ABSTRACT
Introduction: Recurrent metastatic choriocarcinoma is a rare disease with historically limited guidance in the literature regarding standardized treatment protocols. In this case report, we review the course of a patient with recurrent metastatic choriocarcinoma re-treated with single agent pembrolizumab.Our patient was initially diagnosed with metastatic choriocarcinoma (FIGO Stage IV, WHO Score 13) after presenting for evaluation of amenorrhea. She received standard treatment with etoposide, methotrexate, actinomycin D, cyclophosphamide and vincristine (EMA-CO), with a complete response. However, she recurred one year later. Molecular profiling of a chest wall tumor demonstrated strong expression of programmed cell death ligand 1 (PD-L1), and she was started on treatment with single agent pembrolizumab achieving a complete response. Unfortunately, she recurred again 6 months following completion of treatment. She was re-treated with pembrolizumab for 2 years with complete response after 25 cycles and is currently without evidence of disease. She has been followed on surveillance, with no evidence of disease for more than 24 months following treatment. Conclusion: This case represents the first to our knowledge to discuss re-treatment with pembrolizumab for relapsed choriocarcinoma after achieving a complete response.
ABSTRACT
BACKGROUND: Tumor lysis syndrome (TLS) is an extremely rare complication of solid tumors and is more frequently observed in patients with hematologic malignancies. This report describes a novel approach to the management of a rare case of TLS in metastatic gestational trophoblastic neoplasia (GTN). CASE: A 17-year-old female presented 8 weeks postpartum with severe anemia, thyrotoxicosis, and elevated serum beta-human chorionic gonadotropin (beta-hCG). Imaging studies confirmed metastatic GTN to the lungs. The patient developed grade 4 TLS after the first cycle of etoposide, methotrexate, dactinomycin, cyclophosphamide, and vincristine (EMA-CO). She did not respond to standard treatment of aggressive hydration and allupurinol and continued to be in renal failure with elevated uric acid. A single dose of recombinant urate oxidase, rasburicase, rendered the uric acid level undetectable in 3 days and completely reversed the renal failure, avoiding hemodialysis. Three more cycles of EMA-CO were then administered. Subsequently, the patient developed congestive heart failure and was switched to single-agent actinomycin-D. Beta-hCG became negative after 5 cycles, and her ejection fraction returned to baseline. CONCLUSION: This is a rare case of TLS in the setting of metastatic GTN. To our knowledge this is the first reported case of utilizing rasburicase for the management of TLS in GTN.
