Extragenital adenosarcoma: A case report, review of the literature, and management discussion

Huang, Gloria S.; Arend, Rebecca C.; Sakaris, Antoinette; Hébert, Tiffany; Goldberg, Gary L.

doi:10.1016/j.ygyno.2009.07.033

Cited by 26 publications

(34 citation statements)

References 23 publications

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“…14 Alternatives include a combination of ifosfamide and cisplatin, or gemcitabine with docetaxel. 7 Adjuvant therapy with aromatase inhibitors has also been described. 15 Adjuvant radiotherapy may be included as part of a combined regimen if there is an increased recurrence risk, although there is a paucity of evidence for this.…”

Section: Discussionmentioning

confidence: 99%

“…This aggressive behavior may be explained by the tumor's free access to the peritoneal cavity, with transcoelomic spread not hampered by the myometrium, compared to an uterine adenosarcoma. 7 A poorer prognosis may also be attributable to the increased risk of local complications at extrauterine sites, such as bowel obstruction; later presentation and, therefore, more advanced disease at diagnosis; and greater difficulty in achieving complete surgical clearance. 6,17 Second, the absence of underlying endometriosis confers a worse prognosis.…”

Section: Discussionmentioning

confidence: 99%

“…Extrauterine locations are less common and include the ovary, cervix, vagina, adnexae, pelvic peritoneum, rectovaginal septum, and intestinal serosa. [3][4][5][6][7] This article reports 2 cases of extrauterine Müllerian adenosarcoma arising from pelvic endometriosis, and discusses the clinical and histopathologic features of this exceedingly rare condition.…”

Section: Introductionmentioning

confidence: 99%

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Two Cases of Extrauterine Müllerian Adenosarcoma Arising from Pelvic Endometriosis

Yuan

Ching

Kuei

et al. 2019

Journal of Gynecologic Surgery

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Background: Adenosarcoma arises most commonly in the uterus. Rarely, it may arise from extrauterine sites, such as the pelvic peritoneum and rectovaginal septum. Two cases of extrauterine Müllerian adenosarcoma arising from pelvic endometriosis are discussed. Cases and Results: Case 1: A 63-year-old woman presented with vaginal bleeding and a rectovaginal nodule. She underwent an extended hysterectomy with resection of a rectovaginal mass. Histology revealed endometriotic tissue. She returned 21 months later with abdominal distension, and underwent resection of large pelvic abdominal masses. Histology showed an adenosarcoma with sarcomatous overgrowth. Her disease progressed despite chemotherapy, and she died 5 months later. Case 2: A 70-year-old woman with previous breast cancer presented with urinary retention. Computed tomography showed a 13-cm pelvic mass. She underwent total abdominal hysterectomy and salpingo-oophorectomy. Intraoperatively, dense pelvic endometriosis was seen. Histology showed a stage 1C borderline endometrioid adenofibromatous tumor. She also underwent resection of a 10-cm incidentally discovered pelvic mass 18 months later. Histology showed a low-grade adenosarcoma arising from pelvic endometriosis. She was started on letrozole, and remained well postoperatively. Conclusions: Compared to uterine adenosarcoma, an extrauterine location confers a higher mortality and recurrence risk. Higher stromal grade or sarcomatous overgrowth worsen the prognosis, whereas an association with endometriosis is protective. The mainstay of treatment is surgery. Some surgeons recommend fertilitysparing surgery when there is no sarcomatous overgrowth, while others advocate radical surgery with adjuvant chemotherapy or radiotherapy. Further research is needed to define screening, treatment, and follow-up better. Clinicians should consider this rare diagnosis in any woman presenting with a pelvic mass with a history of endometriosis. (J GYNECOL SURG 35:194

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Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

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Two Cases of Extrauterine Müllerian Adenosarcoma Arising from Pelvic Endometriosis

Yuan

Ching

Kuei

et al. 2019

Journal of Gynecologic Surgery

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“…Haematogenous metastases occur in 33% of extragenital cases compared with 2% of uterine primaries. Mortality rates are 40% and 10% in extrauterine and (8) uterine adenosarcomas, respectively (8)(9)(10). The reasons for the poorer prognosis of extragenital adenosarcoma have not been explored yet.…”

Section: Discussionmentioning

confidence: 99%

“…In their review, Huang et al (10) suggested that the presence of endometriosis might confer a better prognosis in patients with extragenital adenosarcoma. Extragenital adenosarcoma may develop from malignant transformation of endometriosis or originate from multipotential pelvic mesothelial cells (7).…”

Section: Discussionmentioning

confidence: 99%

Extragenital Müllerian Adenosarcoma in the Pouch of Douglas

Karateke¹,

Kahramanoğlu²,

Bilgiç³

2014

Balkan Med J

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Müllerian adenosarcoma is a mixed epithelial-mesenchymal neoplasm that originates from the Müllerian duct and is characterised by benign epithelial gland and malignant stromal components as active participants in the neoplastic process (1). The uterine corpus is the most common primary site but Müllerian adenosarcoma has been reported to arise in the ovary, cervix, vagina, pelvic peritoneum, pouch of Douglas, broad ligament, bladder, and colon (2-5). Although adenosarcomas are generally low-grade malignancies, adenosarcoma with sarcomatous overgrowth is a highly aggressive tumour. These tumours are frequently associated with postoperative recurrence or metastases and a fatal outcome, even in earlystage disease (6).We present a case of Müllerian adenosarcoma located in the pouch of Douglas and report the clinical and pathological findings. This is the second case reported in the literature that was treated by total excision of the tumour, and no recurrence was seen during the subsequent 24-month period. CASE PRESENTATIONA 26-year-old, gravida 0, para 0 woman with lower abdominal distension and left lower quadrant pain was seen in our hospital. She had no significant medical or family history. Her menstruation cycles were regular and she had used no medications. According to her history, she had no sexual relationship of any kind. Physical examination revealed a large, palpable mass in both lower quadrants up to the level of the umbilicus, with associated tenderness. Laboratory studies showed a minimally elevated cancer antigen 125 (CA-125) level of 36.2 U/ mL. All other laboratory tests, including carcinogenic antigen (CA 19-9), carcinoembriogenic antigen (CEA), and alphafetoprotein (AFP) were within normal limits.Transabdominal ultrasonography revealed a diffuse, free pelvic fluid and a complex adnexal multiloculated cystic mass measuring 10x8 cm, with solid areas and internal echoes.One day later, computed tomography (CT) of the chest, abdomen, and pelvis demonstrated a 23x11 cm, solid/cystic mass arising in the left ovary with mural nodularity, which extended from the pelvis in the midline towards the umbilicus.Magnetic resonance imaging (MRI) of the abdomen and pelvis was performed 3 days later. The MRI confirmed a predominantly solid left adnexal mass, which measured 15.4x11.2x1 cm (Figure 1). Both CT scan and MRI excluded any distant metastatic spread.The patient underwent surgery with the presumed diagnosis of an ovarian malignancy. Laparotomy revealed an 18 cm cystic mass surrounded by a very thin wall in the pouch of Douglas. The mass was adherent to the sigmoid colon and the pelvic sidewall. Bilateral ovaries, the fallopian tubes, and upper abdomen appeared normal. The mass was removed

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Gynecologic Sarcomas

Rahaman,

Cohen

2017

Holland‐Frei Cancer Medicine

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Overview Sarcomas are extremely rare and account for less than 1.5% of gynecologic cancers. Carcinosarcoma and leiomyosarcoma each account for 35–40% of uterine sarcomas, with endometrial stromal sarcoma (ESS) accounting for 10–15% and other sarcomas including adenosarcomas comprising 5–10%. Uterine carcinosarcoma should be classified as a metaplastic carcinoma of the uterus. Most adenosarcomas and ESSs have good prognosis and respond to hormonal therapy. Undifferentiated endometrial sarcoma (UES) and adenosarcomas with sarcomatous overgrowth are rare and have poor prognosis and require chemotherapy. ESS are histologically and clinically distinct from UES and each have distinct gene rearrangements. More than 50% of Stage I leiomyosarcomas (LMSs) and carcinosarcomas patients will recur. Chemotherapy is required for advanced or recurrent disease. In LMSs, the active drugs are doxorubicin, ifosfamide, gemcitabine, and docetaxel. For uterine carcinosarcomas, the drugs of choice are ifosfamide, cisplatin/carboplatin, and paclitaxel. Adjuvant radiation therapy may provide locoregional control in select cases of uterine carcinosarcomas. Hormonal therapy, including progestational agents, GnRH analogues, and aromatase inhibitors, has a role in the treatment of advanced or recurrent low‐grade ESSs and adenosarcomas.

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Extragenital adenosarcoma: A case report, review of the literature, and management discussion

Cited by 26 publications

References 23 publications

Two Cases of Extrauterine Müllerian Adenosarcoma Arising from Pelvic Endometriosis

Two Cases of Extrauterine Müllerian Adenosarcoma Arising from Pelvic Endometriosis

Extragenital Müllerian Adenosarcoma in the Pouch of Douglas

Gynecologic Sarcomas

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