Background: Current therapy for relapsed/refractory neuroblastoma is not evidence based. Long-term disease control is poor and there is a need for randomised controlled trials (RCT). BEACON-Neuroblastoma is the first randomized European study for relapsed/refractory neuroblastoma and is a critical element of the SIOPEN/ITCC strategy for the development of new agents
Patients and Methods: Factorial phase 2, European, open-label, RCT (EudraCT 2012-000072-42) sponsored by the University of Birmingham, UK, that will evaluate the role of bevacizumab (B) and three backbone chemotherapy regimens (temozolomide (T), irinotecan(I)-temozolomide and topotecan(To)-temozolomide) relapsed/refractory neuroblastoma. The trial incorporates molecular characterisation, functional imaging to elucidate the role of anti-angiogenic therapy, measurement of neuroblastoma mRNAs and is due to be completed in 2017.
Results: As of January 2016, 65 patients have been randomized to T (16), BT (17), IT (15), BIT (16) and BTTo (1) from 26 sites in 8 European countries. Patients had refractory (35), early (22) or late (8) relapsed neuroblastoma. Median age was 5.7 (range 1.6-21.2) years and MYCN amplification was present in 22%. Thirty nine of 50 cases with available data had measurable disease per RECIST, 49 had MIBG positive disease and 24 had bone marrow involvement. Most common sites of disease were bone, bone marrow and primary tumor. Progression-free survival (PFS) at 6 and 12 months is 57% (95% CI: 40%-70%) and 36% (CI: 18%-55%); overall survival is 77% (CI: 61% to 87%) and 52% (CI: 29%-71%) respectively. 6-monthPFS is: 50% (CI: 25%-71%) and 63% (CI: 41%-79%) for relapsed versus refractory neuroblastoma; 51% (CI: 19%-76%) and 57% (CI: 36%-72%) for MYCN amplified versus non-amplified patients.
Conclusions: The BEACON-Neuroblastoma trial has established a network and infrastructure to evaluate new drugs efficiently in Europe and will provide reliable evidence on treatments for relapsed/refractory NBL. The results will inform the design of future relapse and frontline studies.