Case Presentation: We discuss the case of a 38-year-old female who presented in 2012 with a 15-cm-large cyst of the right ovary, with negative cervical smear cytology. Right adnexectomy was performed laparoscopically, and metastatic cystic squamous cell carcinoma probably originating from the endocervix was reported. One month later, radical hysterectomy (Piver type I) and left adnexectomy were performed; a 15-mm-large high endocervical papillary squamous cell carcinoma was diagnosed. There is no evidence of local recurrence or distant metastasis to date.

Conclusion: This case shows that these kind of (primary) cervical and (secondary) ovarian tumors are almost always discontinuous, without any evidence of direct invasion from cervix to ovary, and it also supports the notion that in cases of cervical cancer originating from the endocervix or cervical canal and protruding into or involving the isthmus, the prevalence of ovarian metastasis is higher. This unique case is a good example of unintentionally—and fortunately—diagnosed squamous cell carcinoma of endocervical origin with unilateral ovarian metastasis.