نتایج جستجو برای: hypoperistalsis

تعداد نتایج: 66  

Journal: :Fetal diagnosis and therapy 2015
Lea Tuzovic Sha Tang Russell S Miller Luis Rohena Layla Shahmirzadi Kelly Gonzalez Xiang Li Charles A LeDuc Jiancheng Guo Ashley Wilson Ashley Mills Kenneth Glassberg Heidi Rotterdam Antonia R Sepulveda Wenqi Zeng Wendy K Chung Kwame Anyane-Yeboa

OBJECTIVE To identify the molecular basis for prenatally suspected cases of megacystis microcolon intestinal hypoperistalsis syndrome (MMIHS) (MIM 249210) in 3 independent families with clinical and radiographic evidence of MMIHS. METHODS Whole-exome sequencing (WES) and Sanger sequencing of the ACTG2 gene. RESULTS We identified a novel heterozygous de novo missense variant in ACTG2 c.770G>...

Journal: :Diseases of the esophagus : official journal of the International Society for Diseases of the Esophagus 2011
S Roman A Hot N Fabien J-F Cordier P Miossec J Ninet F Mion

Esophageal involvement occurs in about 80% of patients with systemic sclerosis, with a marked diminution of peristaltic pressures in the distal two-thirds of the esophagus. Our aims were to more fully characterize esophageal motility disorders in systemic sclerosis using high-resolution manometry (HRM) and to determine predictive factors of esophageal involvement. Fifty-one patients (46 females...

2013
Yun Chen Ya-Hui Tsai Yuan-An Liu Shih-Hua Lee Sheng-Hong Tseng Shiue-Cheng Tang

Two-dimensional (2D) histopathology is the standard analytical method for intestinal biopsied tissues; however, the role of 3-dimensional (3D) imaging system in the analysis of the intestinal tissues is unclear. The 3D structure of the crypt organoids from the intestinal stem cell culture and intestinal tissues from the donors and recipients after intestinal transplantation was observed using a...

Journal: :Human molecular genetics 2016
Danny Halim Robert M W Hofstra Luca Signorile Rob M Verdijk Christine S van der Werf Yunia Sribudiani Rutger W W Brouwer Wilfred F J van IJcken Niklas Dahl Joke B G M Verheij Clarisse Baumann John Kerner Yolande van Bever Niels Galjart Rene M H Wijnen Dick Tibboel Alan J Burns Françoise Muller Alice S Brooks Maria M Alves

Megacystis Microcolon Intestinal Hypoperistalsis Syndrome (MMIHS) is a rare congenital disorder, in which heterozygous missense variants in the Enteric Smooth Muscle actin γ-2 (ACTG2) gene have been recently identified. To investigate the mechanism by which ACTG2 variants lead to MMIHS, we screened a cohort of eleven MMIHS patients, eight sporadic and three familial cases, and performed immunoh...

Journal: :Proceedings of the National Academy of Sciences of the United States of America 2017
Danny Halim Michael P Wilson Daniel Oliver Erwin Brosens Joke B G M Verheij Yu Han Vivek Nanda Qing Lyu Michael Doukas Hans Stoop Rutger W W Brouwer Wilfred F J van IJcken Orazio J Slivano Alan J Burns Christine K Christie Karen L de Mesy Bentley Alice S Brooks Dick Tibboel Suowen Xu Zheng Gen Jin Tono Djuwantono Wei Yan Maria M Alves Robert M W Hofstra Joseph M Miano

Megacystis microcolon intestinal hypoperistalsis syndrome (MMIHS) is a congenital visceral myopathy characterized by severe dilation of the urinary bladder and defective intestinal motility. The genetic basis of MMIHS has been ascribed to spontaneous and autosomal dominant mutations in actin gamma 2 (ACTG2), a smooth muscle contractile gene. However, evidence suggesting a recessive origin of th...

2014
Michael F. Wangler Claudia Gonzaga-Jauregui Tomasz Gambin Samantha Penney Timothy Moss Atul Chopra Frank J. Probst Fan Xia Yaping Yang Steven Werlin Ieva Eglite Liene Kornejeva Carlos A. Bacino Dustin Baldridge Jeff Neul Efrat Lev Lehman Austin Larson Joke Beuten Donna M. Muzny Shalini Jhangiani Richard A. Gibbs James R. Lupski Arthur Beaudet

Megacystis-microcolon-intestinal hypoperistalsis syndrome (MMIHS) is a rare disorder of enteric smooth muscle function affecting the intestine and bladder. Patients with this severe phenotype are dependent on total parenteral nutrition and urinary catheterization. The cause of this syndrome has remained a mystery since Berdon's initial description in 1976. No genes have been clearly linked to M...

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