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Case Study
A Rare Case of Intramural Müllerian Adenosarcoma Arising from Adenomyosis of the Uterus
Sun-Jae Lee, Ji Y. Park
J Pathol Transl Med. 2017;51(4):433-440.   Published online June 29, 2017
DOI: https://doi.org/10.4132/jptm.2017.06.11
  • 8,559 View
  • 144 Download
  • 12 Web of Science
  • 13 Crossref
AbstractAbstract PDF
Müllerian adenosarcomas usually arise as polypoid masses in the endometrium of post-menopausal women. Occasionally, these tumors arise in the cervix, vagina, broad and round ligaments, ovaries and rarely in extragenital sites; these cases are generally associated with endometriosis. We experienced a rare case of extraendometrial, intramural adenosarcoma arising in a patient with adenomyosis. A 40-year-old woman presented with sudden-onset suprapubic pain. The imaging findings suggested leiomyoma with cystic degeneration in the uterine fundus. An ill-defined ovoid tumor with hemorrhagic degeneration, measuring 7.5 cm in diameter, was detected. The microscopic findings showed glandular cells without atypia and a sarcomatous component with pleomorphism and high mitotic rates. There was no evidence of endometrial origin. To recognize that adenosarcoma can, although rarely, arise from adenomyosis is important to avoid overstaging and inappropriate treatment.

Citations

Citations to this article as recorded by  
  • Whether surgical procedure can improve the prognosis of endometrial cancer arising in adenomyosis (EC-AIA)? a systematic review and meta-analysis
    Yi Sun, Shitong Lin, Weijia Wu, Fangfang Nie, Yuchen Liu, Jing Wen, Xiaoran Cheng, Qianwen Liu, Yuanpei Wang, Fang Ren
    International Journal of Surgery.2024;[Epub]     CrossRef
  • Mullerian adenosarcoma accidentally detected and coexisting with cervical carcinoma in situ: a rare case report
    Xuemei Qing, Min Xie, Hongying Guo, Liying Zhang, Jiatian Ye, Yong Zhang, Ying Ma
    Frontiers in Oncology.2024;[Epub]     CrossRef
  • Mucinous carcinoma originating from uterine adenomyosis: a case report
    Satoshi Ohira, Ryota Tachibana, Sayaka Yasaki, Koji Tsunemi, Natsuki Uchiyama, Eri Ikeda, Kenji Sano
    Journal of Medical Case Reports.2023;[Epub]     CrossRef
  • Endometrial Cancer Arising in Adenomyosis (EC-AIA): A Systematic Review
    Antonio Raffone, Diego Raimondo, Manuela Maletta, Antonio Travaglino, Federica Renzulli, Daniele Neola, Umberto De Laurentiis, Francesco De Laurentiis, Mohamed Mabrouk, Manuel Maria Ianieri, Renato Seracchioli, Paolo Casadio, Antonio Mollo
    Cancers.2023; 15(4): 1142.     CrossRef
  • Adenomyosis and Its Possible Malignancy: A Review of the Literature
    Liviu Moraru, Melinda-Ildiko Mitranovici, Diana Maria Chiorean, Raluca Moraru, Laura Caravia, Andreea Taisia Tiron, Ovidiu Simion Cotoi
    Diagnostics.2023; 13(11): 1883.     CrossRef
  • Case Report: Uterine Adenosarcoma With Sarcomatous Overgrowth and Malignant Heterologous Elements
    Yunuén I. García-Mendoza, Mario Murguia-Perez, Aldo I. Galván-Linares, Saulo Mendoza-Ramírez, Norma L. García-Salinas, Julio G. Moctezuma-Ramírez, Blanca O. Murillo-Ortiz, Luis Jonathan Bueno-Rosario, Marco A. Olvera-Olvera, Guillermo E. Corredor-Alonso
    Frontiers in Medicine.2022;[Epub]     CrossRef
  • Adenomyosis as a Risk Factor for Myometrial or Endometrial Neoplasms—Review
    Maria Szubert, Edward Kozirog, Jacek Wilczynski
    International Journal of Environmental Research and Public Health.2022; 19(4): 2294.     CrossRef
  • Uterine adenosarcoma arising from a subserosal adenomyoma: A case report
    Shazia Fakhar, Tehreem Zahid, Yamina Ishtiaq
    Gynecologic Oncology Reports.2022; 40: 100957.     CrossRef
  • Uterine Adenosarcoma Originating in Adenomyosis: Report of an Extremely Rare Phenomenon and Review of Published Literature
    Karen L. Talia, Yael Naaman, W. Glenn McCluggage
    International Journal of Gynecological Pathology.2021; 40(4): 342.     CrossRef
  • Uterine adenosarcoma. Report of 5 cases and review of literature
    I.V. Barinova, I.N. Voloshchuk, A.A. Fedorov, N.V. Puchkova, S.N. Buyanova, M.A. Chechneva, A.A. Popov, O.V. Kapitanova, N.I. Kondrikov
    Arkhiv patologii.2021; 83(3): 25.     CrossRef
  • New Aspects of Sarcomas of Uterine Corpus—A Brief Narrative Review
    Stoyan Kostov, Yavor Kornovski, Vesela Ivanova, Deyan Dzhenkov, Dimitar Metodiev, Rafał Watrowski, Yonka Ivanova, Stanislav Slavchev, Dimitar Mitev, Angel Yordanov
    Clinics and Practice.2021; 11(4): 878.     CrossRef
  • Uterine Adenosarcoma with Sarcomatous Overgrowth: A Case Report of Aggressive Disease in a 16-Year-Old Girl and a Literature Review
    Hanyuan Liu, Zhen Shen, Dabao Wu, Ying Zhou
    Journal of Pediatric and Adolescent Gynecology.2018; 31(4): 426.     CrossRef
  • Uterine Adenosarcoma
    Uwe A. Ulrich, Dominik Denschlag
    Oncology Research and Treatment.2018; 41(11): 693.     CrossRef
Case Report
Malignant Mixed Mullerian Tumor Arising from Adenomyosis of Uterus.
Yoon Ju Kim, Jung Il Suh
Korean J Pathol. 1998;32(12):1098-1100.
  • 1,669 View
  • 10 Download
AbstractAbstract
Malignant mixed mullerian tumor (MMMT) is an unusual tumor composed of malignant epithelial and nonepithelial components in the same lesion and is subdivided into homologous and heterologous types. Epidemiologically, these tumors are associated with prior pelvic irradiation, functioning ovarian lesions, exogenous estrogen therapy and rarely endometriosis. We experienced a case of uterine MMMT which arose from adenomyosis in a 47-year-old woman who had no specific past medical history. The posterior uterine corpus showed a 3.5x3.0x2.0 cm sized, relatively well defined tumor mass within the background of the adenomyosis. The tumor was composed of well differentiated endometrial adenocarcinoma and sarcomatous stroma with foci of rhabdomyosarcomatous differentiation confirmed by immunohistochemical and electron microscopic studies. Through the immunohistochemical study, both the epithelial and nonepithelial components were positive for cytokeratin and it suggested that the sarcomatous area originated from metaplasia of the adenocarcinoma component. From the overall findings, it is regarded as an uterine heterologous MMMT which arose from adenomyosis.
Original Article
Adenocarcinoma Arising in Adenomyosis.
Young Il Yang, In Sook Lim, Jong Eun Joo
Korean J Pathol. 1995;29(2):272-274.
  • 1,640 View
  • 13 Download
AbstractAbstract PDF
Adenocarcinoma in adenomyosis is unusual and it is mostly associated with adenocarcinoma in the endometrial mucosa. In contrast, adenocarcinoma arising in adenomyosis without endometrial adenocarcinoma is extremely rare and it suggests that it arises de novo from adenomyosis. We report a case of adenocarcinoma arising in adenomyosis in 44-year-old woman. The endometrial cavity contained a polypoid lesion with atypical hyperplasia, but no evidence of adenocarcinoma in the endometrial mucosa. Simple, complex and atypical hyperplasia associated with well differentiated adenocarcinoma was also noted in the areas of adenomyosis.
Case Report
Low-grade Uterine Endometrial Stromal Sarcoma Resembling Ovarian Sex-Cord Tumor : A case report.
Mee Yon Cho, Kyu Rae Kim, Woo Hee Jung, Hyeon Joo Jeong, Kyi Beum Lee
Korean J Pathol. 1991;25(5):476-480.
  • 1,572 View
  • 10 Download
AbstractAbstract
A case of low-grade endometrial stromal sarcoma resembling ovarian sex-cord tumor in the uterus of a 43-year-old woman is described. This tumor belongs to the group II category of uterine tumors resembling ovarian sex-cord tumor described by Clement and Scully, and the epithelial-like elements show prominent smooth muscle differentiation, proved by immunoreactivity for desmin and actin. The patient did not receive any adjuvant therapy; she is alive and well without recurrence 8 months postoperatively.

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