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Background: Spiradenocylindroma is a benign tumor of skin adnexal origin with overlapping features of two distinct neoplasms: spiradenoma and cylindroma. This cutaneous tumor typically presents on the head and neck and extracutaneous presentations are uncommon. The presentation described below involves a spiradenocylindroma within a mature ovarian teratoma is very rare. Aim: The aim of this article is to portray the diagnostic process of this unusual spiradenocylindroma presentation. Case presentation: A 65 year-old female with a left adnexal mass underwent ultrasonography and magnetic resonance imaging (MRI) which showed a left ovarian multiseptated lesion, with mural calcifications and projections into the mass. Excisional surgery was performed and histopathological examination revealed a spiradenocylindroma. Conclusion: Spiradenocylindroma is rare, hard to identify, and often misdiagnosed. Our study described the process of diagnosis and depicts the rare presentation of this lesion arising within a mature teratoma.