Immune checkpoint inhibitors (ICIs) are a new type of antitumor drug which can achieve antitumor goals by blocking the binding of immune checkpoints to their ligands, thereby enhancing the activity of T cells. Meanwhile, ICIs block the binding of immune checkpoints to their ligands, disrupting the immune tolerance of T cells to self-antigens, which may lead to a series of immune-related adverse events (irAEs). Immune checkpoint inhibitor-induced hypophysitis (IH) is a relatively rare irAE. Due to the lack of specificity in clinical manifestations, it is difficult to accurately diagnose IH in a timely manner in clinical practice. However, the risk of adverse events, especially IH, for patients receiving ICIs has not been adequately investigated. Missed or delayed diagnosis may lead to poor prognosis or even adverse clinical outcomes. In this article, we summarize the epidemiology, pathogenesis, clinical manifestations, diagnosis and treatment of IH.