characterization of the recombinant hPDS STAS domain lacking its intrinsic variable sequence (STAS-ΔIVS). We report a...

2015
Nathalie Falk Marlene Lösl Nadja Schröder Andreas Gießl Gang Dong William Tsang

Cilia and flagella are highly conserved and important microtubule-based organelles that project from the surface of eukaryotic cells and act as antennae to sense extracellular signals. Moreover, cilia have emerged as key players in numerous physiological, developmental, and sensory processes such as hearing, olfaction, and photoreception. Genetic defects in ciliary proteins responsible for cili...

2009
Susanne A Schneider Kailash P Bhatia

BACKGROUND Dystonia is a hyperkinetic movement disorder defined by involuntary sustained muscle spasms and unusual postures. Etiologically, dystonic syndromes can be broadly divided into primary and secondary forms, dystonia-plus syndromes and heredodegenerative forms. In particular, diagnosis of secondary dystonic syndromes can be challenging in view of the variety of causes. PURPOSE The pur...

Journal: :Journal of medical genetics 1999
A Murgia E Orzan R Polli M Martella C Vinanzi E Leonardi E Arslan F Zacchello

Mutations in the gap junction protein connexin 26 (Cx26) gene (GJB2) seem to account for many cases of congenital sensorineural hearing impairment, the reported prevalence being 34-50% in autosomal recessive cases and 10-37% in sporadic cases. The hearing impairment in these patients has been described as severe or profound. We have studied 53 unrelated subjects with congenital non-syndromic se...

Journal: :Human molecular genetics 2003
Xue Zhong Liu Xiao Mei Ouyang Xia Juan Xia Jing Zheng Arti Pandya Fang Li Li Lin Du Katherine O Welch Christine Petit Richard J H Smith Bradley T Webb Denise Yan Kathleen S Arnos David Corey Peter Dallos Walter E Nance Zheng Yi Chen

Prestin, a membrane protein that is highly and almost exclusively expressed in the outer hair cells (OHCs) of the cochlea, is a motor protein which senses membrane potential and drives rapid length changes in OHCs. Surprisingly, prestin is a member of a gene family, solute carrier (SLC) family 26, that encodes anion transporters and related proteins. Of nine known human genes in this family, th...

Journal: :Journal of medical genetics 1997
E Gausden B Coyle J A Armour R Coffey A Grossman G R Fraser R M Winter M E Pembrey P Kendall-Taylor D Stephens L M Luxon P D Phelps W Reardon R Trembath

Pendred syndrome is the association between congenital sensorineural deafness and goitre. The disorder is characterised by the incomplete discharge of radioiodide from a primed thyroid following perchlorate challenge. However, the molecular basis of the association between hearing loss and a defect in organification of iodide remains unclear. Pendred syndrome is inherited as an autosomal recess...

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