Endocervical adenocarcinoma in situ (AIS) with ovarian and pulmonary involvement: report of a case and review of the literature suggesting a “seed and soil hypothesis”

Purpose Cervical cancer metastases to the ovary may occur with advanced tumor stage, deep cervical stromal involvement and corpus involvement. Endocervical adenocarcinoma in situ (AIS) with ovarian involvement is exceptionally rare with about twelve reported cases. Methods Here we present a case of endocervical AIS with ovarian and pulmonary involvement 39 months after the initial diagnosis. The characteristics of that case were compared and summarized with the eleven previously published cases. Results The patients’ age ranged between 30 and 40 years (median 37.4 years). The time interval between the diagnosis of AIS and ovarian involvement was 26.7 months (range 2–84 months). Majority of the patients are alive without evidence of disease after a median time of 63.4 months (range 9–156 months). All reported cases were positive for high-risk HPV which was associated with strong p16 expression on immunohistochemistry. Conclusions The ovarian involvement by endocervical AIS suggests the concept of a transtubal spread of the neoplastic cervical cells with or without previous colonization within the endometrium without evidence of invasive growth, suggesting a seed and soil spread of the disease. In cases with ovarian involvement by the AIS and without additional extragenital spread, the prognosis may be favorable.