• Brain 1H-MRS showed pathological lactate accumulation in one third of DM1 patients• Lactate was higher in DM1 with higher white matter lesion load and cortical atrophy• Muscle 31P-MRS showed oxidative mitochondrial metabolism deficits in DM1• Muscle mitochondrial ATP production was slower in more clinically affected DM1• In vivo multi-organ evidence of oxidative metabolism deficit in DM1 was provided

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