Our study provides some useful insights for immunotherapy of MTM subtypes: patients with CMTM6-high/PD-L1+ status may benefit from the dual blockade of PD-L1 and CMTM6; patients with one positive immune marker (PD-L1 or CMTM6) may need corresponding immune blockage to improve efficacy; for patients with no positive immune maker, immunotherapy targeting other markers, such as PD-L2 or CMTM4, may be needed. This evidence concerns the gene CMTM4 and X-linked myotubular myopathy.