A case of undifferentiated carcinoma arising from adenomyosis

Malignant transformation of adenomyosis is rare, with endometrioid carcinoma being the most common histological type. Herein, we report the first extremely aggressive case of undifferentiated carcinoma arising from adenomyosis. A 59-year-old Japanese woman was referred to our hospital with complaints of general fatigue, abdominal fullness, and loss of appetite. Contrast-enhanced computed tomography (CT) and integrated whole-body positron emission tomography/CT using 18F-fluorodeoxyglucose revealed multiple para-aortic and pelvic lymph node swellings. Contrast-enhanced magnetic resonance imaging revealed a 3-cm degenerated intramural uterine tumor. The patient underwent total hysterectomy, bilateral salpingo-oophorectomy, para-aortic and pelvic lymphadenectomy, and partial omentectomy for the treatment. Pathologically, the patient was diagnosed with undifferentiated carcinoma arising from adenomyosis. Despite adjuvant chemotherapy, the patient died of the disease 7 months after the initial surgery. Further investigation of similar cases may reveal the clinical features of this disease. Similar content being viewed by others Data availability The data are available on the request to the corresponding author.

Bulun SE, Yildiz S, Adli M, Wei JJ (2021) Adenomyosis pathogenesis: insights from next-generation sequencing. Hum Reprod Update 27:1086–1097. https://doi.org/10.1093/humupd/dmab017 Koike N, Tsunemi T, Uekuri C, Akasaka J, Ito F, Shigemitsu A et al (2013) Pathogenesis and malignant transformation of adenomyosis (review). Oncol Rep 29:861–867. https://doi.org/10.3892/or.2012.2184 Wang J, Wang Q, Wang W, Yang J, Xia J, Wei Y (2023) Endometrioid adenocarcinoma arising in adenomyosis in a patient with pelvic organ prolapse-case report. BMC Womens Health 23:150. https://doi.org/10.1186/s12905-023-02310-6 Munro MG (2020) Classification and reporting systems for adenomyosis. J Minim Invasive Gynecol 27:296–308. https://doi.org/10.1016/j.jmig.2019.11.013 Sam M, Raubenheimer M, Manolea F, Aguilar H, Mathew RP, Patel VH et al (2020) Accuracy of findings in the diagnosis of uterine adenomyosis on ultrasound. Abdom Radiol (NY) 45:842–850. https://doi.org/10.1007/s00261-019-02231-9 Puppa G, Shozu M, Perin T, Nomura K, Gloghini A, Campagnutta E et al (2007) Small primary adenocarcinoma in adenomyosis with nodal metastasis: A case report. BMC Cancer 7:103. https://doi.org/10.1186/1471-2407-7-103 Chao X, Wu M, Ma S, Tan X, Zhong S, Bi Y et al (2020) The clinicopathological characteristics and survival outcomes of endometrial carcinoma coexisting with or arising in adenomyosis: A pilot study. Sci Rep 10:5984. https://doi.org/10.1038/s41598-020-63065-w Raffone A, Raimondo D, Maletta M, Travaglino A, Renzulli F, Neola D et al (2023) Endometrial Cancer Arising in Adenomyosis (EC-AIA): A Systematic Review. Cancers (Basel) 15:1142. https://doi.org/10.3390/cancers15041142 Baba A, Yamazoe S, Dogru M, Ogawa M, Takamatsu K, Miyauchi J (2016) Clear cell adenocarcinoma arising from adenomyotic cyst: A case report and literature review. J Obstet Gynaecol Res 42:217–223. https://doi.org/10.1111/jog.12866 Kiuchi K, Hasegawa K, Kanamori A, Machida H, Kojima M, Fukasawa I (2016) Carcinosarcoma arising from uterine adenomyosis: A case report. J Obstet Gynaecol Res 42:358–362. https://doi.org/10.1111/jog.12901 Moraru L, Mitranovici MI, Chiorean DM, Moraru R, Caravia L, Tiron AT et al (2023) Adenomyosis and Its Possible Malignancy: A Review of the Literature. Diagnostics (Basel) 13:1883. https://doi.org/10.3390/diagnostics13111883 Colman HI, Rosenthal AH (1959) Carcinoma developing in areas of adenomyosis. Obstet Gynecol 14:342–348 Machida H, Maeda M, Cahoon SS, Scannell CA, Garcia-Sayre J, Roman LD et al (2017) Endometrial cancer arising in adenomyosis versus endometrial cancer coexisting with adenomyosis: Are these two different entities? Arch Gynecol Obstet 295:1459–1468. https://doi.org/10.1007/s00404-017-4375-z Talia KL, Naaman Y, McCluggage WG (2021) Uterine adenosarcoma originating in adenomyosis: report of an extremely rare phenomenon and review of published literature. Int J Gynecol Pathol 40:342–348. https://doi.org/10.1097/PGP.0000000000000709 Reinhold C, Tafazoli F, Mehio A, Wang L, Atri M, Siegelman ES et al (1999) Uterine adenomyosis: endovaginal US and MR imaging features with histopathologic correlation. Radiographics 19:S147–S160. https://doi.org/10.1148/radiographics.19.suppl_1.g99oc13s147 Manfredi R, Mirk P, Maresca G, Margariti PA, Testa A, Zannoni GF et al (2004) Local-regional staging of endometrial carcinoma: role of MR imaging in surgical planning. Radiology 231:372–378. https://doi.org/10.1148/radiol.2312021184

We would like to thank Editage (www.editage.jp) for English language editing. Author information Authors and Affiliations Corresponding author Ethics declarations Conflict of interest None declared. Additional information Publisher’s note Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations. About this article Cite this article Aoyagi, Y., Nasu, K., Nishida, H. et al. A case of undifferentiated carcinoma arising from adenomyosis. Int Canc Conf J (2026). https://doi.org/10.1007/s13691-026-00874-2 Received: Accepted: Published: Version of record: DOI: https://doi.org/10.1007/s13691-026-00874-2