Literature DB >> 27476540

Severe neurological manifestations in an Egyptian patient with a novel frameshift mutation in the Glutaryl-CoA dehydrogenase gene.

Ahmed Moseilhy1, Magdy M Hassan2, Heba S A El Abd3, Shaimaa A Mohammad4, Rajaa El Bekay5, Ussama M Abdel-Motal6, Allal Ouhtit7, Osama K Zaki8,9, Hatem Zayed10.   

Abstract

To characterize an Egyptian patient with glutaric acidemia type I (GA I) and to identify the causative mutation(s) that may be responsible for the disease phenotype. MRI was performed on the patient using the 1.5 T magnet, biochemical analysis was carried out using gas chromatography/mass spectrometry on the patient's dried blood spot, and the patient's organic acids were measured in dried blood and a urine sample using MS/MS and GC/MS, respectively. Total RNA was isolated from the patient's peripheral blood, and the synthesized cDNA was bi-directionally sequenced. The patient exhibited clinical features and MRI findings compatible with a diagnosis of GA I. The abnormal elevation of organic acids in the urine supported the presence of glutaryl-CoA dehydrogenase deficiency. Gene sequencing revealed a novel homozygous frameshift mutation, c.644_645insCTCG; p.(Pro217Leufs*14), in exon 8 of the GCDH gene. The present study revealed a novel frameshift mutation responsible for a severe GA I phenotype in an Egyptian patient. This novel mutation will ultimately contribute to a better understanding of the molecular pathology of the disease and shed light on the intricacies of the genotype-phenotype correlation of GA I disease.

Entities:  

Keywords:  GC/MS; Glutaric acidemia type I; Glutaryl-CoA dehydrogenase; MRI; MS/MS

Mesh:

Substances:

Year:  2016        PMID: 27476540     DOI: 10.1007/s11011-016-9879-x

Source DB:  PubMed          Journal:  Metab Brain Dis        ISSN: 0885-7490            Impact factor:   3.584


  22 in total

1.  Use of guidelines improves the neurological outcome in glutaric aciduria type I.

Authors:  Jana Heringer; S P Nikolas Boy; Regina Ensenauer; Birgit Assmann; Johannes Zschocke; Inga Harting; Thomas Lücke; Esther M Maier; Chris Mühlhausen; Gisela Haege; Georg F Hoffmann; Peter Burgard; Stefan Kölker
Journal:  Ann Neurol       Date:  2010-11       Impact factor: 10.422

2.  Guideline for the diagnosis and management of glutaryl-CoA dehydrogenase deficiency (glutaric aciduria type I).

Authors:  S Kölker; E Christensen; J V Leonard; C R Greenberg; A B Burlina; A P Burlina; M Dixon; M Duran; S I Goodman; D M Koeller; E Müller; E R Naughten; E Neumaier-Probst; J G Okun; M Kyllerman; R A Surtees; B Wilcken; G F Hoffmann; P Burgard
Journal:  J Inherit Metab Dis       Date:  2007-01-03       Impact factor: 4.982

3.  Outcome of the first 3-years of a DNA-based neonatal screening program for glutaric acidemia type 1 in Manitoba and northwestern Ontario, Canada.

Authors:  C R Greenberg; A N Prasad; L A Dilling; J R G Thompson; J C Haworth; B Martin; P Wood-Steiman; L E Seargeant; B Seifert; F A Booth; C Prasad
Journal:  Mol Genet Metab       Date:  2002-01       Impact factor: 4.797

4.  Natural history, outcome, and treatment efficacy in children and adults with glutaryl-CoA dehydrogenase deficiency.

Authors:  Stefan Kölker; Sven F Garbade; Cheryl R Greenberg; James V Leonard; Jean-Marie Saudubray; Antonia Ribes; H Serap Kalkanoglu; Allan M Lund; Begoña Merinero; Moacir Wajner; Mónica Troncoso; Monique Williams; John H Walter; Jaume Campistol; Milagros Martí-Herrero; Melissa Caswill; Alberto B Burlina; Florian Lagler; Esther M Maier; Bernd Schwahn; Aysegul Tokatli; Ali Dursun; Turgay Coskun; Ronald A Chalmers; David M Koeller; Johannes Zschocke; Ernst Christensen; Peter Burgard; Georg F Hoffmann
Journal:  Pediatr Res       Date:  2006-04-26       Impact factor: 3.756

5.  Correlation of genotype and phenotype in glutaryl-CoA dehydrogenase deficiency.

Authors:  E Christensen; A Ribes; B Merinero; J Zschocke
Journal:  J Inherit Metab Dis       Date:  2004       Impact factor: 4.982

6.  Glutaric Acidemia Type 1-Clinico-Molecular Profile and Novel Mutations in GCDH Gene in Indian Patients.

Authors:  Neerja Gupta; Pawan Kumar Singh; Manoj Kumar; Shivaram Shastri; Sheffali Gulati; Atin Kumar; Anuja Agarwala; Seema Kapoor; Mohandas Nair; Savita Sapra; Sudhisha Dubey; Ankur Singh; Punit Kaur; Madhulika Kabra
Journal:  JIMD Rep       Date:  2015-03-12

7.  Type I glutaric aciduria, part 1: natural history of 77 patients.

Authors:  Kevin A Strauss; Erik G Puffenberger; Donna L Robinson; D Holmes Morton
Journal:  Am J Med Genet C Semin Med Genet       Date:  2003-08-15       Impact factor: 3.908

8.  Glutaric aciduria type I: outcome following detection by newborn screening.

Authors:  S Bijarnia; V Wiley; K Carpenter; J Christodoulou; C J Ellaway; B Wilcken
Journal:  J Inherit Metab Dis       Date:  2008-08-09       Impact factor: 4.982

9.  Newborn screening for glutaric aciduria type I in Victoria: treatment and outcome.

Authors:  Avihu Boneh; Miriam Beauchamp; Maureen Humphrey; Jemma Watkins; Heidi Peters; Joy Yaplito-Lee
Journal:  Mol Genet Metab       Date:  2008-04-14       Impact factor: 4.797

10.  Decline of acute encephalopathic crises in children with glutaryl-CoA dehydrogenase deficiency identified by newborn screening in Germany.

Authors:  Stefan Kölker; Sven F Garbade; Nikolas Boy; Esther M Maier; Thomas Meissner; Chris Mühlhausen; Julia B Hennermann; Thomas Lücke; Johannes Häberle; Jochen Baumkötter; Wolfram Haller; Edith Muller; Johannes Zschocke; Peter Burgard; Georg F Hoffmann
Journal:  Pediatr Res       Date:  2007-09       Impact factor: 3.756

View more
  3 in total

1.  Computational modelling approaches as a potential platform to understand the molecular genetics association between Parkinson's and Gaucher diseases.

Authors:  D Thirumal Kumar; Hend Ghasan Eldous; Zainab Alaa Mahgoub; C George Priya Doss; Hatem Zayed
Journal:  Metab Brain Dis       Date:  2018-07-06       Impact factor: 3.584

2.  Genotype-phenotype correlation in 18 Egyptian patients with glutaric acidemia type I.

Authors:  Ahmed Mosaeilhy; Magdy M Mohamed; George Priya Doss C; Heba S A El Abd; Radwa Gamal; Osama K Zaki; Hatem Zayed
Journal:  Metab Brain Dis       Date:  2017-04-07       Impact factor: 3.584

3.  Clinical, biochemical, neuroradiological and molecular characterization of Egyptian patients with glutaric acidemia type 1.

Authors:  Hatem Zayed; Hamed El Khayat; Hoda Tomoum; Ola Khalifa; Ehab Siddiq; Shaimaa A Mohammad; Radwa Gamal; Zumin Shi; Ahmed Mosailhy; Osama K Zaki
Journal:  Metab Brain Dis       Date:  2019-05-06       Impact factor: 3.584

  3 in total

北京卡尤迪生物科技股份有限公司 © 2022-2023.