BACKGROUNDSimultaneous intracranial and testicular germ cell tumors (GCTs) are extremely rare, leading to a lack of adequate experience in their treatment. Therefore, the authors report a case of this kind of GCT.OBSERVATIONSA 5-year-old boy was admitted to the hospital with headache and vomiting. Computed tomography and magnetic resonance imaging suggested the possibility of a GCT in the pineal region. The value of the serum tumor marker alpha-fetoprotein (AFP) was 5,396.1 μg/L, and β-human chorionic gonadotropin levels were within the normal range. Subsequently, the tumor was removed, and the final pathological result was a mixed GCT. Therefore, chemotherapy and radiation were added. However, the authors found a testicular tumor on ultrasound at the same time, and pathology after surgery suggested a mature cystic teratoma. Following treatment, the patient recovered well, and AFP levels dropped to normal values.LESSONSTo the authors’ knowledge, this report is the fourth case of simultaneous intracranial and testicular GCTs and the first case of a simultaneous mixed GCT in the pineal region and mature teratoma of the testis. A combination of surgery, chemotherapy, and radiation therapy for mixed GCTs in the pineal region and surgical excision for testicular reproductive cell tumors are effective in these patients, but long-term monitoring is required.