Cilostazol in a Child With Moyamoya Disease

Moyamoya disease is characterized by progressive stenosis of the terminal internal carotid arteries and their branches with secondary angiogenesis of lenticulostriate collateral vessels. Although surgical revascularization decreases the risk of ischemic and hemorrhagic strokes, there remains little evidence demonstrating safety and efficacy of nonsurgical interventions in pediatric moyamoya. Recent data support the use of cilostazol in adult moyamoya, but cilostazol safety and efficacy in pediatric moyamoya remains unknown. We report the case of a 9-year-old girl diagnosed with MMD following a large right MCA stroke who experienced frequent transient neurologic events consisting of dysarthria and left-sided drooling after pial synangiosis. After unsuccessful trials of antiseizure medications and blood pressure augmentation, the events ultimately abated after the addition of cilostazol. Episodes recurred but again subsided after cilostazol dosing adjustment. This case suggests that cilostazol may be safe and effective for management of pediatric MMD and highlights the need for further investigation.