We retrospectively reviewed the medical records of 214 MM patients corresponding to 278 therapies (109 BRAF/MEK inhibitors and 169 PD1 blockades) performed in a time span between January 2012 and December 2018, 195 cases as first line, and second or further lines for the remaining 83 ones. The gene discussed is MAP2K7; the disease is Miyoshi myopathy.