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Extraoseus Ewing‘s Sarcoma, Primary Affection of Uterine Cervix –  Case Report


Authors: O. Bílek 1;  M. Holánek 1;  M. Zvaríková 1;  P. Fabian 2;  B. Robešová 3;  M. Procházková 4;  D. Adámková Krákorová 1
Authors place of work: Klinika komplexní onkologické péče, Masarykův onkologický ústav, Brno 1;  Oddělení klinické a experimentální patologie, Masarykův onkologický ústav, Brno 2;  Centrum molekulární bio logie a genové terapie, Interní hematologická a onkologická klinika LF MU a FN Brno 3;  Oddělení radiologie, Masarykův onkologický ústav, Brno 4
Published in the journal: Klin Onkol 2015; 28(4): 284-287
Category: Case Report
doi: https://doi.org/10.14735/amko2015284

Summary

Background:
Ewing‘s sarcoma is usually dia­gnosed in adolescents and young adults, peak of incidence is around 15 years of age. Primary localization is mostly in the skeleton of long bones and chest wall. Primary extraosseous involvement rarely occurs, incidence increases with age.

Case:
We present a case report of a 57‑year‑ old patient with locally advanced tumors of the cervix, clinical stage IIB. Due to histological and molecular genetic examination revealing EWS‑ERG fusion gene, Ewing‘s sarcoma was dia­gnosed. CT revealed pathological pelvic lymphadenopathy and multiple pulmonary bilateral methastases, scintigraphy did not prove any affection of skeleton. The patient underwent a two‑stage intensive chemotherapy regimens VIDE (vincristine, ifosfamide, doxorubicin, etoposide) and VAI (vincristine, actinomycin D, ifosfamide). During the second phase, concomitant radiotherapy of pelvis was aplied. According to PET/ CT, complete remission was achieved. Whole‑ lung irradiation was applied in consolidation of the result.

Conclusion:
Primary Ewing‘s sarcoma of the cervix is an extremely rare disease. To our knowledge, only 12 cases was presented until this time. The average age at time of dia­gnosis was 35 years. Unlike the previous reports, we initially dia­gnosed distant metastases. The treatment was led according to the protocol Ewing 2008 designed for primary skeletal Ewing‘s sarcoma. Currently, 18 months after the therapy, the patient is without signs of disease. However, long‑term follow-up is necessary.

Key words:
Ewing‘s sarcoma –  uterine cervix –  cytogenetics – EWS-ERG – chemotherapy – radiotherapy

The authors declare they have no potential conflicts of interest concerning drugs, products, or services used in the study.

The Editorial Board declares that the manuscript met the ICMJE “uniform requirements” for biomedical papers.

Submitted:
3. 6. 2015

Accepted:
25. 7. 2015


Zdroje

1. Lin PP, Wang Y, Lozano G. Mesenchymal stem cells and the origin of Ewing’s sarcoma. Sarcoma 2011: 276463. doi: 10.1155/ 2011/ 276463.

2. Bleyer WA, Barr RD. Cancer in adolescents and young adults, bone cancer. New York: Springer‑ Verlag 2007: 203– 215.

3. Zöllner, S, Dirksen U, Jürgens H et al. Renal Ewing tumors. Ann Oncol 2013; 24(9): 2455– 2461. doi: 10.1093/ annonc/ mdt215.

4. Hwang, SK, Kim DK, Park SI. Primary Ewing‘s sarcoma of the lung. Korean J Thorac Cardiovasc Surg 2014; 47(1): 47– 50.

5. Kim HS, Kim S, Min YD et al. Ewing‘s sarcoma of the stomach; rare case of Ewing‘s sarcoma and suggestion of new treatment strategy. J Gastric Cancer 2012; 12(4): 258– 261. doi: 10.5230/ jgc.2012.12.4.258.

6. Ozaki Y, Miura Y, Koganemaru S et al. Ewing sarcoma of the liver with multilocular cystic mass formation: a case report. BMC Cancer 2015; 15(1): 16. doi: 10.1186/ s12885‑ 015‑ 1017‑ 3.

7. Sharma P, Bakshi H, Chheda Y et al. Primary Ewing’ssarcoma of penis –  a rare case report. Indian J Surg Oncol 2011; 2(4): 332– 333. doi: 10.1007/ s13193‑ 011‑ 0112‑ 4.

8. Hayes‑ Jordan A, Anderson PM. The dia­gnosis and management of desmoplastic small round cell tumor: a review. Curr Opin Oncol 2011; 23(4): 385– 389. doi: 10.1097/ CCO.0b013e3283477aab.

9. Kavalar R, Pohar Marinšek Z, Jereb B et al. Prognostic value of immunohistochemistry in the Ewing’s sarcoma family of tumors. Med Sci Monit 2009; 15(8): CR442– CR452.

10. Rocchi A, Manara MC, Sciandra M et al. CD99 inhibits neural differentiation of human Ewing sarcoma cells and thereby contributes to oncogenesis. J Clin Invest 2010; 120(3): 668– 680. doi: 10.1172/ JCI36667.

11. Kim SH, Choi EY, Shin YK et al. Generation of cells with Hodgkin’s and Reed‑ Sternberg phenotype through downregulation of CD99 (Mic2). Blood 1998; 92(11): 4287– 4295.

12. Hahn JH, Kim MK, Choi EY et al. CD99 (MIC2) regulates the LFA‑ 1/ ICAM‑1- mediated adhesion of lymphocytes, and its gene encodes both positive and negative regulators of cellular adhesion. J Immunol 1997; 159(5): 2250– 2258.

13. Procházka P, Vícha A, Kodet R et al. Nádory ze skupiny Ewingova sarkomu –  molekulární bio­logie a genetika. Klin Onkol 2007; 20(2): 205– 208.

14. Bajčiová V, Štěrba J, Tomášek J et al. Nádory adolescentů a mladých dospělých. Praha: Grada 2011: 108– 114.

15. De Alava, E, Kawai A, Healey JH et al. EWS‑ FLI1 fusion transcript structure is an independent determinant of prognosis in Ewing‘s sarcoma. J Clin Oncol 1998; 16(4): 1248– 1255.

16. Le Deley MC, Delattre O, Schaefer KL et al. Impact of EWS‑ ETS Psion type on disease progression in Ewing’s sarcoma/ peripheral primitive neuroectodermal tumor: prospective results from the cooperative Euro‑E.W.I.N.G. 99 trial. J Clin Oncol 2010; 28(12): 1982– 1988. doi: 10.1200/ JCO.2009.23.3585.

17. Van Doorninck JA, Ji L, Schaub B et al. Current treatment protocols have eliminated the prognostic advantage of type 1 fusions in Ewing sarcoma: a report from the Children’s Oncology Group. J Clin Oncol 2010; 28: 1989– 1994. doi: 10.1200/ JCO.2009.24.5845.

18. Orr WS, Denbo JW, Billups C et al. Analysis of prog­nostic factors in extraosseous Ewing sarcoma family of tumors: review of St. Jude Children’s Research Hospital experience. Ann Oncol 2012; 19(12): 3816– 3822. doi: 10.1245/ s10434‑ 012‑ 2458‑ 4.

19. Ladenstein R, Pötschger U, Le Deley MC et al. Primary disseminated multifocal Ewing sarcoma: results of the Euro‑EWING 99 trial. J Clin Oncol 2010; 28(20): 3284– 3291. doi: 10.1200/ JCO.2009.22.9864.

20. Thacker MM, Temple HT, Scully SP. Current treatment for Ewing‘s sarcoma. Expert Rev Anticancer Ther 2005; 5(2): 319– 331.

21. Spiller M, Bisogno G, Ferrari A et al. Prognostic factors in localized extraosseous Ewing family tumors. Pediatr Blood Cancer 2006; 46(10): A‑ PD.024, 434.

22. Le Deley MC, Paulussen M, Lewis I et al. Cyclophosphamide compared with ifosfamide in consolidation treatment of standard‑ risk ewing sarcoma: results of the randomized noninferiority Euro‑EWING99-R1 trial. J Clin Oncol 2014; 32(23): 2440– 2448. doi: 10.1200/ JCO. 2013.54.4833.

23. Hou MM, Xi MR, Yang KX. A rare case of extraosseous Ewing sarcoma primarily arising in the ovary. Chin Med J2013; 126(23): 4597.

24. Park JY, Lee S, Kang HJ et al. Primary Ewing‘s sarcoma –  primitive neuroectodermal tumor of the uterus: a case report and literature review. Gynecol Oncol 2007; 106(2): 427– 432.

25. Farley J, O‘Boyle JD, Heaton J et al. Extraosseous Ewing sarcoma of the vagina. Obstet Gynecol 2000; 96(5 Pt 2): 832– 834.

26. Russin VL, Valente PT, Hanjani P. Psammoma bodies in neuroendocrine carcinoma of the uterine cervix. Acta Cytol 1987; 31(6): 791– 795.

27. Sato S, Yajima A, Kimura N et al. Peripheral neuroepithelioma (peripheral primitive neuroectodermal tumor) of the uterine cervix. Tohoku J Exp Med 1996; 180(2): 187– 195.

28. Horn LC, Fischer U, Bilek K. Primitive neuroectodermal tumor of the cervix uteri. A case report. Gen Dia­gn Pathol 1997; 142(3– 4): 227– 230.

29. Cenacchi G, Pasquinelli G, Montanaro L et al. Primary endocervical extraosseous Ewing’s sarcoma/ PNET. Int J Gynecol Pathol 1998; 17(1): 83– 88.

30. Pauwels P, Ambros P, Hattinger C et al. Peripheral primitive neuroectodermal tumour of the cervix. Virchows Arch 2000; 436(1): 68– 73.

31. Tsao AS, Roth LM, Sandler A et al. Cervical primitive neuroectodermal tumor. Gynecol Oncol 2001; 83(1): 138– 142.

32. Malpica A, Moran CA. Primitive neuroectodermal tumor of the cervix: a clinicopathologic and immunohistochemical study of two cases. Ann Dia­gn Pathol 2002; 6(5): 281– 287.

33. Goda JS, Nirah B, Mayur K. Primitive neuroectodermal tumour of the cervix: a rare entity. Internet J Radiol 2007; 6: 3.

34. Snijders‑ Keilholz A, Ewing P, Seynaeve C et al. Primitive neuroectodermal tumor of the cervix uteri: a case report –  changing concepts in therapy. Gynecol Oncol 2005; 98(3): 516– 519.

35. Farzaneh F, Rezvani H, Boroujeni PT et al. Primitive neuroectodermal tumor of the cervix: a case report. J Med Case Rep 2011; 5: 489.

36. Li B, Ouyang L, Han X et al. Primary primitive neuroectodermal tumor of the cervix. Onco Targets Ther 2013; 6: 707– 711. doi: 10.2147/ OTT.S45889.

Štítky
Paediatric clinical oncology Surgery Clinical oncology

Článok vyšiel v časopise

Clinical Oncology

Číslo 4

2015 Číslo 4
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