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Mitochondrial cytopathy

MedGen UID:
419940
Concept ID:
C2931928
Disease or Syndrome
Synonym: MITOCHONDRIAL CYTOPATHY
SNOMED CT: Mitochondrial cytopathy (240096000)
 
OMIM®: 530000

Definition

Kearns-Sayre syndrome (KSS) is a mitochondrial encephalomyopathy characterized by progressive external ophthalmoplegia, pigmentary retinopathy, and at least 1 of the following: heart block, cerebellar ataxia, or a cerebrospinal fluid protein level above 100 mg/dl. Serum and cerebrospinal fluid lactate and pyruvate levels are increased, and muscle biopsy shows ragged-red fibers. Onset is before 20 years of age (summary by Tanji et al., 1999; Pineda et al., 2006). [from OMIM]

Professional guidelines

PubMed

Ahmed ST, Craven L, Russell OM, Turnbull DM, Vincent AE
Neurotherapeutics 2018 Oct;15(4):943-953. doi: 10.1007/s13311-018-00674-4. PMID: 30406383Free PMC Article
El-Hattab AW, Adesina AM, Jones J, Scaglia F
Mol Genet Metab 2015 Sep-Oct;116(1-2):4-12. Epub 2015 Jun 15 doi: 10.1016/j.ymgme.2015.06.004. PMID: 26095523
Karaa A, Goldstein A
Pediatr Diabetes 2015 Feb;16(1):1-9. Epub 2014 Oct 20 doi: 10.1111/pedi.12223. PMID: 25330715

Recent clinical studies

Etiology

Gazdikova K, Fojtova A, Ticha L
Bratisl Lek Listy 2022;123(9):659-671. doi: 10.4149/BLL_2022_106. PMID: 36039885
Alves CAPF, Gonçalves FG, Grieb D, Lucato LT, Goldstein AC, Zuccoli G
Top Magn Reson Imaging 2018 Aug;27(4):219-240. doi: 10.1097/RMR.0000000000000173. PMID: 30086109
Emma F, Salviati L
Nephrol Ther 2017 Apr;13 Suppl 1:S23-S28. doi: 10.1016/j.nephro.2017.01.014. PMID: 28577739
Dominic EA, Ramezani A, Anker SD, Verma M, Mehta N, Rao M
Heart 2014 Apr;100(8):611-8. Epub 2014 Jan 21 doi: 10.1136/heartjnl-2013-304657. PMID: 24449718
Emma F, Bertini E, Salviati L, Montini G
Pediatr Nephrol 2012 Apr;27(4):539-50. Epub 2011 Jun 9 doi: 10.1007/s00467-011-1926-6. PMID: 21656172Free PMC Article

Diagnosis

Viering DHHM, Vermeltfoort L, Bindels RJM, Deinum J, de Baaij JHF
J Am Soc Nephrol 2023 Nov 1;34(11):1875-1888. Epub 2023 Sep 6 doi: 10.1681/ASN.0000000000000224. PMID: 37678265Free PMC Article
Lu JQ, Tarnopolsky MA
Mitochondrion 2021 Jan;56:52-61. Epub 2020 Nov 19 doi: 10.1016/j.mito.2020.11.005. PMID: 33220502
Alves CAPF, Gonçalves FG, Grieb D, Lucato LT, Goldstein AC, Zuccoli G
Top Magn Reson Imaging 2018 Aug;27(4):219-240. doi: 10.1097/RMR.0000000000000173. PMID: 30086109
El-Hattab AW, Scaglia F
Cell Calcium 2016 Sep;60(3):199-206. Epub 2016 Mar 4 doi: 10.1016/j.ceca.2016.03.003. PMID: 26996063
Hall AM, Unwin RJ, Hanna MG, Duchen MR
QJM 2008 Oct;101(10):755-66. Epub 2008 May 16 doi: 10.1093/qjmed/hcn060. PMID: 18487272

Therapy

Harmon KK, Stout JR, Fukuda DH, Pabian PS, Rawson ES, Stock MS
Nutrients 2021 May 27;13(6) doi: 10.3390/nu13061825. PMID: 34071875Free PMC Article
Finsterer J, Zarrouk Mahjoub S
Expert Opin Drug Metab Toxicol 2012 Jan;8(1):71-9. Epub 2011 Dec 7 doi: 10.1517/17425255.2012.644535. PMID: 22149023
Koga Y, Povalko N, Nishioka J, Katayama K, Yatsuga S, Matsuishi T
Biochim Biophys Acta 2012 May;1820(5):608-14. Epub 2011 Sep 14 doi: 10.1016/j.bbagen.2011.09.005. PMID: 21944974
Muravchick S
Adv Drug Deliv Rev 2008 Oct-Nov;60(13-14):1553-60. Epub 2008 Jul 4 doi: 10.1016/j.addr.2008.03.019. PMID: 18647627
Tarnopolsky MA
Subcell Biochem 2007;46:183-204. doi: 10.1007/978-1-4020-6486-9_10. PMID: 18652078

Prognosis

Son JS, Seo GH, Kim YM, Kim GH, Jin HK, Bae JS, Im HJ, Yoo HW, Lee BH
Medicine (Baltimore) 2022 Feb 4;101(5):e28793. doi: 10.1097/MD.0000000000028793. PMID: 35119049Free PMC Article
Fervenza FC, Gavrilova RH, Nasr SH, Irazabal MV, Nath KA
Am J Kidney Dis 2019 Feb;73(2):273-277. Epub 2018 Oct 8 doi: 10.1053/j.ajkd.2018.06.032. PMID: 30309714
Brown DA, Perry JB, Allen ME, Sabbah HN, Stauffer BL, Shaikh SR, Cleland JG, Colucci WS, Butler J, Voors AA, Anker SD, Pitt B, Pieske B, Filippatos G, Greene SJ, Gheorghiade M
Nat Rev Cardiol 2017 Apr;14(4):238-250. Epub 2016 Dec 22 doi: 10.1038/nrcardio.2016.203. PMID: 28004807Free PMC Article
El-Hattab AW, Scaglia F
Cell Calcium 2016 Sep;60(3):199-206. Epub 2016 Mar 4 doi: 10.1016/j.ceca.2016.03.003. PMID: 26996063
Wang LC, Lee WT, Tsai WY, Tsau YK, Shen YZ
Pediatr Neurol 2000 May;22(5):403-6. doi: 10.1016/s0887-8994(00)00128-4. PMID: 10913735

Clinical prediction guides

Zhao X, Li Y, Yu J, Teng H, Wu S, Wang Y, Zhou H, Li F
Metabolism 2024 Jun;155:155913. Epub 2024 Apr 11 doi: 10.1016/j.metabol.2024.155913. PMID: 38609039
Viering DHHM, Vermeltfoort L, Bindels RJM, Deinum J, de Baaij JHF
J Am Soc Nephrol 2023 Nov 1;34(11):1875-1888. Epub 2023 Sep 6 doi: 10.1681/ASN.0000000000000224. PMID: 37678265Free PMC Article
Son JS, Seo GH, Kim YM, Kim GH, Jin HK, Bae JS, Im HJ, Yoo HW, Lee BH
Medicine (Baltimore) 2022 Feb 4;101(5):e28793. doi: 10.1097/MD.0000000000028793. PMID: 35119049Free PMC Article
Dominic EA, Ramezani A, Anker SD, Verma M, Mehta N, Rao M
Heart 2014 Apr;100(8):611-8. Epub 2014 Jan 21 doi: 10.1136/heartjnl-2013-304657. PMID: 24449718
Scaglia F
Dev Disabil Res Rev 2010;16(2):136-43. doi: 10.1002/ddrr.115. PMID: 20818728

Recent systematic reviews

Viering DHHM, Vermeltfoort L, Bindels RJM, Deinum J, de Baaij JHF
J Am Soc Nephrol 2023 Nov 1;34(11):1875-1888. Epub 2023 Sep 6 doi: 10.1681/ASN.0000000000000224. PMID: 37678265Free PMC Article
Kabunga P, Lau AK, Phan K, Puranik R, Liang C, Davis RL, Sue CM, Sy RW
Int J Cardiol 2015 Feb 15;181:303-10. Epub 2014 Dec 13 doi: 10.1016/j.ijcard.2014.12.038. PMID: 25540845

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