Benign metastatic leiomyoma: literature review
https://doi.org/10.17650/1994-4098-2025-21-2-115-122
Abstract
The review systematises literature data on pathogenesis, clinical course and treatment of benign metastasising leiomyoma. Benign metastatic leiomyoma is a rare disease, occurring in women of reproductive period, characterised by the presence of multiple benign indolent smooth-muscle mitotically inactive tumours (metastases) of ectopic localisation with a history of uterine leiomyoma and no other tumour process. It is presented in various forms: peritoneal leiomyomatosis, intravenous leiomyomatosis involvement of parenchymal organs, bones, skeletal muscles. Currently, there are no standards for the treatment of patients with benign metastasising leiomyoma. Treatment depends on localisation of metastases and clinical manifestations of the disease: dynamic observation, hormone therapy or surgical treatment.
About the Authors
Kh. B. KotivRussian Federation
Khristina Bogdanovna Kotiv
68 Leningradskaya St., Pesochnyy Settlement, Saint Petersburg 197758
41 Kirochnaya St., Saint Petersburg 191015
Competing Interests:
The authors declare no conflict of interest
E. M. Khmelevskaia
Russian Federation
68 Leningradskaya St., Pesochnyy Settlement, Saint Petersburg 197758
41 Kirochnaya St., Saint Petersburg 191015
Competing Interests:
The authors declare no conflict of interest
I. V. Berlev
Russian Federation
68 Leningradskaya St., Pesochnyy Settlement, Saint Petersburg 197758
41 Kirochnaya St., Saint Petersburg 191015
Competing Interests:
The authors declare no conflict of interest
References
1. Adamyan L.V., Andreeva E.N., Artymuk N.V. et al. Uterine fibroids: diagnostics, treatment and rehabilitation. Clinical guidelines for patient management. Moscow: Izd-vo Nauchnogo tsentra akusherstva, ginekologii i perinatologii im. V.I. Kulakova, 2015. (In Russ.).
2. Pechetov A.A., Lednev A.N., Ratnikova N.K., Volchanskiy D.A. Benign metastatic uterine leiomyoma with lung involvement: problems of diagnosis and treatment. Khirurgiya. Zhurnal im. N.I. Pirogova = Surgery. Journal named after N.I. Pirogov 2020;(9):85–8. (In Russ.).
3. Steiner P.E. Metastasizing fibroleiomyoma of the uterus: Report of a case and review of the literature. Am J Pathol 1939;15(1):89–110.7.
4. Robboy S.J., Bentley R.C., Butnor K., Anderson M.C. Pathology and pathophysiology of uterine smooth-muscle tumors. Environ Health Perspect 2000;108(Suppl 5):779–84. DOI: 10.1289/ehp.00108s5779
5. Savostikova M.V., Levchenko N.E., Laktionov K.P. et al. Mesenchymal tumors of the uterine body. Onkoginekologiya = Oncogynecology 2014;(3):11–22. (In Russ.).
6. Awonuga A.O., Rotas M., Imudia A.N. et al. Recurrent benign metastasizing leiomyoma after hysterectomy and bilateral salpingo oophorectomy. Arch Gynecol Obstet 2008;278:373–6.
7. Al-Talib A., Tulandi T. Pathophysiology and possible iatrogenic cause of leiomyomatosis peritonealis disseminata. Gynecol Obstet Invest 2010;69(4):239–4.
8. Abell M.R., Littler E.R. Benign metastasizing uterine leiomyoma. Multiple lymph nodal metastases. Cancer 1975;36(6):2206–13.
9. Taubert H.D., Wissner S.E., Haskins A.L. Leiomyomatosis peritonealis disseminata: An unusual complication of genital leiomyomata. Obstet Gynecol 1965;25:561–74.
10. Danikas D., Goudas V.T., Rao C.V., Brief D.K. Luteinizing hormone receptor expression in leiomyomatosis peritonealis disseminata. Obstet Gynecol 2000;95(6 Pt 2):1009–11.
11. Nucci M.R., Drapkin R., Dal Cin P. et al. Distinctive cytogenetic profile in benign metastasizing leiomyoma: Pathogenetic implications. Am J Surg Pathol 2007;31(5):737–43.
12. Rivera J.A., Christopoulos S., Small D., Trifiro M. Hormonal manipulation of benign metastasizing leiomyomas: Report of two cases and review of the literature. J Clin Endocrinol Metab 2004;89(7):3183–8. DOI: 10.1210/jc.2003-032021
13. Bekkers R.L., Willemsen W.N., Schijf C.P. et al. Leiomyomatosis peritonealis disseminata: Does malignant transformation occur? A literature review. Gynecol Oncol 1999.
14. Herrero J., Kamali P., Kirschbaum M. Leiomyomatosis peritonealis disseminata associated with endometriosis: A case report and literature review. Eur J Obstet Gynecol Reprod Biol 1998;76(2):189–91.
15. Hoynck van Papendrecht H.P., Gratama S. Leiomyomatosis peritonealis disseminata. Eur J Obstet Gynecol Reprod Biol 1983;14(4):251–9.
16. Arai T., Yasuda Y., Takaya T., Shibayama M. Natural decrease of benign metastasizing leiomyoma. Chest 2000;117(3):921–2.
17. Egberts J.H., Schafmayer C., Bauerschlag D.O. et al. Benign abdominal and pulmonary metastasizing leiomyoma of the uterus. Arch Gynecol Obstet 2006;274(5):319–22.
18. Wentling G.K., Sevin B.U., Geiger X.J., Bridges M.D. Benign metastasizing leiomyoma responsive to megestrol: Case report and review of the literature. Int J Gynecol Cancer 2005;15:1213–7.
19. Funakoshi Y., Sawabata N., Takeda S. et al. Pulmonary benign metastasizing leiomyoma from the uterus in a postmenopausal woman: Report of a case. Surg Today 2004;34(1):55–7.
20. Fan R., Feng F., Yang H. et al. Pulmonary benign metastasizing leiomyomas: A case series of 23 patients at a single facility. BMC Pulm Med 2020;20(1):292.
21. Barnaś E., Książek M., Raś R. et al. Benign metastasizing leiomyoma: A review of current literature in respect to the time and type of previous gynecological surgery. PLoS One 2017;12(4):e0175875. DOI: 10.1371/journal.pone.0175875
22. Kayser K., Zink S., Schneider T. et al. Benign metastasizing leiomyoma of the uterus: Documentation of clinical, immunohistochemical and lectin-histochemical data of ten cases. Virchows Arch 2000;437(3):284–92. DOI: 10.1007/s004280000207
23. Kotiv Kh.B., Khmelevskaya E.M., Nyuganen A.O. et al. Clinical case: Bilateral lung damage by benign metastatic leiomyoma. Moscow, 2025. (In Russ.).
24. Rao A.V., Wilson J., Sylvester K. Pulmonary benign metastasizing leiomyoma following hysterectomy: A clinicopathologic correlation. J Thorac Oncol 2008;3(6):674–6. DOI: 10.1097/JTO.0b013e3181757a46
25. Koh D.M., Burn P.R., King D.M. Benign metastasizing leiomyoma with intracaval leiomyomatosis. Br J Radiol 2000;73(868):435–7. DOI: 10.1259/bjr.73.868.10844871
26. Takemura G., Takatsu Y., Kaitani K. et al. Metastasizing uterine leiomyoma. A case with cardiac and pulmonary metastasis. Pathol Res Pr 1996;192(6):622–9. DOI: 10.1016/S0344-0338(96)80116-6
27. Jo J.H., Lee J.H., Kim D.C. et al. A case of benign metastasizing leiomyoma with multiple metastasis to the soft tissue, skeletal muscle, lung and breast. Korean J Intern Med 2006;21(3):199–201. DOI: 10.3904/kjim.2006.21.3.199
28. Mauduit M., Langouet Q., Rouzé S. et al. Post-menopausal pulmonary leiomyoma metastasis: An unexpected finding. Indian J Thorac Cardiovasc Surg 2018;34(4):513–5. DOI: 10.1007/s12055-018-0676-5
29. Huang L., Shi G., Wang Q. et al. Pulmonary and mediastinum metastasis of uterine leiomyoma: A case report. Med Baltim 2019;98(49):e18276. DOI: 10.1097/MD.0000000000018276
30. Yoon G., Kim T.J., Sung C.O. et al. Benign metastasizing leiomyoma with multiple lymph node metastasis: A case report. Cancer Res Treat 2011;43(2):131–3. DOI: 10.4143/crt.2011.43.2.131
31. André D., Gouveia F., Luís H. et al. Benign metastasizing leiomyoma – a case of benign metastasis. JRSM Open 2021. DOI: 10.1177/20542704211064482
32. Consamus E.N., Reardon M.J., Ayala A.G. et al. Metastasizing leiomyoma to heart. Methodist Debakey Cardiovasc J 2014;10(4):251–4. DOI: 10.14797/mdcj-10-4-251
33. Meddeb M., Chow R.D., Whipps R., Haque R. The heart as a site of metastasis of benign metastasizing leiomyoma: Case report and review of the literature. Case Rep Cardiol 2018. DOI: 10.1155/2018/7231326
34. Reis Soares R., Ferber Drumond L., Soares da Mata D. et al. Cardiac metastasizing leiomyoma: A case report. Int J Surg Case Rep 2020;7:647–50. DOI: 10.1016/j.ijscr.2020.11.095
35. Efared B., Atsame-Ebang G., Sani R. et al. Unexpected pulmonary tumor: Metastasis from a benign uterine leiomyoma in a post menopausal woman. A case report. BMC Res Notes 2017;10(1):662. DOI: 10.1186/s13104-017-2998-6
36. Ferreira A., Malheiro M., Martins A. Spinal cord compression secondary to benign metastasizing leiomyoma. Cureus 2022;14(2):e21845. DOI: 10.7759/cureus.21845
37. Otsuka S., Yanazume S., Mizuno M. et al. Metastatic leiomyoma following menopause: A case report and review of literature. Cureus 2022;14(11):e31549. DOI: 10.7759/cureus.31549
38. Karnib M., Rhea I., Elliott R. et al. Benign metastasizing leiomyoma in the heart of a 45-year-old woman. Tex Heart Inst J 2021;48(1):e197066. DOI: 10.14503/THIJ-19-7066
39. Khan M., Faisal A., Ibrahim H. et al. Pulmonary benign metastasizing leiomyoma: A case report. Respir Med Case Rep 2018;24:117–21. DOI: 10.1016/j.rmcr.2018.04.017
40. Choe Y.H., Jeon S.Y., Lee Y.C. et al. Benign metastasizing leiomyoma presenting as multiple cystic pulmonary nodules: A case report. BMC Women Health 2017;17(1):81. DOI: 10.1186/s12905-017-0435-6
41. Kang M.W., Kang S.K., Yu J.H. et al. Benign metastasizing leiomyoma: Metastasis to rib and vertebra. Ann Thorac Surg 2011;91(3):924–6. DOI: 10.1016/j.athoracsur.2010.08.030
42. Berti A.F., Santillan A., Velasquez L.A. Benign metastasizing leiomyoma of the cervical spine 31 years after uterine leiomyoma resection. J Clin Neurosci 2015;22(9):1491–2. DOI: 10.1016/j.jocn.2015.02.031
43. Awonuga A.O., Shavell V.I., Imudia A.N. et al. Pathogenesis of benign metastasizing leiomyoma: A review. Obstet Gynecol Surv 2010;6(3):189–95. DOI: 10.1097/OGX.0b013e3181d60f93
44. Kuo T., London S.N., Dinh T.V Endometriosis occurring in leiomyomatosis peritonealis disseminata: Ultrastructural study and histogenetic consideration. Am J Surg Pathol 1980;4(2): 197–204. DOI: 10.1097/00000478-198004000-00012
45. Hoetzenecker K., Ankersmit H.J., Aigner C. et al. Consequences of a wait-and-see strategy for benign metastasizing leiomyomatosis of the lung. Ann Thorac Surg 2009;87:613–4. DOI: 10.1016/j.athoracsur.2008.06.052
46. Hague W.M., Abdulwahid N.A., Jacobs H.S., Craft I. Use of LHRH analogue to obtain reversible castration in a patient with benign metastasizing leiomyoma. Br J Obstet Gynaecol 1986;93(5):455–60.
47. Cohen J.D., Robins H.I. Response of “benign” metastasizing leiomyoma to progestin withdrawal. Case report. Eur J Gynaecol Oncol 1993;14:44, 45.
48. Motegi M., Takayanagi N., Sando Y. et al A case of so-called benign metastasizing leiomyoma responsive to progesterone. Nihon Kyobu Shikkan Gakkai Zasshi 1993;31:890–5.
49. Ando H., Kusunoki S., Ota T. et al. Long-term efficacy and safety of aromatase inhibitor use for leiomyomatosis peritonealis disseminata. J Obstet Gynaecol Res 2017;43(9):1489–92. DOI: 10.1111/jog.13376
50. Vilos G.A., Allaire C., Laberge P.Y., Leyland N. The management of uterine leiomyomas. J Obstet Gynaecol Can 2015;37(2):157–78.
51. Altinoz M.A., Ozpinar A., Elmaci I. Reproductive epidemiology of glial tumors may reveal novel treatments: High-dose progestins or progesterone antagonists as endocrino-immune modifiers against glioma. Neurosurg Rev 2018;173:20–30. DOI: 10.1007/s10143-018-0953-1
52. Jautzke G., Müller-Ruchholtz E., Thalmann U. Immunohistological detection of estrogen and progesterone receptors in multiple and well differentiated leiomyomatous lung tumors in women with uterine leiomyomas (so-called benign metastasizing leiomyomas). A report on 5 cases. Pathol Res Pr 1996;192(3):215–23. DOI: 10.1016/S0344-0338(96)80224-X
53. Lewis E.I., Chason R.J., DeCherney A.H. et al. Novel hormone treatment of benign metastasizing leiomyoma: An analysis of five cases and literature review. Fertil Steril 2013;99(7):2017–24. DOI: 10.1016/j.fertnstert.2013.01.147
54. Safrai M., Chill H.H., Reuveni Salzman A., Shushan A. Selective progesterone receptor modulators for the treatment of uterine leiomyomas. Obstet Gynecol 2017;130(2):315–8. DOI: 10.1097/AOG.0000000000002143
55. Reis F.M., Bloise E., Ortiga-Carvalho T.M. Hormones and pathogenesis of uterine fibroids. Best Pr Res Clin Obstet Gynaecol 2016;34:13–24. DOI: 10.1016/j.bpobgyn.2015.11.015
56. Kortekaas K.E., Pelikan H.M.P. Non-surgical intervention for retroperitoneal lymphogenic and pulmonary metastases of a benign leiomyoma: Treatment with ulipristal acetate. BMJ Case Rep 2018;2018:bcr2017222693. DOI: 10.1136/bcr-2017-222693
57. Jiang J., He M., Hu X. et al. Deep sequencing reveals the molecular pathology characteristics between primary uterine leiomyoma and pulmonary benign metastasizing leiomyoma. Clin Transl Oncol 2018;20(8):1080–6. DOI: 10.1007/s12094-018-1847-y
58. Ottlakan A., Borda B., Lazar G. et al. Treatment decision based on the biological behavior of pulmonary benign metastasizing leiomyoma. J Thorac Dis 2016;8(8):E672–6. DOI: 10.21037/jtd.2016.06.61
Review
For citations:
Kotiv Kh.B., Khmelevskaia E.M., Berlev I.V. Benign metastatic leiomyoma: literature review. Tumors of female reproductive system. 2025;21(2):115-122. (In Russ.) https://doi.org/10.17650/1994-4098-2025-21-2-115-122