Two cases presented with a myalgia-hiperCKemia syndrome: patient P42-Myo104 carrying a homozygous VUS c.1186A > G in exon 14 of the DYSF gene and patient P33-Myo095 with a heterozygous recurrent pathogenic variant c.5979dupA on exon 53. This evidence concerns the gene DYSF and Myalgia.