Mitochondrial depletion syndrome type 3: the Lebanese variant
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Frontiers Media SA
Abstract
Introduction: Mitochondrial DNA depletion syndrome type 3 is an emerging disorder linked to variants in the deoxyguanosine kinase gene, which encodes for mitochondrial maintenance. This autosomal recessive disorder is frequent in the Middle East and North Africa. Diagnosis is often delayed due to the non-specificity of clinical presentation with cerebro-hepatic deterioration. The only therapeutic option is liver transplantation, although the value of this remains debatable. Methods: We describe the clinical, biochemical, and molecular profiles of Lebanese patients with this rare disorder. We also present a review of all cases from the Middle East and North Africa. Results: All Lebanese patients share a unique mutation, unreported in other populations. Almost half of patients worldwide originate from the Middle East and North Africa, with cases reported from only 7 of the 21 countries in this region. Clinical presentation is heterogeneous, with early-onset neurological and hepatic signs. Liver failure and lactic acidosis are constants. Several variants can be identified in each population; a unique c.235C>T p. (Gln79*) pathogenic variant is found in Lebanese patients. Outcome is poor, with death before 1 year of age. Conclusion: The pathogenic nonsense variant c.235C>T p. (Gln79*) in the deoxyguanosine kinase gene may be considered a founder mutation in Lebanon. Further genotypic delineation of this devastating disorder in populations with high consanguinity rates is needed. Copyright © 2023 Majdalani, Yazbeck, El Harake, Samaha and Karam.
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Keywords
Dguok, Lactic acidosis, Lebanon, Liver transplantation, Mena, Mitochondrial depletion, Mtdna maintenance defects, Neonatal liver failure, 4 hydroxyphenyllactic acid, 4 hydroxyphenylpyruvic acid, Alanine aminotransferase, Alpha fetoprotein, Amino acid, Ammonia, Aspartate aminotransferase, Bilirubin, Bilirubin glucuronide, Deoxyguanosine kinase, Fresh frozen plasma, Galactose 1 phosphate uridylyltransferase, Gamma glutamyltransferase, Glucose, Lactic acid, N acetyltyrosine, Phenylalanine, Succinylacetone, Tyrosine, Unclassified drug, Article, Biochemical analysis, Blood clotting disorder, Case report, Cholestasis, Clinical article, Clinical feature, Cryoprecipitate, Developmental delay, Failure to thrive, Female, Gene mutation, Genetic screening, Genetic variability, Hepatomegaly, High throughput sequencing, Human, Hypertransaminasemia, Hypoglycemia, Infant, International normalized ratio, Intrauterine growth retardation, Jaundice, Lebanese, Liver failure, Male, Microcephaly, Mitochondrial dna depletion syndrome, Mitochondrial dna depletion syndrome type 3, Muscle hypotonia, Newborn, Nonsense mutation, Nuclear magnetic resonance spectroscopy, Nystagmus, Psychomotor disorder, Retrospective study, Tandem mass spectrometry, Tyrosinemia, Vomiting, Whole exome sequencing