Abstract:
RATIONALE:Tolosa-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 CONCERNS:We experienced two patients with THS that recurred twice while tapering or after ceasing corticosteroid administration. DIAGNOSIS:Both patients were diagnosed as recurrent THS. INTERVENTIONS:Methotrexate was treated with a combination of corticosteroid after THS recurred twice with corticosteroid therapy alone. OUTCOMES:After 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.
journal_name
Medicine (Baltimore)journal_title
Medicineauthors
Kwon HS,Kim TY,Kim JH,Bae JH,Koh SH,Choi H,Lee KY,Lee YJ,Kim YS,Kim HYdoi
10.1097/MD.0000000000019882subject
Has Abstractpub_date
2020-04-01 00:00:00pages
e19882issue
17eissn
0025-7974issn
1536-5964pii
00005792-202004240-00071journal_volume
99pub_type
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