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Primary yolk sac tumor of the endometrium: a case report and review of the literatures

Abstract

Purpose

Yolk sac tumor (YST) is a malignant tumor derived from germ cells and usually occurs in the gonads. Extra-gonadal YST is most commonly seen in the vagina of children, but rarely in the cervix, vulva and endometrium. Primary YST of endometrium was extremely rare, standard treatment was still controversial and no guideline was established so far. The aim of the present study was to provide a comprehensive understanding and systematic thought for the management of primary YST of endometrium.

Methods

A systematic research of the literature was conducted in Scopus, PubMed database and Cochrane Library, including case reports and case series. We summarized clinical characteristics, treatments and prognosis of all collected cases. We collected data regarding patients, serum AFP level, initial symptoms, surgical information, postoperative chemotherapy and radiotherapy. A new case was also discussed.

Results

We found only 26 cases have been reported previously. We reported a new case of primary endometrial YST in a 27-year-old woman, and in this case, we creatively performed bilaterally ovarian preservation and used DC (docetaxel and carboplatin) regimen of postoperative chemotherapy, we achieved a relatively good prognosis during the follow-up period of 14 months.

Conclusion

Primary YST of endometrium, kind of highly malignant germ cell tumors, was extremely rare, of which initial symptom is usually abnormal vaginal bleeding. Standard treatment was still controversial and no guideline was established so far. Surgery combining with postoperative chemotherapy was considered effective for treatment of primary endometrial YST. Decision on whether to preserve ovaries in young patient with early stage needs careful consideration, comprehensive preoperative assessment and full communication. Intraoperative biopsy and strict postoperative follow-up are recommended. However, standard chemotherapy regimen and feasibility of postoperative radiotherapy remains to be discussed.

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References

  1. Smith HO, Berwick M, Verschraegen CF, Wiggins C, Lansing L, Muller CY, Qualls CR (2006) Incidence and survival rates for female malignant germ cell tumors. Obstet Gynecol 107(5):1075–1085. https://doi.org/10.1097/01.AOG.0000216004.22588.ce

    Article  PubMed  Google Scholar 

  2. Pasternack T, Shaco-Levy R, Wiznitzer A, Piura B (2008) Extraovarian pelvic yolk sac tumor: case report and review of published work. J Obstet Gynaecol Res 34(4 Pt 2):739–744. https://doi.org/10.1111/j.1447-0756.2008.00725.x

    Article  PubMed  Google Scholar 

  3. Clement PB, Young RH, Scully RE (1988) Extraovarian pelvic yolk sac tumors. Cancer 62(3):620–626. https://doi.org/10.1002/1097-0142(19880801)62:3%3c620:aid-cncr2820620330%3e3.0.co;2-p

    CAS  Article  PubMed  Google Scholar 

  4. Tao T, Yang J, Cao D, Guo L, Chen J, Lang J, Shen K (2012) Conservative treatment and long-term follow up of endodermal sinus tumor of the vagina. Gynecol Oncol 125(2):358–361. https://doi.org/10.1016/j.ygyno.2011.12.430

    Article  PubMed  Google Scholar 

  5. Amant F, Mirza MR, Koskas M, Creutzberg CL (2018) Cancer of the corpus uteri. Int J Gynaecol Obstet 143(Suppl 2):37–50. https://doi.org/10.1002/ijgo.12612

    Article  PubMed  Google Scholar 

  6. Pileri S, Martinelli G, Serra L, Bazzocchi F (1980) Endodermal sinus tumor arising in the endometrium. Obstet Gynecol 56(3):391–396

    CAS  PubMed  Google Scholar 

  7. Joseph MG, Fellows FG, Hearn SA (1990) Primary endodermal sinus tumor of the endometrium—a clinicopathological, immunocytochemical, and ultrastructural-study. Cancer 65(2):297–302. https://doi.org/10.1002/1097-0142(19900115)65:2%3c297:Aid-Cncr2820650219%3e3.0.Co;2-E

    CAS  Article  PubMed  Google Scholar 

  8. Ohta M, Sakakibara K, Mizuno K, Kano T, Matsuzawa K, Tomoda Y, Nakashima N, Ogawa T (1988) Successful treatment of primary endodermal sinus tumor of the endometrium. Gynecol Oncol 31(2):357–364

    CAS  Article  Google Scholar 

  9. Spatz A, Bouron D, Pautier P, Castaigne D, Duvillard P (1998) Primary yolk sac tumor of the endometrium: a case report and review of the literature. Gynecol Oncol 70(2):285–288. https://doi.org/10.1006/gyno.1998.5036

    CAS  Article  PubMed  Google Scholar 

  10. Patsner B (2002) Primary endodermal sinus tumor of the endometrium presenting as “recurrent” endometrial adenocarcinoma—reply. Gynecol Oncol 84(1):184. https://doi.org/10.1006/gyno.2001.6240

    Article  Google Scholar 

  11. Oguri H, Sumitomo R, Maeda N, Fukaya T, Moriki T (2006) Primary yolk sac tumor concomitant with carcinosarcoma originating from the endometrium: case report. Gynecol Oncol 103(1):368–371. https://doi.org/10.1016/j.ygyno.2006.04.024

    Article  PubMed  Google Scholar 

  12. Shokeir MO, Noel SM, Clement PB (1996) Malignant mullerian mixed tumor of the uterus with a prominent alpha-fetoprotein-producing component of yolk sac tumor. Mod Pathol 9(6):647–651

    CAS  PubMed  Google Scholar 

  13. Rossi R, Stacchiotti D, Bernardini MG, Calvieri G, Lo Voi R (2011) Primary yolk sac tumor of the endometrium: a case report and review of the literature. Am J Obstet Gynecol 204(4):e3–e4. https://doi.org/10.1016/j.ajog.2010.12.014

    Article  PubMed  Google Scholar 

  14. Qzler A, Dogan S, Mamedbeyli G, Rahatli S, Haberal AN, Dursun P, Ayhan A (2015) Primary yolk sac tumor of endometrium: report of two cases and review of literature. J Exp Ther Oncol 11(1):5–9

    PubMed  Google Scholar 

  15. Wang C, Li G, Xi L, Gu M, Ma D (2011) Primary yolk sac tumor of the endometrium. Int J Gynaecol Obstet 114(3):291–293. https://doi.org/10.1016/j.ijgo.2011.03.020

    Article  PubMed  Google Scholar 

  16. Ji M, Lu Y, Guo L, Feng F, Wan X, Xiang Y (2013) Endometrial carcinoma with yolk sac tumor-like differentiation and elevated serum beta-hCG: a case report and literature review. OncoTargets Ther 6:1515–1522. https://doi.org/10.2147/OTT.S51983

    CAS  Article  Google Scholar 

  17. Abhilasha N, Bafna UD, Pallavi VR, Rathod PS, Krishnappa S (2014) Primary yolk sac tumor of the endometrium: a rare entity. Indian J Cancer 51(4):446. https://doi.org/10.4103/0019-509X.175315

    CAS  Article  PubMed  Google Scholar 

  18. Damato S, Haldar K, McCluggage WG (2016) Primary endometrial yolk sac tumor with endodermal-intestinal differentiation masquerading as metastatic colorectal adenocarcinoma. Int J Gynecol Pathol 35(4):316–320. https://doi.org/10.1097/PGP.0000000000000236

    CAS  Article  PubMed  Google Scholar 

  19. Ravishankar S, Malpica A, Ramalingam P, Euscher ED (2017) Yolk sac tumor in extragonadal pelvic sites: still a diagnostic challenge. Am J Surg Pathol 41(1):1–11. https://doi.org/10.1097/PAS.0000000000000722

    Article  PubMed  Google Scholar 

  20. Nogales-Fernandez F, Silverberg SG, Bloustein PA, Martinez-Hernandez A, Pierce GB (1977) Yolk sac carcinoma (endodermal sinus tumor): ultrastructure and histogenesis of gonadal and extragonadal tumors in comparison with normal human yolk sac. Cancer 39(4):1462–1474

    CAS  Article  Google Scholar 

  21. Talerman A, Haije WG, Baggerman L (1978) Serum alphafetoprotein (AFP) in diagnosis and management of endodermal sinus (yolk sac) tumor and mixed germ cell tumor of the ovary. Cancer 41(1):272–278

    CAS  Article  Google Scholar 

  22. Leary AF, Quinn M, Fujiwara K, Coleman RL, Kohn E, Sugiyama T, Glasspool R, Ray-Coquard I, Colombo N, Bacon M, Zeimet A, Westermann A, Gomez-Garcia E, Provencher D, Welch S, Small W, Millan D, Okamoto A, Stuart G, Ochiai K, participants of the Fifth Ovarian Cancer Consensus C (2017) Fifth Ovarian Cancer Consensus Conference of the Gynecologic Cancer InterGroup (GCIG): clinical trial design for rare ovarian tumours. Ann Oncol 28(4):718–726. https://doi.org/10.1093/annonc/mdw662

    CAS  Article  PubMed  Google Scholar 

  23. Ayhan A, Taskiran C, Bozdag G, Altinbas S, Altinbas A, Yuce K (2005) Endodermal sinus tumor of the ovary: the Hacettepe University experience. Eur J Obstet Gynecol Reprod Biol 123(2):230–234. https://doi.org/10.1016/j.ejogrb.2005.04.021

    Article  PubMed  Google Scholar 

  24. Zanetta G, Bonazzi C, Cantu M, Binidagger S, Locatelli A, Bratina G, Mangioni C (2001) Survival and reproductive function after treatment of malignant germ cell ovarian tumors. J Clin Oncol 19(4):1015–1020. https://doi.org/10.1200/JCO.2001.19.4.1015

    CAS  Article  PubMed  Google Scholar 

  25. Low JJ, Perrin LC, Crandon AJ, Hacker NF (2000) Conservative surgery to preserve ovarian function in patients with malignant ovarian germ cell tumors. A review of 74 cases. Cancer 89(2):391–398

    CAS  Article  Google Scholar 

  26. Brewer M, Gershenson DM, Herzog CE, Mitchell MF, Silva EG, Wharton JT (1999) Outcome and reproductive function after chemotherapy for ovarian dysgerminoma. J Clin Oncol 17(9):2670–2675. https://doi.org/10.1200/JCO.1999.17.9.2670

    CAS  Article  PubMed  Google Scholar 

  27. de La Motte Rouge T, Pautier P, Duvillard P, Rey A, Morice P, Haie-Meder C, Kerbrat P, Culine S, Troalen F, Lhomme C (2008) Survival and reproductive function of 52 women treated with surgery and bleomycin, etoposide, cisplatin (BEP) chemotherapy for ovarian yolk sac tumor. Ann Oncol 19(8):1435–1441. https://doi.org/10.1093/annonc/mdn162

    Article  Google Scholar 

  28. National Comprehensive Cancer Network (2019) (NCCN) Clinical practice guidelines in oncology. Ovarian cancer including fallopian tube cancer and primary peritoneal cancer, Version 1. 2019. https://www.nccn.org/professionals/physician_gls/f_guidelines.asp. Accessed 8 Mar 2019

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Funding

This study was funded by Qilu Hospital of Shandong University in China.

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Authors

Contributions

TL: data collection, data analysis, formal analysis and manuscript writing/editing. LQ: data collection, formal analysis and manuscript writing. YM: paper assessment. GL: formal analysis. XZ: manuscript revising. PL: manuscript revising. Tao Lu and Liping Qi contributed equally.

Corresponding authors

Correspondence to Xiaolei Zhang or Peishu Liu.

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The authors report no conflict of interest.

Ethical approval

All the procedures performed in studies involving human participants were in accordance with the ethical standards of Ethics Committee of Qilu Hospital of Shandong University and with the 1964 Helsinki Declaration and its later amendments or comparable ethical standards. The publication of this case report got permission of the patient.

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This article does not contain any studies with human participants or animals performed by any of the authors.

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Informed consent for publication of this case report was received and personally signed from the patient on 2019-4-10.

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Lu, T., Qi, L., Ma, Y. et al. Primary yolk sac tumor of the endometrium: a case report and review of the literatures. Arch Gynecol Obstet 300, 1177–1187 (2019). https://doi.org/10.1007/s00404-019-05309-3

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  • DOI: https://doi.org/10.1007/s00404-019-05309-3

Keywords

  • Yolk sac tumor
  • Endodermal sinus tumor
  • α-Fetoprotein
  • Ovarian preservation
  • Endocrine function preservation
  • Endometrium