Comparative biochemical studies of ATPases in cells from patients with the T8993G or T8993C mitochondrial DNA mutations
- PMID: 9870208
- DOI: 10.1023/a:1005418718299
Comparative biochemical studies of ATPases in cells from patients with the T8993G or T8993C mitochondrial DNA mutations
Abstract
We performed comparative biochemical studies in cultured fibroblast mitochondria from patients with the T8993G or the T8993C point mutations in the ATPase 6 gene of mitochondrial DNA. We found that ATP production was much more severely decreased in cells from patients with the T8993G mutation than in those from patients with the T8993C mutation. Kinetic studies suggest that both mutations affect only the F0 sector of the mitochondrial ATPase complex. We conclude that these two mutations, which result in the substitution of different amino acids at the same site of the ATPase, result in an enzyme with different biochemical characteristics.
Similar articles
-
Clinical and biochemical characteristics in patients with a high mutant load of the mitochondrial T8993G/C mutations.Am J Med Genet A. 2006 Apr 15;140(8):863-8. doi: 10.1002/ajmg.a.31194. Am J Med Genet A. 2006. PMID: 16532470
-
Oligomycin induces a decrease in the cellular content of a pathogenic mutation in the human mitochondrial ATPase 6 gene.J Biol Chem. 1999 Apr 2;274(14):9386-91. doi: 10.1074/jbc.274.14.9386. J Biol Chem. 1999. PMID: 10092618
-
Clinical heterogeneity associated with the mitochondrial DNA T8993C point mutation.Pediatr Res. 1996 May;39(5):914-7. doi: 10.1203/00006450-199605000-00028. Pediatr Res. 1996. PMID: 8726250
-
Structure and function of the ATPase-ATP synthase complex of mitochondria as compared to chloroplasts and bacteria.Biochimie. 1986 Mar;68(3):367-74. doi: 10.1016/s0300-9084(86)80003-7. Biochimie. 1986. PMID: 2874838 Review.
-
Mitochondrial diseases and ATPase defects of nuclear origin.Biochim Biophys Acta. 2004 Jul 23;1658(1-2):115-21. doi: 10.1016/j.bbabio.2004.04.012. Biochim Biophys Acta. 2004. PMID: 15282182 Review.
Cited by
-
Tall stature and progressive overweight in mitochondrial encephalopathy.J Inherit Metab Dis. 2003;26(7):720-2. doi: 10.1023/b:boli.0000005647.71704.25. J Inherit Metab Dis. 2003. PMID: 14707524
-
The pathogenic m.8993 T > G mutation in mitochondrial ATP6 gene prevents proton release from the subunit c-ring rotor of ATP synthase.Hum Mol Genet. 2021 Apr 27;30(5):381-392. doi: 10.1093/hmg/ddab043. Hum Mol Genet. 2021. PMID: 33600551 Free PMC article.
-
Biochemical analysis of respiratory function in cybrid cell lines harbouring mitochondrial DNA mutations.Biochem J. 2004 Dec 1;384(Pt 2):287-93. doi: 10.1042/BJ20040561. Biochem J. 2004. PMID: 15324306 Free PMC article.
-
Variability of Clinical Phenotypes Caused by Isolated Defects of Mitochondrial ATP Synthase.Physiol Res. 2024 Aug 31;73(Suppl 1):S243-S278. doi: 10.33549/physiolres.935407. Epub 2024 Jul 17. Physiol Res. 2024. PMID: 39016153 Free PMC article.
-
Variants in Human ATP Synthase Mitochondrial Genes: Biochemical Dysfunctions, Associated Diseases, and Therapies.Int J Mol Sci. 2024 Feb 13;25(4):2239. doi: 10.3390/ijms25042239. Int J Mol Sci. 2024. PMID: 38396915 Free PMC article. Review.
References
Publication types
MeSH terms
Substances
Grants and funding
LinkOut - more resources
Full Text Sources