Rapid enlargement of endometrial stromal sarcoma after uterine fibroid embolization for presumed adenomyosis: a case report and literature review

Uterine sarcomas have rarely been diagnosed after uterine artery embolization. It remains unclear whether the diagnostic work-up is required prior to such embolization to prevent a missed diagnosis of sarcomas and a delay in providing definitive treatment. Because of the rarity and heterogeneity of endometrial stromal neoplasms, little is known about their epidemiology, pathogenesis, and molecular pathology. The authors report a case of low-grade endometrial stromal sarcoma (ESS) diagnosed after uterine fibroid embolization. Although they performed laparoscopic biopsy of the rapidly growing uterine mass, they could not detect the ESS. Although rare, ESS should be considered in the differential diagnosis of uterine fibroid enlargement. It is essential to assess the risk of malignancy by taking into account the patient's clinical symptoms, results of the physical exam, and imaging findings prior to uterine artery embolization. Pathologic diagnosis should include an adequate biopsy sample and the use of molecular genetic testing.

Endometrial stromal sarcoma; Uterine leiomyoma; Embolization.

[1] Noventa M., Gizzo S., Conte L., Dalla Toffola A., Litta P., Saccardi C.: “Fertility sparing surgery in young women affected by endometrial stromal sarcoma: an oncologic dilemma or a reliable option? review of literature starting from a peculiar case”. Onco Targets Ther., 2015, 8, 29.

[2] Franquemont D.W., Frierson H.F., Jr., Mills S.E.: “An immunohistochemical study of normal endometrial stroma and endometrial stromal neoplasms. Evidence for smooth muscle differentiation”. Am. J. Surg. Pathol., 1991, 15, 861.

[3] Rauh-Hain J.A., del Carmen M.G.: “Endometrial stromal sarcoma: a systematic review”. Obstet. Gynecol., 2013, 122, 676.

[4] Rothmund R., Hartkopf A., Joachim C., Walter C.B., Wallwiener M., Kraemer B., et al.: “Clinical characteristics, pathological reevaluation, surgical management and adjuvant therapy of patients with endometrial stromal tumors”. Arch. Gynecol. Obstet., 2014, 290, 1195.

[5] Kurjak A., Shalan H., Sosic A., Benic S., Zudenigo D., Kupesic S., et al.: “Endometrial carcinoma in postmenopausal women: evaluation by transvaginal color Doppler ultrasonography”. Am. J. Obstet. Gynecol., 1993, 169, 1597.

[6] Lee C.H., Nucci M.R.: “Endometrial stromal sarcoma - the new genetic paradigm”. Histopathology, 2015, 67, 1.

[7] Chien J.C., Hsieh S.C., Lee R.C., Chen C.Y., Cheng C.J., Chan W.P.: “Endometrial stromal sarcoma mimicking submucosal myoma protruding to the vagina: MRI findings”. Eur. J. Gynaecol. Oncol., 2005, 26, 657.

[8] Buzaglo K., Bruchim I., Lau S.K., Ferenczy A., Tulandi T., Gotlieb W.H.: “Sarcoma post-embolization for presumed uterine fibroids”. Gynecol. Oncol., 2008, 108, 244.

[9] Chang K.L., Crabtree G.S., Lim-Tan S.K., Kempson R.L., Hendrickson M.R.: “Primary uterine endometrial stromal neoplasms. A clinicopathologic study of 117 cases”. Am. J. Surg. Pathol., 1990, 14, 415.

[10] Chiang S., Ali R., Melnyk N., McAlpine J.N., Huntsman D.G., Gilks C.B., et al.: “Frequency of known gene rearrangements in endometrial stromal tumors”. Am. J. Surg. Pathol., 2011, 35, 1364.

[11] Lee C.H., Marino-Enriquez A., Ou W., Zhu M., Ali R.H., Chiang S., et al.: “The clinicopathologic features of YWHAE-FAM22 endometrial stromal sarcomas: a histologically high-grade and clinically aggressive tumor”. Am. J. Surg. Pathol., 2012, 36, 641.

[12] Stewart C.J., Leung Y.C., Murch A., Peverall J.: “Evaluation of fluorescence in-situ hybridization in monomorphic endometrial stromal neoplasms and their histological mimics: a review of 49 cases”. Histopathology, 2014, 65, 473.

[13] He L., Li J.D., Xiong Y., Huang X., Huang L., Lin J.X., et al.: “Clinicopathological and molecular markers associated with prognosis and treatment effectiveness of endometrial stromal sarcoma: a retrospective study in China”. Arch. Gynecol. Obstet., 2014, 289, 383.

[14] Hrzenjak A., Dieber-Rotheneder M., Moinfar F., Petru E., Zatloukal K.: “Molecular mechanisms of endometrial stromal sarcoma and undifferentiated endometrial sarcoma as premises for new therapeutic strategies”. Cancer Lett., 2014, 354, 21.

[15] Park J.Y., Sung C.O., Jang S.J., Song S.Y., Han J.H., Kim K.R.: “Pulmonary metastatic nodules of uterine low-grade endometrial stromal sarcoma: histopathological and immunohistochemical analysis of 10 cases”. Histopathology, 2013, 63, 833.

[16] Goldberg J., Burd I., Price F.V., Worthington-Kirsch R.: “Leiomyosarcoma in a premenopausal patient after uterine artery embolization”. Am. J. Obstet. Gynecol., 2004, 191, 1733.

[17] Kim S.N., Park S.W., Sohn I.S., Lee J.Y., Lee S.-J., Kwon H.S., et al.: “Endometrial stromal sarcoma diagnosed after uterine fibroid embolization”. Eur. J. Radiol. Extra, 2009, 70, e33.