Skip to main content
Top
Published in: BMC Cancer 1/2011

Open Access 01-12-2011 | Case report

Cervical Mullerian Adenosarcoma with heterologous sarcomatous overgrowth: a fourth case and review of literature

Authors: Tito Silvio Patrelli, Salvatore Gizzo, Stefania Di Gangi, Giorgia Guidi, Mario Rondinelli, Giovanni Battista Nardelli

Published in: BMC Cancer | Issue 1/2011

Login to get access

Abstract

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.
Appendix
Available only for authorised users
Literature
1.
go back to reference D'Angelo E, Prat J: Uterine sarcomas: a review. Gynecol Oncol. 2010, 116 (1): 131-9. 10.1016/j.ygyno.2009.09.023.CrossRefPubMed D'Angelo E, Prat J: Uterine sarcomas: a review. Gynecol Oncol. 2010, 116 (1): 131-9. 10.1016/j.ygyno.2009.09.023.CrossRefPubMed
2.
go back to reference Brooks SE, Zhan M, Cote T, Baquet CR: Surveillance, epidemiology, and end results analysis of 2677 cases of uterine sarcoma 1989-1999. Gynecol Oncol. 2004, 93 (1): 204-8. 10.1016/j.ygyno.2003.12.029.CrossRefPubMed Brooks SE, Zhan M, Cote T, Baquet CR: Surveillance, epidemiology, and end results analysis of 2677 cases of uterine sarcoma 1989-1999. Gynecol Oncol. 2004, 93 (1): 204-8. 10.1016/j.ygyno.2003.12.029.CrossRefPubMed
3.
go back to reference Nordal RR, Thoresen SO: Uterine sarcomas in Norway 1956-1992: incidence, survival and mortality. Eur J Cancer. 1997, 33 (6): 907-11. 10.1016/S0959-8049(97)00040-3.CrossRefPubMed Nordal RR, Thoresen SO: Uterine sarcomas in Norway 1956-1992: incidence, survival and mortality. Eur J Cancer. 1997, 33 (6): 907-11. 10.1016/S0959-8049(97)00040-3.CrossRefPubMed
4.
go back to reference Harlow BL, Weiss NS, Lofton S: The epidemiology of sarcomas of the uterus. J Natl Cancer Inst. 1986, 76 (3): 399-402.PubMed Harlow BL, Weiss NS, Lofton S: The epidemiology of sarcomas of the uterus. J Natl Cancer Inst. 1986, 76 (3): 399-402.PubMed
5.
go back to reference Prat J: FIGO staging for uterine sarcomas. Int J Gynaecol Obstet. 2009, 104 (3): 177-8. 10.1016/j.ijgo.2008.12.008. Erratum in: Int J Gynaecol Obstet. 2009 Sep;106(3):277CrossRefPubMed Prat J: FIGO staging for uterine sarcomas. Int J Gynaecol Obstet. 2009, 104 (3): 177-8. 10.1016/j.ijgo.2008.12.008. Erratum in: Int J Gynaecol Obstet. 2009 Sep;106(3):277CrossRefPubMed
6.
go back to reference Clement PB, Scully RE: Müllerian adenosarcoma of the uterus. A clinicopathologic analysis of ten cases of a distinctive type of müllerian mixed tumor. Cancer. 1974, 34 (4): 1138-49. 10.1002/1097-0142(197410)34:4<1138::AID-CNCR2820340425>3.0.CO;2-9.CrossRefPubMed Clement PB, Scully RE: Müllerian adenosarcoma of the uterus. A clinicopathologic analysis of ten cases of a distinctive type of müllerian mixed tumor. Cancer. 1974, 34 (4): 1138-49. 10.1002/1097-0142(197410)34:4<1138::AID-CNCR2820340425>3.0.CO;2-9.CrossRefPubMed
7.
go back to reference Clement PB, Scully RE: Mullerian adenosarcoma of the uterus: a clinicopathologic analysis of 100 cases with a review of the literature. Hum Pathol. 1990, 21 (4): 363-81. 10.1016/0046-8177(90)90198-E.CrossRefPubMed Clement PB, Scully RE: Mullerian adenosarcoma of the uterus: a clinicopathologic analysis of 100 cases with a review of the literature. Hum Pathol. 1990, 21 (4): 363-81. 10.1016/0046-8177(90)90198-E.CrossRefPubMed
8.
go back to reference Major FJ, Blessing JA, Silverberg SG, Morrow CP, Creasman WT, Currie JL, Yordan E, Brady MF: Prognostic factors in early-stage uterine sarcoma. A Gynecologic Oncology Group study. Cancer. 1993, 1571 (4 Suppl): 1702-9.CrossRef Major FJ, Blessing JA, Silverberg SG, Morrow CP, Creasman WT, Currie JL, Yordan E, Brady MF: Prognostic factors in early-stage uterine sarcoma. A Gynecologic Oncology Group study. Cancer. 1993, 1571 (4 Suppl): 1702-9.CrossRef
9.
go back to reference Piura B, Rabinovich A, Meirovitz M, Yanai-Inbar I: Mullerian adenosarcoma of the uterus: case report and review of literature. Eur J Gynaecol Oncol. 2000, 21 (4): 387-90.PubMed Piura B, Rabinovich A, Meirovitz M, Yanai-Inbar I: Mullerian adenosarcoma of the uterus: case report and review of literature. Eur J Gynaecol Oncol. 2000, 21 (4): 387-90.PubMed
10.
go back to reference Kaku T, Silverberg SG, Major FJ, Miller A, Fetter B, Brady MF: Adenosarcoma of the uterus: a Gynecologic Oncology Group clinicopathologic study of 31 cases. Int J Gynecol Pathol. 1992, 11 (2): 75-88. 10.1097/00004347-199204000-00001.CrossRefPubMed Kaku T, Silverberg SG, Major FJ, Miller A, Fetter B, Brady MF: Adenosarcoma of the uterus: a Gynecologic Oncology Group clinicopathologic study of 31 cases. Int J Gynecol Pathol. 1992, 11 (2): 75-88. 10.1097/00004347-199204000-00001.CrossRefPubMed
11.
go back to reference Verschraegen CF, Vasuratna A, Edwards C, Freedman R, Kudelka AP, Tornos C, Kavanagh JJ: Clinicopathologic analysis of mullerian adenosarcoma: the M.D. Anderson Cancer Center experience. Oncol Rep. 1998, 5 (4): 939-44.PubMed Verschraegen CF, Vasuratna A, Edwards C, Freedman R, Kudelka AP, Tornos C, Kavanagh JJ: Clinicopathologic analysis of mullerian adenosarcoma: the M.D. Anderson Cancer Center experience. Oncol Rep. 1998, 5 (4): 939-44.PubMed
12.
go back to reference Fleming NA, Hopkins L, de Nanassy J, Senterman M, Black AY: Mullerian adenosarcoma of the cervix in a 10-year-old girl: case report and review of the literature. J Pediatr Adolesc Gynecol. 2009, 22 (4): e45-51. 10.1016/j.jpag.2008.06.001.CrossRefPubMed Fleming NA, Hopkins L, de Nanassy J, Senterman M, Black AY: Mullerian adenosarcoma of the cervix in a 10-year-old girl: case report and review of the literature. J Pediatr Adolesc Gynecol. 2009, 22 (4): e45-51. 10.1016/j.jpag.2008.06.001.CrossRefPubMed
13.
go back to reference Orenstein HH, Richart RM, Fenoglio CM: Müllerian adenosarcoma of the uterus: literature review, case report, and ultrastructural observations. Ultrastruct Pathol. 1980, 1 (2): 189-200. 10.3109/01913128009141416.CrossRefPubMed Orenstein HH, Richart RM, Fenoglio CM: Müllerian adenosarcoma of the uterus: literature review, case report, and ultrastructural observations. Ultrastruct Pathol. 1980, 1 (2): 189-200. 10.3109/01913128009141416.CrossRefPubMed
14.
go back to reference McCluggage WG: Mullerian adenosarcoma of the female genital tract. Adv Anat Pathol. 2010, 17 (2): 122-9. 10.1097/PAP.0b013e3181cfe732.CrossRefPubMed McCluggage WG: Mullerian adenosarcoma of the female genital tract. Adv Anat Pathol. 2010, 17 (2): 122-9. 10.1097/PAP.0b013e3181cfe732.CrossRefPubMed
15.
go back to reference Reed NS: Uterine sarcomas--the biggest challenge?. Clin Oncol (R Coll Radiol). 2002, 14 (1): 50-3.CrossRef Reed NS: Uterine sarcomas--the biggest challenge?. Clin Oncol (R Coll Radiol). 2002, 14 (1): 50-3.CrossRef
16.
go back to reference Norris HJ, Taylor HB: Postirradiation sarcomas of the uterus. Obstet Gynecol. 1965, 26 (5): 689-94.PubMed Norris HJ, Taylor HB: Postirradiation sarcomas of the uterus. Obstet Gynecol. 1965, 26 (5): 689-94.PubMed
17.
go back to reference Press MF, Scully RE: Endometrial "sarcomas" complicating ovarian thecoma, polycystic ovarian disease and estrogen therapy. Gynecol Oncol. 1985, 21 (2): 135-54. 10.1016/0090-8258(85)90246-X.CrossRefPubMed Press MF, Scully RE: Endometrial "sarcomas" complicating ovarian thecoma, polycystic ovarian disease and estrogen therapy. Gynecol Oncol. 1985, 21 (2): 135-54. 10.1016/0090-8258(85)90246-X.CrossRefPubMed
18.
go back to reference Clement PB, Oliva E, Young RH: Mullerian adenosarcoma of the uterine corpus associated with tamoxifen therapy: a report of six cases and a review of tamoxifen-associated endometrial lesions. Int J Gynecol Pathol. 1996, 15 (3): 222-9. 10.1097/00004347-199607000-00006.CrossRefPubMed Clement PB, Oliva E, Young RH: Mullerian adenosarcoma of the uterine corpus associated with tamoxifen therapy: a report of six cases and a review of tamoxifen-associated endometrial lesions. Int J Gynecol Pathol. 1996, 15 (3): 222-9. 10.1097/00004347-199607000-00006.CrossRefPubMed
19.
go back to reference Jones MW, Lefkowitz M: Adenosarcoma of the uterine cervix: a clinicopathological study of 12 cases. Int J Gynecol Pathol. 1995, 14 (3): 223-9. 10.1097/00004347-199507000-00005.CrossRefPubMed Jones MW, Lefkowitz M: Adenosarcoma of the uterine cervix: a clinicopathological study of 12 cases. Int J Gynecol Pathol. 1995, 14 (3): 223-9. 10.1097/00004347-199507000-00005.CrossRefPubMed
20.
go back to reference Kerner H, Lichtig C: Müllerian adenosarcoma presenting as cervical polyps: a report of seven cases and review of the literature. Obstet Gynecol. 1993, 81 (5 Pt 1): 655-9.PubMed Kerner H, Lichtig C: Müllerian adenosarcoma presenting as cervical polyps: a report of seven cases and review of the literature. Obstet Gynecol. 1993, 81 (5 Pt 1): 655-9.PubMed
21.
go back to reference Manoharan M, Azmi MA, Soosay G, Mould T, Weekes AR: Mullerian adenosarcoma of uterine cervix: report of three cases and review of literature. Gynecol Oncol. 2007, 105 (1): 256-60. 10.1016/j.ygyno.2006.12.029.CrossRefPubMed Manoharan M, Azmi MA, Soosay G, Mould T, Weekes AR: Mullerian adenosarcoma of uterine cervix: report of three cases and review of literature. Gynecol Oncol. 2007, 105 (1): 256-60. 10.1016/j.ygyno.2006.12.029.CrossRefPubMed
22.
go back to reference Tang CK, Toker C, Harriman B: Müllerian adenosarcoma of the uterine cervix. Hum Pathol. 1981, 12 (6): 579-81. 10.1016/S0046-8177(81)80073-1.CrossRefPubMed Tang CK, Toker C, Harriman B: Müllerian adenosarcoma of the uterine cervix. Hum Pathol. 1981, 12 (6): 579-81. 10.1016/S0046-8177(81)80073-1.CrossRefPubMed
23.
go back to reference Ramos P, Ruiz A, Carabias E, Piñero I, Garzon A, Alvarez I: Müllerian adenosarcoma of the cervix with heterologous elements: report of a case and review of the literature. Gynecol Oncol. 2002, 84 (1): 161-6. 10.1006/gyno.2001.6423.CrossRefPubMed Ramos P, Ruiz A, Carabias E, Piñero I, Garzon A, Alvarez I: Müllerian adenosarcoma of the cervix with heterologous elements: report of a case and review of the literature. Gynecol Oncol. 2002, 84 (1): 161-6. 10.1006/gyno.2001.6423.CrossRefPubMed
24.
go back to reference "World Health Organization Classification of Tumors. Pathology and Genetics. Tumours of the breast and female genital organs". Edited by: Tavassoli FA, Devilee P. 2003, Lyon: IARC Press, 247-248. "World Health Organization Classification of Tumors. Pathology and Genetics. Tumours of the breast and female genital organs". Edited by: Tavassoli FA, Devilee P. 2003, Lyon: IARC Press, 247-248.
25.
go back to reference Zaloudek CJ, Norris HJ: Adenofibroma and adenosarcoma of the uterus: a clinicopathologic study of 35 cases. Cancer. 1981, 1548 (2): 354-66.CrossRef Zaloudek CJ, Norris HJ: Adenofibroma and adenosarcoma of the uterus: a clinicopathologic study of 35 cases. Cancer. 1981, 1548 (2): 354-66.CrossRef
26.
go back to reference Park HM, Park MH, Kim YJ, Chun SH, Ahn JJ, Kim CI, Sung SH, Han WS, Kim SC: Mullerian adenosarcoma with sarcomatous overgrowth of the cervix presenting as cervical polyp: a case report and review of the literature. Int J Gynecol Cancer. 2004, 14 (5): 1024-9. 10.1111/j.1048-891X.2004.014546.x.CrossRefPubMed Park HM, Park MH, Kim YJ, Chun SH, Ahn JJ, Kim CI, Sung SH, Han WS, Kim SC: Mullerian adenosarcoma with sarcomatous overgrowth of the cervix presenting as cervical polyp: a case report and review of the literature. Int J Gynecol Cancer. 2004, 14 (5): 1024-9. 10.1111/j.1048-891X.2004.014546.x.CrossRefPubMed
27.
go back to reference Comunoğlu N, Comunoğlu C, Başsüllü N, Somunkiran A, Calay Z: Müllerian adenosarcoma with sarcomatous overgrowth of the cervix: unusual large polypoid mass. Ups J Med Sci. 2007, 112 (1): 67-72. 10.3109/2000-1967-096.CrossRefPubMed Comunoğlu N, Comunoğlu C, Başsüllü N, Somunkiran A, Calay Z: Müllerian adenosarcoma with sarcomatous overgrowth of the cervix: unusual large polypoid mass. Ups J Med Sci. 2007, 112 (1): 67-72. 10.3109/2000-1967-096.CrossRefPubMed
28.
go back to reference Duggal R, Nijhawan R, Aggarwal N, Sikka P: Mullerian adenosarcoma (heterologous) of the cervix with sarcomatous overgrowth: a case report with review of literature. J Gynecol Oncol. 2010, 21 (2): 125-8. 10.3802/jgo.2010.21.2.125.CrossRefPubMedPubMedCentral Duggal R, Nijhawan R, Aggarwal N, Sikka P: Mullerian adenosarcoma (heterologous) of the cervix with sarcomatous overgrowth: a case report with review of literature. J Gynecol Oncol. 2010, 21 (2): 125-8. 10.3802/jgo.2010.21.2.125.CrossRefPubMedPubMedCentral
29.
go back to reference Soslow RA, Ali A, Oliva E: Mullerian adenosarcomas: an immunophenotypic analysis of 35 cases. Am J Surg Pathol. 2008, 32 (7): 1013-21. 10.1097/PAS.0b013e318161d1be.CrossRefPubMed Soslow RA, Ali A, Oliva E: Mullerian adenosarcomas: an immunophenotypic analysis of 35 cases. Am J Surg Pathol. 2008, 32 (7): 1013-21. 10.1097/PAS.0b013e318161d1be.CrossRefPubMed
30.
go back to reference Gallardo A, Prat J: Mullerian adenosarcoma: a clinicopathologic and immunohistochemical study of 55 cases challenging the existence of adenofibroma. Am J Surg Pathol. 2009, 33 (2): 278-88. 10.1097/PAS.0b013e318181a80d.CrossRefPubMed Gallardo A, Prat J: Mullerian adenosarcoma: a clinicopathologic and immunohistochemical study of 55 cases challenging the existence of adenofibroma. Am J Surg Pathol. 2009, 33 (2): 278-88. 10.1097/PAS.0b013e318181a80d.CrossRefPubMed
31.
go back to reference Blom R, Guerrieri C: Adenosarcoma of the uterus: a clinicopathologic, DNA flow cytometric, p53 and mdm-2 analysis of 11 cases. Int J Gynecol Cancer. 1999, 9 (1): 37-43. 10.1046/j.1525-1438.1999.09885.x.CrossRefPubMed Blom R, Guerrieri C: Adenosarcoma of the uterus: a clinicopathologic, DNA flow cytometric, p53 and mdm-2 analysis of 11 cases. Int J Gynecol Cancer. 1999, 9 (1): 37-43. 10.1046/j.1525-1438.1999.09885.x.CrossRefPubMed
32.
go back to reference Chen KT: Rhabdomyosarcomatous uterine adenosarcoma. Int J Gynecol Pathol. 1985, 4 (2): 146-52. 10.1097/00004347-198506000-00006.CrossRefPubMed Chen KT: Rhabdomyosarcomatous uterine adenosarcoma. Int J Gynecol Pathol. 1985, 4 (2): 146-52. 10.1097/00004347-198506000-00006.CrossRefPubMed
33.
go back to reference Krivak TC, Seidman JD, McBroom JW, MacKoul PJ, Aye LM, Rose GS: Uterine adenosarcoma with sarcomatous overgrowth versus uterine carcinosarcoma: comparison of treatment and survival. Gynecol Oncol. 2001, 83 (1): 89-94. 10.1006/gyno.2001.6334.CrossRefPubMed Krivak TC, Seidman JD, McBroom JW, MacKoul PJ, Aye LM, Rose GS: Uterine adenosarcoma with sarcomatous overgrowth versus uterine carcinosarcoma: comparison of treatment and survival. Gynecol Oncol. 2001, 83 (1): 89-94. 10.1006/gyno.2001.6334.CrossRefPubMed
34.
go back to reference Daya DA, Scully RE: Sarcoma botryoides of the uterine cervix in young women: a clinicopathological study of 13 cases. Gynecol Oncol. 1988, 29 (3): 290-304. 10.1016/0090-8258(88)90228-4.CrossRefPubMed Daya DA, Scully RE: Sarcoma botryoides of the uterine cervix in young women: a clinicopathological study of 13 cases. Gynecol Oncol. 1988, 29 (3): 290-304. 10.1016/0090-8258(88)90228-4.CrossRefPubMed
35.
go back to reference Gal D, Kerner H, Beck D, Peretz BA, Eyal A, Paldi E: Mullerian adenosarcoma of the uterine cervix. Gynecol Oncol. 1988, 31 (3): 445-53. 10.1016/S0090-8258(88)80030-1.CrossRefPubMed Gal D, Kerner H, Beck D, Peretz BA, Eyal A, Paldi E: Mullerian adenosarcoma of the uterine cervix. Gynecol Oncol. 1988, 31 (3): 445-53. 10.1016/S0090-8258(88)80030-1.CrossRefPubMed
36.
go back to reference Clement PB: Müllerian adenosarcomas of the uterus with sarcomatous overgrowth. A clinicopathological analysis of 10 cases. Am J Surg Pathol. 1989, 13 (1): 28-38. 10.1097/00000478-198901000-00004.CrossRefPubMed Clement PB: Müllerian adenosarcomas of the uterus with sarcomatous overgrowth. A clinicopathological analysis of 10 cases. Am J Surg Pathol. 1989, 13 (1): 28-38. 10.1097/00000478-198901000-00004.CrossRefPubMed
37.
go back to reference Hallak M, Peipert JF, Heller PB, Sedlacek TV, Schauer GM: Mullerian adenosarcoma of the uterus with sarcomatous overgrowth. J Surg Oncol. 1992, 51 (1): 68-70. 10.1002/jso.2930510117.CrossRefPubMed Hallak M, Peipert JF, Heller PB, Sedlacek TV, Schauer GM: Mullerian adenosarcoma of the uterus with sarcomatous overgrowth. J Surg Oncol. 1992, 51 (1): 68-70. 10.1002/jso.2930510117.CrossRefPubMed
38.
go back to reference Rovirosa A, Ascaso C, Ordi J, Abellana R, Arenas M, Lejarcegui JA, Pahisa J, Puig-Tintoré LM, Mellado B, Armenteros B, Iglesias X, Biete A: Is vascular and lymphatic space invasion a main prognostic factor in uterine neoplasms with a sarcomatous component? A retrospective study of prognostic factors of 60 patients stratified by stages. Int J Radiat Oncol Biol Phys. 2002, 152 (5): 1320-9.CrossRef Rovirosa A, Ascaso C, Ordi J, Abellana R, Arenas M, Lejarcegui JA, Pahisa J, Puig-Tintoré LM, Mellado B, Armenteros B, Iglesias X, Biete A: Is vascular and lymphatic space invasion a main prognostic factor in uterine neoplasms with a sarcomatous component? A retrospective study of prognostic factors of 60 patients stratified by stages. Int J Radiat Oncol Biol Phys. 2002, 152 (5): 1320-9.CrossRef
39.
go back to reference Fukunaga M, Ishihara A, Ushigome S: Extrauterine low-grade endometrial stromal sarcoma: report of three cases. Pathol Int. 1998, 48 (4): 297-302. 10.1111/j.1440-1827.1998.tb03909.x.CrossRefPubMed Fukunaga M, Ishihara A, Ushigome S: Extrauterine low-grade endometrial stromal sarcoma: report of three cases. Pathol Int. 1998, 48 (4): 297-302. 10.1111/j.1440-1827.1998.tb03909.x.CrossRefPubMed
Metadata
Title
Cervical Mullerian Adenosarcoma with heterologous sarcomatous overgrowth: a fourth case and review of literature
Authors
Tito Silvio Patrelli
Salvatore Gizzo
Stefania Di Gangi
Giorgia Guidi
Mario Rondinelli
Giovanni Battista Nardelli
Publication date
01-12-2011
Publisher
BioMed Central
Published in
BMC Cancer / Issue 1/2011
Electronic ISSN: 1471-2407
DOI
https://doi.org/10.1186/1471-2407-11-236

Other articles of this Issue 1/2011

BMC Cancer 1/2011 Go to the issue
Webinar | 19-02-2024 | 17:30 (CET)

Keynote webinar | Spotlight on antibody–drug conjugates in cancer

Antibody–drug conjugates (ADCs) are novel agents that have shown promise across multiple tumor types. Explore the current landscape of ADCs in breast and lung cancer with our experts, and gain insights into the mechanism of action, key clinical trials data, existing challenges, and future directions.

Dr. Véronique Diéras
Prof. Fabrice Barlesi
Developed by: Springer Medicine