Adenocarcinoma in situ (AIS) of the endocervix is typically confined to the cervix, but may be extensive. We report 2 cases of extensive AIS-one with intraendometrial spread-that recurred after cone biopsy and were associated with ovarian metastases. In both cases, primary and metastatic tumors were positive for human papillomavirus 16. Both patients are currently disease-free and one has had no recurrence after a follow-up period of 145 months. Extensive, recurring AIS is a rare variant that may be unique for its risk of coincident ovarian involvement. However, ovarian spread, albeit rare, does not invariably result in an adverse outcome.