Possible link between congenital uterine malformations and epitheliod trophoblastic tumor

In: Open Journal of Obstetrics and Gynecology · 2012 · vol. 02(03) , pp. 227–229 · doi:10.4236/ojog.2012.23046 · W2029027127
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This report describes a case of epithelioid trophoblastic tumor in a patient with a history of congenital uterine septum, suggesting a possible link between uterine malformations and ETT development.

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This paper reports a clinical case in a 41-year-old patient with irregular bleeding and a history of congenital uterine septum treated previously by Tompkins operation. Despite serum hCG levels under the upper limit, D&C endometrial samples showed epithelioid trophoblastic tumor, confirmed by immunostaining, and the authors performed prompt abdominal hysterectomy with ovaries preserved. The paper proposes, under the mullerianosis theory of endometriosis, that aberrant differentiation or migration during fetal uterine organogenesis could leave ectopic cells that, in an apoptosis-prone environment, may initiate epithelioid trophoblastic tumor, and it notes that further studies are needed to assess this link. Relevance to endometriosis: the authors explicitly invoke the mullerianosis theory of endometriosis to propose a mechanistic connection between embryologic ectopic cells and epithelioid trophoblastic tumor, though the paper’s main focus is a single ETT case with a proposed pathogenetic hypothesis.

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Abstract

Background: Epitheloid trophoblast tumor (ETT) is a tenuous type of gestational trophoblastic disease. Although clinically ETT behave in variable fashion, it is considered as a potentially malignant tumor, fatal malignancy occurs in 10% of the cases. Case and Conclusion: A 41 years old patient, who previously underwent Tompkins operation because of congenital uterine septum was admitted to our clinic due to irregular bleeding. Despite of serum hCG levels under the upper limit of normal, histopathology analysis of D&C obtained endometrial samples confirmed the presence of ETT by immunostainings. Henceforth we performed prompt abdominal hysterectomy and preserved the normal structured ovaries. According to the müllerianosis theory of endometriosis aberrant differentiation or migration could cause spreading of cells throughout the fetal organogenesis of the uterus. These cells in an apoptosis week environment might correlate with the initiation of ETT. As a conclusion we suggest further studies to assess the link between ectopic cells and ETT, to gain a better understanding in the pathomechanism of the tumor.
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Possible link between congenital uterine malformations and epitheliod trophoblastic tumor () Balint Farkas, Krisztina Kovacs, Miklos Vizer, Jozsef Bodis, Peter Gocze Department of Obstetrics and Gynecology, University of Pecs Medical Center, Pécs, Hungary. Department of Pathology, University of Pecs, Pécs, Hungary. DOI: 10.4236/ojog.2012.23046 PDF HTML 4,089 Downloads 6,441 Views Citations Department of Obstetrics and Gynecology, University of Pecs Medical Center, Pécs, Hungary. Department of Pathology, University of Pecs, Pécs, Hungary. DOI: 10.4236/ojog.2012.23046 PDF HTML 4,089 Downloads 6,441 Views Citations Background: Epitheloid trophoblast tumor (ETT) is a tenuous type of gestational trophoblastic disease. Although clinically ETT behave in variable fashion, it is considered as a potentially malignant tumor, fatal malignancy occurs in 10% of the cases. Case and Conclusion: A 41 years old patient, who previously underwent Tompkins operation because of congenital uterine septum was admitted to our clinic due to irregular bleeding. Despite of serum hCG levels under the upper limit of normal, histopathology analysis of D&C obtained endometrial samples confirmed the presence of ETT by immunostainings. Henceforth we performed prompt abdominal hysterectomy and preserved the normal structured ovaries. According to the müllerianosis theory of endometriosis aberrant differentiation or migration could cause spreading of cells throughout the fetal organogenesis of the uterus. These cells in an apoptosis week environment might correlate with the initiation of ETT. As a conclusion we suggest further studies to assess the link between ectopic cells and ETT, to gain a better understanding in the pathomechanism of the tumor. Share and Cite: Farkas, B. , Kovacs, K. , Vizer, M. , Bodis, J. and Gocze, P. (2012) Possible link between congenital uterine malformations and epitheliod trophoblastic tumor. Open Journal of Obstetrics and Gynecology, 2, 227-229. doi: 10.4236/ojog.2012.23046. Conflicts of Interest The authors declare no conflicts of interest. | [1] | Seckl, M.J., Sebire, N.J. and Berkowitz, R.S. (2010) Gestational trophoblastic disease. Lancet, 376, 717-729. doi:10.1016/S0140-6736(10)60280-2 | | [2] | Shih, I.M. and Kurman, R.J. (1998) Epithelioid trophoblastic tumor: A neoplasm distinct from choriocarcinoma and placental site trophoblastic tumor simulating carcinoma. The American Journal of Surgical Pathology, 22, 1393-1403. doi:10.1097/00000478-199811000-00010 | | [3] | Shih, I.M. (2007) Gestational trophoblastic neoplasia-pathogenesis and potential therapeutic targets. Lancet Oncology, 8, 642-650. doi:10.1016/S1470-2045(07)70204-8 | | [4] | Kimberly, H., Allison, M.D., Jason, E., Love, M.D., Rochelle, L. and Garcia, M.D. (2006) Epithelioid trophoblastic tumor: Review of a rare neoplasm of the chorionic-type intermediate trophoblast. Archives of Pathology & Laboratory Medicine, 130, 1875-1877. | | [5] | Lee, D.M., Osathanondh, R., Yeh, J. (1998) Localisation of Bcl-2 in the human fetal müllerian tract. Fertil Steril, 70, 135-140. doi:10.1016/S0015-0282(98)00126-5 | | [6] | Klattig, J. and Englert, C. (2007) The Mullerian duct: Recent insight into its development and regression. Sexual Development, 1, 271-278. doi:10.1159/000108929 | | [7] | Mai, K.T., Yazdi, H.M., Perkins, D.G., Parks, W. (1998) Development of endometriosis from embryonic duct remnants. Human Pathology, 29, 319-322. doi:10.1016/S0046-8177(98)90110-1 | | [8] | Palmer, J.E., Macdonald, M., Wells, M., Path, F.R.C., Hancock, B.W. and Tidy, J.A. (2008) Epitheliod trophoblast tumor. A review of the Literature. The Journal of Reproductive Medicine, 53, 465-475. | | [9] | Allison, K.H., Love, J.E. and Garcia, R.L. (2006) Epitheloid trophoblastic tumor: Review of a rare neoplasm of the chorionic type intermediate trophoblast. Archives of Pathology & Laboratory Medicine, 130, 1875-1877. | | [10] | Fadare, O., Parkash, V., Carcangiu, M.L., et al. (2006) Epitheloid trophoblast tumor: Clinicopathologic features with an emphasis on uterine cervical involvement. Modern Pathology; 19, 75-82. doi:10.1038/modpathol.3800485 | | [11] | Gocze, M.P., Beamer, G.W., De Jong, H.F. and Freeman, A.D. (1997) Hormone synthesis and responsiveness of spontaneous granulose cell tumors in (SWR × SWXJ-9) F1 mice. Gynecology Oncology, 65, 143-148. doi:10.1006/gyno.1997.4635 | | [12] | Ajithkumar, T.V., Abraham, E.K., Rejnishkumar, R. and Minimole, A.L. (2003) Placental site trophoblastic tumor. Obstetrical & Gynecological Survey, 58, 484-488. doi:10.1097/01.OGX.0000077466.40895.32 | | [13] | Driscoll, E.G. (1984) Placental site chorioma, the neoplasm of the implantation site trophoblast. The Journal of Reproductive Medicine, 29, 821-825. | | [14] | Knox, S., Brooks, S.E., et al. (2002) Choriocarcinoma and epithelial trophoblastic tumor: Successful treatment of relapse with hysterectomy and high-dose chemotherapy with peripheral stem cell support: A case report. Gynecologic Oncology, 85, 204-208. | | [15] | Kurman, R.J. (2002) Blaustein’s pathology of the female genital tract. 5th Edition, Springer, New York. | Copyright © 2026 by authors and Scientific Research Publishing Inc. This work and the related PDF file are licensed under a Creative Commons Attribution 4.0 International License.

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