نتایج جستجو برای: ciliopathy

تعداد نتایج: 423  

Journal: :Human molecular genetics 2012
Silvio Alessandro Di Gioia Stef J F Letteboer Corinne Kostic Dikla Bandah-Rozenfeld Lisette Hetterschijt Dror Sharon Yvan Arsenijevic Ronald Roepman Carlo Rivolta

Retinitis pigmentosa (RP) is a retinal degenerative disease characterized by the progressive loss of photoreceptors. We have previously demonstrated that RP can be caused by recessive mutations in the human FAM161A gene, encoding a protein with unknown function that contains a conserved region shared only with a distant paralog, FAM161B. In this study, we show that FAM161A localizes at the base...

Journal: :Human molecular genetics 2011
Katharina Hopp Christina M Heyer Cynthia J Hommerding Susan A Henke Jamie L Sundsbak Shail Patel Priyanka Patel Mark B Consugar Peter G Czarnecki Troy J Gliem Vicente E Torres Sandro Rossetti Peter C Harris

Meckel syndrome (MKS) is an embryonic lethal, autosomal recessive disorder characterized by polycystic kidney disease, central nervous system defects, polydactyly and liver fibrosis. This disorder is thought to be associated with defects in primary cilia; therefore, it is classed as a ciliopathy. To date, six genes have been commonly associated with MKS (MKS1, TMEM67, TMEM216, CEP290, CC2D2A an...

2014
David Asante Nicola L. Stevenson David J. Stephens

Cytoplasmic dynein-2 is the motor for retrograde intraflagellar transport (IFT), and mutations in dynein-2 are known to cause skeletal ciliopathies. Here, we define for the first time the composition of the human cytoplasmic dynein-2 complex. We show that the proteins encoded by the ciliopathy genes WDR34 and WDR60 are bona fide dynein-2 intermediate chains and are both required for dynein-2 fu...

Journal: :Journal of the American Society of Nephrology : JASN 2014
Rannar Airik Gisela G Slaats Zhi Guo Anna-Carina Weiss Naheed Khan Amiya Ghosh Toby W Hurd Simon Bekker-Jensen Jacob M Schrøder Steve J Elledge Jens S Andersen Andreas Kispert Maddalena Castelli Alessandra Boletta Rachel H Giles Friedhelm Hildebrandt

Nephronophthisis-related ciliopathies (NPHP-RCs) are developmental and degenerative kidney diseases that are frequently associated with extrarenal pathologies such as retinal degeneration, obesity, and intellectual disability. We recently identified mutations in a gene encoding the centrosomal protein SDCCAG8 as causing NPHP type 10 in humans. To study the role of Sdccag8 in disease pathogenesi...

2016
Jigna V. Raja M. L. Asha G. Arun Kumar Anupama V. Sattigeri Diksha Malhotra

For all intents and purposes, craniofacial development is initiated as soon as the anteroposterior axis of an embryo is established. Although the neural crest receives a significant amount of attention, craniofacial tissue has more patterning information than other tissues of the body. New studies have further clarifi ed the contribution of ciliary epithelia as a source of patterning informatio...

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