Choriocarcinoma of testes is a very rare tumor with poor prognosis, usually presenting with high serum level of human chorionic gonadotropin (hCG>50,000 mIU/ml) and advanced hematogenous metastases. Data with salvage chemotherapy has been sparse, with few long-term survivors. Between April 1996 and October 2004, 184 patients with germ cell tumor were treated at Indiana University with salvage high-dose chemotherapy (HDCT) with autologous peripheral blood stem cell transplant. Thirteen had pure choriocarcinoma or choriocarcinoma syndrome (normal testes by palpation and ultrasound, normal serum alpha-fetoprotein, advanced hematogenous metastases and high level hCG). All patients had progressed following one or two lines of cisplatin combination therapy. HDCT regimen was carboplatin 700 mg/m(2) and etoposide 750 mg/m(2) intravenously given for 3 consecutive days. A second course was given after hematopoietic recovery, usually 3-4 weeks later. The median survival was 19 months (range 5-90). Six patients (46%) are alive and continuously disease free (cNED) at a median follow-up of 37 months (range 19-75). One additional patient who relapsed after HDCT and was treated with third line chemotherapy followed by two surgical resections of choriocarcinoma is currently alive NED at +90 months from HDCT. Long-term disease-free survival and potential cure is possible with HDCT in choriocarcinoma patients that progressed after standard cisplatin combination therapy.