Fertility-sparing management of atypical polypoid adenomyoma in a premenopausal woman: a two-case report and literature review

Hysteroscopic treatment assisted by photodynamic diagnosis for atypical polypoid adenomyoma: A report of two cases

Atypical polypoid adenomyoma (APA) is a rare uterine premalignant lesion mainly occurring in premenopausal and nulliparous women. Although hysteroscopic resection (HR) has showed promising results, the conservative management of APA in young women is not standardized, and few data are available in the literature. We aimed to assess oncologic outcomes of the conservative treatment of APA. A multicenter observational retrospective cohort study was performed including all patients with APA who underwent conservative treatment from January 2006 to June 2020. Rates of each oncologic outcome (i.e. initial complete response, persistence, progression to cancer, recurrence, long-term treatment success, and treatment failure) were calculated for all conservative treatment together and separately. Twenty-five patients were included. Conservative treatments consisted of HR alone (n=14) and HR+progestin (n=11). Overall, 24 (96%) patients showed initial complete response, of which 21 (84%) showed long-term treatment success; four (16%) patients had progression to cancer, of which two (8%) first recurred as APA. Long-term treatment success was achieved in 13 of 14 (92.9%) patients with HR alone and 8 of 11 (72.3%) with HR+progestin. Conservative treatment appears to be a safe option in women with APA. The four-steps HR might be considered as the first-line conservative approach, while the addition of progestin does not seem to improve oncologic outcomes. However, the risk of progression to cancer highlights the need for a close and long-term follow up with ultrasonography and hysteroscopic biopsies, and for hysterectomy in patients not desiring pregnancy.

Atypical polypoid adenomyoma (APA) is a rare benign tumor frequently diagnosed in young women that may coexist with or progress to atypical endometrial hyperplasia (EAH) or endometrioid endometrial carcinoma (EEC). This study aimed to investigate which subset of patients with APA are prone to concurrent or subsequent EAH or EEC, evaluate the necessity of progestin treatment in patients with APA only after achieving a complete response (CR) through hysteroscopic lesion resection, and assess the impact of concurrent APA on the fertility-preserving treatment of EAH or EEC. This retrospective single-center study analyzed 86 patients with APA treated at the Obstetrics and Gynecology Hospital of Fudan University between January 2010 and October 2021. Patients with EAH or EEC only who underwent fertility-preserving treatment during the same period were matched in a 2:1 ratio with patients with concurrent APA and EAH or EEC. The clinicopathological characteristics, treatments, and prognosis were analyzed. The median patient age was 31 years (range 21-47 years). Among the 86 included patients, nine underwent total hysterectomy, 62 received conservative treatment, and the remaining 15 were lost to follow-up. A comparison of the 16 patients with APA only versus the 58 patients with APA and concurrent or subsequent EAH or EEC revealed that a homeostasis model assessment of insulin resistance (HOMA-IR) of > 2.2 (P = 0.047) and high-density lipoprotein (HDL) concentration of < 1.2 mmol/L (P = 0.028) were independent risk factors for EAH or EEC in patients with APA. Among the 17 patients with APA only who received conservative treatment and achieved a CR after hysteroscopic lesion resection, 13 received hormone treatment for a median duration of 6.3 months. The median follow-up time for these 17 patients was 49.0 months, during which no recurrence of APA was observed, but six patients developed endometrial hyperplastic diseases. Regarding the impact of concurrent APA on fertility-preserving treatment for EAH or EEC, the median time to achieve a CR was 24.0 weeks (95% confidence interval [CI]: 23.0-40.4) in the APA group and 26.0 weeks (95% CI: 24.3-32.3) in the non-APA group (P = 0.424). There were no significant differences between the two groups in the outcomes of fertility-preserving treatment. Patients with APA only may still develop endometrial hyperplastic diseases after complete resection of the lesion under hysteroscopy to achieve a CR, particularly those with a HOMA-IR of > 2.2 or HDL concentration of < 1.2 mmol/L. Concurrent APA did not affect the efficacy of fertility-preserving treatment in patients with EAH or EEC.

An atypical polypoid adenomyoma (APAM) is a benign mixed epithelial and mesenchymal uterine tumor. Patients typically present with hypermenorrhea or abnormal uterine bleeding, and the tumor is most commonly found in nulliparous or infertile women of reproductive age. The natural pathological course of an APAM remains unclear because of the rarity of the disease. The coexistence or sequential development of low-grade malignant endometrial tumors has been reported; however, reports of a mixed carcinoma arising from an APAM have not been published yet. In this report, the authors describe the case of a woman with a mixed carcinoma of the uterus arising from an APAM. This case extends our knowledge of the natural pathological course of an APAM and its etiological relationship with a mixed carcinoma of the uterus, which may contribute to the development of management strategies for an APAM.

Atypical polypoid adenomyoma is an uncommon uterine lesion which can coexist with endometrial atypical hyperplasia and/or cancer. Atypical polypoid adenomyoma affects premenopausal women in most cases, but it shows high recurrence rate if conservatively treated. To date, the management of patients is based on low-quality evidence and is not standardized. Our primary aim was to explore the optimal management of atypical polypoid adenomyoma, with particular regard to the fertility-sparing approach. The secondary aim was to define clinicopathologic features of atypical polypoid adenomyoma. Medline, Embase, Web of Sciences, Scopus, ClinicalTrial.gov, OVID, Google Scholar and Cochrane Library were searched for studies reporting outcomes of atypical polypoid adenomyoma treatments. Univariate comparisons among outcomes of fertility-sparing treatments (rates of initial response, progression, recurrence, final complete response, pregnancy) were performed with Fisher's exact test (α=.05). Eleven retrospective studies with 237 patients were included; 85.5% of patients were premenopausal and 62.9% were nulliparous. Atypical polypoid adenomyoma coexisted with atypical hyperplasia in 5.5% of cases and with endometrial cancer in 5.9%. Overall risks of recurrence and progression to cancer were 28.9% and 16.6%, respectively. Fertility-sparing treatments included hormonal therapy with or without maintenance, hysteroscopic transcervical resection, dilation and curettage, and hormonal therapy combined with transcervical resection or dilation and curettage. Transcervical resection showed significantly higher initial response rates (P from <0.001 to 0.023) than any other treatment. Transcervical resection and transcervical resection+hormonal therapy showed significantly lower progression rates (P<0.001), and higher final complete response rates (P<0.001) than any other treatment. No significant differences were found in the rates of pregnancy (P=0.533 - 0.647) or recurrence (P=0.052 - 0.475). Among the different transcervical resection techniques, the 4-step transcervical resection showed significantly lower rates of progression (P=0.002) and recurrence (P=0.013) than other techniques. Limitations to our results were the retrospective design of the studies and the relatively small sample size, due to the rarity of atypical polypoid adenomyoma. Based on its effectiveness and safety, transcervical resection may be the first-line fertility-sparing treatment for atypical polypoid adenomyoma. In particular, 4-step transcervical resection showed the best results. Given the risk of recurrence, progression and coexistent atypical hyperplasia or cancer, follow-up biopsies are advisable. When fertility preservation is not required, hysterectomy might be advisable.

Objective: To provide a reference for the diagnosis and treatment of atypical polypoid adenomyoma (APA). Methods: This was a retrospective study of 203 APA patients from 2011 to 2021. The clinicopathological characteristics, treatments, and prognosis were analyzed. Results: The average age at diagnosis of APA patients was 39.30 ± 11.01 years, and premenopausal women accounted for 81.3%. Abnormal uterine bleeding or menorrhagia were the most common clinical manifestations of APA. The uterine fundus (78.3%), followed by the lower segment of the uterus (11.8%), was the most common location of the APA lesions. Abnormal blood vessels were seen on the surface of 28 APA tumors. APA can coexist with atypical endometrial hyperplasia (18.2%) and endometrial cancer (10.8%). Immunohistochemical analysis was performed on 99 samples. In the glandular component, ER (94.8%), PR (94.8%), Ki-67 (51.5%), p53 (45.6%), PTEN (18.8%), and mismatch repair proteins (96.4%) were positively expressed. Stromal immunophenotype expression was exhibited as follows: CD10-(89.5%), p16+(86.9%), h-caldesmon-(66.7%), Desmin+(75%), and Vimentin+(88.9%). Fifty-five APA patients received TCR, and 33 of them received adjuvant therapy after the operation. The postoperative recurrence rate (9.1% vs. 36.4%, p < 0.05) and malignant transformation rate (3.0% vs. 18.2%, p < 0.05) of the treated group were significantly lower than the untreated group. Conclusions: APA usually occurs in women of childbearing age, and the diagnosis is based on pathological morphology. APA has a low malignant potential, and those who have fertility requirements can undergo conservative TCR treatment, supplemented by progesterone treatment after surgery and close follow-up. Total hysterectomy is the treatment of choice for APA patients with atypical endometrial hyperplasia around the lesion.

An atypical polypoid adenomyoma (APAM) is classified as a benign, mixed epithelial and mesenchymal tumor. It is a broad-based polypoid tumor that rarely arises in the uterus of sexually mature women. We report a case of a woman diagnosed with an APAM. A 40-year-old nulligravida with a chief complaint of dysfunctional vaginal bleeding was found to have a 2 cm tumor in the uterine lower body by an ultrasound examination. An endometrial biopsy detected an APAM and atypical endometrial hyperplasia (AEH). We performed a hysteroscopic transcervical resection (TCR) as well as a dilation and curettage. Endoscopy revealed a 2 cm broad-based polypoid tumor. We could not identify a clear borderline between the normal muscle layer and the APAM. We resected as much of the tumor as possible. The pathological diagnosis was APAM and AEH. The patient wished to preserve her fertility, therefore we administered medroxyprogesterone therapy for the AEH postoperatively. Two months postoperatively, residual APAM was suspected by endometrial biopsy and MRI, therefore we performed a repeat TCR. We could not clearly identify tumor tissue; however, we resected the suspicious area detected by the MRI. The pathological diagnosis was APAM. No recurrence occurred 10 months postoperatively. Although a fertility-preserving procedure for an APAM has not been established, TCR appears to be the procedure of choice for resection under direct vision.

Fertility-sparing treatment for atypical polypoid adenomyoma.

Five unusual polypoid lesions of the endometrium which we term atypical polypoid adenomyomas are described; these growths occurred in premenopausal women. Each of the polyps was characterized by irregular atypical glands with squamous metaplasia and a cellular, smooth muscle mesenchyme, having a pattern to be distinguished from infiltrating adenocarcinoma or a malignant mixed müllerian tumor. Electron microscopy in one case confirmed the presence of a well-differentiated smooth muscle component. These lesions were focal and noninvasive. Two of the three patients who had hysterectomies has small residual lesions confined to the endometrium. Follow-up from 4 to 24 months showed no recurrence. One patient had a second D & C reported as normal; she subsequently became pregnant. The benign behavior of these unusual polyps suggests that atypical adenomyomas can be managed conservatively in the premenopausal woman without the necessity of a hysterectomy.

MRI and Transvaginal Ultrasound Findings of Atypical Polypoid Adenomyoma: A Case Report

Atypical polypoid adenomyoma (APAM) is a rare uterine tumor that occurs predominantly in premenopausal women. The clinical management of APAMs remains to be established. We report the case of a 46-year-old nulliparous patient who underwent hysteroscopic transcervical resection (TCR) for an atypical polypoid adenomyoma or adenofibroma. As the cervical myoma involved the uterus, the tumor (3.6×2.4 cm) could not be resected completely because of the risk of perforation of the uterus. The patient chose watchful waiting instead of our advice of hysterectomy or further treatment. The residual tumor measured 3.8×1.5 cm after TCR and had grown to 4.8×1.0 cm 6 months later. Furthermore, endometrial biopsy revealed that the APAM had low malignant potential. Subsequently, she underwent total laparoscopic hysterectomy and bilateral salpingo-oophorectomy. The final diagnosis was APAM.

Background: Atypical polypoid adenomyomas (APAMs) are tumors of the uterine cavity. APAMs are difficult to distinguish from endometrial polyps or submucosal myomas. An APAM is composed of atypical endometrial glands surrounded by smooth muscle.Case: A 33-year-old woman had dysmenorrhea and hypermenorrhea. An ultrasound and MRI revealed a 2-cm tumor in the uterine cavity. The histologic diagnosis of a specimen obtained by hysteroscopic resection showed an APAM. After 1 year, an APAM recurred. Multiple polypoid lesions were resected through a hysteroscope.Conclusion: Although difficult to diagnosis; however, a pathologic diagnosis can be made on APAMs following hysteroscopic resection.

Atypical polypoid adenomyoma (APA) of the uterus is a rare neoplasm that predominantly occurs in premenopausal woman. It typically appears as a polypoid mass with a mixture of hyperintensity and hypointensity on T2-weighted MRI. We report a case of a 45-year-old woman with multiple APAs (33 and 22 mm). She underwent hysteroscopic resection. The diagnosis of APAs was histologically made by irregularly proliferating atypical endometrial glands that were admixed with a stroma. This is the first case of multiple APAs with radiological findings. It is important to recognize the possible multiplicity of APA for proper diagnosis.

Management of atypical polypoid adenomyoma (APAM) is complicated because it can sometimes coexist with atypical endometrial hyperplasia (AEH) or endometrioid adenocarcinoma. It is often difficult to assess myometrial invasion in APAM complicated with endometrial cancer. We encountered three patients who, contrary to magnetic resonance imaging, did not have myometrial invasion on hysteroscopic transcervical resection (TCR) and therefore could have fertility preserved, and consequently could become pregnant. We removed the polypoid lesion and a 3-5 mm-thick layer of the normal inner membrane at the root of the polypoid lesion, and then performed total curettage. Several pathological diagnostic procedures were then carried out on each of these resected specimens. Thereafter, high-dose medroxyprogesterone acetate (MPA) was initiated. All three patients underwent hysteroscopic transcervical tumor resection. The pathological diagnoses were as follows: patient 1, G1 endometrioid adenocarcinoma (EMG1) + APAM; patients 2,3, AEH + APAM. No findings of myometrial invasion in the resected root specimen were observed in any patient. In all cases, high-dose MPA was initiated. After the disappearance of tumors, each patient achieved pregnancy. Complications such as placenta accreta were not observed at the time of delivery. In patients with APAM and AEH or EMG1, TCR may aid accurate diagnosis when myometrial invasion is unclear on diagnostic imaging.

Triple assessment of breast is based on clinical examination, histological and radiological study where in our study, the pattern of breast swelling in a premenopausal and postmenopausal woman was compared. A total of 150 female patients with palpable breast lump went through clinical examination, FNAC, USG breast while mammogram &amp; core needle biopsy was done in selected patients. Histopathological examination of the specimen was done for all post-operative patients. Benign &amp; malignant breast lesion accounted for 82(54.6%) &amp; 68(45.3%) respectively. Based on the incidence of benign breast lump &amp; malignant breast lump among pre menopausal women, it was 79% &amp; 21% respectively while in post menopausal women it was 21.9% &amp; 78.1%. The most common type of breast lump among the pre and post menopausal women was fibroadenoma 56.9% &amp; Invasive ductal carcinoma 59.3% respectively. The most common benign breast lump &amp; malignant breast lesion based on the histological pattern among the premenopausal women was fibroadenoma 72% &amp; Invasive ductal carcinoma 50% while in postmenopausal women it was fibroadenosis 71.4% &amp; Invasive Ductal Carcinoma 76%. Based on the hormonal status(ER/PR) of pre and postmenopausal women positivity was seen majorly in post menopausal women 82.3% compared to premenopausal women 17.7%. In our study, the malignant breast disease was more common in post menopausal, while benign breast disease was common in pre menopausal women. Triple assessment in evaluating a breast lump is per se a reliable &amp; valid tool in the armamentarium of treating clinician with very high diagnostic accuracy.