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Journal of Thoracic Oncology:
doi: 10.1097/JTO.0b013e31819cce6c
Case Report

Primary Pulmonary Choriocarcinoma Presenting with a Hemothorax

Seol, Hyun-Joo MD*; Lee, Ju-Han MD†; Lee, Ki Yeol MD‡; Kim, Je-Hyeong MD§; Lee, Nak-Woo MD∥; Park, Hyung-Joo MD¶

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*Department of Obstetrics and Gynecology, College of Medicine, Kyunghee University; Departments of †Pathology, ‡Diagnostic Radiology, §Pulmonology, ∥Obstetrics and Gynecology, and ¶Thoracic Surgery, College of Medicine, Korea University, Seoul, Korea.

Disclosure: The authors declare no conflict of interest.

Address for correspondence: Nak-Woo Lee, MD, PhD, Department of Obstetrics and Gynecology, Korea University Ansan Hospital, 516, Gojan-dong, Danwon-gu, Ansan-si, Gyeonggi-do, 425-707, Korea. E-mail: nwlee@korea.ac.kr

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Abstract

Primary pulmonary choriocarcinoma is an extremely rare condition that is difficult to diagnose. In this report, we describe a case of primary pulmonary choriocarcinoma that presented with a hemothorax, initially diagnosed as an ectopic pregnancy. The patient was treated successfully with surgery followed by combination chemotherapy. In rare instances such as this, choriocarcinoma can originate in sites outside the genital tract; a thorough and thoughtful evaluation will ensure an accurate diagnosis and prompt treatment.

Choriocarcinoma usually arises in the uterine cavity. Although this may metastasizes to the lung, primary pulmonary choriocarcinoma is extremely rare. The clinical manifestations of pulmonary choriocarcinoma vary from an asymptomatic state to hemoptysis, chest pain, or dyspnea, according to the site or pattern of the tumor. A few cases of choriocarcinoma presenting with a hemothorax have been reported, but most of these cases involved metastatic tumors.1 We report a highly unusual case of primary pulmonary choriocarcinoma that presented with a hemothorax.

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CASE REPORT

A 19-year-old woman was admitted to the emergency room for chest pain that had developed suddenly 2 hours before admission. She was hemodynamically stable and had decreased breath sounds, particularly in the right lower lung field. Her last menstrual period had occurred 5 months before presentation and a qualitative urine beta human chorionic gonadotrophin (β-hCG) test was positive. However, a transvaginal ultrasound could detect neither an intrauterine nor extrauterine gestational sac. Chest radiograph and computed tomography showed a substantial effusion in the right pleural cavity and chest tube insertion yielded a bloody effusion, which confirmed the diagnosis of hemothorax. Follow-up computed tomography demonstrated a thin-walled mass measuring 2.5 cm in the right lower lobe of the lung (Figure 1). The serum level of β-hCG was 4247 mIU/ml. Endometrial curettage was performed and no gestational tissue was found. The patient was preoperatively diagnosed with an ectopic gestation in the abdominal cavity involving the diaphragm that resulted in a hemothorax, and therefore underwent diagnostic pelviscopy and thoracoscopy. Bilateral ovaries and salpinges were found to be intact without any evidence of ectopic pregnancy or pelvic tumor. During thoracoscopy, a mass was found in the right lower lung near the diaphragm. Frozen pathologic examination suggested that it was a choriocarcinoma. A thoracotomy was performed for complete removal of the tumor because its extensive adhesion to the pleura impaired thoracoscopic resection of the mass. The final pathologic examination confirmed the lesion as a primary pulmonary choriocarcinoma (Figure 2) and the patient received three courses of the multiagent etoposide, methotrexate, actinomycin, cyclophosphamide, vincristine chemotherapy regimen (etoposide 200 mg/m2/d on days 1 and 2, methotrexate 300 mg/m2/d on day 1, actinomycin 0.5 mg/d on days 1 and 2, cyclophosphamide 600 mg/m2/d on day 8, and vincristine 1.0 mg/m2/d on day 8). After a year, her serum β-hCG levels were within the normal range.

Figure 1
Figure 1
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Figure 2
Figure 2
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DISCUSSION

Primary pulmonary choriocarcinoma is extremely rare, with only 23 cases reported in the literature.2 To our knowledge, a primary pulmonary choriocarcinoma presenting with a hemothorax has never been reported. Although the origin of primary pulmonary choriocarcinomas is controversial, several theories about the pathogenesis of this tumor have been proposed, such as differentiation of the pulmonary epithelium into trophoblastic structures or metastatic emboli of gestational trophoblastic tissue undergoing spontaneous regression.3 In our case, we excluded metastatic gestational choriocarcinoma because the pelviscopy revealed grossly normal pelvic organs and endometrial tissues. The present case is an unusual type of primary pulmonary choriocarcinoma that manifested as a hemothorax. Although it was originally erroneously diagnosed as an ectopic pregnancy, the patient was treated successfully by surgical resection and combination chemotherapy.

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REFERENCES

1. Sudduth CD, Strange C, Campbell BA, Sahn SA. Metastatic choriocarcinoma of the lung presenting as hemothorax. Chest 1991;99:527–528.

2. Yamamoto S, Tanaka H, Takeo H, Yasuda K, Mastukuma S. Primary pulmonary choriocarcinoma combined with adenocarcinoma. Pathol Int 2006;56:402–407.

3. Aparicio J, Oltra A, Martinez-Moragon E, Llorca C, Gomez- Aldaravi L, Pastor M. Extragonadal nongestational choriocarcinoma involving the lung: a report of three cases. Respiration 1996;63:251–253.

Keywords:

Choriocarcinoma; Primary pulmonary Choriocarcinoma; Hemothorax

© 2009International Association for the Study of Lung Cancer

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