BACKGROUND: The presence of human papilloma virus (HPV) 33 and possible myoepithelial differentiation in an adenoid cystic carcinoma of the cervix (ACC) Ib1 and the clinical outcome are reported. CASE: The case is an ACC, stage Ib1 harboring HPV 33, which was treated by radical hysterectomy and lymphadenectomy only. Human papilloma virus analysis of the tumor was done by means of nonisotopic in situ hybridization and polymerase chain reaction. Additionally immunoelectron microscopy was performed. CONCLUSION: Our patient had a vault recurrence 12 months after class III (Meigs) hysterectomy. The cancer tissue was totally excised, the lymphnodes (n = 31) were negative, and there was no lympho-vascular invasion and no perineural infiltration. The primary tumor displayed HPV 33 integration and features suggesting myoepithelial differentiation. We would recommend a low threshold for the use of radiotherapy in the management of similar findings.