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Essential thrombocytosis (ET) is an uncommon pediatric hematologic disorder that can result in thrombotic complications, including cerebral venous sinus thrombosis (CVST). Although CVST associated with ET is exceedingly rare, it can be devastating to the patient. We here report a pediatric case of CVST associated with ET. The patient was treated with hydroxyurea and warfarin, which was later replaced by low-dose aspirin. Platelet counts were well controlled after 16 months of follow-up, and no further thrombotic events occurred. Mucositis was the main adverse effect of treatment.