Literature DB >> 28360385

Molecular analysis of glycogen storage disease type Ia in Iranian Azeri Turks: identification of a novel mutation.

Shekari Khaniani Mahmoud1, Aziz Khorrami, Mandana Rafeey, Robabeh Ghergherehchi, Mansoori Derakhshan Sima.   

Abstract

Glycogen storage diseases (GSDs) are caused by abnormalities in enzymes that are involved in the regulation of gluconeogenesis and glycogenolysis. GSD I, an autosomal recessive metabolic disorder, is the most common GSD and has four subtypes. Here, we examined GSD Ia caused by the defective glucose-6-phosphatase catalytic (G6PC) gene. We investigated the frequency of GSD Ia and clarified its molecular aspect in patients with the main clinical and biochemical characteristics of GSD, including 37 unrelated patients with a mean age of three years at the time of diagnosis. All patients belonged to the Azeri Turkish population. Hypoglycaemia and hypertriglyceridaemia were the most frequent laboratory findings. Mutations were detected by performing direct sequencing. Mutation analysis of the G6PC gene revealed that GSD Ia accounted for 11% in GSD patients with involvement of liver. Three patients were homozygous for R83C mutation. In addition, a novel stop mutation, Y85X, was identified in a patient with the typical features of GSD Ia.

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Year:  2017        PMID: 28360385     DOI: 10.1007/s12041-016-0734-y

Source DB:  PubMed          Journal:  J Genet        ISSN: 0022-1333            Impact factor:   1.166


  21 in total

Review 1.  Molecular genetics of type 1 glycogen storage disease.

Authors:  A R Janecke; E Mayatepek; G Utermann
Journal:  Mol Genet Metab       Date:  2001-06       Impact factor: 4.797

2.  Glycogen storage disease: report of 17 cases from southern India.

Authors:  Abraham Koshy; Kannan Ramaswamy; Marjorie Correa; Swarna Rekha
Journal:  Indian J Gastroenterol       Date:  2006 Jul-Aug

Review 3.  Mutations in the glucose-6-phosphatase-alpha (G6PC) gene that cause type Ia glycogen storage disease.

Authors:  Janice Y Chou; Brian C Mansfield
Journal:  Hum Mutat       Date:  2008-07       Impact factor: 4.878

4.  Identification of mutations in the glucose-6-phosphatase gene in Czech and Slovak patients with glycogen storage disease type ia, including novel mutations K76N, V166A and 540del5.

Authors:  L Kozák; H Francová; E Hrabincová; S Stastná; K Pesková; M Elleder
Journal:  Hum Mutat       Date:  2000-07       Impact factor: 4.878

5.  Glycogen storage disease type I: diagnosis and phenotype/genotype correlation.

Authors:  Dietrich Matern; Hans Hermann Seydewitz; Deeksha Bali; Christine Lang; Yuan-Tsong Chen
Journal:  Eur J Pediatr       Date:  2002-07-27       Impact factor: 3.183

6.  Transmembrane topology of glucose-6-phosphatase.

Authors:  C J Pan; K J Lei; B Annabi; W Hemrika; J Y Chou
Journal:  J Biol Chem       Date:  1998-03-13       Impact factor: 5.157

Review 7.  Glycogen storage diseases: new perspectives.

Authors:  Hasan Ozen
Journal:  World J Gastroenterol       Date:  2007-05-14       Impact factor: 5.742

8.  Glycogen storage disease type III diagnosis and management guidelines.

Authors:  Priya S Kishnani; Stephanie L Austin; Pamela Arn; Deeksha S Bali; Anne Boney; Laura E Case; Wendy K Chung; Dev M Desai; Areeg El-Gharbawy; Ronald Haller; G Peter A Smit; Alastair D Smith; Lisa D Hobson-Webb; Stephanie Burns Wechsler; David A Weinstein; Michael S Watson
Journal:  Genet Med       Date:  2010-07       Impact factor: 8.822

9.  Liver storage disease in Iran: a ten year study of liver biopsies in Children Medical Center Hospital in Tehran-Iran.

Authors:  Farzaneh Motamed; Maryam Monajemzadeh; Soroush Seifirad; Mandana Ashrafi; Abbas Rasti; Fatemeh Mahjoub
Journal:  Hepat Mon       Date:  2011-08       Impact factor: 0.660

10.  Three novel mutations of the G6PC gene identified in Chinese patients with glycogen storage disease type Ia.

Authors:  Bi-Xia Zheng; Qian Lin; Mei Li; Yu Jin
Journal:  Eur J Pediatr       Date:  2014-07-01       Impact factor: 3.183

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  1 in total

1.  Genotype-phenotype correlation and description of two novel mutations in Iranian patients with glycogen storage disease 1b (GSD1b).

Authors:  Maryam Eghbali; Maryam Abiri; Saeed Talebi; Zahra Noroozi; Marjan Shakiba; Parastoo Rostami; Hosein Alimadadi; Mehri Najafi; Fatemeh Yazarlou; Ali Rabbani; Mohammad Hossein Modarressi
Journal:  Orphanet J Rare Dis       Date:  2020-01-31       Impact factor: 4.123

  1 in total

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