Although the use of ipilimumab monotherapy front-line for metastatic melanoma (MM) or in the adjuvant setting for resected Stage III melanoma has been largely replaced by PD-1 inhibitors, we feel that CTLA-4 inhibitors, particularly in combination with PD-1 blockade, will continue to be widely used in multiple cancer types. The gene discussed is CTLA4; the disease is Miyoshi myopathy.