The rationale included: 1) MTM1, DNM2, BIN1, and SPEG mutations cause CNM (5, 19, 27, 28); 2) mouse models of CNM genes show an abnormal triad structure and defective calcium handling in skeletal muscles (7, 19, 29, 30); and 3) in this study we show that DNM2 levels were increased in SPEG-deficient mice and SPEG-β interacted with DNM2. Here, SPEG is linked to centronuclear myopathy.