Objective: Surgical treatment options are limited for medically and endovascular refractory cerebral venous sinus thrombosis (CVT). We describe the technical feasibility of open, direct sinus thrombectomy in a pediatric patient with medically and endovascular refractory CVT. Methods: A 15-year-old patient with superior sagittal sinus (SSS) thrombosis refractory to medical and endovascular treatment experienced clinical deterioration with the progression of bifrontal hematomas and cerebral edema. The patient was treated surgically with a decompressive hemicraniectomy and burr hole SSS thrombectomy with a Fogarty balloon and direct sinus injection of tissue plasminogen activator. Results: Surgical treatment with decompressive craniotomy and direct sinus thrombectomy was technically feasible with excellent intracranial pressure control, and radiological recanalization of the superior sagittal sinus with a good clinical outcome. Conclusion: Direct surgical sinus thrombectomy is a potential concomitant treatment strategy with decompressive craniectomy for medically refractory CVT in a pediatric setting.