Silent Intruders: A Case Report of Concurrent Atypical Endometrial Polyp with Cervical Intraepithelial Neoplasia

A 53-year-old multiparous, postmenopausal female of Asian ethnicity, belonging to lower socioeconomic status, presented to the gynecological outpatient department at our center with complaints of per vaginal bleeding on and off for 2 months. She had attained menopause 2 years ago. She was a nonsmoker, nonalcoholic, reported to have a single sexual partner. The bleeding was controlled initially on over-the-counter medications, but later, she experienced recurrent, heavier bouts of bleeding in the last 7 days. Previously, her menstrual cycles were occasionally heavier around the time of menopause. However, she did not undergo any formal evaluation. The patient did not report any medical or surgical illnesses. There was no history of malignancy in the family members. On examination, she had a mild degree of anemia. Her body mass index was 23.9 kg/m 2 . The abdominal examination did not reveal any abnormality. On bimanual examination, an 8 cm × 7 cm cervical fibroid could be felt through the external os. The uterine size on per vaginal examination was corresponding to 14 weeks of gestation. Adnexa was difficult to palpate due to forniceal obliteration. A 2 dimensional-transvaginal ultrasound pelvis followed by a 3 tesla magnetic resonance imaging (MRI) was done [ Figure 1 ]. MRI images described the presence of cervical fibroid (FIGO-VIII) arising from the posterior lip of the cervix and focal adenomyosis in the fundal myometrium with no diffusion restriction with endometrial thickness 8 mm (mixed with blood clots). All the blood investigations were within normal limits. Cervical cytology was negative for intraepithelial lesion/malignancy. An outpatient endometrial sampling with pipelles could not yield an adequate sample for interpretation. (a) Three Tesla-magnetic resonance imaging pelvis showing a well-defined T1, T2 isotense lesion 7.7 cm × 6.5 cm × 6.6 cm arising from the posterior lip of the cervix (marked in **), an intramural fundal fibroid (marked in *), blood products (marked with arrow), (b) shows endomyometrial junction well maintained, (c) enhancing lesion under the fibroid possible adenomyotic changes (marked with arrow) The patient underwent a total abdominal hysterectomy (TAH) and bilateral salpingo-oophorectomy (BSO). Intraoperative findings – the specimen (uterus + cervix) measured 12 cm × 6 cm × 5 cm. The cut section showed an isolated, sessile endometrial polyp, and the endometrium appeared atrophic [ Figure 2 ]. Cervical fibroid 8 cm × 7 cm was seen in the endocervical canal and was found attached to the anterior cervical canal. Final histopathological examination revealed AEH/EIN confined to endometrial polyp and background of atrophic endometrium, as noted in Figure 3 . In addition, histology of the cervix revealed CIN Grade-1. The postoperative recovery of the patient was uneventful. The patient was counseled regarding regular follow-up. (a) Total abdominal hysterectomy + bilateral salpingo-oophorectomy specimen showing cervical fibroid (separated), (b) Gross finding of the cut-open specimen showing a sessile polyp near posterior fundic region Histopathology showing atypical endometrial hyperplasia/endometrioid intraepithelial neoplasia, (a) Low power view of the endometrial polyp (inset) with closely packed glands and absent intervening stroma (×100; H and E stain), (b) The atypical glands are arranged back to back with nuclear stratification (×400; H and E stain)

Endometrial polyps are one of the most common causes of postmenopausal bleeding that need further evaluation. Majority of these polyps run a benign etiology, but malignant transformation occurs infrequently, increasing with age. Atypical endometrial polyps tend to appear in the background of endometrial hyperplasia and in women with underlying risk factors.[ 1 ] Only a few reports of endometrial malignancy confined to polyp have been reported with background atrophic endometrium.[ 2 3 ] Moreover, a co-existence in terms of Atypical endometrial polyp atypical endometrial hyperplasia/Endometrioid intraepithelial neoplasia (AEH/EIN) with cervical intraepithelial neoplasia (CIN) in a background of atrophic endometrium has never been described. We present a unique case of such an association underlying a large cervical fibroid in a 53-year-old presenting with postmenopausal bleeding.

To date, there are cases reporting concomitant occurrence of atypical endometrial changes confined to polyp in the background of endometrial hyperplasia/endometrial cancer (EC).[ 4 5 ] To the best of our knowledge, AEH/EIN entity confined to the polyp in the background of atrophic endometrium concurrent with CIN, as in our case, has never been reported. Rarely, a hyperplasia, either complex or atypical in type, is identified within a polyp in a biopsy or polypectomy specimen.[ 1 ] The diagnosis of AEH/EIN in patients without the risk factors, including obesity, unopposed estrogen therapy, diabetes, and nulliparity, needs careful consideration. The patient described did not have any underlying risk factors and neither any obvious radiological imaging findings suggesting premalignant changes. As per the recent EIN guidelines, the diagnosis can be established using routinely stained (hematoxylin and eosin) sections and a standard light microscope. Lesion dimension of a minimum of 1 mm, crowding of the glands that exceeds the area of stroma, and the changes in cytology relative to background after exclusion of benign polyps, secretory endometrium and cancers confirm the AEH/EIN diagnosis. In our case, AEH/EIN (size 2 mm) was arising in the endometrial polyp in an atrophic endometrium. An evident, tortuous, superficial vascularization can be indicative of atypical features in the endometrial polyp apart from glandular architectural abnormalities.[ 5 ] Expectantly, AEH, being premalignant, carries a substantial risk of progression to endometrioid EC. A TAH and BSO is the definitive treatment for AEH/EIN.[ 6 ] Pipelle’s biopsy shows a remarkable sensitivity and specificity in the diagnosis of patients with abnormal uterine bleeding but is less useful in the background of atrophic endometrium.[ 7 8 ] Hysteroscopic evaluation is preferable to pipelle biopsy in women with postmenopausal bleeding. In our case, owing to the size and location of the cervical fibroid, hysteroscopy was not feasible for an inadequate Pipelle’s biopsy sample. “Insufficient” sample on outpatient endometrial sampling should be dealt with caution.[ 9 ] A recent study by Geels et al . in 2012[ 3 ] concluded that endometrial carcinoma may arise in an atrophic endometrium through unique pathways but may not follow the hypothesized progression model for type I tumors. Prabhakaran et al ., in 2022, reported a diagnosis of endometrial carcinoma confined to a polyp in a postmenopausal female with atrophic endometrium.[ 2 ] This was like our case. However, there were no concurrent CIN-1 changes. It is well known that pap smear to evaluate cervical pathology has a low sensitivity 30%–87%.[ 10 ] Diagnosing CIN-1 in hysterectomy specimens is not an uncommon sequence. In our case, the pap smear though was negative for intraepithelial lesion/malignancy reported CIN-1 in microscopic sections. However, the co-existence of AEH/EIN with background atrophic endometrium and CIN-1 is a first of its kind.

Routine screening for cervical cancer, although essential, is not commonly employed especially before planning a major surgery. This case reiterates the fact that pap smear evaluation is mandatory in all cases. Furthermore, a histopathological evaluation suggesting lesser degrees of CIN must always be reported, as this could contribute to the actual prevalence of this condition. Although a postoperative diagnosis, this incidental histopathological finding deserves reporting to highlight the significance of uncommon “silent intruders.” The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient has given her consent for her images and other clinical information to be reported in the journal. The patient understand that name and initial will not be published and due efforts will be made to conceal identity, but anonymity cannot be guaranteed. There are no conflicts of interest.