نتایج جستجو برای: microcolon

تعداد نتایج: 82  

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: :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: :Archives of disease in childhood 2003
A Mehta K Hussain

Transient hyperinsulinism (HI) has been reported in infants born to mothers with diabetes mellitus, in infants subjected to perinatal asphyxia, or intrauterine growth restriction, in those born with Beckwith-Weidemann syndrome (BWS), and in those with rhesus haemolytic disease. The aetiology and mechanisms that underlie transient HI in some of these conditions remain poorly understood. Little i...

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...

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