Literature DB >> 23968935

Mitochondrial depletion syndromes in children and adults.

Josef Finsterer1, Uwe Ahting.   

Abstract

To highlight differences between early-onset and adult mitochondrial depletion syndromes (MDS) concerning etiology and genetic background, pathogenesis, phenotype, clinical presentation and their outcome. MDSs most frequently occur in neonates, infants, or juveniles and more rarely in adolescents or adults. Mutated genes phenotypically presenting with adult-onset MDS include POLG1, TK2, TyMP, RRM2B, or PEO1/twinkle. Adult MDS manifest similarly to early-onset MDS, as myopathy, encephalo-myopathy, hepato-cerebral syndrome, or with chronic progressive external ophthalmoplegia (CPEO), fatigue, or only minimal muscular manifestations. Diagnostic work-up or treatment is not at variance from early-onset cases. Histological examination of muscle may be normal but biochemical investigations may reveal multiple respiratory chain defects. The outcome appears to be more favorable in adult than in early-onset forms. Mitochondrial depletion syndromes is not only a condition of neonates, infants, or juveniles but rarely also occurs in adults, presenting with minimal manifestations or manifestations like in the early-onset forms. Outcome of adult-onset MDS appears more favorable than early-onset MDS.

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Year:  2013        PMID: 23968935     DOI: 10.1017/s0317167100014852

Source DB:  PubMed          Journal:  Can J Neurol Sci        ISSN: 0317-1671            Impact factor:   2.104


  14 in total

Review 1.  The emergence of the mitochondrial genome as a partial regulator of nuclear function is providing new insights into the genetic mechanisms underlying age-related complex disease.

Authors:  Martin P Horan; David N Cooper
Journal:  Hum Genet       Date:  2013-12-04       Impact factor: 4.132

Review 2.  Genetic Counselling for Maternally Inherited Mitochondrial Disorders.

Authors:  Joanna Poulton; Josef Finsterer; Patrick Yu-Wai-Man
Journal:  Mol Diagn Ther       Date:  2017-08       Impact factor: 4.074

3.  Whole exome sequencing identifies a homozygous POLG2 missense variant in an infant with fulminant hepatic failure and mitochondrial DNA depletion.

Authors:  Hemant Varma; Phyllis L Faust; Alejandro D Iglesias; Stephen M Lagana; Karen Wou; Michio Hirano; Salvatore DiMauro; Mahesh M Mansukani; Kirsten E Hoff; Peter L Nagy; William C Copeland; Ali B Naini
Journal:  Eur J Med Genet       Date:  2016-08-31       Impact factor: 2.708

4.  POLG1-related Mitochondrial Disorder with MNGIE- and Leigh-like Features.

Authors:  Josef Finsterer
Journal:  Ann Indian Acad Neurol       Date:  2020-06-10       Impact factor: 1.383

5.  Analysis of Human Mitochondrial DNA Content by Southern Blotting and Nonradioactive Probe Hybridization.

Authors:  Joel H Wheeler; Carolyn K J Young; Matthew J Young
Journal:  Curr Protoc Toxicol       Date:  2019-04-14

6.  A Combined Model of Human iPSC-Derived Liver Organoids and Hepatocytes Reveals Ferroptosis in DGUOK Mutant mtDNA Depletion Syndrome.

Authors:  Jingyi Guo; Lifan Duan; Xueying He; Shengbiao Li; Yi Wu; Ge Xiang; Feixiang Bao; Liang Yang; Hongyan Shi; Mi Gao; Lingjun Zheng; Huili Hu; Xingguo Liu
Journal:  Adv Sci (Weinh)       Date:  2021-03-08       Impact factor: 16.806

7.  Atheromatosis of the Scalp: A Novel Feature of Chronic Progressive External Ophthalmoplegia Plus Due to a Single Mitochondrial DNA Deletion.

Authors:  Josef Finsterer
Journal:  Cureus       Date:  2021-12-23

Review 8.  Genes and Pathways Involved in Adult Onset Disorders Featuring Muscle Mitochondrial DNA Instability.

Authors:  Naghia Ahmed; Dario Ronchi; Giacomo Pietro Comi
Journal:  Int J Mol Sci       Date:  2015-08-05       Impact factor: 5.923

9.  Mitochondrial disorder caused Charles Darwin's cyclic vomiting syndrome.

Authors:  Josef Finsterer; John Hayman
Journal:  Int J Gen Med       Date:  2014-01-08

Review 10.  Syndromes associated with mitochondrial DNA depletion.

Authors:  Célia Nogueira; Ligia S Almeida; Claudia Nesti; Ilaria Pezzini; Arnaldo Videira; Laura Vilarinho; Filippo M Santorelli
Journal:  Ital J Pediatr       Date:  2014-04-03       Impact factor: 2.638

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