{"paper_id":"4067cc67-b6d7-44ca-b9e3-c53c21b03f68","body_text":"Abstract\nIntroduction\nParaneoplastic coagulation disorders associated with benign and malignant gynecological tumors have been reported. However, reports on cutaneous purpural manifestation of paraneoplastic coagulopathy in cases of gynecological tumors are extremely limited.\nCase report\nA 40-year-old woman was referred due to generalized ecchymosis 7 years after laparoscopic-assisted myomectomy. Coagulogram was markedly deranged with severe thrombocytopenia. After hematological disorders were excluded, image diagnostic modalities showed multiple intramyometrial heterogeneous mass lesions with extensive intratumoral hemorrhage. Laparoscopic-assisted vaginal hysterectomy was performed with supplementation of coagulation factors and platelets. Histological examination showed intramyometrial low-grade endometrial stromal sarcoma (ESS) coexistent with adenomyosis. Coagulation disorder immediately disappeared after hysterectomy and the postoperative course was uneventful. Recurrence of either ESS or coagulopathy has not been noted 15 months postoperatively, to date.\nConclusions\nThis case illustrates a rare but potentially life-threatening consumptive coagulopathy caused by intramyometrial low-grade ESS that was assumed to secondarily arise from stromal cells of adenomyosis developed in postmyomectomy scar.\nSimilar content being viewed by others\nReferences\nNorris HJ, Taylor HB (1966) Mesenchymal tumors of the uterus. I. A clinical and pathological study of 53 endometrial stromal tumors. Cancer 19:755–766\nShah JP, Bryant CS, Kumar S, Ali-Fehmi R, Malone JM Jr, Morris RT (2008) Lymphadenectomy and ovarian preservation in low-grade endometrial stromal sarcoma. Obstet Gynecol 112:1102–1108\nUeda M, Otsuka M, Hatakenaka M, Torii Y (2000) Uterine endometrial stromal sarcoma located in uterine myometrium: MRI appearance. Eur Radiol 10:780–782\nToprak U, Paşaoğlu E, Karademir MA, Gülbay M (2004) Sonographic, CT, and MRI findings of endometrial stromal sarcoma located in the myometrium and associated with peritoneal inclusion cyst. AJR Am J Roentgenol 182:1531–1533\nHarris MG, Bannatyne P, Russell P, Atkinson K, Rickard KA, Kronenberg H (1982) Chronic consumptive coagulopathy with a uterine leiomyoma. Aust N Z J Obstet Gynaecol 22:54–58\nCaputo R, Kanbour-Shakir A (1993) Chronic disseminated intravascular coagulation caused by a uterine leiomyoma. A case report. J Reprod Med 38:737–740\nNakamura Y, Kawamura N, Ishiko O, Ogita S (2002) Acute disseminated intravascular coagulation developed during menstruation in an adenomyosis patient. Arch Gynecol Obstet 267:110–112\nKitao K, Ohara N, Funakoshi T, Moriyama T, Morita H, Kitazawa S, Maruo T (2005) Consumptive coagulopathy that developed in a pregnant woman with degenerated uterine leiomyoma: case report. Clin Exp Obstet Gynecol 32:250\nMatsushita H, Kurabayashi T, Tomita M, Yamamoto Y, Aoki Y, Tanaka K (2001) Disseminated intravascular coagulation associated with intratumoral hemorrhage of ovarian cancer. Gynecol Obstet Invest 51:274–276\nRai P, Rajaram S, Goel N, Ayalur Gopalakrishnan R, Agarwal R, Mehta S (2008) A rare paraneoplastic presentation of a functional ovarian tumor. J Pelvic Med Surg 14:405–407\nThiers BH, Sahn RE, Callen JP (2009) Cutaneous manifestations of internal malignancy. CA Cancer J Clin 59:73–98\nTakeda A, Sakai K, Mitsui T, Nakamura H (2007) Wound retraction system for gasless laparoscopic-assisted myomectomy with a subcutaneous abdominal wall-lift method. J Minim Invasive Gynecol 14:240–246\nChu PG, Arber DA, Weiss LM, Chang KL (2001) Utility of CD10 in distinguishing between endometrial stromal sarcoma and uterine smooth muscle tumors: an immunohistochemical comparison of 34 cases. Mod Pathol 14:465–471\nPanganamamula UR, Harmanli OH, Isik-Akbay EF, Grotegut CA, Dandolu V, Gaughan JP (2004) Is prior uterine surgery a risk factor for adenomyosis? Obstet Gynecol 104:1034–1038\nOstrzenski A (1998) Extensive iatrogenic adenomyosis after laparoscopic myomectomy. Fertil Steril 69:143–145\nConflict of interest statement\nNone.\nAuthor information\nAuthors and Affiliations\nCorresponding author\nRights and permissions\nAbout this article\nCite this article\nTakeda, A., Imoto, S., Mori, M. et al. Paraneoplastic consumptive coagulopathy related to intramyometrial low-grade endometrial stromal sarcoma coexistent with adenomyosis diagnosed 7 years after laparoscopic-assisted myomectomy. Arch Gynecol Obstet 282, 665–670 (2010). https://doi.org/10.1007/s00404-010-1376-6\nReceived:\nAccepted:\nPublished:\nIssue date:\nDOI: https://doi.org/10.1007/s00404-010-1376-6","source_license":"CC0","license_restricted":false}