Immune checkpoint inhibitors (ICIs), have emerged to the forefront of management for various advanced cancers, such as melanoma, lung cancer and renal cell carcinoma. Immune checkpoints such as CTLA-4 and PD-1 serve to inhibit T cell activation and signaling; therefore through blockade of these pathways, ICIs promote anti-tumour immune activation. However, as a result of T cell disinhibition, ICIs have been reported to cause immune related adverse events (irAEs) affecting numerous organ systems. One of the most serious and potentially life-threatening irAE is inflammatory myositis. Myositis, which generally presents with progressive proximal muscle weakness and elevated serum creatine kinase (CK), has been reported in <1% of patients who have received ICI therapy. A rare cause of elevated CK is adrenal insufficiency, which has been reported in up to 6% of ICI users. Here we report a case of ICI-related hypophysitis related myopathy that was initially misdiagnosed as ICI-associated inflammatory myositis. This case illustrates the importance of considering a wide differential when assessing hyperCKemia in the setting of ICI use.