Volume 3, Issue 6, December 2018, Page: 72-77
Gestational Ovarian Choriocarcinoma: A Review of the Literature and Presentation of a Perplexing Case
Emily Bryer DO, Department of Internal Medicine, Pennsylvania Hospital, Philadelphia, Pennsylvania, USA
David Henry MD, Department of Hematology and Oncology, Pennsylvania Hospital, Philadelphia, Pennsylvania, USA
Received: Mar. 22, 2019;       Accepted: Apr. 28, 2019;       Published: May 27, 2019
DOI: 10.11648/j.ijcocr.20180306.11      View  28      Downloads  10
Gestational trophoblastic tumors are a heterogeneous and aggressive group of neoplasms. Choriocarcinoma, an extremely rare subset of gestational trophoblastic tumor, is often metastatic at diagnosis and typically follows a pregnancy that is either ectopic or molar. We present a critical review of choriocarcinomas with an emphasis on choriocarcinomas of ovarian origin which are even more uncommon with an incidence estimated at 1 per 369 million. This manuscript also includes a case of a woman with polycystic ovarian syndrome who required assisted conception to conceive her daughter and then developed a rare extrauterine gestational ovarian choriocarcinoma eighteen months following her antecedent healthy pregnancy with uncomplicated delivery. Many aspects of this presentation are unique, even for such an extraordinary tumor; some of these include the absence of vaginal bleeding, the presence of unilateral pulmonary metastases, and an overwhelmingly positive serum β-hCG with a negative urine β-hCG. While such a discrepancy in urine and serum β-hCG has been reported in gestational trophoblastic disease following molar pregnancies, to our knowledge it has not yet been reported in choriocarcinomas following a normal pregnancy.
Gestational Trophoblastic Disease, Ovarian Choriocarcinoma, Beta-Human Chorionic Gonadotropin, Pulmonary Metastases
To cite this article
Emily Bryer DO, David Henry MD, Gestational Ovarian Choriocarcinoma: A Review of the Literature and Presentation of a Perplexing Case, International Journal of Clinical Oncology and Cancer Research. Vol. 3, No. 6, 2018, pp. 72-77. doi: 10.11648/j.ijcocr.20180306.11
Copyright © 2018 Authors retain the copyright of this article.
This article is an open access article distributed under the Creative Commons Attribution License (http://creativecommons.org/licenses/by/4.0/) which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.
Shen X, Kuang Y-P. The Clinical Outcomes of In Vitro Fertilization/Intracytoplasmic Sperm Injection Treatments in 43 Women with a History of Gestational Trophoblastic Disease. Reprod Dev Med. 2018; 2 (1): 38. doi: 10.4103/2096-2924.232879.
Stockton L, Green E, Kaur B, De Winton E. Non-Gestational Choriocarcinoma with Widespread Metastases Presenting with Type 1 Respiratory Failure in a 39-Year-Old Female: Case Report and Review of the Literature. Case Rep Oncol. 2018; 11 (1): 151-158. doi: 10.1159/000486639.
Mahantappa H, Belur S, Agarwal A. Gestational ovarian choriocarcinoma: A case report. Arch Clin Exp Surg. 2018; 8 (1): 1. doi: 10.5455/aces.20180314041954.
Lurain JR. Gestational trophoblastic disease I: Epidemiology, pathology, clinical presentation and diagnosis of gestational trophoblastic disease, and management of hydatidiform mole. Am J Obstet Gynecol. 2010; 203 (6): 531-539. doi: 10.1016/j.ajog.2010.06.073.
Cole LA. O-glycosylation of proteins in the normal and neoplastic trophoblast. Trophobl Res 1987; 2: 139–48.
Cole LA et al. Gestational trophoblastic diseases: Pathophysiology of hyperglycosylated hCG. Gynecologic Oncology. 2006; 102: 145-150. doi: 10.1016/j.ygyno.2005.12.047.
Seibre NJ, Seckl MJ. Gestational trophoblastic disease. Management of Unintended and Abnormal Pregnancy. 2009. Blackwell Publishing. ISBN: 9781405176965.
Kohorn E. Negotiating a staging and risk factor scoring system for gestational trophoblastic neoplasia. A progress report. J Reprod Med. 2002. 47 (6): 445-50.
May T, Goldstein DP, Berkowitz RS. Current Chemotherapeutic Management of Patients with Gestational Trophoblastic Neoplasia. Chemother Res Pract. 2011; 2011: 1-12. doi: 10.1155/2011/806256.
El-Helw LM, Hancock BW. Treatment of metastatic gestational trophoblastic neoplasia. Lancet Oncol. 2007; 8 (8): 715-724. doi: 10.1016/S1470-2045 (07) 70239-5.
Sakamoto Y, Takei Y, Fujiwara H, Machida S, Taneichi A, Suzuki M. Gestational choriocarcinoma with uterine serosal metastasis mimicking ruptured ectopic pregnancy: A case report. Oncol Lett. 2015; 9 (5): 2185-2188. doi: 10.3892/ol.2015.2999.
Hariharan C, Jajoo SS, Khemka AS. A rare case of choriocarcinoma following a normal pregnancy and delivery. International Journal of Recent Surgical and Medical Sciences. 2017; 3 (1): 64-66.
Dilek S, Pata O, Tok E, Polat A. Extraovarian nongestational choriocarcinoma in a postmenopausal woman. Int J Gynecol Cancer. 2004 Sep–Oct; 14 (5): 1033–5.
Gerson RF, Lee EY, Gorman E. Primary extrauterine ovarian choriocarcinoma mistaken for ectopic pregnancy: Sonographic imaging findings. Am J Roentgenol. 2007; 189 (5): 1164. doi: 10.2214/AJR.05.0814.
Elliott MM, Kardana A, Lustbader JW, Cole LA. Carbohydrate and peptide structure of the alpha- and beta-subunits of human chorionic gonadotropin from normal and aberrant pregnancy and choriocarcinoma. Endocrine 1997; 7: 15.
Exman P et al. Primary ovarian choriocarcinoma: individual DNA polymorphic analysis as a strategy to confirm diagnosis and treatment. Rare Tumors. 2013; 5 (24): 89-92.
Mangla M, Singla D, Kaur H, Sharma S. Unusual clinical presentations of choriocarcinoma: A systematic review of case reports. Taiwan J Obstet Gynecol. 2017; 56 (1): 1-8. doi: 10.1016/j.tjog. 2015.05.011.
Chen M, Yang J, Lin M. An unusual gestational choriocarcinoma occurring primarily on the surface of a subserous leiomyoma. BJOG: An International Journal of Obstetrics and Gynaecology. 2004; 111 (7): 188-19.
Jia N, Chen Y, Tao X, Ou E, Lu X, Feng W. A gestational choriocarcinoma of the ovary diagnosed by DNA polymorphic analysis: A case report and systematic review of the literature. J Ovarian Res. 2017; 10 (1): 1-8. doi: 10.1186/s13048-017-0334-3.
Witzleben C, Bruninga G. Infantile choriocarcinoma: a characteristic syndrome. J Pediatr. 1963; 73: 374–8.
Gerson RF, Lee EY, Gorman E. Primary extrauterine ovarian choriocarcinoma mistaken for ectopic pregnancy: sonographic imaging findings. American Journal of Radiology. 2007; 189: W280-W283.
Dehner LP. Gestational and nongestational trophoblastic neoplasia. A historic and pathobiologic survey. Am J Surg Pathol. 1980; 4: 43–58.
Fox H. Gestational trophpoblastic disease. British Medical Journal. 1997; 314: 1363. Doi: 10.1136/bmj.314.7091.1363
Bates M, Everard J, Wall L, Horsman JM, Hancock BW. Is there a relationship between treatment for infertility and gestational trophoblastic disease? Hum Reprod. 2004; 19 (2): 365-367. doi: 10.1093/humrep/deh068.
Palmer JR. Advances in the epidemiology of gestational trophoblastic disease. J Reproductive Medicine. 1994. 39 (3): 155-162.
Palmer JR et al. Oral contraceptive use and risk of gestational trophoblastic tumors. J Natl Cancer Inst. 1999. 91 (7): 635-640.
Flam F, Lundstrom V, Lindstedt J, Silfversward C. Choriocarcinoma of the fallopian tube associated with induced superovulation in an IVF program; a case report. Eur J Obstet Gynecol Reprod Biol. 1989; 33 (2): 183-186. doi: 10.1016/0028-2243 (89) 90212-8.
Lin LH, Fushida K, Hase EA, et al. Gestational Tubal Choriocarcinoma Presenting as a Pregnancy of Unknown Location following Ovarian Induction. Case Rep Obstet Gynecol. 2018; 2018: 1-6. doi: 10.1155/2018/4705192.
Kállén B, Finnström O, Lindam A, Nilsson E, Nygren KG, Otterblad Olausson P. Malignancies among women who gave birth after in vitro fertilization. Hum Reprod. 2011; 26 (1): 253-258. doi: 10.1093/humrep/deq307.
Tsukamoto N et al. Choriocarcinoma occurring within the normal placenta with breast metastasis. Journal of Gynecologic Oncology. 1981; 11 (3): 348-363. doi: 10.1016/0090-8258 (81) 90049-4
Brewer JI, Mazur MT. Gestational choriocarcinoma. Its origin in the placenta during seemingly normal pregnancy. Am J Surg Pathol. 1981. 3: 267-277.
Hayashi S, Yoshiro A, Tomita S, et al. Primary non-gestational pure choriocarcinoma arising in the ovary: A case report and literature review. Oncol Lett. 2015; 9 (5): 2109-2111. doi: 10.3892/ol.2015.2985.
Vance RP, Geisinger KR. Pure nongestational choriocarcinoma of the ovary. Cancer. 1985: (56): 2321-2325.
Lurain JR, Sand PK, Brewer JI. Choriocarcinoma associated with ectopic pregnancy. Obstet Gynecol. 1986; 68: 286 – 287.
Rawal G, Ahluwalia C, Yadav AK, Dhawan I. Case Report Primary pure non-gestational choriocarcinoma of ovary: a rare case report. 2017; 6 (9): 4166-4169.
Lv L, Yang K, Wu H, Lou J, Peng Z. Pure choriocarcinoma of the ovary: A case report. J Gynecol Oncol. 2011; 22 (2): 135-139. doi: 10.3802/jgo. 2011.22.2.135.
Axe SR, Klein VR, Woodruff JD. Choriocarcinoma of the ovary. Obstet Gynecol 1985; 66: 111-4.
Lorigan PC, Grierson AJ, Goepel JR, Coleman RE, Goyns MH. Gestational choriocarcinoma of the ovary diagnosed by analysis of tumour DNA. Cancer Lett. 1996; 104 (1): 27-30. doi: 10.1016/0304-3835 (96) 04219-X.
Mood N, Samadi N, Rahimi-Moghaddam P et al. Pure ovarian choriocarcinoma: report of two cases. J Res Med Sci. 2009; 14 (5): 327-330.
Biswas S, Rodeck CH. Plasma prolactin levels during pregnancy. Br J Obstet Gynaecol. 1976; 83 (9): 683-687.
Brant WE, Helms C. Fundamentals of Diagnostic Radiology. Wolters Kluwer Health; 2012. https://books.google.com/books?id=uVeUWa3-1o8C.
Lloyd R V. Endocrine Pathology:: Differential Diagnosis and Molecular Advances. Springer New York; 2010. https://books.google.com/books?id=Je9QTWRH6s8C.
Brotherton J. Comparison of human chorionic gonadotrophin in human urine and amniotic fluid as estimated by a polyclonal and by a specific monoclonal assay in very early pregnancy. Hum Reprod. 1989; 4 (7): 837-842. doi: 10.1093/oxfordjournals.humrep.a136997.
Pang YP, Rajesh H, Tan LK. Molar pregnancy with false negative urine hCG: The hook effect. Singapore Med J. 2010; 51 (3) e58-e61.
Korevaar TIM, Steegers EAP, de Rijke YB, et al. Reference ranges and determinants of total hCG levels during pregnancy: The Generation R Study. Eur J Epidemiol. 2015; 30 (9): 1057-1066. doi: 10.1007/s10654-015-0039-0.
Browse journals by subject