• Hyuk Sung Kwon, Tae Yoon Kim, Ji Hyun Kim, Jeong Hoon Bae, Seong-Ho Koh, Hojin Choi, Kyu-Yong Lee, Young Joo Lee, Young Seo Kim, and Hyun Young Kim.
• Department of Neurology.
• Medicine (Baltimore). 2020 Apr 1; 99 (17): e19882.

RationaleTolosa-Hunt syndrome (THS) is rare condition characterized by painful ophthalmoplegia that usually responds well to corticosteroid. About a half of THS patients experience recurrence within intervals of months to years from initial presentation. Recurrence is more common in younger patients, and can be ipsilateral, contralateral, or bilateral. Cyclosporine, azathioprine, methotrexate, mycophenolate mofetil, infliximab, and radiotherapy can be considered as second-line treatment. However, there is insufficient evidence for treatments preventing recurrence of THS.Patient ConcernsWe experienced two patients with THS that recurred twice while tapering or after ceasing corticosteroid administration.DiagnosisBoth patients were diagnosed as recurrent THS.InterventionsMethotrexate was treated with a combination of corticosteroid after THS recurred twice with corticosteroid therapy alone.OutcomesAfter adding methotrexate to the steroid regimen, their symptoms were successfully regulated and ceased to recur LESSONS:: These cases add to the evidence for the use of methotrexate as a second-line therapeutic agent for those patients with recurrent THS attacks. Further studies are in need to prove the risk and benefits of second-line treatments in THS.

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