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Mesonephric-like adenocarcinoma (MLA) of the ovary is a recently recognized, rare malignancy with aggressive clinical behavior, and is thought to originate from Mullerian epithelium with mesonephric transdifferentiation. Emerging evidence suggests that MLA may be classified as an endometriosis-associated neoplasm. The presence of a sarcomatous component within MLA is extremely rare, with common differential diagnoses including the spindle cell component of MLA, carcinosarcoma, as well as mixed Mullerian adenocarcinoma and adenosarcoma. Herein, we report a 58-year-old Chinese woman with bilateral ovarian solid-cystic masses. The left ovarian mass comprised a biphasic tumor with a predominantly high-grade sarcomatous component displaying heterologous mesenchymal differentiation, including liposarcoma, rhabdomyosarcoma and chondrosarcoma-like areas, with a null-type p53 expression. The epithelial component ranged from a bland appearance in areas diagnostic of adenosarcoma to a clearly invasive carcinoma, both with mesonephric-like phenotype, being negative for estrogen receptor, progesterone receptor, and Wilms' tumor 1, variably positive for paired box gene 8, GATA binding protein 3, and thyroid transcription factor 1, with a wild-type p53 expression. The differing p53 expression between the epithelial and sarcomatous elements mitigated against a diagnosis of carcinosarcoma. The right ovarian mass showed endometriosis with focal direct evidence of the development of malignancy within a benign endometriotic cyst, exhibiting the identical immunoprofile of MLA but originating as another malignancy. To the best of our knowledge, this case represents the first reported case of synchronous bilateral ovarian MLAs with separate origins, from high-grade Mullerian adenosarcoma and endometriosis respectively, which broadens the morphologic spectrum of MLA and provides further evidence supporting the Mullerian origin theory., Competing Interests: The authors declare no conflict of interest., (Copyright © 2023 by the International Society of Gynecological Pathologists.)

This was a 57-year-old woman who presented with mild discomfort in the right groin. Physical examination revealed a mass in the right groin, and by ultrasound, the mass was hypoechoic and solid with some internal vascularity. The clinical differential diagnosis included lymphoma and others. The mass was excised for pathologic evaluation. Gross examination of the specimen revealed a 3 × 2.4 × 2 cm, solid and cystic mass. Microscopically, it was a biphasic tumor consisting of carcinomatous and sarcomatous components. The tumor was seen contiguous with endometriosis and atypical endometrioid hyperplasia. The histologic findings were consistent with malignant mixed Mullerian tumor (MMMT) arising from endometriosis in the right groin. The tumor involved the resection margin. Subsequent chest/abdominal/pelvic computed tomography did not reveal evidence of tumors, and diagnostic peritoneal/pelvic laparoscopy did not show diseases. Postoperatively, the patient received 6 cycles of chemotherapy consisting of carboplatin and paclitaxel, followed by radiation in the right groin. Malignant transformation from endometriosis occurs in less than 1% of endometriosis cases, and about 80% of the transformed tumors occur in the ovaries. The most commonly transformed malignant tumors are endometrioid and clear cell carcinomas, with rare adenosarcoma and endometrial stromal sarcoma reported. To our knowledge, we are reporting the first case of MMMT arising from endometriosis in the groin.

Background: Müllerian adenosarcoma is a rare malignancy. These tumors occur mainly in the uterus, but also in extrauterine locations, usually related to endometriosis. Because of their rarity, there is limited data on optimal management strategies., Case Presentation: We present a 44-year-old woman with a history of endometriosis who consults for chronic pelvic pain. In the imaging tests, a heterogeneous mass is observed that impresses endometriosis, encompassing the uterus and left appendage. Surgery is performed by finding an extrauterine adenosarcoma that affected the uterus, ovary and bladder wall., Conclusion: This is a rare case but should be considered in a patient with atypical clinical characteristics or preoperative pathology, so we show the diagnostic and therapeutic strategies carried out for the resolution of the case.

We report a case of a primary vaginal adenosarcoma with sarcomatous overgrowth in a postmenopausal 58-year-old woman with recurrent endometriosis. In the past 5 years she underwent several biopsies of a polypoid lesion on the vaginal cuff, and the last histologic examination of the biopsy showed an adenosarcoma with "sarcomatous overgrowth" in a background of endometriosis. There was no evidence of distant metastatic disease on the diagnostic workup, and we performed a laparoscopy to remove the pelvic mass. We reviewed the literature on the electronic databases Medline, Embase, and Science Direct on articles published in English from 1990 to 2015. We identified 5 articles in which the surgical treatment was performed via a laparotomic approach. The present case is the first in the literature to report feasibility of laparoscopic treatment for this kind of pathology with a detailed description of the surgical technique., (Copyright © 2016 AAGL. Published by Elsevier Inc. All rights reserved.)

Reported here is a case of Müllerian adenosarcoma of the ovary which contained a primitive neuroectodermal tissue component within the stroma. The adenosarcoma coexisted with clear cell adenocarcinoma in an endometriotic cyst. The patient was a 33-year-old woman with a large unilocular endometriotic cyst of the right ovary. On the internal wall of the cyst, both a plaque-like protrusion with a papillary surface and a dome-like mural nodule were noted. The former exhibited features of Müllerian adenosarcoma, and the latter showed those of clear cell adenocarcinoma. In the deeper portion of adenosarcoma, teratoma-like tissue which contained various tissue components including primitive neuroectodermal tissue was found. The presence of primitive neuroectodermal tissue in the stroma of adenosarcoma suggested the diagnosis of 'adenosarcoma with neuroectodermal differentiation' ('teratoid adenosarcoma'), although the possibility of the incidental occurrence of an immature teratoma could not be completely excluded., (© 2011 The Authors. Pathology International © 2011 Japanese Society of Pathology and Blackwell Publishing Asia Pty Ltd.)

Objective: To explore the role of long-standing hormone replacement therapy (HRT) in the malignant transformation of endometriosis., Design: Short case series. Three cases of women with pelvic clearance receiving long-standing HRT studied in detail., Setting: Teaching hospital in the United Kingdom (Gynaecological Cancer Centre) (Institutional Review Board approval was not obtained as it was not deemed necessary, this being a case series)., Patient(s): Women with a history of pelvic clearance for endometriosis and longstanding HRT., Intervention(s): HRT., Main Outcome Measure(s): Malignant transformation of endometriosis., Result(s): Long-standing HRT in all three women with pelvic clearance for endometriosis resulted in malignant transformation of residual endometriosis many years after the initial surgery. All cases presented with a new pelvic lesion., Conclusion(s): The diagnosis of malignant transformation needs to be considered in women with a history of endometriosis and with long-term HRT use in whom a new pelvic lesion is detected. The risk of malignant transformation in women with endometriosis after pelvic clearance receiving HRT needs to be explored further. Surveillance with CA-125 and imaging in such cases to predict recurrence or malignant transformation needs to be studied further in a research setting.

Objective: To investigate the clinicopathologic features of atypical endometriosis (AEM) and to discuss the relationship between AEM and tumors., Methods: A retrospective analysis was performed of 163 cases of AEM, which were retrieved from the Department of Pathology files at the Tianjin Central Obstetrics and Gynecology Hospital between Jan 2004 and Dec 2006. The pathologic changes of AEM including its glandular epithelium, stroma, background and the conditions coexisting with tumor were observed., Results: The AEM accounted for 4.38% (163/3724) of the endometriosis (EM) cases. Of 172 AEM foci of 163 patients, 168 were in the ovary, and the other 4 were in the fallopian tube, cervix and uterine serosa respectively. The rate of ovarian AEM was 6.81% of endometriosis. AEM associated with tumour was found in 26 cases (15.95%) and among 27 of ovarian AEM, 15 were malignant, 9 borderline and 3 benign. The AEM epithelia were mainly arranged in the form of surface epithelium. They present with characteristic features of moderate to marked pleomorphism, epithelial tufting and bud structures by microscopy. The walls of AEM cyst were presented with three layers of epithelium, endometrioid stroma and fibrosis-collagen. The endometrioid stroma were usually thin in contrast to the fibro-collagen tissue, which was often thick with scarred background. The transformation from AEM to tumor was found in most of the malignant tumors (14/15, 93%)., Conclusions: AEM lesions hold some features of both EM and tumor, which may have a relatively higher potential for tumorigenesis and canceration. The process of damage and repair in EM foci during a long course may play a role in the development of EM into AEM and finally into tumor.

A case of müllerian adenosarcoma arising in ovarian endometriosis is reported in which the whole of the mesenchymal component exhibited striking deciduoid morphology, a phenomenon that has not been previously described. The patient was not taking hormonal preparations. We discuss the differential diagnosis and the possible pathogenesis of the deciduoid stromal alteration.

Extrauterine mullerian adenosarcoma is rare and is thought to arise from endometriotic deposits or pluripotent mesothelial and mesenchymal cells of the pelvic cavity. Concomitant colon-rectal neoplasm was reported but the relationship between extrauterine adenosarcoma and colon tumor was not studied. We describe an extrauterine adenosarcoma with a concomitant rectal tubulovillous adenoma. The patient had a long-term history of endometriosis and unopposed estrogen therapy. Immunohistochemical study was performed to investigate the origin and nature of the adenosarcoma. Immunostaining provided the evidence to distinguish between rectal tumor and adenosarcoma. Extrauterine adenosarcoma may arise from endometriosis, and hormone replacement therapy may have some role in the malignant transformation process. Adenosarcoma should be considered in the differential diagnosis of a new pelvic tumor in a patient with a history of endometriosis.

We present a case of a liver endometrioma in a postmenopausal woman. After failed management with leuprolide acetate, the mass was resected and contained focal areas of mullerian adenosarcoma. This is a rare case of mullerian adenosarcoma that appeared to arise within an endometrioma of the liver.

Objective: To present a rare case of endometrial stromal sarcoma arising in endometriosis of the rectovaginal septum., Design: Case report., Setting: Academic tertiary referral center for endometriosis treatment., Patient(s): A 50-year-old woman with a history of endometriosis presented with catamenial rectal pain and deep dyspareunia. Imaging findings suggested new endometriotic lesions in the rectovaginal space., Intervention(s): Total hysterectomy, salpingo-oophorectomy, and excision of the lesion in the rectovaginal septum were performed. Although extemporary pathology confirmed endometriosis, the final histologic diagnosis was extrauterine adenosarcoma in the rectovaginal septum. Two years later, recurrence of the malignancy occurred and was treated by resecting the new perirectal mass. Subsequent radiotherapy and chemotherapy were administered., Main Outcome Measure(s): Imaging findings at follow-up evaluation., Result(s): The patient was in good health for 2 years after the initial surgery, when she developed a new lesion at the site of the previous resection. The histologic appearance of the lesion was consistent with recurrence of the tumor. After postoperative therapy, the patient is now without evidence of disease., Conclusion(s): Malignant transformation of endometriosis should be considered in the differential diagnosis of a new pelvic lesion in a patient with a history of endometriosis.

During the last 15-year period in our laboratory, 14 cases of malignant change of endometriosis were diagnosed in women 37-70 years of age. Seven cases were adenocarcinomas, one case was an adenosarcoma and six cases presented the morphology of endometrial stromal sarcoma. Eleven cases developed in various sites of the genital system (5/11 in the ovaries, 3/11 in the uterus 2/11 in the vagina, 1/11 in the tubal wall) and three cases in extragenital sites (1/3 in the omentum, 1/3 in the pelvis, 1/3 in the colon). Immunohistochemical investigation for the expression of cytokeratines, vimentin, hormonal receptors, E-cadherin, desmine and smooth muscle actin was performed by a streptavidin-biotin method. The immunopathology profile and follow-up results after therapy are presented.

Background: Primary adenosarcoma arising in vaginal endometriosis poses a diagnostic challenge, especially in superficial vaginal biopsies., Case: A 56-year-old woman, with a prior diagnosis of ovarian endometriosis, presented with a rapidly enlarging mass of the vaginal vault. Two prior biopsies were benign and showed endometriosis. The third vaginal biopsy revealed benign endometriotic glands cuffed by a cellular stroma with moderate cytologic atypia, a histological appearance diagnostic of Müllerian adenosarcoma. A 16-cm vaginal mass that had infiltrated the pelvic structures was resected., Conclusions: Close clinical follow-up of extrauterine endometriosis and clinical-pathologic correlation is necessary. Histological features such as cellular stromal cuffing around benign endometriotic glands are critical in arriving at a timely diagnosis of adenosarcoma in patients with persistent extrauterine endometriosis, even in superficial vaginal biopsies.

Objective: To report laparoscopic management of 15 patients with infiltrative bladder wall endometriosis and to report a case of endometrioid adenosarcoma., Design: Prospective chart review., Setting: Referral center for endometriosis., Patient(s): Fifteen women with infiltrating endometriosis of the bladder., Intervention(s): Laparoscopic segmental cystectomy and pathologic review of endometriotic bladder nodules in 15 patients., Main Outcome Measure(s): Location and characteristics of endometriotic bladder nodules., Result(s): Laparoscopic and cystoscopic evaluation confirmed that the endometriotic lesions were penetrating through the bladder wall. In 8 patients, the lesions were located in the dome of the bladder. In the remaining 7, the lesions were in the posterior wall, above the trigone. It was possible to treat all the lesions by performing a laparoscopic partial cystectomy. No intraoperative complications occurred. Deeply infiltrating endometriosis was confirmed on histologic evaluation in 14 cases. One patient was diagnosed with endometriosis on frozen section, but the final pathology revealed an adenosarcoma of the bladder., Conclusion(s): Surgical excision of deeply infiltrating endometriosis of the bladder wall can be performed laparoscopically and offers the benefit of a definitive pathologic diagnosis to rule out an occult malignancy.

We report a primary peritoneal mullerian adenosarcoma with sarcomatous overgrowth associated with endometriosis in a 50-year-old female. The tumor formed multiple peritoneal masses occupying the pelvis and subdiaphragmatic space. Histologically, the tumor was composed of benign-appearing mullerian glands surrounded by a sarcomatous stroma. In addition, one perisplenic mass was largely devoid of the epithelial component and was of higher grade than the remaining lesions. Multiple foci of endometriosis were associated with the pelvic masses. To our knowledge, primary peritoneal mullerian adenosarcoma with sarcomatous overgrowth associated with endometriosis has not been previously reported.

The clinicopathologic features of neoplasms arising in gastrointestinal endometriosis have not been well characterized. In this series, we report 17 cases of gastrointestinal endometriosis complicated by neoplasms (14 cases) or precancerous changes (three cases). Four patients, one of whom also had hypermenorrhea, presented with chronic abdominal pain and five had obstructive symptoms; one of these also had rectal bleeding. One patient presented with an acute abdomen and fecal peritonitis, one had vaginal bleeding, and one had a progressive change in bowel habits. Nine patients had a long history of endometriosis, 11 patients had had hysterectomies, and eight of these had also received unopposed estrogen therapy. The lesions involved the rectum (6), sigmoid (6), colon, unspecified (2), and small intestine (3), and comprised 8 endometrioid adenocarcinomas (EA), 4 mullerian adenosarcomas (MAS), 1 endometrioid stromal sarcoma (ESS), 1 endometrioid adenofibroma of borderline malignancy (EBA) with carcinoma in situ, 2 atypical hyperplasias (AH), and one endometrioid adenocarcinoma in situ (ACIS). The tumors ranged in size from 2 to 15 cm and all involved the serosa and muscularis propria. Two tumors extended into the mucosa, with mucosal ulceration in one. Follow-up was available in 11 cases. One patient with EA was dead of disease at 1 year, one had two recurrences at 1 and 2 years, and three were alive with no evidence of disease (ANED) at 9 months to 13 years (mean, 68 mos). The patient with the EBA was ANED at 3 months. Two patients with MAS were ANED at 2 and 3 years. The patient with ESS had a recurrence at 3 years and was ANED 6 years after her original diagnosis. One woman with AH was ANED at 60 months and the patient with ACIS was ANED at 16 months. One of the carcinomas was originally misdiagnosed as a primary intestinal adenocarcinoma. The pathologist should be aware of the possibility of a tumor of genital tract type when evaluating intestinal neoplasms in females, particularly if they have a history of endometriosis and have received unopposed estrogen therapy.

This clinicopathologic study of primary Mullerian tumors of the bowel arising in foci of endometriosis is based on six new cases and an analysis of 17 previously reported cases. Varieties of Mullerian tumors occur in the bowel; the most common types are endometrioid carcinoma, followed by various mixed Mullerian tumors and stromal sarcomas. Seventy-eight percent develop in the rectosigmoid colon, the remaining in the cecum or ileum. Those in the latter area tend to be sarcomas or mixed Mullerian tumors. Certain architectural growth characteristics, derived from precursor endometriosis, are common to most endometriosis-associated intestinal tumors (EAITs). Seventy percent of EAITs occur in the outer bowel wall. Transmural tumors tend to form luminal polyps and assume an hourglass shape. Metachronous or synchronous Mullerian tumors occur in 39% of cases. Seventy percent of women with EAITs are in their mid 30s to early 50s. Common presenting symptoms are abdominal or pelvic pain, melena, and an abdominal or pelvic mass. Documented in 26% of patients is a history of prolonged unopposed estrogen therapy. Only 28.5% of cases die of their tumors, but follow-up is less than 5 years in all but 2 patients.

Objective: Malignant transformation of endometriosis has been well documented. Endometrioid adenocarcinoma is the most common malignancy to occur in this setting, although other carcinomas and rarely stromal tumors can be seen. We present the first case in the literature of adenosarcoma, a rare mixed mullerian or mesodermal tumor, arising in extrauterine vaginal endometriosis., Case: A 42-year-old woman underwent multiple medical therapies and surgeries for aggressive endometriosis. A pelvic exenteration was abandoned secondary to severe fibrosis, and low-dose radiotherapy was used to control bleeding from vaginal endometriosis. The pathologic diagnosis of recurrent endometriosis was confirmed multiple times over her 4-year course. Excision of a recurrent vaginal mass revealed adenosarcoma with heterologous elements., Conclusion: It is important to biopsy or excise recurrent endometriosis, as malignant transformation can occur, giving rise to epithelial, stromal, or mixed epithelial-mesenchymal tumors., (Copyright 2000 Academic Press.)

We report a case of adenosarcoma arising in hepatic endometriosis. Both CT and MR scans demonstrated a huge heterogeneous mass containing septated, thick-walled cystic lesions. After enlarged right hepatectomy, the patient was asymptomatic with no abnormalities at liver and abdominal CT scan at 2-year follow-up.