Immune checkpoint inhibitors (ICIs) have been recently proposed as a strategy for treating anti-malignant neoplasms. However, this treatment leads to immune-related adverse events (irAEs) such as autoimmune endocrinopathy. Early diagnosis and appropriate treatment of ICI-related hypophysitis are essential as it can manifest as a life-threatening condition due to an adrenal crisis. In this review, we summarize the pathogenesis, risk factors, diagnostic processes, clinical characteristics, and its current management. In particular, we discuss the different aspects of anti-CTLA-4 antibody-related and anti-PD-1/anti-PD-L1 antibody-related hypophysitis. We also propose key points for early detection and diagnosis by identifying the target group that should be monitored more carefully. Specific methods of hormone replacement therapy have also been described. We hope that this review will lead to a better understanding and management of this rare but serious condition during cancer treatment and further elucidate the pathophysiology of pituitary autoimmunity.