Background: Cervical adenocarcinoma in situ (AIS) is the known precursor condition of cervical adenocarcinoma. The aim of this study is to evaluate treatment choices for AIS in terms of residual disease, recurrence and progression to invasive cancer. Methods: This is a retrospective cohort study conducted at the Mercy Hospital for Women in Melbourne, Australia. Women diagnosed with AIS on histology between 2000 and 2013 were included. Patient records were reviewed for notes on symptoms, cytology, colposcopy and biopsy reports, initial treatment, any subsequent treatment, outcome and follow-up. Results: A total of 114 patients were included: 87 patients were treated with one or more fertility-sparing procedures; 25 patients received a subsequent hysterectomy, one a trachelectomy and one chemo-and radiotherapy. Residual disease was found in 31% (9/29) after a previous procedure with positive margins. In 13.3% (2/15) residual disease was diagnosed despite clear surgical margins. The mean follow-up time was 78.6 months (range 7.9-183). Two patients (2/87, 2.3%) who primarily received fertilitysparing treatment had recurrent AIS. None of the patients progressed to invasive cancer. Conclusion: Both positive and negative margins after initial treatment must be carefully considered in each case. With negative margins, fertility-sparing surgery should be a treatment choice available for all women, not only for those who want to preserve fertility. Fertility-sparing surgery requires regular follow-up.