Thyrotropin-secreting adenoma (TSHoma) is the least common type of pituitary adenoma, these patients often present with symptoms of hyperthyroidism. When TSHoma patients combined with autoimmune hypothyroidism, it is critically difficult to diagnose for the specific confusion in the results of thyroid function test.One middle-aged male patient was presented with a sellar tumor on cranial MRI for headache symptoms. After hospitalization, a significant increase in thyrotropin (TSH) was revealed by the endocrine tests, while free thyronine (FT3) and free thyroxine (FT4) decreased, and the diffuse destruction of thyroid gland was revealed by thyroid ultrasound. Based on the endocrine test results, the patient was diagnosed as autoimmune hypothyroidism. After the multidisciplinary discussion, the pituitary adenoma was removed by endoscopic transnasal surgery, until the tumor was completely excised, for which TSHoma was revealed by postoperative pathology. A significant decrease of TSH was revealed by the postoperative thyroid function tests, the treatment for autoimmune hypothyroidism was conducted. After 20 months of follow-up, the thyroid function of patient had been improved significantly.When the thyroid function test results of patients with TSHoma are difficult to interpret, the possibility of combined primary thyroid disease should be considered. TSHoma combined with autoimmune hypothyroidism is rare, which is difficult to diagnose. The multidisciplinary collaborative treatment could help to improve the outcomes of treatment.© 2023. The Author(s).