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Journal of Minimally Invasive Gynecology 2016We 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... (Review)
Review
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.
Topics: Adenosarcoma; Cell Transformation, Neoplastic; Endometriosis; Feasibility Studies; Female; Humans; Laparoscopy; Middle Aged; Recurrence; Ultrasonography, Doppler, Color; Vaginal Neoplasms
PubMed: 27041653
DOI: 10.1016/j.jmig.2016.03.019 -
The American Journal of Surgical... Oct 2002Forty cases of mesodermal adenosarcoma of the ovary occurred in women 30-84 years of age (mean 54 years). Abdominal discomfort and distension were the usual complaints.... (Review)
Review
Forty cases of mesodermal adenosarcoma of the ovary occurred in women 30-84 years of age (mean 54 years). Abdominal discomfort and distension were the usual complaints. All the patients were treated with an oophorectomy, which was accompanied by a hysterectomy in 85%, a contralateral oophorectomy in 65%, and nonsurgical therapy in 28%. Tumor rupture occurred at or before the operation in 67% of the cases. Twenty-six tumors were stage I, 11 stage II, and 3 stage III. The tumors were unilateral in 97.5% of the cases and 5.5-50 cm (mean 14 cm) in greatest dimension; most of the tumors were predominantly solid but contained numerous small cysts. Microscopic examination revealed sarcomatous overgrowth in 12 tumors. Sex cord-like elements were present in six tumors (including four with sarcomatous overgrowth) and heterologous elements in five (including two with sarcomatous overgrowth). The highest mitotic index of the sarcomatous component was 1-25 (mean 6) mitotic figures per 10 high power fields. Only 6 of 26 women (23%) who were followed postoperatively for > or=5 years were free of tumor. In the other 20 patients recurrent tumor appeared at 0.4-6.6 years (mean 2.6 years) after operation as pure sarcoma (low grade or high grade) or adenosarcoma (with or without sarcomatous overgrowth). Eight women had additional recurrences, and four women had blood-borne metastases. One patient was alive at 15.7 years after the excision of pulmonary metastases. The 5-, 10-, and 15-year survival rates were 64%, 46%, and 30%, respectively. Age <53 years, tumor rupture, a high grade, and the presence of high-grade sarcomatous overgrowth appeared to be associated with recurrence or extraovarian spread. Ovarian adenosarcomas have a worse prognosis than uterine adenosarcomas, presumably because of the greater ease of peritoneal spread. Many of the tumors caused problems in differential diagnosis.
Topics: Adenosarcoma; Adult; Aged; Aged, 80 and over; Diagnosis, Differential; Disease-Free Survival; Female; Humans; Hysterectomy; Middle Aged; Ovarian Neoplasms; Ovariectomy; Survival Analysis
PubMed: 12360039
DOI: 10.1097/00000478-200210000-00001 -
Pathology International Nov 1995A new case is reported of müllerian adenosarcoma, presenting as a 'benign cervical polyp' protruding through the vulva of a 44 year-old woman admitted with abnormal... (Review)
Review
A new case is reported of müllerian adenosarcoma, presenting as a 'benign cervical polyp' protruding through the vulva of a 44 year-old woman admitted with abnormal vaginal bleeding. This report emphasizes the importance of a careful examination of the stroma and special features of the entrapped glands in order to contribute to an earlier and proper diagnosis. The literature is reviewed and the probable histogenesis of this tumor and differential diagnosis with embryonal rhabdomysarcoma (sarcoma botryoides), adenofibroma, malignant mesodermal tumor and carcinosarcoma is discussed.
Topics: Adenosarcoma; Adult; Cell Differentiation; Diagnosis, Differential; Female; Humans; Mixed Tumor, Mullerian; Uterine Cervical Neoplasms
PubMed: 8581155
DOI: 10.1111/j.1440-1827.1995.tb03412.x -
Gynecologic Oncology Aug 2003Primary adenosarcoma arising in vaginal endometriosis poses a diagnostic challenge, especially in superficial vaginal biopsies. (Review)
Review
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.
Topics: Adenosarcoma; Endometriosis; Female; Humans; Middle Aged; Mixed Tumor, Mullerian; Vaginal Neoplasms
PubMed: 12893226
DOI: 10.1016/s0090-8258(03)00266-x -
Annals of Coloproctology Oct 2014A Müllerian adenosarcoma is an extremely rare tumor characterized by a stromal component of usually low-grade malignancy and by a benign glandular epithelial component....
A Müllerian adenosarcoma is an extremely rare tumor characterized by a stromal component of usually low-grade malignancy and by a benign glandular epithelial component. A Müllerian adenosarcoma occurs mainly in the uterus, but also in extrauterine locations. Extrauterine Müllerian adenosarcomas are thought to arise from endometriotic deposits. A 36-year-old female presented to Daegu Catholic University Medical Center with a symptom of loose stool for several months. The imaging studies revealed a rectal mass, so she underwent a laparoscopic low anterior resection. Although extemporary pathology revealed an inflammatory myofibroblastic tumor, the final histologic diagnosis was a Müllerian adenosarcoma arising from rectal endometriosis. To our knowledge, except a concomitant rectal villotubular adenoma, cases of Müllerian adenosarcomas arising the rectal wall are rare. An adenosarcoma arising from endometriosis should be considered in the differential diagnosis of a pelvic mass, even one appearing in rectal wall, because ectopic endometrial tissue exists everywhere.
PubMed: 25360431
DOI: 10.3393/ac.2014.30.5.232 -
Gynecologic Oncology Jan 2002Müllerian adenosarcoma (MA) is a rare neoplasm composed of benign epithelial and malignant stromal components. Its location in the cervix and the presence of... (Review)
Review
BACKGROUND
Müllerian adenosarcoma (MA) is a rare neoplasm composed of benign epithelial and malignant stromal components. Its location in the cervix and the presence of heterologous elements are extremely infrequent (to our knowledge, only 14 more cases have been reported in the English literature). We describe another case of MA of the uterine cervix with heterologous elements and review the clinical and pathological features of these tumors.
CASE REPORT
A nulliparous 25-year-old woman was admitted to the hospital because of metrorrhagia and recurrent masses, dependent on the cervix, initially considered endocervical polyps. Microscopically, the last curettage performed revealed a tumor composed of two elements, epithelial and mesenchymal. The epithelial elements were benign endocervical type glands, and the mesenchymal were sarcomatous, containing minor foci of cartilage. A diagnosis of endocervical heterologous adenosarcoma was reached, and a total hysterectomy and bilateral salpingo-oophorectomy were performed. The patient is alive 2 years after the surgical procedure. Neither chemotherapy nor other adjuvant therapies have been administered, and she is clinically free of disease at the moment.
CONCLUSION
Because MA with heterologous elements seems to appear at the earliest stages of the reproductive lifespan in women, commonly with a history of recurrent polyps, and because its malignant potential is uncertain, gynecologists and pathologists should be aware and think about the possibility of this tumor. Long-term follow-up is essential and so is the accumulation of individual cases to provide further experience with these unusual neoplasms.
Topics: Adenosarcoma; Adult; Female; Humans; Mixed Tumor, Mullerian; Uterine Cervical Neoplasms
PubMed: 11748995
DOI: 10.1006/gyno.2001.6423 -
Gynecologic Oncology Reports Feb 2022To examine clinicopathologic characteristics and oncologic outcomes of patients diagnosed with Mullerian adenosarcoma and to evaluate ovarian preservation as a practical...
OBJECTIVE
To examine clinicopathologic characteristics and oncologic outcomes of patients diagnosed with Mullerian adenosarcoma and to evaluate ovarian preservation as a practical management option in early-stage disease.
METHODS
A retrospective review was performed of 31 patients treated for uterine, ovarian, or cervical adenosarcoma at our institution between 1/2000-3/2020. Recurrence-free survival (RFS) and overall survival (OS) were analyzed with Kaplan-Meier estimates, the log-rank test, and Cox proportional hazards regression.
RESULTS
Median age was 51 years (IQR: 41-68). Primary sites included uterine corpus (n = 23, 74.2%), uterine cervix (n = 7, 22.6%), and ovary (n = 1, 3.2%). Surgical management primarily consisted of total hysterectomy +/- bilateral adnexectomy +/- lymph node dissection. Fifteen (48.1%) patients underwent lymph node dissection; no patients had positive nodes. Ovaries were preserved in 6 (19.4%). Twenty-two (71.0%) patients received no adjuvant therapy, 4 (12.9%) received chemotherapy, 1 (3.2%) received chemoradiation, and 3 (9.7%) received hormonal therapy. Sarcomatous overgrowth (p = 0.04), high grade histology (p = 0.002), and greater depth of myometrial invasion (p = 0.001) were associated with decreased RFS. None of the 6 patients with ovarian preservation had recurrences. At last follow up, 21 patients (67.7%) had no evidence of disease, 7 (22.6%) were deceased due to disease, and 3 (9.7%) were deceased due to non-cancerous reasons.
CONCLUSIONS
Uterine adenosarcoma appears to have a relatively good prognosis, especially in the absence of risk factors, such as sarcomatous overgrowth, high grade histology, and deep myometrial invasion. Ovarian preservation may be a feasible management option with non-inferior outcomes for premenopausal women with early-stage disease. Future studies including larger patient cohorts are needed for this rare disease.
PubMed: 35005157
DOI: 10.1016/j.gore.2021.100913 -
Ultrastructural Pathology 1997Four cases of Mullerian adenosarcoma were studied by light and electron microscopy and immunohistochemistry. All 4 cases showed the histologic characteristics of... (Review)
Review
Four cases of Mullerian adenosarcoma were studied by light and electron microscopy and immunohistochemistry. All 4 cases showed the histologic characteristics of adenosarcoma with benign endometrial glands and a malignant stroma. Ultrastructurally, the epithelial component in all cases had the appearance of proliferative endometrial glands, and the malignant mesenchymal cells showed features of endometrial stroma. A distinct basal lamina separating glands from stroma was present. In addition, 2 of the cases showed extensive smooth muscle differentiation which was associated with sarcomatous overgrowth. The smooth muscle features were confirmed by immunohistochemistry. Multiple theories of the histogenesis of this tumor are discussed.
Topics: Adenosarcoma; Adult; Aged; Aged, 80 and over; Cell Differentiation; Female; Humans; Middle Aged; Mixed Tumor, Mullerian; Muscle, Smooth; Uterine Neoplasms
PubMed: 9029768
DOI: 10.3109/01913129709023249 -
Cureus Feb 2021Background and objective Müllerian adenosarcomas (MA) are rare biphasic tumors with benign epithelial and sarcomatous stromal components. There is very limited cohort...
Background and objective Müllerian adenosarcomas (MA) are rare biphasic tumors with benign epithelial and sarcomatous stromal components. There is very limited cohort study data on MA in the South Asian countries and no such study has been attempted in Pakistan. Our aim was to evaluate the clinicopathological characteristics of MA and to review the published literature on the condition. Additionally, we also analyzed the impact of various prognostic factors on the overall survival (OS) of patients with MA. Materials and methods This was a retrospective observational study performed at the Shaukat Khanum Memorial Hospital and Research Centre, Lahore from 2003 to 2020. A total of 59 histologically confirmed cases of MA were included in the study and critically reviewed. Results The mean age of the patients was 54 ±16 years, and the most common tumor location was the uterine corpus (48, 81.4%), followed by the cervix (eight, 13.6%), ovary (two, 3.4%), and vagina (one, 1.7%). Sarcomatous overgrowth (SO) was seen in 22 (37.3%) patients, and high-grade cytology was observed in 18 (30.5%) patients. Furthermore, lymphovascular invasion (LVI) was present in six (10.2%) patients, and myometrial invasion was noted in 25 (42.4%) patients. The follow-up details of 29 patients were available, and death was recorded in 13 (44.8%) patients with a median OS of three years. Conclusion MA is a rare and diagnostically challenging entity due to its wide differential diagnosis. It is essential to take note of different morphological features such as SO, cytological features, LVI, and heterologous differentiation because of their significant prognostic impact.
PubMed: 33747657
DOI: 10.7759/cureus.13360 -
BMC Cancer Jun 2011Uterine sarcomas are relatively rare tumors that account for approximately 1-3% of female genital tract malignancies and between 4-9% of uterine cancers. Less than 8% of... (Review)
Review
BACKGROUND
Uterine sarcomas are relatively rare tumors that account for approximately 1-3% of female genital tract malignancies and between 4-9% of uterine cancers. Less than 8% of all cases are Mullerian adenosarcoma, a distinctive uterine neoplasm characterized by a benign, but occasionally atypical, epithelial and a malignant, usually low-grade, stromal component, both of which should be integral and neoplastic constituents of the tumor. Mullerian adenosarcoma with sarcomatous overgrowth (MASO) is a very aggressive variant, associated with post-operative recurrence, metastases, even when diagnosed in early stage.
CASE PRESENTATION
We present a fourth MASO case derived from uterine cervix in a 72-year-old woman with metrorrhagia and a polypoid mass protruding through the cervical ostium. Total abdominal hysterectomy, bilateral salpingo-oophorectomy, systematic pelvic lymph node dissection, omental biopsy and appendectomy were performed. Surgery treatment was associated with adjuvant whole-pelvis radiation (45 Gy) and adjuvant chemotherapy (cisplatin/ifosfamide). After nine months of follow up, the patient was free of tumor.
CONCLUSIONS
The rarity of MASO of the cervix involves a management difficult. Most authors recommend total abdominal hysterectomy, usually accompanied by bilateral salpingo-oophorectomy. There is no common agreement on staging by lymphadenectomy during primary surgery and adjuvant chemo-radio therapy.
Topics: Adenosarcoma; Aged; Antineoplastic Combined Chemotherapy Protocols; Appendectomy; Chemotherapy, Adjuvant; Cisplatin; Female; Humans; Hysterectomy; Ifosfamide; Lymph Node Excision; Mixed Tumor, Mullerian; Neoplasm Invasiveness; Omentum; Ovariectomy; Prognosis; Radiotherapy, Adjuvant; Remission Induction; Salpingectomy; Uterine Cervical Neoplasms
PubMed: 21663687
DOI: 10.1186/1471-2407-11-236