A 43-year-old woman, previously misdiagnosed as having primary hyperthyroidism and treated with antithyroid drugs, presented to us with overt hyperthyroidism, high levels of thyroid hormones and elevated thyroid-stimulating hormone (TSH). Μagnetic resonance imaging (MRI) revealed a pituitary microadenoma extending suprasellarly. The patient responded favorably to initial treatment with somatostatin analogs for 2 years but due to the escape phenomenon, TSH levels escalated and hyperthyroidism relapsed. Transsphenoidal adenomectomy was applied but recurrence was again observed due to incomplete tumor removal. Gamma knife radiosurgery was finally employed 5.5 years ago, resulting in complete disease remission without evidence of long-term complications to date. Thyrotropin-secreting adenomas (TSHomas) are rare with an estimated prevalence of about one case per million. We retrieved from the literature 14 cases of TSHomas treated by gamma knife radiosurgery and compared the outcomes. Our results demonstrate the efficacy and safety of gamma knife radiosurgery for achieving remission in most of the cases, suggesting validation of this technique as an effective treatment option for the management of recurrent TSHomas.