Skip to main content
Top
Published in: Journal of Medical Case Reports 1/2022

Open Access 01-12-2022 | Gestational Trophoblastic Disease | Case report

Severe thyrotoxicosis as initial presentation of gastric choriocarcinoma: a case report

Authors: Nicole M. Iñiguez-Ariza, Dalia Cuenca, Juvenal Franco-Granillo, Alberto Villalobos-Prieto, Janet Pineda-Díaz, Javier Baquera-Heredia

Published in: Journal of Medical Case Reports | Issue 1/2022

Login to get access

Abstract

Background

Extragonadal choriocarcinoma is rare and can be associated with hyperthyroidism when producing very high levels of human chorionic gonadotropin.

Case presentation

A 62-year-old Hispanic female presented with a 3-week history of shortness of breath, palpitations, extreme weakness, new-onset hot flashes, and right flank pain. Her physical examination was remarkable for tachycardia, hepatomegaly, hyperreflexia, and tremor; goiter was absent. Laboratory studies revealed increased lactate dehydrogenase, alkaline phosphatase, suppressed thyroid stimulating hormone, very elevated T4, and absent thyroid stimulating immunoglobulin. 18F-fluorodeoxyglucose positron emission tomography-computed tomography exhibited hepatomegaly with multiple large fluorodeoxyglucose-avid liver masses and a focus of fluorodeoxyglucose avidity in the stomach with no structural correlate. A thyroid scan (99mTcO 4 ) showed diffusely increased tracer uptake. She was started on propranolol and methimazole. Upon stabilization of severe thyrotoxicosis, upper endoscopy was performed, showing a ~ 5 cm bleeding lesion in the greater stomach curvature body; biopsy was consistent with choriocarcinoma; beta-human chorionic gonadotropin hormone was 2,408,171 mIU/mL. The patient received methotrexate followed by etoposide and cisplatin. Methimazole was titrated down, and upon liver failure the medication was stopped. The thyrotoxicosis was effectively controlled with antithyroid drug and concurrent chemotherapy. At ~ 1.5 months after initial diagnosis, the patient died due to bleeding/acute liver failure with coagulation defects followed by multiple organ failure.

Conclusions

Severe thyrotoxicosis can represent an unusual initial presentation of metastatic choriocarcinoma in the setting of extreme elevation of beta-human chorionic gonadotropin. Primary gastric choriocarcinoma is an aggressive malignancy with very poor outcomes. The co-occurrence of severe thyrotoxicosis with advanced primary gastric choriocarcinoma and imminent liver failure complicates management options.
Literature
1.
go back to reference Walkington L, Webster J, Hancock BW, Everard J, Coleman RE. Hyperthyroidism and human chorionic gonadotrophin production in gestational trophoblastic disease. Br J Cancer. 2011;104(11):1665–9.CrossRefPubMedPubMedCentral Walkington L, Webster J, Hancock BW, Everard J, Coleman RE. Hyperthyroidism and human chorionic gonadotrophin production in gestational trophoblastic disease. Br J Cancer. 2011;104(11):1665–9.CrossRefPubMedPubMedCentral
2.
go back to reference Abu-Rustum NR, Yashar CM, Bean S, Bradley K, Campos SM, Chon HS, et al. Gestational trophoblastic neoplasia, version 2.2019, NCCN clinical practice guidelines in oncology. J Natl Compr Canc Netw. 2019;17(11):1374–91.CrossRefPubMed Abu-Rustum NR, Yashar CM, Bean S, Bradley K, Campos SM, Chon HS, et al. Gestational trophoblastic neoplasia, version 2.2019, NCCN clinical practice guidelines in oncology. J Natl Compr Canc Netw. 2019;17(11):1374–91.CrossRefPubMed
3.
go back to reference Seckl MJ, Sebire NJ, Fisher RA, Golfier F, Massuger L, Sessa C, et al. Gestational trophoblastic disease: ESMO clinical practice guidelines for diagnosis, treatment and follow-up. Ann Oncol. 2013;24(Suppl 6):vi39-50.CrossRefPubMed Seckl MJ, Sebire NJ, Fisher RA, Golfier F, Massuger L, Sessa C, et al. Gestational trophoblastic disease: ESMO clinical practice guidelines for diagnosis, treatment and follow-up. Ann Oncol. 2013;24(Suppl 6):vi39-50.CrossRefPubMed
4.
go back to reference Kobayashi A, Hasebe T, Endo Y, Sasaki S, Konishi M, Sugito M, et al. Primary gastric choriocarcinoma: two case reports and a pooled analysis of 53 cases. Gastric Cancer. 2005;8(3):178–85.CrossRefPubMed Kobayashi A, Hasebe T, Endo Y, Sasaki S, Konishi M, Sugito M, et al. Primary gastric choriocarcinoma: two case reports and a pooled analysis of 53 cases. Gastric Cancer. 2005;8(3):178–85.CrossRefPubMed
6.
go back to reference Ajay VarunReddy TKR. Primary gastric choriocarcinoma with paraneoplastic hyperthyroidism: a rare case report. J Dental Med Sci. 2016;15(6):78–80. Ajay VarunReddy TKR. Primary gastric choriocarcinoma with paraneoplastic hyperthyroidism: a rare case report. J Dental Med Sci. 2016;15(6):78–80.
7.
go back to reference Yoon JH, Kim MS, Kook EH, Ahn SH, Jeong SY, Han MS, et al. Primary gastric choriocarcinoma: two case reports and review of the literatures. Cancer Res Treat. 2008;40(3):145–50.CrossRefPubMedPubMedCentral Yoon JH, Kim MS, Kook EH, Ahn SH, Jeong SY, Han MS, et al. Primary gastric choriocarcinoma: two case reports and review of the literatures. Cancer Res Treat. 2008;40(3):145–50.CrossRefPubMedPubMedCentral
9.
go back to reference Liu AY, Chan WY, Ng EK, Zhang X, Li BC, Chow JH, et al. Gastric choriocarcinoma shows characteristics of adenocarcinoma and gestational choriocarcinoma: a comparative genomic hybridization and fluorescence in situ hybridization study. Diagn Mol Pathol. 2001;10(3):161–5.CrossRefPubMed Liu AY, Chan WY, Ng EK, Zhang X, Li BC, Chow JH, et al. Gastric choriocarcinoma shows characteristics of adenocarcinoma and gestational choriocarcinoma: a comparative genomic hybridization and fluorescence in situ hybridization study. Diagn Mol Pathol. 2001;10(3):161–5.CrossRefPubMed
10.
go back to reference Liu Z, Mira JL, Cruz-Caudillo JC. Primary gastric choriocarcinoma: a case report and review of the literature. Arch Pathol Lab Med. 2001;125(12):1601–4.CrossRefPubMed Liu Z, Mira JL, Cruz-Caudillo JC. Primary gastric choriocarcinoma: a case report and review of the literature. Arch Pathol Lab Med. 2001;125(12):1601–4.CrossRefPubMed
11.
go back to reference Ross DS, Burch HB, Cooper DS, Greenlee MC, Laurberg P, Maia AL, et al. 2016 American Thyroid Association Guidelines for diagnosis and management of hyperthyroidism and other causes of thyrotoxicosis. Thyroid. 2016;26(10):1343–421.CrossRefPubMed Ross DS, Burch HB, Cooper DS, Greenlee MC, Laurberg P, Maia AL, et al. 2016 American Thyroid Association Guidelines for diagnosis and management of hyperthyroidism and other causes of thyrotoxicosis. Thyroid. 2016;26(10):1343–421.CrossRefPubMed
12.
go back to reference Hershman JM. Physiological and pathological aspects of the effect of human chorionic gonadotropin on the thyroid. Best Pract Res Clin Endocrinol Metab. 2004;18(2):249–65.CrossRefPubMed Hershman JM. Physiological and pathological aspects of the effect of human chorionic gonadotropin on the thyroid. Best Pract Res Clin Endocrinol Metab. 2004;18(2):249–65.CrossRefPubMed
13.
go back to reference Korevaar TI, Steegers EA, de Rijke YB, Schalekamp-Timmermans S, Visser WE, Hofman A, et al. Reference ranges and determinants of total hCG levels during pregnancy: the Generation R Study. Eur J Epidemiol. 2015;30(9):1057–66.CrossRefPubMedPubMedCentral Korevaar TI, Steegers EA, de Rijke YB, Schalekamp-Timmermans S, Visser WE, Hofman A, et al. Reference ranges and determinants of total hCG levels during pregnancy: the Generation R Study. Eur J Epidemiol. 2015;30(9):1057–66.CrossRefPubMedPubMedCentral
14.
go back to reference Meister LH, Hauck PR, Graf H, Carvalho GA. Hyperthyroidism due to secretion of human chorionic gonadotropin in a patient with metastatic choriocarcinoma. Arq Bras Endocrinol Metabol. 2005;49(2):319–22.CrossRefPubMed Meister LH, Hauck PR, Graf H, Carvalho GA. Hyperthyroidism due to secretion of human chorionic gonadotropin in a patient with metastatic choriocarcinoma. Arq Bras Endocrinol Metabol. 2005;49(2):319–22.CrossRefPubMed
15.
go back to reference Sotello D, Rivas AM, Test VJ, Lado-Abeal J. Choriocarcinoma presenting with thyrotoxicosis. Proc (Bayl Univ Med Cent). 2016;29(1):42–3.PubMedPubMedCentral Sotello D, Rivas AM, Test VJ, Lado-Abeal J. Choriocarcinoma presenting with thyrotoxicosis. Proc (Bayl Univ Med Cent). 2016;29(1):42–3.PubMedPubMedCentral
16.
go back to reference Hsieh TY, Hsu KF, Kuo PL, Huang SC. Uterine choriocarcinoma accompanied by an extremely high human chorionic gonadotropin level and thyrotoxicosis. J Obstet Gynaecol Res. 2008;34(2):274–8.CrossRefPubMed Hsieh TY, Hsu KF, Kuo PL, Huang SC. Uterine choriocarcinoma accompanied by an extremely high human chorionic gonadotropin level and thyrotoxicosis. J Obstet Gynaecol Res. 2008;34(2):274–8.CrossRefPubMed
17.
go back to reference Morley JE, Jacobson RJ, Melamed J, Hershman JM. Choriocarcinoma as a cause of thyrotoxicosis. Am J Med. 1976;60(7):1036–40.CrossRefPubMed Morley JE, Jacobson RJ, Melamed J, Hershman JM. Choriocarcinoma as a cause of thyrotoxicosis. Am J Med. 1976;60(7):1036–40.CrossRefPubMed
18.
go back to reference Odell WD, Bates RW, Rivlin RS, Lipsett MB, Hertz R. Increased thyroid function without clinical hyperthyroidism in patients with choriocarcinoma. J Clin Endocrinol Metab. 1963;23:658–64.CrossRefPubMed Odell WD, Bates RW, Rivlin RS, Lipsett MB, Hertz R. Increased thyroid function without clinical hyperthyroidism in patients with choriocarcinoma. J Clin Endocrinol Metab. 1963;23:658–64.CrossRefPubMed
19.
go back to reference Soutter WP, Norman R, Green-Thompson RW. The management of choriocarcinoma causing severe thyrotoxicosis. Two case reports. Br J Obstet Gynaecol. 1981;88(9):938–43.CrossRefPubMed Soutter WP, Norman R, Green-Thompson RW. The management of choriocarcinoma causing severe thyrotoxicosis. Two case reports. Br J Obstet Gynaecol. 1981;88(9):938–43.CrossRefPubMed
20.
go back to reference Yoshimura M, Hershman JM. Thyrotropic action of human chorionic gonadotropin. Thyroid. 1995;5(5):425–34.CrossRefPubMed Yoshimura M, Hershman JM. Thyrotropic action of human chorionic gonadotropin. Thyroid. 1995;5(5):425–34.CrossRefPubMed
21.
go back to reference Yoshimura M, Pekary AE, Pang XP, Berg L, Goodwin TM, Hershman JM. Thyrotropic activity of basic isoelectric forms of human chorionic gonadotropin extracted from hydatidiform mole tissues. J Clin Endocrinol Metab. 1994;78(4):862–6.PubMed Yoshimura M, Pekary AE, Pang XP, Berg L, Goodwin TM, Hershman JM. Thyrotropic activity of basic isoelectric forms of human chorionic gonadotropin extracted from hydatidiform mole tissues. J Clin Endocrinol Metab. 1994;78(4):862–6.PubMed
22.
go back to reference Inaguma S, Wang Z, Lasota J, Sarlomo-Rikala M, McCue PA, Ikeda H, et al. Comprehensive immunohistochemical study of programmed cell death ligand 1 (PD-L1): analysis in 5536 cases revealed consistent expression in trophoblastic tumors. Am J Surg Pathol. 2016;40(8):1133–42.CrossRefPubMedPubMedCentral Inaguma S, Wang Z, Lasota J, Sarlomo-Rikala M, McCue PA, Ikeda H, et al. Comprehensive immunohistochemical study of programmed cell death ligand 1 (PD-L1): analysis in 5536 cases revealed consistent expression in trophoblastic tumors. Am J Surg Pathol. 2016;40(8):1133–42.CrossRefPubMedPubMedCentral
23.
go back to reference Veras E, Kurman RJ, Wang TL, Shih IM. PD-L1 expression in human placentas and gestational trophoblastic diseases. Int J Gynecol Pathol. 2017;36(2):146–53.CrossRefPubMedPubMedCentral Veras E, Kurman RJ, Wang TL, Shih IM. PD-L1 expression in human placentas and gestational trophoblastic diseases. Int J Gynecol Pathol. 2017;36(2):146–53.CrossRefPubMedPubMedCentral
24.
go back to reference Ochi M, Miyamoto S, Terada Y, Furuhata Y, Awano N, Izumo T, et al. The significant antitumor activity of nivolumab in lung adenocarcinoma with choriocarcinomatous features. Intern Med. 2018;57(12):1773–7.CrossRefPubMedPubMedCentral Ochi M, Miyamoto S, Terada Y, Furuhata Y, Awano N, Izumo T, et al. The significant antitumor activity of nivolumab in lung adenocarcinoma with choriocarcinomatous features. Intern Med. 2018;57(12):1773–7.CrossRefPubMedPubMedCentral
25.
go back to reference Raghavapuram R, Veerankutty FH, Anandakumar M. Primary choriocarcinoma of the stomach. A case report and review of the literature. Indian J Surg Oncol. 2016;7(1):119–23.CrossRefPubMedPubMedCentral Raghavapuram R, Veerankutty FH, Anandakumar M. Primary choriocarcinoma of the stomach. A case report and review of the literature. Indian J Surg Oncol. 2016;7(1):119–23.CrossRefPubMedPubMedCentral
Metadata
Title
Severe thyrotoxicosis as initial presentation of gastric choriocarcinoma: a case report
Authors
Nicole M. Iñiguez-Ariza
Dalia Cuenca
Juvenal Franco-Granillo
Alberto Villalobos-Prieto
Janet Pineda-Díaz
Javier Baquera-Heredia
Publication date
01-12-2022
Publisher
BioMed Central
Published in
Journal of Medical Case Reports / Issue 1/2022
Electronic ISSN: 1752-1947
DOI
https://doi.org/10.1186/s13256-022-03343-5

Other articles of this Issue 1/2022

Journal of Medical Case Reports 1/2022 Go to the issue