Synchronous placental site trophoblastic tumor and choriocarcinoma

Birama Robby Indraprasta; Brahmana Askandar Tjokroprawiro

doi:10.15562/bmj.v12i3.4718

Synchronous placental site trophoblastic tumor and choriocarcinoma

Birama Robby Indraprasta ,

Brahmana Askandar Tjokroprawiro ,

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DOI: https://doi.org/10.15562/bmj.v12i3.4718 |
Published: 2023-09-05

Abstract

Link of Video Abstract: https://youtu.be/bx7KdiTKiOQ

Introduction: Gestational trophoblastic neoplasia (GTN) comprises a series of malignancies characterized by abnormal proliferation of fetal trophoblastic tissue. GTN lesions refer to a class of malignant lesions that have distinct histopathologic features, including invasive hydatidiform moles, choriocarcinomas, placental site trophoblastic tumors (PSTT), and epithelioid trophoblastic tumor (ETT). Most often, they occur coexist separately, but there are also cases of mixed GTN with a combination of histologic PSTT, ETT, or choriocarcinoma. PSTT is a rare form of gestational trophoblastic disease (GTD). The percent incidence of this disease is 0.23% of total GTD and 1-2% of total GTN. The incidence rate of choriocarcinoma is also limited. Data collection on the incidence of mixed GTN has been more challenging due to the rarity of this disease.

Case Description: We report a case of a 26-year-old female patient with choriocarcinoma and PSTT. The patient’s beta-human chorionic gonadotropin (β-hCG) levels remained elevated despite treatment with single-agent chemotherapeutic regimen. She underwent a series of chemotherapy treatments and two surgeries were required. The histological examination of the uterine tumor revealed a choriocarcinoma whilst the hysterectomy thereafter showed a PSTT. After the hysterectomy the β-hCG level continued to rise. Due to chemotherapy resistance, EMACO (etoposide, methotrexate, actinomycin-D, cyclophosphamide, vincristine) was changed to EP-EMA (Etoposide, cisplatin, etoposide, methotrexate, actinomycin-D). After two administrations of EP-EMA, β-hCG levels normalized.

Conclusion: This is a rare case of mixed GTN of choriocarcinoma and PSTT. Mixed GTN should be considered when chemotherapy is not effective in the treatment of GTN. These cases need to be reported to improve the diagnosis and management of patients.

References

Chaves MM, Maia T, Cunha TM, Veiga VF. Placental site trophoblastic tumour: the rarest subtype of gestational trophoblastic disease. BMJ Case Rep. 2020;13(10). e235756.
Froeling FEM, Ramaswami R, Papanastasopoulos P, Kaur B, Sebire NJ, Short D, et al. Intensified therapies improve survival and identification of novel prognostic factors for placental-site and epithelioid trophoblastic tumours. Br J Cancer. 2019;120(6):587–94.
Colecchi C, Partemi S, Minelli N, Cascini F, Rossi R, Fulcheri E, et al. Placental Site Trophoblastic Tumor with lung metastases as cause of death in a young patient: a case report. Placenta. 2011;32(12):1060–3.
Zhao J, Lv WG, Feng FZ, Wan XR, Liu JH, Yi XF, et al. Placental site trophoblastic tumor: A review of 108 cases and their implications for prognosis and treatment. Gynecol Oncol.;142(1):102–8.
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):531–9.
Kong Y, Tao G, Zong L, Yang J, Wan X, Wang W, et al. Diagnosis and management of mixed gestational trophoblastic neoplasia: a study of 16 cases and a review of the literature. Front Oncol. 2019;9:493635.
Alexander AL, Strohl AE, Maniar KP, Lurain JR. Placental site trophoblastic tumor: Successful treatment of 13 cases. Gynecol Oncol reports. 2020;32:100548.
Behtash N, Karimi Zarchi M. Placental site trophoblastic tumor. J Cancer Res Clin Oncol. 2008;134(1):1–6.
Ngan HYS, Seckl MJ, Berkowitz RS, Xiang Y, Golfier F, Sekharan PK, et al. Update on the diagnosis and management of gestational trophoblastic disease. Int J Gynaecol Obstet. 2018;143 Suppl 2:79–85.
Yang C, Li J, Zhang Y, Xiong H, Sheng X. Epithelioid trophoblastic tumor coexisting with choriocarcinoma around an abdominal wall cesarean scar: a case report and review of the literature. J Med Case Rep. 2020;14(1):178.
Gari A. Placental site trophoblastic tumor and choriocarcinoma: An unusual presentation Clinical Pathology. BMC Res Notes. 2015;8(1):1–4.
Zeng C, Rezai S, Hughes AC, Henderson CE, Liu J. Synchronous choriocarcinoma and epithelioid trophoblastic tumor concurring at the cesarean scar: a case report and review of the literature. Case Rep Obstet Gynecol. 2019;2019: 5093938.
Feng X, Wei Z, Zhang S, Du Y, Zhao H. A review on the pathogenesis and clinical management of placental site trophoblastic tumors. Front Oncol. 2019;9:937.
De Nola R, Schönauer LM, Fiore MG, Loverro M, Carriero C, Naro E Di. Management of placental site trophoblastic tumor: Two case reports. Medicine (Baltimore). 2018;97(48):e13439.
Mao TL, Kurman RJ, Huang CC, Lin MC, Shih IM. Immunohistochemistry of choriocarcinoma: an aid in differential diagnosis and in elucidating pathogenesis. Am J Surg Pathol. 2007;31(11):1726–32.
Shih IM. Gestational trophoblastic neoplasia--pathogenesis and potential therapeutic targets. Lancet Oncol. 2007;8(7):642–50.
Shih IM, Kurman RJ. The pathology of intermediate trophoblastic tumors and tumor-like lesions. Int J Gynecol Pathol. 2001;20(1):31–47.
Aziz MF. Gynecological cancer in Indonesia. J Gynecol Oncol. 2009;20(1):8–10.
Mulawardhana P, Hartono P, Nugroho H, Ayuningtyas A. Death of 43 Indonesian women with ovarian cancer: A case series. Int J Surg Case Rep. 2021;78:391.
Nagai Y, Wakayama A, Suzuki S, Asato K, Hirakawa M, Kudaka W, et al. Metastatic placental site trophoblastic tumour successfully treated with hysterectomy and EMA/CO chemotherapy. J Obstet Gynaecol. 2011;31(1):99–101.
Tsuji Y, Tsubamoto H, Hori M, Ogasawara T, Koyama K. Case of PSTT treated with chemotherapy followed by open uterine tumor resection to preserve fertility. Gynecol Oncol. 2002;87(3):303–7.
Tjokroprawiro BA. Lung metastasis of gynecologic cancers. Bali Med J. 2021;10(2):701–7.

[1] Chaves MM, Maia T, Cunha TM, Veiga VF. Placental site trophoblastic tumour: the rarest subtype of gestational trophoblastic disease. BMJ Case Rep. 2020;13(10). e235756.

[2] Froeling FEM, Ramaswami R, Papanastasopoulos P, Kaur B, Sebire NJ, Short D, et al. Intensified therapies improve survival and identification of novel prognostic factors for placental-site and epithelioid trophoblastic tumours. Br J Cancer. 2019;120(6):587–94.

[3] Colecchi C, Partemi S, Minelli N, Cascini F, Rossi R, Fulcheri E, et al. Placental Site Trophoblastic Tumor with lung metastases as cause of death in a young patient: a case report. Placenta. 2011;32(12):1060–3.

[4] Zhao J, Lv WG, Feng FZ, Wan XR, Liu JH, Yi XF, et al. Placental site trophoblastic tumor: A review of 108 cases and their implications for prognosis and treatment. Gynecol Oncol.;142(1):102–8.

[5] 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):531–9.

[6] Kong Y, Tao G, Zong L, Yang J, Wan X, Wang W, et al. Diagnosis and management of mixed gestational trophoblastic neoplasia: a study of 16 cases and a review of the literature. Front Oncol. 2019;9:493635.

[7] Alexander AL, Strohl AE, Maniar KP, Lurain JR. Placental site trophoblastic tumor: Successful treatment of 13 cases. Gynecol Oncol reports. 2020;32:100548.

[8] Behtash N, Karimi Zarchi M. Placental site trophoblastic tumor. J Cancer Res Clin Oncol. 2008;134(1):1–6.

[9] Ngan HYS, Seckl MJ, Berkowitz RS, Xiang Y, Golfier F, Sekharan PK, et al. Update on the diagnosis and management of gestational trophoblastic disease. Int J Gynaecol Obstet. 2018;143 Suppl 2:79–85.

[10] Yang C, Li J, Zhang Y, Xiong H, Sheng X. Epithelioid trophoblastic tumor coexisting with choriocarcinoma around an abdominal wall cesarean scar: a case report and review of the literature. J Med Case Rep. 2020;14(1):178.

[11] Gari A. Placental site trophoblastic tumor and choriocarcinoma: An unusual presentation Clinical Pathology. BMC Res Notes. 2015;8(1):1–4.

[12] Zeng C, Rezai S, Hughes AC, Henderson CE, Liu J. Synchronous choriocarcinoma and epithelioid trophoblastic tumor concurring at the cesarean scar: a case report and review of the literature. Case Rep Obstet Gynecol. 2019;2019: 5093938.

[13] Feng X, Wei Z, Zhang S, Du Y, Zhao H. A review on the pathogenesis and clinical management of placental site trophoblastic tumors. Front Oncol. 2019;9:937.

[14] De Nola R, Schönauer LM, Fiore MG, Loverro M, Carriero C, Naro E Di. Management of placental site trophoblastic tumor: Two case reports. Medicine (Baltimore). 2018;97(48):e13439.

[15] Mao TL, Kurman RJ, Huang CC, Lin MC, Shih IM. Immunohistochemistry of choriocarcinoma: an aid in differential diagnosis and in elucidating pathogenesis. Am J Surg Pathol. 2007;31(11):1726–32.

[16] Shih IM. Gestational trophoblastic neoplasia--pathogenesis and potential therapeutic targets. Lancet Oncol. 2007;8(7):642–50.

[17] Shih IM, Kurman RJ. The pathology of intermediate trophoblastic tumors and tumor-like lesions. Int J Gynecol Pathol. 2001;20(1):31–47.

[18] Aziz MF. Gynecological cancer in Indonesia. J Gynecol Oncol. 2009;20(1):8–10.

[19] Mulawardhana P, Hartono P, Nugroho H, Ayuningtyas A. Death of 43 Indonesian women with ovarian cancer: A case series. Int J Surg Case Rep. 2021;78:391.

[20] Nagai Y, Wakayama A, Suzuki S, Asato K, Hirakawa M, Kudaka W, et al. Metastatic placental site trophoblastic tumour successfully treated with hysterectomy and EMA/CO chemotherapy. J Obstet Gynaecol. 2011;31(1):99–101.

[21] Tsuji Y, Tsubamoto H, Hori M, Ogasawara T, Koyama K. Case of PSTT treated with chemotherapy followed by open uterine tumor resection to preserve fertility. Gynecol Oncol. 2002;87(3):303–7.

[22] Tjokroprawiro BA. Lung metastasis of gynecologic cancers. Bali Med J. 2021;10(2):701–7.

Synchronous placental site trophoblastic tumor and choriocarcinoma

Abstract

References

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