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Year : 2018  |  Volume : 14  |  Issue : 10  |  Page : 803-805

Successful management of a patient with radiological presentation of choriocarcinoma syndrome before induction chemotherapy

Department of Urology and Andrology, Graduate School of Medicine, Kansai Medical University, Osaka, Japan

Date of Web Publication24-Sep-2018

Correspondence Address:
Hidefumi Kinoshita
Department of Urology and Andrology, Graduate School of Medicine, Kansai Medical University, Osaka
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/0973-1482.175433

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 > Abstract 

Choriocarcinoma syndrome is a life-threatening lysis syndrome caused by blood vessel rupture and subsequent tumor bleeding. We describe a case of pretreatment choriocarcinoma syndrome that developed in a 27-year-old man. He underwent a high orchiectomy at a local hospital and was diagnosed with metastatic testicular tumor given the high serum human chorionic gonadotropin levels (943,601 mIU/mL). Thus, he was referred to our institution. Although he had bulky lung metastases and alveolar bleeding, we were able to administer full-dose chemotherapy with etoposide and cisplatin. On day 3 of chemotherapy, he presented with severe hypoxia and worsening of alveolar bleeding. Thus, he underwent tracheal intubation at the Intensive Care Unit. Full-dose chemotherapy was continued, and the patient was extubated upon improvement. He is currently alive and continuing treatment at another hospital.

Keywords: Alveolar bleeding, choriocarcinoma syndrome, full-dose chemotherapy, metastatic testicular tumor, radiological presentation

How to cite this article:
Yoshida T, Hayami Y, Yoshida K, Kinoshita H, Matsuda T. Successful management of a patient with radiological presentation of choriocarcinoma syndrome before induction chemotherapy. J Can Res Ther 2018;14, Suppl S3:803-5

How to cite this URL:
Yoshida T, Hayami Y, Yoshida K, Kinoshita H, Matsuda T. Successful management of a patient with radiological presentation of choriocarcinoma syndrome before induction chemotherapy. J Can Res Ther [serial online] 2018 [cited 2020 Nov 28];14:803-5. Available from: https://www.cancerjournal.net/text.asp?2018/14/10/803/175433

 > Introduction Top

Choriocarcinoma syndrome is a life-threatening tumor lysis syndrome subtype, with mortality rates as high as 80%. Most of the cases are reported with extraordinarily elevated human chorionic gonadotropin (hCG) values and multiple metastases. Tumor lysis involves the rupture of blood vessels, causing bleeding from the tumor, and subsequently, choriocarcinoma syndrome.[1] This report describes the successful treatment of severe choriocarcinoma syndrome caused by pulmonary metastatic testicular tumor using a multimodal therapy with subsequent respiratory intensive care.

 > Case Report Top

A 27-year-old man with a body surface area of 1.68 m2 was referred to our hospital after undergoing a left high orchiectomy. The pathological diagnosis of the left testicular mass was necrotic tumor tissue. He was diagnosed with germ cell tumor and vanishing primary tumor based on the hCG level of 943,601 mIU/mL and lactate dehydrogenase level of 2812 U/I. The alpha-fetoprotein level was 2 ng/mL or less. Computed tomography (CT) revealed multiple lung metastases and alveolar bleeding around the tumors [Figure 1]a. Oxygen saturation was 91% at room air, leading to a diagnosis of subclinical acute respiratory distress syndrome (ARDS) secondary to choriocarcinoma syndrome.
Figure 1: Computed tomography revealed multiple lung tumors and alveolar bleeding, (a) pretreatment, (b) during treatment

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This patient had poor prognosis according to the International Germ Cell Consensus Classification. Therefore, although bulky lung metastases and alveolar bleeding were observed, we recommended that the patient should undergo therapy with full-dose etoposide and cisplatin (EP), considering the likelihood of further deterioration of his respiratory condition. Bleomycin was avoided as first-line chemotherapy because it can cause lung toxicity as an adverse effect.

On day 3 of EP therapy, the patient complained of severe dyspnea and presented with worsening alveolar bleeding [Figure 1]b. PaO2 also decreased to 55.1 mmHg in room air. In the Intensive Care Unit, the patient was immediately placed under mechanical ventilation and was treated intensively to improve his systemic condition. After judging his condition as stable, we considered he could tolerate further chemotherapy. Thus, we continued to administer full-dose EP on days 4 and 5 of the EP regimen without delay or dose reduction.

Respiratory function gradually improved after two courses of EP therapy. Two courses of bleomycin, EP (BEP) therapy were administered while the patient remained under mechanical ventilation. Serum hCG decreased are shown in [Figure 2]. After four courses of EP and BEP, the serum hCG level decreased to 238.3 mIU/mL. Then, the patient was extubated and weaned off ventilatory support. The patient received four courses of paclitaxel, ifosfamide, and cisplatin and two courses of irinotecan and nedaplatin (CPT/N) as second- and third-line chemotherapies. Serum hCG level decreased to 4.7 mIU/mL and residual lung metastases had decreased in CT images after CPT/N therapy. The patient improved and continued to undergo additional chemotherapy for the residual disease at another hospital. The patient is currently alive and is receiving further chemotherapy at another hospital.
Figure 2: Changes in serum human chorionic gonadotropin levels during mechanical ventilation

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 > Discussion Top

Choriocarcinoma syndrome occurs in patients with numerous metastatic tumors and very high serum hCG level. The symptoms vary according to the sites of metastases. The most commonly reported site of onset of choriocarcinoma syndrome is the lung. In the present case, tumor lysis and vessel destruction caused alveolar bleeding and subsequent progression to ARDS. According to previous reports, almost patients with the same condition as ours died of severe ARDS.[2],[3] Bleeding from intestinal or liver metastases was also reported. Most reports showed that choriocarcinoma syndrome occurs typically on day 2 or 3 of the first induction chemotherapy.[4],[5],[6],[7] Although very rarely, pretreatment onset has also been reported.[3] In our case, there was radiological evidence of pretreatment choriocarcinoma syndrome with alveolar hemorrhage. Thus, taking measures to prevent the onset of this syndrome is important.

Adequate preparation for the management of patients who are likely to develop ARDS at an intensive care setting, mainly by providing mechanical ventilation support, may be the most important way to overcome choriocarcinoma syndrome. In the present case, because a CT before induction therapy already showed choriocarcinoma syndrome with alveolar hemorrhage, we expected that any chemotherapy could induce severe ARDS, which actually occurred on day 3 of EP. However, we were able to rapidly and successfully manage the serious condition. The stable condition of the patient allowed the administration of further chemotherapy.

To prevent this fatal syndrome, modified BEP regimes have been reported. According to the European Expert Consensus meeting, the reduced dose of the first chemotherapy is recommended for patients at high-risk of this syndrome. It is recommended to begin the administration of EP therapy for 2 days only, followed by full-dose BEP or Vp-16 ifosfamide cisplatin therapy beginning on day 11.[8] The concept of this treatment modification was based on the findings by Massard et al.,[9] who showed that ARDS secondary to choriocarcinoma syndrome was drastically decreased from 13/15 (86.7%) to 3/10 (30%), and the mortality decreased from 10/15 (66.7%) to 2/10 (20%) after modified BEP treatment.

However, there are some issues of the modified regime that still need clarification. The oncologic outcome of this regime compared with the conventional regime was uncertain; a consensus on the selection criteria of high-risk patients that might benefit more from the modified rather than the conventional treatment is required. Thus, it is necessary to conduct prospective studies to clarify the adequate risk classification and oncological outcome of the modified regime. Moreover, Massard et al.[9] reported that the mortality of ARDS was 10/13 (76.9%) with the conventional regime and 2/3 (66.7%) with the modified regime. Once ADRS secondary to choriocarcinoma syndrome occurred, it was questionable whether the modified regime reduced mortality or not. The mainstay of therapy for ADRS is the management of the underlying disorder causing it; therefore, we might be able to successfully treat him with full-dose conventional chemotherapy, while maintaining a stable respiratory condition under mechanical ventilation.

 > Conclusion Top

While strict whole-body control was required to manage choriocarcinoma syndrome, we were able to successfully administer a multidisciplinary treatment, controlling the respiratory status by mechanical ventilation, without the need to decrease the chemotherapy dose.

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Conflicts of interest

There are no conflicts of interest.

 > References Top

Logothetis CJ, Samuels ML, Selig DE, Johnson DE, Swanson DA, von Eschenbach AC. Primary chemotherapy followed by a selective retroperitoneal lymphadenectomy in the management of clinical stage II testicular carcinoma: A preliminary report. J Urol 1985;134:1127-30.  Back to cited text no. 1
Kobatake K, Kato M, Mita K. Advanced testicular cancer associated with life-threatening tumour lysis syndrome and choriocarcinoma syndrome. Can Urol Assoc J 2015;9:62-4.  Back to cited text no. 2
McGowan MP, Pratter MR, Nash G. Primary testicular choriocarcinoma with pulmonary metastases presenting as ARDS. Chest 1990;97:1258-9.  Back to cited text no. 3
Kawai K, Takaoka E, Naoi M, Mori K, Minami M, Shimazui T, et al. A case of metastatic testicular cancer complicated by tumour lysis syndrome and choriocarcinoma syndrome. Jpn J Clin Oncol 2006;36:665-7.  Back to cited text no. 4
Kandori S, Kawai K, Fukuhara Y, Joraku A, Miyanaga N, Shimazui T, et al. Acase of metastatic testicular cancer complicated by pulmonary hemorrhage due to choriocarcinoma syndrome. Int J Clin Oncol 2010;15:611-4.  Back to cited text no. 5
Tatokoro M, Kawakami S, Sakura M, Kobayashi T, Kihara K, Akamatsu H. Successful management of life-threatening choriocarcinoma syndrome with rupture of pulmonary metastatic foci causing hemorrhagic shock. Int J Urol 2008;15:263-4.  Back to cited text no. 6
Kirch C, Blot F, Fizazi K, Raynard B, Theodore C, Nitenberg G. Acute respiratory distress syndrome after chemotherapy for lung metastases from non-seminomatous germ-cell tumors. Support Care Cancer 2003;11:575-80.  Back to cited text no. 7
Beyer J, Albers P, Altena R, Aparicio J, Bokemeyer C, Busch J, et al. Maintaining success, reducing treatment burden, focusing on survivorship: Highlights from the third European consensus conference on diagnosis and treatment of germ-cell cancer. Ann Oncol 2013;24:878-88.  Back to cited text no. 8
Massard C, Plantade A, Gross-Goupil M, Loriot Y, Besse B, Raynard B, et al. Poor prognosis nonseminomatous germ-cell tumours (NSGCTs): Should chemotherapy doses be reduced at first cycle to prevent acute respiratory distress syndrome in patients with multiple lung metastases? Ann Oncol 2010;21:1585-8.  Back to cited text no. 9


  [Figure 1], [Figure 2]


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