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Table of Contents
CASE REPORT
Year : 2019  |  Volume : 11  |  Issue : 1  |  Page : 109-112  

Acute pulmonary hemorrhage associated with metastatic testicular choriocarcinoma in a 46-year-old incarcerated male


1 Alabama College of Osteopathic Medicine, Dothan, Alabama, USA
2 Department of Urology, Hahnemann University Hospital, Philadelphia, Pennsylvania, USA
3 Department of Urology, Einstein Medical Center, Philadelphia, Pennsylvania, USA

Date of Submission22-Apr-2018
Date of Acceptance11-Dec-2018
Date of Web Publication21-Jan-2019

Correspondence Address:
Miss. Hali Pearce
105 Devon Avenue, Bellmawr, NJ 08031
USA
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DOI: 10.4103/UA.UA_51_18

PMID: 30787584

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   Abstract 

Pure testicular choriocarcinoma is a rare histological subtype of germ cell tumor (GCT) and typically presents with distant metastases and aggressive features leading to a generally poor prognosis. Unique to choriocarcinoma among GCT histological subtypes is the propensity of spontaneous hemorrhage into metastatic lesions. We report a case of pure testicular choriocarcinoma in a 46-year-old male with postoperative acute pulmonary hemorrhage secondary to tumor invasion of the lungs, and the subsequent management of his disease with a discussion of relevant literature.

Keywords: Choriocarcinoma, germ cell tumor, pulmonary hemorrhage, testicular cancer


How to cite this article:
Pearce H, Edwards DC, Levy JA, McGreen BH, Mackovick L, Brennan M, McHugh M, Mapow B, Schanne FJ, Belkoff L. Acute pulmonary hemorrhage associated with metastatic testicular choriocarcinoma in a 46-year-old incarcerated male. Urol Ann 2019;11:109-12

How to cite this URL:
Pearce H, Edwards DC, Levy JA, McGreen BH, Mackovick L, Brennan M, McHugh M, Mapow B, Schanne FJ, Belkoff L. Acute pulmonary hemorrhage associated with metastatic testicular choriocarcinoma in a 46-year-old incarcerated male. Urol Ann [serial online] 2019 [cited 2019 Apr 24];11:109-12. Available from: http://www.urologyannals.com/text.asp?2019/11/1/109/250554


   Introduction Top


Primary testicular tumors are the most common solid malignant tumor in young men in the United States.[1] Germ cell tumors (GCTs) are the most common subtype of testicular malignancies. While normally identified as components of a mixed GCT, choriocarcinoma as an isolated histology comprises only 1% of GCT.[2] Choriocarcinoma syndrome (CS), a rare and lethal complication, presents as hemorrhage from metastatic sites and can manifest at any time during treatment, but frequently after the initiation of chemotherapy.[3]


   Case Report Top


A 46-year-old incarcerated male presented to the emergency department with the primary complaint of nausea, vomiting, and weight loss. His medical history was significant for a diagnosis of schizophrenia and history of right-sided cryptorchidism, status postinguinal orchiopexy at the age of 13 years. He had a 20 pack-year smoking history. He denied hematuria, dysuria, fevers, or chills. Overall, he was a poor historian of questionable mental status. On physical examination, it was noted that he had a large, firm, immobile right-sided scrotal mass that was tender to examination. The patient reported that this mass had been present and enlarging over the previous several months.

Initial laboratory values revealed a mild leukocytosis (13.0 × 103), anemia (9.1 g/dL), normal alpha -fetoprotein (AFP) (1.0 ng/mL), elevated lactate dehydrogenase (LDH) (715 U/L), and elevated beta-human chorionic gonadotropin (b-HCG) (136,017 mIU/mL). Scrotal ultrasound revealed a 12.5 cm heterogeneous scrotal mass with central necrosis [Figure 1]a. Chest X-ray revealed diffuse pulmonary metastases [Figure 1]b. Magnetic resonance imaging (MRI) of the brain was ultimately performed for questionable altered mental status and revealed a small enhancing lesion [Figure 1]c.
Figure 1: Testicular choriocarcinoma. Preoperative imaging. (a) Scrotal ultrasonography reveals 12.5 cm heterogeneous scrotal mass with central necrosis. (b) Chest X-ray reveals diffuse pulmonary metastases. (c) Magnetic resonance imaging brain reveals a small enhancing lesion

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He underwent open right radical inguinal orchiectomy, which was complicated by his previous inguinal surgery and dense adherence to the right hemiscrotum. Gross examination demonstrated a large, centrally necrotic lesion [Figure 2]a and [Figure 2]b. Final pathology revealed pure choriocarcinoma with a positive inferior scrotal margin. Noted on histopathology were a complete replacement of parenchyma with tumor and epididymal invasion [Figure 2]c, multinucleated syncytiotrophoblasts [Figure 2]d, and tumor emboli [Figure 2]e. Postoperatively, the patient developed respiratory distress requiring non-rebreather mask oxygenation and was weaned to nasal cannula over the following days. This was initially attributed to history of smoking and the large pulmonary metastatic burden. On postoperative day (POD) #2, he was started on etoposide, ifosfamide, and cisplatin chemotherapy. However, he remained tachycardic with increasingly worse oxygen saturation. CT angiogram was performed to evaluate for pulmonary embolism (PE), which demonstrated a singular, small left lower lobe PE, determined to be an unlikely contributor to his profound hypoxemia [Figure 3]. On duplex ultrasound, no lower extremity venous thrombosis was identified. Nevertheless, heparin anticoagulation was initiated. The same day, he developed severe hemoptysis and increased oxygen requirements. Anticoagulation was held until hemoptysis resolved, at which point, he was transitioned to apixaban. Ultimately, during a subsequent admission, he was transitioned to an inferior vena cava filter secondary to gastrointestinal bleeding, suspected to be from potential gastrointestinal metastases.
Figure 2: Testicular choriocarcinoma. Pathology/gross specimen. (a) Right testicle, gross. (b) Gross right testicle, incised to reveal large, centrally necrotic lesion. (c) Histopathology reveals complete replacement of parenchyma with tumor and epididymal invasion. (d) Histopathology reveals multinucleated syncytiotrophoblasts. (e) Histopathology reveals tumor emboli

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Figure 3: Testicular choriocarcinoma. Postoperative Imaging. Computed tomography imaging of the chest reveals left lower lobe pulmonary embolism (arrow)

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bHCG levels were followed over the subsequent months. On POD #14, bHCG was 34,238 mIU/mL, 1590 mIU/mL at 1 month, and 74 mIU/mL at 3 months. The patient continued on the previous chemotherapy regimen and is currently on his fourth cycle with no further hemorrhagic events.


   Discussion and Management Top


The incidence of testicular cancer has increased during the last century, but testicular tumors are still a rare occurrence. Nevertheless, primary testicular tumors are the most common solid malignant tumor in young men in the US.[1] According to the National Cancer Institute's Surveillance, Epidemiology, and End Results Program, testicular cancer has an annual incidence of 5.7 cases per 100,000 people. In 2014, there was an estimated prevalence of 251,194 men living with testicular cancer in the US. Despite its prevalence, testicular cancer has a 95.1% 5-year survival rate.[4] GCTs are the most common subtype of testicular cancers. The incidence rates of GCTs in white males and black males are 3.7 and 0.9/100,000 per year, respectively.[2]

Testicular choriocarcinoma is classified as a subtype of nonseminomatous GCT (NSGCT). Choriocarcinoma is rarely identified in its pure form and is commonly found as a component of a mixed GCT. Pure testicular choriocarcinomas make up 1% of GCT cases.[2] Testicular choriocarcinoma is significantly more aggressive and chemo-refractory than other subtypes of testicular cancer, with a 5-year survival rate <80%.[5] Testicular cancer primarily affects men between the ages of 20 and 34 years; however, the majority of choriocarcinoma cases affect men between the ages of 25 and 30 years. Choriocarcinoma has a high likelihood of early metastasis, primarily to the lungs, liver, and brain. The lungs are involved in almost 100% of metastatic cases.[6]

Typical presenting features of a choriocarcinoma include a man in his third to the fourth decade with a testicular mass and elevated serum bHCG. Signs and symptoms may include bilateral tender gynecomastia, hematogenous metastases (most commonly to the lungs bilaterally, presenting with a “snowstorm” appearance on chest X-ray), hyperthyroidism and/or hemoptysis.[3]

A rare and lethal complication of the disease, CS is defined as hemorrhage from metastatic sites. If the metastases are in the lung, pulmonary hemorrhage, and acute pulmonary decompensation can result due to tumor invasion into pulmonary vasculature. CS is more likely with higher levels of bHCG.[7] The most common presentation of CS is pulmonary hemorrhage, but hemorrhage can develop in the liver, brain, and small bowel as well. Hemorrhage can occur immediately following chemotherapy, or present as the initial primary complaint.[3]

The current patient had a history of ipsilateral cryptorchidism status postorchidopexy at age thirteen. Cryptorchidism is considered a risk factor for infertility and testicular GCTs.[8] Cryptorchidism is a common diagnosis in newborn male infants, with around 2%–4% diagnosed with unilateral or bilateral cryptorchidism. About 10% of all cases of testicular GCTs occur in men with a history of cryptorchidism, and the risk of developing testicular cancer due to cryptorchidism is increased 5–10 times than that of the general male population.[9] The current guidelines for the management of congenital cryptorchidism is orchiopexy between 6 and 18 months of age if there is not spontaneous resolution.[10]

When presented with a suspicious testicular mass, workup includes a thorough history and physical, AFP, bHCG, and LDH levels, a complete chemistry profile, and testicular ultrasound. Postdiagnostic workup of an NSGCT includes chest/abdominal/pelvic computed tomography, repeat bHCG, LDH, and AFP levels, and brain MRI if indicated. NSGCTs of all stages can be treated with primary chemotherapy using tumor markers to follow response. The standard chemotherapy regimen for poor-risk patients is four cycles of BEP (bleomycin, etoposide, and cisplatin); however, four cycles of VIP (etoposide, cisplatin, and ifosfamide) can be used in patients who cannot tolerate bleomycin due to pulmonary toxicity.[11] If bHCG levels fail to normalize, additional cycles should be implemented. If bHCG levels plateau after several rounds of chemotherapy, the patient has refractory disease and more aggressive treatment should be considered including high-dose chemotherapy with stem-cell rescue. bHCG levels <10 mIU/mL indicate an acceptable response to treatment.[5]


   Conclusion Top


Pure testicular choriocarcinomas are a rare occurrence and can be complicated by the development of CS. Metastatic choriocarcinoma must be treated with a chemotherapeutic regimen, and bHCG should be followed to ensure response to treatment in addition to imaging parameters.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
Adra N, Einhorn LH. Testicular cancer update. Clin Adv Hematol Oncol 2017;15:386-96.  Back to cited text no. 1
    
2.
Lowe K, Paterson J, Armstrong S, Walsh S, Groome M, Mowat C, et al. Metastatic testicular choriocarcinoma: A rare cause of upper GI bleeding. ACG Case Rep J 2015;3:36-8.  Back to cited text no. 2
    
3.
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. 3
    
4.
Cancer Stat Facts: Testis Cancer. National Cancer Institute – Surveillance, Epidemiology and End Results Program. Available from: https://www.seer.cancer.gov/statfacts/html/testis.html. [Last retrieved on 2017 Nov 28].  Back to cited text no. 4
    
5.
Reilley MJ, Pagliaro LC. Testicular choriocarcinoma: A rare variant that requires a unique treatment approach. Curr Oncol Rep 2015;17:2.  Back to cited text no. 5
    
6.
Tinkle LL, Graham BS, Spillane TJ, Barr RJ. Testicular choriocarcinoma metastatic to the skin: An additional case and literature review. Cutis 2001;67:117-20.  Back to cited text no. 6
    
7.
Takahashi T, Kobayashi R. Choriocarcinoma syndrome after resection of primary pulmonary choriocarcinoma: Report of a case. Surg Case Rep 2016;2:122.  Back to cited text no. 7
    
8.
Leslie SW, Villanueva CA. Cryptorchidism. In: StatPearls. Treasure Island (FL): StatPearls Publishing; 2018. Available from: https://www.ncbi.nlm.nih.gov/books/NBK470270/. [Last updated on 2018 Jan 06].  Back to cited text no. 8
    
9.
Ferguson L, Agoulnik AI. Testicular cancer and cryptorchidism. Front Endocrinol (Lausanne) 2013;4:32.  Back to cited text no. 9
    
10.
Niedzielski JK, Oszukowska E, Słowikowska-Hilczer J. Undescended testis – Current trends and guidelines: A review of the literature. Arch Med Sci 2016;12:667-77.  Back to cited text no. 10
    
11.
National Comprehensive Cancer Network. Testicular Cancer (Version 2.2018). https://www.nccn.org/professionals/physician_gls/PDF/testicular.pdf. [Last accessed on 2017 Nov 28].  Back to cited text no. 11
    


    Figures

  [Figure 1], [Figure 2], [Figure 3]



 

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