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. Author manuscript; available in PMC: 2017 Jan 1.
Published in final edited form as: Crit Rev Biochem Mol Biol. 2015 Nov 29;51(1):53–64. doi: 10.3109/10409238.2015.1117056

Appendix Table 1.

Disease-associated amino acid residues of the human mtDNA helicase. These residues were mapped onto the helicase models presented in Figures 2 and 4.

Residue Mutation Associated disease References
R303 R303Q and R303W Autosomal dominant progressive external ophthalmoplegia (Dundar et al., 2012), (Ji et al., 2014)
W315 W315L and W315S Autosomal dominant progressive external ophthalmoplegia (Dundar et al., 2012), (Ji et al., 2014)
A318 A318T Hepatocerebral mtDNA depletion syndrome, infantile-onset spinocerebellar ataxia (Dundar et al., 2012), (Ji et al., 2014)
K319 K319E and K319T Autosomal dominant progressive external ophthalmoplegia (Dundar et al., 2012), (Ji et al., 2014)
R334 R334P and R334Q Autosomal dominant progressive external ophthalmoplegia (Dundar et al., 2012), (Ji et al., 2014)
P335 P335L Autosomal dominant progressive external ophthalmoplegia (Dundar et al., 2012), (Ji et al., 2014)
R354 R354P Autosomal dominant progressive external ophthalmoplegia (Dundar et al., 2012), (Ji et al., 2014)
R357 R357P Autosomal dominant progressive external ophthalmoplegia (Dundar et al., 2012), (Ji et al., 2014)
A359 A359T Autosomal dominant progressive external ophthalmoplegia (Dundar et al., 2012), (Ji et al., 2014)
L360 L360G Autosomal dominant progressive external ophthalmoplegia (Ji et al., 2014)
A362 A362P Autosomal dominant progressive external ophthalmoplegia (Ji et al., 2014)
W363 W363L Autosomal dominant progressive external ophthalmoplegia (Ji et al., 2014)
I367 I367T Autosomal dominant progressive external ophthalmoplegia (Dundar et al., 2012), (Ji et al., 2014)
S369 S369P and S369Y Autosomal dominant progressive external ophthalmoplegia (Dundar et al., 2012), (Ji et al., 2014)
F370 F370C and F370L Autosomal dominant progressive external ophthalmoplegia (Dundar et al., 2012), (Ji et al., 2014)
R374 R374Q and R374W Autosomal dominant progressive external ophthalmoplegia (Dundar et al., 2012), (Ji et al., 2014)
L378 L378P Autosomal dominant progressive external ophthalmoplegia (Ji et al., 2014)
L381 L381P Autosomal dominant progressive external ophthalmoplegia (Dundar et al., 2012), (Ji et al., 2014)
R391 R391H/N585S Perrault syndrome (Morino et al., 2014)
F395 F395L Renal tubulopathy (Prasad et al., 2013)
S426 S426N Autosomal dominant progressive external ophthalmoplegia (Dundar et al., 2012), (Ji et al., 2014)
W441 W441G/V507I Perrault syndrome (Morino et al., 2014)
N448 N448D Autosomal dominant progressive external ophthalmoplegia (Morino et al., 2014)
M455 M455T Autosomal dominant progressive external ophthalmoplegia (Morino et al., 2014)
L456 L456V Infantile-onset spinocerebellar ataxia (Dundar et al., 2012), (Ji et al., 2014)
T457 T457I Hepatocerebral mtDNA depletion syndrome (Dundar et al., 2012), (Ji et al., 2014)
Q458 Q458H Autosomal dominant progressive external ophthalmoplegia (Ji et al., 2014)
A460 A460G and A460P Autosomal dominant progressive external ophthalmoplegia (Ji et al., 2014)
R463 P083S/R463W Infantile-onset spinocerebellar ataxia (Hartley et al., 2012)
W474 W474C and W474S Autosomal dominant progressive external ophthalmoplegia (Dundar et al., 2012), (Ji et al., 2014)
A475 A475D, A475P and A475T Autosomal dominant progressive external ophthalmoplegia (Ji et al., 2014)
F478 F478I Autosomal dominant progressive external ophthalmoplegia (Dundar et al., 2012), (Ji et al., 2014)
E479 E479K Autosomal dominant progressive external ophthalmoplegia (Dundar et al., 2012), (Ji et al., 2014)
F485 F485L Autosomal dominant progressive external ophthalmoplegia (Dundar et al., 2012), (Ji et al., 2014)
V507 W441G/V507I Perrault syndrome (Morino et al., 2014)
Y508 Y508C Infantile-onset spinocerebellar ataxia (Dundar et al., 2012), (Ji et al., 2014)
Y537 Y537H Autosomal dominant progressive external ophthalmoplegia (Ji et al., 2014)
N585 R391H/N585S Perrault syndrome (Morino et al., 2014)