نتایج جستجو برای: mmachc gene

تعداد نتایج: 1141380  

2013
Martin Kömhoff Marcus T. Roofthooft Dineke Westra Thea K. Teertstra Attilio Losito Nicole C. A. J. van de Kar Rolf M. F. Berger

Pulmonary arterial hypertension (PAH) and renal thrombotic microangiopathy (rTMA) are rare diseases in childhood, frequently leading to death and end-stage renal disease, respectively. Their combined occurrence has been reported anecdotally. We investigated the clinical, biochemical, and genetic aspects of 5 children with the rare combination of PAH and rTMA. Onset of disease ranged from 1.5 to...

Journal: :Molecular genetics and metabolism 2008
Célia Nogueira Chiara Aiello Roberto Cerone Esmeralda Martins Ubaldo Caruso Isabella Moroni Cristiano Rizzo Luísa Diogo Elisa Leão Fernando Kok Federica Deodato Maria Cristina Schiaffino Sara Boenzi Olivier Danhaive Clara Barbot Sílvia Sequeira Mattia Locatelli Filippo M Santorelli Graziella Uziel Laura Vilarinho Carlo Dionisi-Vici

Methylmalonic aciduria (MMA) and homocystinuria, cblC type (MIM 277400) is the most frequent inborn error of vitamin B(12). The recent identification of the disease gene, MMACHC, has permitted preliminary genotype-phenotype correlations. We studied 24 Italian and 17 Portuguese patients with cblC defect to illustrate the spectrum of mutations in a southern European population and discuss the imp...

2017
Minguang Chen Jieqiu Zhuang JianHuan Yang Dexuan Wang Qing Yang

RATIONALE Methylmalonic acidemia (MMA) is a common organic acidemia, mainly due to methylmalonyl-CoA mutase (MCM) or its coenzyme cobalamin (VitB12) metabolic disorders. Cobalamin C (CblC) type is the most frequent inborn error of cobalamin metabolism; it can develop symptoms in childhood and often combine multisystem damage, which leads to methylmalonic acid, propionic acid, methyl citrate, an...

Journal: :Human molecular genetics 2015
Lachlan A Jolly Lam Son Nguyen Deepti Domingo Ying Sun Simon Barry Miroslava Hancarova Pavlina Plevova Marketa Vlckova Marketa Havlovicova Vera M Kalscheuer Claudio Graziano Tommaso Pippucci Elena Bonora Zdenek Sedlacek Jozef Gecz

Both gain- and loss-of-function mutations have recently implicated HCFC1 in neurodevelopmental disorders. Here, we extend our previous HCFC1 over-expression studies by employing short hairpin RNA to reduce the expression of Hcfc1 in embryonic neural cells. We show that in contrast to over-expression, loss of Hcfc1 favoured proliferation of neural progenitor cells at the expense of differentiati...

2013
Niels Grarup Patrick Sulem Camilla H. Sandholt Gudmar Thorleifsson Tarunveer S. Ahluwalia Valgerdur Steinthorsdottir Helgi Bjarnason Daniel F. Gudbjartsson Olafur T. Magnusson Thomas Sparsø Anders Albrechtsen Augustine Kong Gisli Masson Geng Tian Hongzhi Cao Chao Nie Karsten Kristiansen Lise Lotte Husemoen Betina Thuesen Yingrui Li Rasmus Nielsen Allan Linneberg Isleifur Olafsson Gudmundur I. Eyjolfsson Torben Jørgensen Jun Wang Torben Hansen Unnur Thorsteinsdottir Kari Stefánsson Oluf Pedersen

Genome-wide association studies have mainly relied on common HapMap sequence variations. Recently, sequencing approaches have allowed analysis of low frequency and rare variants in conjunction with common variants, thereby improving the search for functional variants and thus the understanding of the underlying biology of human traits and diseases. Here, we used a large Icelandic whole genome s...

2013
Josef Finsterer Sinda Zarrouk-Mahjoub

Letter to the Editor With interest we read the article by Esposito et al. about a female child with myopathy and left ventricular hypertrabeculation / noncompaction (LVHT) carrying a mutation in two different genes, the integrin-α7 gene and the myosin heavy chain 7B gene (MYH7B) [1]. We have the following comments and concerns. The authors mention in the discussion that MYL2 and MYL3 mutations ...

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