Background: Endocervical adenocarcinoma in situ (AIS) often occurs in premenopausal patients, and, because ovarian metastasis is exceedingly rare, surgical intervention often involves preservation of the ovaries. Isolated recurrence to the ovary following definitive surgery for AIS is even rarer, with only several cases reported to date. Case: A 48-year-old female presented to her gynecologist complaining of new-onset back pain and urinary frequency. Her history was significant for adenocarcinoma in situ 5 years prior, for which she underwent two conization procedures followed by hysterectomy, which revealed no residual tumors. Evaluation of her current symptoms revealed that she had a large pelvic mass, for which she underwent bilateral oophorectomy and a staging procedure for presumed ovarian cancer. Her final pathology was consistent with a recurrence of cervical adenocarcinoma. Results: After chemoradiotherapy and chemotherapy, she had no evidence of disease at a 2-year follow-up. Conclusions: Although ovarian metastasis in the setting of AIS is rare, and the incidence of isolated ovarian recurrence is even more uncommon, patients should be counseled and monitored closely for this possibility following treatment for AIS.