Context: Atypical polypoid adenomyoma (APAM) is a rare biphasic tumor of the uterus that could be confused, both clinically and citologically, with more aggressive entities, expecially with endometrioid adenocarcinoma or adenocarcinoma of the cervix. Usually it is a benign tumor, but there is a 45% risk of recurrence and a small risk (8,8%) of transition to endometrioid adenocarcinoma. So, even if the standard treatment is hysterectomy, APAM could be treated with more conservative therapy. Objective: Herein, we report a case of APAM of the endocervical canal. Patient: A 48- year-old woman executed cervical Pap smear for screening. That examination revealed the presence of atypical glandular cells of undetermined significance (AGUS). Subsequent LEEP together with multiple biopsies was performed and sent to histopathological examination, which gave a result of endometrial polyp with complex atypical hyperplasia and fragments of adenomyoma. Transvaginal ultrasound examination showed a hypoechogenic area, with low vascularization in the cervix. MRI confirmed the presence of this lesion without adding other details. Intervention: Considering the clinical, cytological, and histopathological aspects, the woman was first suspected to have endometrial or endocervical malignancy, and hysterectomy with bilateral salpingo-oophorectomy was performed. Outcome: No further surgical procedures were performed. The patient didnâ€™t have any postoperative complications. Results: Histopathological examination of the surgical specimen documented the presence of a polypoid biphasic (endometrioid and smooth muscle) proliferation, with cytological atypia of the glandular structures, without the presence of endometrial stroma between the two components. Immunohistochemical staining documented the positivity of hormonal receptors (estrogens and progesterone) and negativity of p16, CD10 and CEA. Conclusions: APAM usually occur in premenopausal or perimenopausal women, frequently in the lower uterine segment or in the histmus. APAM, although rare, should be considered in those cases of AGC in which the cytological, clinical and histopathological aspect does not correspond perfectly, expecially in women who wish to retain uterus, just because in these patients this tumor could be treated with more conservative approaches, like a simple polypectomy and subsequent close follow-up, avoiding radical hysterectomy.