| Literature DB >> 27476540 |
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