MTO1-deficient mouse model mirrors the human phenotype showing complex I defect and cardiomyopathy.

Acoustic startle response measured in another cohort of mice (9 controls and 10 mutants aged 9 weeks) showed an altered curve: there was a highly significant interaction between genotype and startle pulse intensity (F (7,11) = 4.153, p<0.001), and Bonferroni posthoc tests revealed a significantly lower response of mutants at maximum dB (p<0.01, Fig. 2F ).

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The lighter Mto1 mutant females showed a trend to improved performance on an accelerating rotarod whereas mutant males showed impaired performance in advancing trials (data not shown).

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