Cervical Adenofibroma: A Rare Cause for Postmenopausal Bleeding.

A postmenopausal female in her mid-50s presented to our outpatient department with a complaint of postmenopausal bleeding, two episodes for 2 days each, in the past 5 months, soaked 2–3 pads per day. There was no history of foul-smelling discharge, vaginal itching, or postcoital bleeding. There was no history of weight loss or decrease in appetite. No prior cervical cancer screening was done. No history of any other medical or surgical comorbidities. There was no history of any gynecological, breast, or gastrointestinal malignancy in the family. Menstrual cycles were regular, lasting for 3–4 days, soaking 2–3 pads/day, not associated with dysmenorrhea. She had attained menopause 2 years back and the perimenopausal transition was smooth. She was a para 2 lady with previous 2 vaginal deliveries and bilateral tubal ligation. She was a hemodynamically stable, well-built patient. Per abdomen examination was soft, and non-tender, and no organomegaly was noted. On per-speculum examination, multiple polyps were seen arising from the ectocervix and cervical canal (maximum size 1 cm × 1 cm, total mass 5 cm × 5 cm × 3 cm). The cervix was healthy and the vagina was pale and parched. On per vaginal examination, the uterus was the postmenopausal size, and no forniceal fullness was noted. The polyps were firm in consistency and did not bleed on touch. Routine blood investigations were done. On transvaginal ultrasonography, an endometrial thickness of 6 mm was noted and bilateral adnexa was normal. Cervical cytology, endometrial aspiration, and cervical polypectomy were done. Cervical cytology was suggestive of benign cellular atrophy and histopathology of endometrial aspiration came out to be healthy glandular tissue. Cervical polyps demonstrated mixed benign mesenchymal and epithelial components without any atypia or mitotic figures, which was suggestive of adenofibroma of the cervix. Immunohistochemistry suggested a Ki67 score of 1%. The patient was taken up for total extra-fascial hysterectomy with bilateral salpingo-oophorectomy. The postoperative period was uneventful. The uterine specimen had a smooth outer surface and the cut-surface showed multiple intramural fibroids. The cervical canal showed a residual polyp measuring 0.5 cm × 0.5 cm × 0.5 cm with an irregular surface. Histopathology report demonstrated polyp with papillary architecture with endocervical glands surrounded by fibromatous stroma, showing peri-glandular condensation, without cytologic atypia, and margins were free, suggestive of cervical adenofibroma [ Figures 1 and 2 ]. The patient was followed up for 1 year and had no complaint of bleeding or discharge per vagina. A low-power view of cervical tissue with slit-like spaces; s/o adenofibroma or adenosarcoma High-power view of cervical tissue showing leaf like morphology with hypercellular periglandular stroma, but with no evidence of nuclear atypia or mitotic figures; typical of adenofibroma

Adenofibroma of the cervix is an extremely rare benign Mullerian neoplasm which contains both epithelial and mesenchymal elements. In 2014 WHO classification of the female genital tract, it was classified under mixed epithelial/mesenchymal tumor along with adenocarcinoma, however, it is not a separate entity in the current 2020 WHO classification.[ 1 ] Adenosarcoma is much more common than adenofibroma. There is controversy as to whether adenofibroma exists or not. It is also documented that all adenofibromas are in fact low-grade or well-differentiated adenosarcomas since even in the absence of mitotic activity, cases may recur or rarely even metastasize, therefore, a confident diagnosis of adenofibroma cannot be made.[ 2 ] They generally arise from the endometrial cavity, though can arise from the cervix or extra-uterine location.[ 3 ]

Cervical adenofibroma is mostly seen in peri- or postmenopausal women, although occurrence in a younger age group has been reported.[ 3 4 5 ] Less than 10 cases of cervical adenofibroma have been reported till now.[ 3 4 5 6 7 8 9 10 11 ] Most of the adenofibroma arise from uterine endometrium. They present as abnormal uterine bleeding or postmenopausal bleeding from polypoidal mass arising from the uterus/cervix or even extra uterine location. The biggest cohort was studied by Gallardo and Prat where immunohistochemical analyses of 55 cases of uterine adenosarcoma in comparison with benign cases, such as adenofibroma, endometrial polyps, and endometriosis.[ 12 ] There were no typical clinical or imaging characteristics that strongly suggested the diagnosis. Two patients had low-grade adenosarcoma and both were initially underdiagnosed as adenofibroma.[ 12 ] Adenosarcoma and adenofibroma share similar histology but differ mainly in the frequency of mitotic figures found in stroma. Adenosarcoma is diagnosed if the stroma shows atypia, increased peri glandular stromal cells, invasiveness, and mitotic figures ≥4/10 high-power fields.[ 3 ] Immunohistochemistry markers Ki-67, p53, and loss of CD 10 help in differentiating adenosarcomas with sarcomatous overgrowth from adenofibroma.[ 12 ] The exact etiology of cervical adenofibroma remains unclear, some believe that it represents endometriosis with extreme smooth muscle metaplasia.[ 11 ] Whereas others believe it to arise from Müllerian tissues.[ 12 ] It is important to pathologically differentiate these tumors from adenomyomas, adenosarcomas, or carcinosarcomas. Given these overlapping features, accurate diagnosis requires histopathological confirmation, supplemented by immunohistochemical analysis when necessary. A total hysterectomy is a viable option and ensures complete tumor removal. A confident diagnosis of adenofibroma cannot be made on a biopsy or polypectomy specimen because adenosarcoma cannot be excluded unless the whole tumor is available for histological examination.[ 2 ] However, for young women who wish to preserve their fertility, extensive local resection can serve as an alternative to hysterectomy, as long as the integrity of the resection is confirmed and long-term follow-up is feasible.[ 3 ] Given its benign nature, the prognosis is excellent, with no reported cases of malignant transformation or metastasis in the literature. However, a long-term follow-up is advisable to ensure complete excision and to monitor for any residual or recurrent disease.[ 3 4 11 ] While adenofibroma of the cervix is an uncommon and benign entity, its potential to mimic malignancy poses diagnostic challenges. Clinicians and pathologists must maintain awareness of this lesion to prevent overtreatment as well as undertreatment. Future research into its pathogenesis and molecular characteristics may further elucidate its biological behavior and improve diagnostic accuracy. 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 initials will not be published and due efforts will be made to conceal identity, but anonymity cannot be guaranteed. There are no conflicts of interest.