Head and neck osteosarcomas are infrequent and usually present in the third–fourth decades of life. However, they are extremely rare in the pediatric population. Primary involvement of the cranial vault, excluding the mandible and maxilla, is an exceedingly rare phenomenon; thus, the number of clinical studies published in the literature is limited. Because of the anatomy of the head, complete resection may be difficult to achieve. Furthermore, an aggressive surgical approach can cause a significant functional impairment or cosmetic defect. We report the case of a 17-year-old patient with an aggressive recurrence of multiple tumors in the left side of the skull accompanied with a severe headache and complete left-sided vision loss with no metastases at presentation. The recurrence of the tumor was preceded by pregnancy and delivery, which raised the question of whether pregnancy hormones were a provoking factor in the recurrence of the tumor or not.
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