نتایج جستجو برای: arpkd

تعداد نتایج: 183  

Journal: :Current Biology 2001
Hongmin Qin Joel L. Rosenbaum Maureen M. Barr

In this report, we show that the Caenorhabditis elegans gene osm-5 is homologous to the Chlamydomonas gene IFT88 and the mouse autosomal recessive polycystic kidney disease (ARPKD) gene, Tg737. The function of this ARPKD gene may be evolutionarily conserved: mutations result in defective ciliogenesis in worms [1], algae [2], and mice [2, 3]. Intraflagellar transport (IFT) is essential for the d...

Journal: :Proceedings of the National Academy of Sciences of the United States of America 2004
Ming-Zhi Zhang Weiyi Mai Cunxi Li Sae-youll Cho Chuanming Hao Gilbert Moeckel Runxiang Zhao Ingyu Kim Jikui Wang Huaqi Xiong Hong Wang Yasunori Sato Yizhong Wu Yasuni Nakanuma Marusia Lilova York Pei Raymond C Harris Song Li Robert J Coffey Le Sun Dianqing Wu Xing-Zhen Chen Matthew D Breyer Zhizhuang Joe Zhao James A McKanna Guanqing Wu

Mutations of the polycystic kidney and hepatic disease 1 (PKHD1) gene have been shown to cause autosomal recessive polycystic kidney disease (ARPKD), but the cellular functions of the gene product (PKHD1) remain uncharacterized. To illuminate its properties, the spatial and temporal expression patterns of PKHD1 were determined in mouse, rat, and human tissues by using polyclonal Abs and mAbs re...

Journal: :Kidney & blood pressure research 2007
Jacqueline K Phillips Deborah Hopwood Rhonda A Loxley Kamaljit Ghatora Jason D Coombes Ying Sin Tan Joanne L Harrison Douglas J McKitrick Vasyl Holobotvskyy Leonard F Arnolda Gopala K Rangan

BACKGROUND/METHODS We have examined the hypothesis that cyst formation is key in the pathogenesis of cardiovascular disease in a Lewis polycystic kidney (LPK) model of autosomal-recessive polycystic kidney disease (ARPKD), by determining the relationship between cyst development and indices of renal function and cardiovascular disease. RESULTS In the LPK (n = 35), cysts appear at week 3 (1.1 ...

Journal: :American journal of physiology. Renal physiology 2002
Erik M Schwiebert Darren P Wallace Gavin M Braunstein Sandi R King Janos Peti-Peterdi Kazushige Hanaoka William B Guggino Lisa M Guay-Woodford P Darwin Bell Lawrence P Sullivan Jared J Grantham Amanda L Taylor

ATP and its metabolites are potent autocrine agonists that act extracellularly within tissues to affect epithelial function. In polycystic kidneys, renal tubules become dilated and/or encapsulated as cysts, creating abnormal microenvironments for autocrine signaling. Previously, our laboratory has shown that high-nanomolar to micromolar quantities of ATP are released from cell monolayers in vit...

Journal: :Journal of the American Society of Nephrology : JASN 2007
Sandro Rossetti Peter C Harris

The phenotypes that are associated with the common forms of polycystic kidney disease (PKD)--autosomal dominant (ADPKD) and autosomal recessive (ARPKD)--are highly variable in penetrance. This is in terms of severity of renal disease, which can range from neonatal death to adequate function into old age, characteristics of the liver disease, and other extrarenal manifestations in ADPKD. Influen...

2014
Shixuan Wang Maoqing Wu Gang Yao Jingjing Zhang Jing Zhou

FPC (fibrocystin or polyductin) is a single transmembrane receptor-like protein, responsible for the human autosomal recessive polycystic kidney disease (ARPKD). It was recently proposed that FPC undergoes a Notch-like cleavage and subsequently the cleaved carboxy(C)-terminal fragment translocates to the nucleus. To study the functions of the isolated C-tail, we expressed the intracellular doma...

2015
Jean-Bernard Beaudry Sabine Cordi Céline Demarez Sébastien Lepreux Christophe E. Pierreux Frédéric P. Lemaigre Wan-Xi Yang

Biliary cysts in adult patients affected by polycystic liver disease are lined by cholangiocytes that proliferate, suggesting that initiation of cyst formation depends on proliferation. Here, we challenge this view by analyzing cyst-lining cell proliferation and differentiation in Cpk mouse embryos and in livers from human fetuses affected by Autosomal Recessive Polycystic Kidney Disease (ARPKD...

2013
Maoqing Wu Chaozhe Yang Binli Tao Su Bu Lisa M. Guay-Woodford

Cystin is a novel cilia-associated protein that is disrupted in the cpk mouse, a well-characterized mouse model of autosomal recessive polycystic kidney disease (ARPKD). Interestingly, overexpression of the Myc gene is evident in animal models of ARPKD and is thought to contribute to the renal cystic phenotype. Using a yeast two-hybrid approach, the growth suppressor protein necdin, known to mo...

2009
Sadaf Ahmad Ray Choi Quincy Roberts Jennillee Wallace

Polycystic kidney disease is the most common genetic, life-threatening disease, affecting more than 12.5 million people worldwide. Fluid-filled renal cysts that eventually destroy renal tissue and renal function altogether are characteristic of polycystic kidney disease. The autosomal dominant form of the disease which is also the most common form, ADPKD, is linked to mutations in the genes PKD...

نمودار تعداد نتایج جستجو در هر سال

با کلیک روی نمودار نتایج را به سال انتشار فیلتر کنید