Sudden unexpected death in epilepsy (SUDEP) is a devastating complication in patient with epilepsy. The mechanism of SUDEP is unclear, but several risk factors are reported and various studies are ongoing. Cerebral cavernous hemangioma (CCH) is a kind of vascular malformation that can cause seizure. Seizure caused by CCH is usually progress to intractable epilepsy. Dendriform pulmonary ossification (DPO) is an uncommon and rarely diagnosed disease during life, and there have been no reports of SUDEP with DPO. We recently experienced an autopsy case of a young woman dying suddenly and unexpectedly, who had suffered from epilepsy caused by CCH and in one of them DPO was identified. Herein we report that case and provide a literature review.