• Ze-Hua Lai, Kai-Qi Ding, Xuan-Qiang Tu, Yuan-Yue Song, and Li-Li Zeng.
• Department of Neurology and Institute of Neurology, Ruijin Hospital, Shanghai Jiao Tong University School of Medicine, Shanghai, China.
• Medicine (Baltimore). 2023 Sep 8; 102 (36): e34682e34682.

RationaleFew reports of idiopathic hypereosinophilic syndrome exist presenting as ischemic cerebrovascular disease, and the majority are watershed infarction. We report the first case of idiopathic hypereosinophilic syndrome that has clinical features of capsular warning syndrome lasting 6 weeks.Patient ConcernsA 26-year-old man complained of recurrent right limb weakness, accompanying slurred speech, and right facial paresthesia.DiagnosesThe patient was diagnosed with idiopathic hypereosinophilic syndrome (IHES).InterventionsAdequate glucocorticoid and anticoagulant treatments were given.OutcomesThe patient's motor ability improved, and he was discharged 2 weeks later. Muscle strength in the right-side extremities had fully recovered at a 3-month follow-up after discharge.LessonsThis case suggests that idiopathic hypereosinophilic syndrome should be considered as a cause of capsular warning syndrome, and the dose of glucocorticoid and the efficacy evaluation index needs to be reevaluated for the treatment of ischemic cerebrovascular disease associated with idiopathic hypereosinophilic syndrome.Copyright © 2023 the Author(s). Published by Wolters Kluwer Health, Inc.

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