نتایج جستجو برای: ciliopathy

تعداد نتایج: 423  

2015
Haribaskar Ramachandran Konstantin Herfurth Rudolf Grosschedl Tobias Schäfer Gerd Walz Michael Massiah

Glis2/NPHP7 is a transcriptional regulator mutated in type 7 nephronophthisis, an autosomal recessive ciliopathy associated with cystic and fibrotic kidney disease as well as characteristic extrarenal manifestations. While most ciliopathy-associated molecules are found in the cilium, Glis2/NPHP7 presumably localizes to the nucleus. However, the detection of endogenous Glis2/NPHP7 has remained u...

Journal: :Human molecular genetics 2012
Tom R Webb David A Parfitt Jessica C Gardner Ariadna Martinez Dalila Bevilacqua Alice E Davidson Ilaria Zito Dawn L Thiselton Jacob H C Ressa Marina Apergi Nele Schwarz Naheed Kanuga Michel Michaelides Michael E Cheetham Michael B Gorin Alison J Hardcastle

X-linked retinitis pigmentosa (XLRP) is genetically heterogeneous with two causative genes identified, RPGR and RP2. We previously mapped a locus for a severe form of XLRP, RP23, to a 10.71 Mb interval on Xp22.31-22.13 containing 62 genes. Candidate gene screening failed to identify a causative mutation, so we adopted targeted genomic next-generation sequencing of the disease interval to determ...

2016
Hee Gyung Kang Hyun Kyung Lee Yo Han Ahn Je-Gun Joung Jaeyong Nam Nayoung K D Kim Jung Min Ko Min Hyun Cho Jae Il Shin Joon Kim Hye Won Park Young Seo Park Il-Soo Ha Woo Yeong Chung Dae-Yeol Lee Su Young Kim Woong Yang Park Hae Il Cheong

Nephronophthisis-related ciliopathy (NPHP-RC) is a common genetic cause of end-stage renal failure during childhood and adolescence and exhibits an autosomal recessive pattern of inheritance. Genetic diagnosis is quite limited owing to genetic heterogeneity in NPHP-RC. We designed a novel approach involving the step-wise screening of Sanger sequencing and targeted exome sequencing for the genet...

2017
Sheila Castro-Sánchez María Álvarez-Satta Mohamed A Tohamy Sergi Beltran Sophia Derdak Diana Valverde

Ciliopathies are a group of rare disorders characterized by a high genetic and phenotypic variability, which complicates their molecular diagnosis. Hence the need to use the latest powerful approaches to faster identify the genetic defect in these patients. We applied whole exome sequencing to six consanguineous families clinically diagnosed with ciliopathy-like disease, and for which mutations...

Journal: :Human molecular genetics 2013
Amanda C Leightner Cynthia J Hommerding Ying Peng Jeffrey L Salisbury Vladimir G Gainullin Peter G Czarnecki Caroline R Sussman Peter C Harris

Meckel syndrome (MKS) is a lethal disorder associated with renal cystic disease, encephalocele, ductal plate malformation and polydactyly. MKS is genetically heterogeneous and part of a growing list of syndromes called ciliopathies, disorders resulting from defective cilia. TMEM67 mutation (MKS3) is a major cause of MKS and the related ciliopathy Joubert syndrome, although the complete etiology...

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