Literature DB >> 25932215

Two novel mutations of ornithine transcarbamylase gene identified from three Chinese neonates with ornithine transcarbamylase deficiency.

Jing Liu1, Lei Dong2, Yan Wang3, Mei Zhang1.   

Abstract

We aim to analyze the blood metabolic profiling and the gene mutation of ornithine transcarbamylase (OTC) in three neonates with ornithine transcarbamylase deficiency (OTCD). Three neonates with OTCD were included in this study. The profiling of amino acids and acylcarnitine was determined using MS-MS assay. The OTC exons were amplified using PCR amplification. DNA sequencing was performed, based on which mutation analysis of OTC genes was carried out. For the clinical symptoms, all the three neonates showed poor reaction and feeding. In addition, convulsion and neonatal infection were noticed. A remarkable decrease of citrulline concentration was revealed by MS-MS assay. In case 1, a 548A > G substitution was identified in exon 6, which resulted in replacement of cysteine by tyrosine in codon 183. In case 2, a 1016T > G substitution was identified in exon 10, leading to replacement of valine by glycine in codon 339. In case 3, a 995G > C mutation was noted in exon 9, resulting in missense mutation of tryptophane to serine in codon332. Three types of OTC gene mutations were identified in Chinese neonates with OTC deficiency, among which two novel mutations, including 1016T > G and 995G > C, are presented uniquely in our study.

Entities:  

Keywords:  MS-MS assay; Ornithine transcarbamylase deficiency; mutation; ornithine transcarbamylase

Year:  2015        PMID: 25932215      PMCID: PMC4402862     

Source DB:  PubMed          Journal:  Int J Clin Exp Med        ISSN: 1940-5901


  13 in total

1.  Expression of wild-type and mutant human ornithine transcarbamylase genes in Chinese hamster ovary cells and lack of dominant negative effect of R141Q and R40H mutants.

Authors:  L Augustin; M Mavinakere; H Morizono; M Tuchman
Journal:  Pediatr Res       Date:  2000-12       Impact factor: 3.756

2.  Late-onset ornithine transcarbamylase deficiency in male patients: prognostic factors and characteristics of plasma amino acid profile.

Authors:  Eimei Harada; Atsushi Nishiyori; Yasuyuki Tokunaga; Yoriko Watanabe; Norikazu Kuriya; Ryukichi Kumashiro; Tateo Kuno; Ryuichi Kuromaru; Shinichi Hirose; Kotaro Ichikawa; Makoto Yoshino
Journal:  Pediatr Int       Date:  2006-04       Impact factor: 1.524

3.  Identification of 'private' mutations in patients with ornithine transcarbamylase deficiency.

Authors:  M Tuchman; H Morizono; B S Rajagopal; R J Plante; N M Allewell
Journal:  J Inherit Metab Dis       Date:  1997-08       Impact factor: 4.982

4.  Liquid chromatography-tandem mass spectrometry for analysis of acylcarnitines in dried blood specimens collected at autopsy from neonatal intensive care unit.

Authors:  Wen-jun Tu; Fang Dai; Xin-yu Wang; James Jian Ho
Journal:  Chin Med Sci J       Date:  2010-06

5.  Identification of novel mutations in the human ornithine transcarbamylase (OTC) gene of Korean patients with OTC deficiency and transient expression of the mutant proteins in vitro.

Authors:  Gu-Hwan Kim; Jin-Ho Choi; Hyung-Haon Lee; Sangwook Park; Sung-Su Kim; Han-Wook Yoo
Journal:  Hum Mutat       Date:  2006-11       Impact factor: 4.878

6.  Mutations and polymorphisms in the human ornithine transcarbamylase (OTC) gene.

Authors:  Saori Yamaguchi; Lisa L Brailey; Hiroki Morizono; Allen E Bale; Mendel Tuchman
Journal:  Hum Mutat       Date:  2006-07       Impact factor: 4.878

7.  Four newly identified ornithine transcarbamylase (OTC) mutations (D126G, R129H, I172M and W332X) in Japanese male patients with early-onset OTC deficiency.

Authors:  T Matsuura; R Hoshide; K Kiwaki; S Komaki; E Koike; F Endo; K Oyanagi; Y Suzuki; I Kato; K Ishikawa
Journal:  Hum Mutat       Date:  1994       Impact factor: 4.878

8.  Identification of new mutations in the ornithine transcarbamylase (OTC) gene in Korean families.

Authors:  H W Yoo; G H Kim; D H Lee
Journal:  J Inherit Metab Dis       Date:  1996       Impact factor: 4.982

9.  Ornithine transcarbamylase deficiency: a possible risk factor for thrombosis.

Authors:  Lakshmi Venkateswaran; Fernando Scaglia; Valerie McLin; Paula Hertel; Oleg A Shchelochkov; Saul Karpen; Donald Mahoney; Donald L Yee
Journal:  Pediatr Blood Cancer       Date:  2009-07       Impact factor: 3.167

10.  Human ornithine transcarbamylase locus mapped to band Xp21.1 near the Duchenne muscular dystrophy locus.

Authors:  V Lindgren; B de Martinville; A L Horwich; L E Rosenberg; U Francke
Journal:  Science       Date:  1984-11-09       Impact factor: 47.728

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