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Myopathy, lactic acidosis, and sideroblastic anemia

MedGen UID:
373888
Concept ID:
C1838103
Disease or Syndrome
Synonyms: Mitochondrial myopathy and sideroblastic anemia; Myopathy with lactic acidosis and sideroblastic anemia
SNOMED CT: Mitochondrial myopathy with sideroblastic anemia syndrome (724138007); Myopathy, lactic acidosis and sideroblastic anemia (724138007)
Modes of inheritance:
Autosomal recessive inheritance
MedGen UID:
141025
Concept ID:
C0441748
Intellectual Product
Source: Orphanet
A mode of inheritance that is observed for traits related to a gene encoded on one of the autosomes (i.e., the human chromosomes 1-22) in which a trait manifests in individuals with two pathogenic alleles, either homozygotes (two copies of the same mutant allele) or compound heterozygotes (whereby each copy of a gene has a distinct mutant allele).
 
Related genes: PUS1, YARS2
 
Monarch Initiative: MONDO:0000863
OMIM®: 600462
OMIM® Phenotypic series: PS600462
Orphanet: ORPHA2598

Definition

Belongs to the heterogeneous family of metabolic myopathies. It is characterised by progressive exercise intolerance manifesting in childhood, onset of sideroblastic anaemia around adolescence, lactic acidaemia and mitochondrial myopathy. Less than 10 cases have been described so far. A 656C-->T mutation in the nuclear pseudouridine synthase 1 gene (PUS1), localised to 12q24.33, has recently been identified in some patients. Deficient pseudouridylation of mitochondrial tRNAs may be responsible for the oxidative phosphorylation disorder. Transmission is autosomal recessive. [from SNOMEDCT_US]

Term Hierarchy

CClinical test,  RResearch test,  OOMIM,  GGeneReviews,  VClinVar  
Follow this link to review classifications for Myopathy, lactic acidosis, and sideroblastic anemia in Orphanet.

Recent clinical studies

Etiology

Rudaks LI, Watson E, Oboudiyat C, Kumar KR, Sullivan P, Cowley MJ, Davis RL, Sue CM
Am J Med Genet A 2022 Jul;188(7):2226-2230. Epub 2022 Apr 8 doi: 10.1002/ajmg.a.62755. PMID: 35393742Free PMC Article
Zehravi M, Wahid M, Ashraf J, Fatima T
Genet Test Mol Biomarkers 2021 Mar;25(3):218-226. doi: 10.1089/gtmb.2020.0246. PMID: 33734897
Tesarova M, Vondrackova A, Stufkova H, Veprekova L, Stranecky V, Berankova K, Hansikova H, Magner M, Galoova N, Honzik T, Vodickova E, Stary J, Zeman J
Pediatr Blood Cancer 2019 Apr;66(4):e27591. Epub 2018 Dec 26 doi: 10.1002/pbc.27591. PMID: 30588737
Riley LG, Cooper S, Hickey P, Rudinger-Thirion J, McKenzie M, Compton A, Lim SC, Thorburn D, Ryan MT, Giegé R, Bahlo M, Christodoulou J
Am J Hum Genet 2010 Jul 9;87(1):52-9. doi: 10.1016/j.ajhg.2010.06.001. PMID: 20598274Free PMC Article

Diagnosis

Rudaks LI, Watson E, Oboudiyat C, Kumar KR, Sullivan P, Cowley MJ, Davis RL, Sue CM
Am J Med Genet A 2022 Jul;188(7):2226-2230. Epub 2022 Apr 8 doi: 10.1002/ajmg.a.62755. PMID: 35393742Free PMC Article
Beecher G, Fleming MD, Liewluck T
Muscle Nerve 2022 Apr;65(4):374-390. Epub 2022 Jan 5 doi: 10.1002/mus.27474. PMID: 34985130
Sommerville EW, Ng YS, Alston CL, Dallabona C, Gilberti M, He L, Knowles C, Chin SL, Schaefer AM, Falkous G, Murdoch D, Longman C, de Visser M, Bindoff LA, Rawles JM, Dean JCS, Petty RK, Farrugia ME, Haack TB, Prokisch H, McFarland R, Turnbull DM, Donnini C, Taylor RW, Gorman GS
JAMA Neurol 2017 Jun 1;74(6):686-694. doi: 10.1001/jamaneurol.2016.4357. PMID: 28395030Free PMC Article
Shahni R, Wedatilake Y, Cleary MA, Lindley KJ, Sibson KR, Rahman S
Am J Med Genet A 2013 Sep;161A(9):2334-8. Epub 2013 Aug 5 doi: 10.1002/ajmg.a.36065. PMID: 23918765Free PMC Article
Finsterer J
Acta Haematol 2007;118(2):88-98. Epub 2007 Jul 18 doi: 10.1159/000105676. PMID: 17637511

Prognosis

Tesarova M, Vondrackova A, Stufkova H, Veprekova L, Stranecky V, Berankova K, Hansikova H, Magner M, Galoova N, Honzik T, Vodickova E, Stary J, Zeman J
Pediatr Blood Cancer 2019 Apr;66(4):e27591. Epub 2018 Dec 26 doi: 10.1002/pbc.27591. PMID: 30588737
Sommerville EW, Ng YS, Alston CL, Dallabona C, Gilberti M, He L, Knowles C, Chin SL, Schaefer AM, Falkous G, Murdoch D, Longman C, de Visser M, Bindoff LA, Rawles JM, Dean JCS, Petty RK, Farrugia ME, Haack TB, Prokisch H, McFarland R, Turnbull DM, Donnini C, Taylor RW, Gorman GS
JAMA Neurol 2017 Jun 1;74(6):686-694. doi: 10.1001/jamaneurol.2016.4357. PMID: 28395030Free PMC Article
Nakajima J, Eminoglu TF, Vatansever G, Nakashima M, Tsurusaki Y, Saitsu H, Kawashima H, Matsumoto N, Miyake N
J Hum Genet 2014 Apr;59(4):229-32. Epub 2014 Jan 16 doi: 10.1038/jhg.2013.143. PMID: 24430573
Finsterer J
Acta Haematol 2007;118(2):88-98. Epub 2007 Jul 18 doi: 10.1159/000105676. PMID: 17637511

Clinical prediction guides

Riley LG, Heeney MM, Rudinger-Thirion J, Frugier M, Campagna DR, Zhou R, Hale GA, Hilliard LM, Kaplan JA, Kwiatkowski JL, Sieff CA, Steensma DP, Rennings AJ, Simons A, Schaap N, Roodenburg RJ, Kleefstra T, Arenillas L, Fita-Torró J, Ahmed R, Abboud M, Bechara E, Farah R, Tamminga RYJ, Bottomley SS, Sanchez M, Huls G, Swinkels DW, Christodoulou J, Fleming MD
Haematologica 2018 Dec;103(12):2008-2015. Epub 2018 Jul 19 doi: 10.3324/haematol.2017.182659. PMID: 30026338Free PMC Article
Sommerville EW, Ng YS, Alston CL, Dallabona C, Gilberti M, He L, Knowles C, Chin SL, Schaefer AM, Falkous G, Murdoch D, Longman C, de Visser M, Bindoff LA, Rawles JM, Dean JCS, Petty RK, Farrugia ME, Haack TB, Prokisch H, McFarland R, Turnbull DM, Donnini C, Taylor RW, Gorman GS
JAMA Neurol 2017 Jun 1;74(6):686-694. doi: 10.1001/jamaneurol.2016.4357. PMID: 28395030Free PMC Article
Finsterer J
Acta Haematol 2007;118(2):88-98. Epub 2007 Jul 18 doi: 10.1159/000105676. PMID: 17637511
Inbal A, Avissar N, Shaklai M, Kuritzky A, Schejter A, Ben-David E, Shanske S, Garty BZ
Am J Med Genet 1995 Jan 30;55(3):372-8. doi: 10.1002/ajmg.1320550325. PMID: 7726239

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