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Case Report
 
Massive gastro-intestinal haemorrhage in metastatic germ cell tumor from duodenal invasion: a report of two cases
Keywords :
Ankur Gadodia1, Indranil Ghosh2, Gaurav Prakash2, Sanjay Thulkar1, Vinod Raina2
Departments of Radiodiagnosis1 and Medical Oncology,2
Dr. B. R. A. Institute Rotary Cancer Hospital,
All India Institute of Medical Sciences,
New Delhi – 110029, India


Corresponding Author
: Dr. Ankur Gadodia
Email: gadodia_ankur@yahoo.co.in


DOI: http://dx.doi.org/

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48uep6bbph|2000F98CTab_Articles|Fulltext

Testicular germ cell tumor (GCT) is the most common malignancy among men aged 15 to 35 years, accounting for 1-2% of all malignancies.[1] Metastasis from testicular GCTs to the gastrointestinal (GI) tract is rare.[2,3,4] We report two young males with a metastatic mixed germ cell tumor, who had massive gastrointestinal hemorrhage secondary to duodenal invasion by retroperitoneal lymph node mass.


Case 1

A 25 year old man presented with right testicular swelling for 1 month duration. He underwent right orchidectomy at an outside center. Histopathological examination revealed a seminoma and immature teratoma. Subsequently he was referred to our center. On examination, there was a 5 × 5 cm lump in the right hypochondrium which was apparently arising from the retroperitoneum. Chest radiograph showed multiple ill-defined focal nodules. While being evaluated further, he developed massive hemoptysis and respiratory failure, for which he required endotracheal intubation and mechanical ventilation. Contrast-enhanced computed tomography (CECT) scan of chest and abdomen confirmed presence of multiple lung metastases with surrounding hemorrhage and revealed presence of multiple liver metastases and a pericaval mass infiltrating the duodenum. His serum alpha-fetoprotein (AFP), beta-human chorionic gonadotropin (ß-HCG), and lactate dehydrogenase (LDH) levels were 11.94 ng/mL (reference range 0 to 8), 14546 IU/L (normal less than 5), and 898 U/L (reference range 100 to 190), respectively.

The patient was diagnosed as a case of testicular mixed GCT stage IIIC (poor risk) with multiple lung, liver and retroperitoneal metastases. As the patient was very sick and on ventilator, he was given single-agent carboplatin 600 mg, along with packed red blood cell transfusions. Within 48 hours, there was clinical improvement and he was extubated. He was discharged after 3 days with future plan of continuing chemotherapy with bleomycin-etoposide-cisplatin (BEP) regime.

But after ten days, he again presented with history of malena and microcytic hypochromic anemia; hemoglobin was 4.5 g/dl. Serum ß-HCG had increased to >15,000 mIU/ml. Upper gastrointestinal tract endoscopy (UGIE) revealed a large ulcerated friable growth infiltrating the second part of duodenum with active bleeding from the surface. Argon plasma coagulation (APC) was applied over the bleeding site to achieve hemostasis. Endoscopic biopsy revealed choriocarcinoma. Patient was subsequently taken up for angiography and selective embolisation of the feeding vessels was done (Figure 1a & b). There was no further GI bleed and he has been transfusion-independent since the procedure. Thereafter he received BEP chemotherapy in reduced doses (cisplatin 40 mg/m2, etoposide 200 mg/m2 and bleomycin 30 IU) to avoid rapid tumor necrosis. He has tolerated the therapy well and clinically the abdominal mass has reduced in size. BEP chemotherapy has been planned for follow up visit.

Case 2

A 21 year old male presented with right testicular swelling of 4 months duration and jaundice for 20 days. On examination, there was 10 × 10 cm mass in the epigastric region. Serum bilirubin was 12 mg/dl (conjugated fraction 10 mg/dl). CECT scan of chest and abdomen revealed multiple pulmonary metastases, multiple large retroperitoneal lymph nodes displacing head of pancreas and gastric antrum, and compression of the common bile duct. He underwent rightsided high inguinal orchidectomy and histopathological examination revealed mixed germ cell tumor (embryonal cell carcinoma and yolk cell tumor).

Post orchidectomy serum AFP, ß-HCG and LDH levels were> 350 ng/mL, 1929 IU/L and 468 IU/L respectively. Final diagnosis of mixed testicular GCT stage IIIC was made. Percutaneous transhepatic biliary drainage was performed to relieve the biliary obstruction Due to hyperbilirubinemia and poor performance status, he was given low-dose single agent cisplatin (20 mg per day for 5 days). Because of marginal clinical response, same treatment was repeated and the patient discharged while waiting for the serum bilirubin to come down. He presented after three weeks with malena and anaemia. UGIE showed large friable growth with multiple clots in second part of duodenum. Argon plasma coagulation was used over bleeding points and packed red blood cells were transfused. In spite of above, the patient had an episode of massive upper GI bleeding with hemorrhagic shock. He was resuscitated with intravenous fluids, blood and plasma transfusions. Gel-foam embolisation of feeding vessel was planned but no feeding vessel could be identified on digital subtraction angiography. After recovery he was treated with first cycle of BEP regimen, following which the epigastric mass reduced by 50% and there was no further GI bleed. Further plan for this patient includes BEP chemotherapy

Discussion

Testicular GCTs are often metastatic to distant sites including retroperitoneal lymph nodes, lungs and liver. Clinically apparent gastrointestinal involvement from metastatic testicular cancer occurs in less than 5% of cases and is more commonly associated with non seminomatous germ cell tumours (NSGCT) than with seminomas.[2,3,4,5,6,7,8,9] However in a post-mortem study gastrointestinal metastases were found in 27% of cases.[5] Patients with intestinal metastases can present with perforation, obstruction and bleeding at diagnosis or during treatment. The sites of GI involvement include duodenum, jejunum, ileum, stomach, oesophagus, colon, and pancreas, with duodenum being the most common site.[4,5,6]

The close anatomical proximity between retroperitoneal nodes and duodenum may explain the high frequency of duodenal involvement.[9] In both patients who presented here, GI bleed was due to local invasion of duodenum by retroperitoneal lymph node mass. Onset was after the diagnosis of metastatic testicular GCT and while on chemotherapy for the same. Chemotherapy induced tumor necrosis might have potentiated bleeding in both cases.

Compared with other GCTs, choriocarcinoma frequently spreads hematogenously rather than through the lymphatic system.[1] It is the most aggressive GCT and has the tendency to outgrow its blood supply resulting in ulceration, necrosis, bleeding and perforation. In the first case, the primary tumor revealed seminoma and immature teratoma, whereas duodenal mass showed choriocarcinoma. A markedly high serum ß-HCG at baseline suggested that choriocarcinoma was a significant component of the tumor from the outset. As the primary tumor did not show any foci of choriocarcinoma, possibly a transformation in histology of the tumor to choriocarcinoma occurred at the metastatic site, which resulted in increased susceptibility to necrosis and bleed.

The outcome of the patients of metastatic GCT with GI involvement has been shown to be quite poor, and in one series, 4/5 patients died from unresponsive or progressive disease.[6] Management of such patients mostly requires multimodality approach, including endoscopic techniques, chemotherapy and angio-embolisation.

Obstructive jaundice has rarely been reported as a complication of metastatic GCT. Unsuspected testicular seminoma, metastatic to retroperitoneal lymph node has been reported to cause obstructive jaundice and mimic primary pancreatic cancer in an elderly male.[10] In analogy to obstructive jaundice caused by other solid-organ malignancies, management involves biliary drainage procedures and chemotherapy in reduced doses. As cisplatin and carboplatin are excreted through liver, they may be used safely in these patients. But in a chemo responsive tumor like GCT, whether chemotherapy alone can result in relief of the obstruction is not clear. In present patient we used biliary drainage and single age cisplatin initially, followed by BEP chemotherapy after normalising of serum bilirubin.

These two cases serve to illustrate several points: 1) GI haemorrhage in a young male may represent an underlying metastatic GCT; 2) endoscopic techniques and selective angiographic embolisation play an important role in controlling intestinal bleeding in such patients; and 3) there is increased risk of recurrent GI hemorrhage during the initial phase of therapy, likely due to chemotherapy induced tumor necrosis. The importance of a multidisciplinary management for these patients, encompassing the fields of medical oncology, urology, gastroenterology and interventional radiology, is also highlighted.

References

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  2. Cicin I, Ozyilmaz F, Karagol H, Yalcin F, Uzunoglu S, Kaplan M. Massive upper gastrointestinal bleeding from pure metastatic choriocarcinoma in patient with mixed germ cell tumor with subclinical intestinal metastasis. Urology. 2009;73:443.e15–7.
  3. Harikumar R, Harish K, Aravindan KP, Thomas V. Testicular choriocarcinoma with gastric metastasis presenting as hematemesis. Indian J Gastroenterol. 2004;23:223–4.
  4. Plukker JT, Schraffordt Koops H, Sleijfer DT, Oosterhuis JW, van der Jagt E. Intestinal haemorrhages in patients with a nonseminomatous testicular tumor. Cancer. 1991;68:2630–2.
  5. Bredael JJ, Vugrin D, Whitmore WF Jr. Autopsy findings in 154 patients with germ cell tumors of the testis. Cancer. 1982;50:548–51.
  6. Nord C, Fossa SD, Giercksky KE. Gastrointestinal presentation of germ cell malignancy. Eur Urol. 2000;38:721–4.
  7. Shariat SF, Duchene D, Kabbani W, Mucher Z, Lotan Y. Gastrointestinal hemorrhage as first manifestation of metastatic testicular tumor. Urology. 2005;66:1319.
  8. Varadarajulu S, Ramsey WH. Hematemesis as the initial presentation of testicular cancer. Am J Gastroenterol. 2000;12:3678–9.
  9. Nakamura A, Ikeda Y, Morishita S, Sato Y, Matsumoto M, Inomoto T, et al. Upper gastrointestinal bleeding arising from metastatic testicular tumor. J Gastroenterol. 1997;32:650–3.
  10. Wehrschütz M, Stöger H, Ploner F, Hofmann G, Wolf G, Höfler G, et al. Seminoma metastases mimicking primary pancreatic cancer. Onkologie. 2002;25:371–3.