TY - JOUR
T1 - Rationale for irradiation of persisting oligo-skeletal metastases to improve survival of metastatic neuroblastoma patients with a poor response to chemotherapy
T2 - A retrospective study
AU - Rossillon, Lea
AU - Edeline, Véronique
AU - Agrigoroaie, Laurentiu
AU - Pasqualini, Claudia
AU - Berlanga, Pablo
AU - Bolle, Stephanie
AU - Dufour, Christelle
AU - Valteau-Couanet, Dominique
N1 - Publisher Copyright:
© 2024 The Author(s). Pediatric Blood & Cancer published by Wiley Periodicals LLC.
PY - 2025/1/1
Y1 - 2025/1/1
N2 - Background: Persistent metaiodobenzylguanidine (mIBG)-positive skeletal metastases post induction in high-risk neuroblastoma correlate with a poor outcome. The aim of this study was to investigate the potential rationale for a prospective randomized study evaluating the impact on event-free survival of the irradiation of residual oligo-skeletal metastases. Procedure: Patients over 1 year with a stage M neuroblastoma treated between 2000 and 2020 at Gustave Roussy were identified. Patients with a positive mIBG scan at diagnosis and persistent skeletal metastases after high-dose chemotherapy (HDC) were included. Data were retrospectively collected and mIBG scans reviewed by two nuclear medicine physicians. Results: Persistent skeletal uptake after HDC was observed in 30/201 patients (15%). Four patients reached a complete response at the end of maintenance treatment and did not relapse (median follow-up [FU] 8 years [1.8–11.8]), while two patients had progressive disease during maintenance. Among the 24 patients with persistent skeletal uptakes at the end of treatment, seven had a persistent response (median FU 8.2 years [4–15.6]). Median SIOPEN (International Society of Paediatric Oncology European Neuroblastoma) scores post consolidation and at the end of treatment were, respectively, 2 [1–6] and 2 [0–4] for patients with persistent responses compared to 4 [1–28] and 2 [1–17] for patients with progressive diseases. Median SIOPEN score at progression was 34 [2–56]. Conclusions: Our study underlines that only a minority of patients had persistent skeletal mIBG-positive scans after HDC. Recurrence mainly occurred in disease sites present at diagnosis that cleared with chemotherapy. On-therapy control of the disease is the main challenge. These results highlight the complexity of conducting a randomized study exploring this strategy.
AB - Background: Persistent metaiodobenzylguanidine (mIBG)-positive skeletal metastases post induction in high-risk neuroblastoma correlate with a poor outcome. The aim of this study was to investigate the potential rationale for a prospective randomized study evaluating the impact on event-free survival of the irradiation of residual oligo-skeletal metastases. Procedure: Patients over 1 year with a stage M neuroblastoma treated between 2000 and 2020 at Gustave Roussy were identified. Patients with a positive mIBG scan at diagnosis and persistent skeletal metastases after high-dose chemotherapy (HDC) were included. Data were retrospectively collected and mIBG scans reviewed by two nuclear medicine physicians. Results: Persistent skeletal uptake after HDC was observed in 30/201 patients (15%). Four patients reached a complete response at the end of maintenance treatment and did not relapse (median follow-up [FU] 8 years [1.8–11.8]), while two patients had progressive disease during maintenance. Among the 24 patients with persistent skeletal uptakes at the end of treatment, seven had a persistent response (median FU 8.2 years [4–15.6]). Median SIOPEN (International Society of Paediatric Oncology European Neuroblastoma) scores post consolidation and at the end of treatment were, respectively, 2 [1–6] and 2 [0–4] for patients with persistent responses compared to 4 [1–28] and 2 [1–17] for patients with progressive diseases. Median SIOPEN score at progression was 34 [2–56]. Conclusions: Our study underlines that only a minority of patients had persistent skeletal mIBG-positive scans after HDC. Recurrence mainly occurred in disease sites present at diagnosis that cleared with chemotherapy. On-therapy control of the disease is the main challenge. These results highlight the complexity of conducting a randomized study exploring this strategy.
KW - irradiation
KW - mIBG
KW - neuroblastoma
KW - persistent oligo-skeletal metastases
UR - http://www.scopus.com/inward/record.url?scp=85206199109&partnerID=8YFLogxK
U2 - 10.1002/pbc.31350
DO - 10.1002/pbc.31350
M3 - Article
AN - SCOPUS:85206199109
SN - 1545-5009
VL - 72
JO - Pediatric Blood and Cancer
JF - Pediatric Blood and Cancer
IS - 1
M1 - e31350
ER -