Primary multiple endocrine insufficiency during immune checkpoint inhibitor treatment: A case report

Immunotherapy with immune checkpoint inhibitors (ICI) has shown promising activity against many tumor types. However, they can also induce a wide array of immune-related adverse events, ranging from mild to fatal. Primary 3 endocrine gland insufficiency during treatment with ICI has rarely been reported.

We report the case of a 33-year-old man with Ewing sarcoma who was treated with toripalimab as a second-line treatment. Approximately 11 months after initiating treatment, the patient developed subclinical hypothyroidism, which was followed by adrenal insufficiency and hypogonadism 6 months later. Consequently, the decision was made to discontinue ICI therapy and initiate hormone replacement therapy to manage endocrine deficiencies.

Serum adrenocorticotropic hormone, thyroid stimulating hormone, and prolactin levels increased significantly, while cortisol, estradiol, and testosterone levels decreased (Table 1). The patient had negative findings on the pituitary MRI.

As part of the management strategy, ICI therapy was ceased and hormone replacement therapy was commenced to address endocrine deficiencies.

After hormone replacement therapy, his symptoms improved and follow-up examinations showed normalization of hormone levels.

Clinicians should be aware of the potential of immune checkpoint inhibitor therapy to cause endocrine dysfunction. Prompt recognition and management of these adverse events are crucial for patient health and quality of life.