Extra-gonadal pure yolk sac tumor of the ear is very rare. We report a case of a yolk sac tumor of the ear and review the English literature in PubMed. The initial complaint was a mass protruding out of the external auditory canal. Computed tomography (CT) showed a large irregularly enhancing isodense mass lesion measuring 42*16 mm in the right external auditory canal, the right mastoid process, and extending to the right back parapharyngeal space with unclear border. Laboratory studies revealed that serum alpha fetoprotein (AFP) was significantly elevated at 664.60 ng/ml (range, 0 to 25 ng/ml), and neuron-specific enolase (NSE) was 28.3 ng/ml (range, 0 to 16.3 ng/ml). After finishing 6 cycles of chemotherapy, the patient underwent a total resection of yolk sac tumor of the ear. In addition, we review the English literature of the yolk sac tumor of the ear.
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