نتایج جستجو برای: cockayne syndrome

تعداد نتایج: 621994  

Journal: :Mechanisms of Ageing and Development 2013

Journal: :The Journal of neuroscience : the official journal of the Society for Neuroscience 2016
Robert N Rainey Sum-Yan Ng Juan Llamas Gijsbertus T J van der Horst Neil Segil

UNLABELLED Cisplatin is a common and effective chemotherapeutic agent, yet it often causes permanent hearing loss as a result of sensory hair cell death. The causes of sensitivity to DNA-damaging agents in nondividing cell populations, such as cochlear hair and supporting cells, are poorly understood, as are the specific DNA repair pathways that protect these cells. Nucleotide excision repair (...

Journal: :Medicina oral, patologia oral y cirugia bucal 2006
María de la Luz Arenas-Sordo Edgar Hernández-Zamora Luis Alberto Montoya-Pérez Beatriz Catalina Aldape-Barrios

Cockayne s syndrome is a genetic disorder with a recessive autosomal inheritance, described first by Cockayne in 1936. Patients with this syndrome present failure to thrive, short stature, premature aging, neurological alterations, photosensitivity, delayed eruption of the primary teeth, congenitally absent of some permanent teeth, partial macrodontia, atrophy of the alveolar process and caries...

Journal: :AJNR. American journal of neuroradiology 2010
M Koob V Laugel M Durand H Fothergill C Dalloz F Sauvanaud H Dollfus I J Namer J-L Dietemann

CS is an autosomal recessive multisystem disorder, which is mainly characterized by neurologic and sensory impairment, cachectic dwarfism, and photosensitivity. We describe the neuroimaging features (MR imaging, ¹H-MR spectroscopy, and CT) in the various clinical subtypes of CS from a cohort of genetically and biochemically proved cases. Hypomyelination, calcifications, and brain atrophy were t...

2013
Iltaeg Cho Pei-Fang Tsai Robert J. Lake Asjad Basheer Hua-Ying Fan

The Cockayne syndrome complementation group B (CSB) protein is essential for transcription-coupled DNA repair, and mutations in CSB are associated with Cockayne syndrome--a devastating disease with complex clinical features, including the appearance of premature aging, sun sensitivity, and numerous neurological and developmental defects. CSB belongs to the SWI2/SNF2 ATP-dependent chromatin remo...

Journal: :Proceedings of the National Academy of Sciences of the United States of America 2016
Morten Scheibye-Knudsen Anne Tseng Martin Borch Jensen Karsten Scheibye-Alsing Evandro Fei Fang Teruaki Iyama Sanjay Kumar Bharti Krisztina Marosi Lynn Froetscher Henok Kassahun David Mark Eckley Robert W Maul Paul Bastian Supriyo De Soumita Ghosh Hilde Nilsen Ilya G Goldberg Mark P Mattson David M Wilson Robert M Brosh Myriam Gorospe Vilhelm A Bohr

Cockayne syndrome is a neurodegenerative accelerated aging disorder caused by mutations in the CSA or CSB genes. Although the pathogenesis of Cockayne syndrome has remained elusive, recent work implicates mitochondrial dysfunction in the disease progression. Here, we present evidence that loss of CSA or CSB in a neuroblastoma cell line converges on mitochondrial dysfunction caused by defects in...

2014
Shanshan Yu Liyuan Chen Lili Ye Lingna Fei Wei Tang Yujiao Tian Qian Geng Xin Yi Jiansheng Xie

Cockayne syndrome (CS) is a rare autosomal recessive disorder, the primary manifestations of which are poor growth and neurologic abnormality. Mutations of the ERCC6 and ERCC8 genes are the predominant cause of Cockayne syndrome, and the ERCC6 gene mutation is present in approximately 65% of cases. The present report describes a case of Cockayne syndrome in a Chinese family, with the patients c...

Journal: :Brazilian Journal of Medicine and Human Health 2014

2014
Robert J. Lake Erica L. Boetefuer Pei-Fang Tsai Jieun Jeong Inchan Choi Kyoung-Jae Won Hua-Ying Fan

Cockayne syndrome is an inherited premature aging disease associated with numerous developmental and neurological defects, and mutations in the gene encoding the CSB protein account for the majority of Cockayne syndrome cases. Accumulating evidence suggests that CSB functions in transcription regulation, in addition to its roles in DNA repair, and those defects in this transcriptional activity ...

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