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- Title
Validation of the mIBG skeletal SIOPEN scoring method in two independent high-risk neuroblastoma populations: the SIOPEN/HR-NBL1 and COG-A3973 trials.
- Authors
Ladenstein, Ruth; Lambert, Bieke; Pötschger, Ulrike; Castellani, Maria-Rita; Lewington, Valerie; Bar-Sever, Zvi; Oudoux, Aurore; Śliwińska, Anna; Taborska, Katerina; Biassoni, Lorenzo; Yanik, Gregory A.; Naranjo, Arlene; Parisi, Marguerite T.; Shulkin, Barry L.; Nadel, Helen; Gelfand, Michael J.; Matthay, Katherine K.; Park, Julie R.; Kreissman, Susan G.; Valteau-Couanet, Dominique
- Abstract
Background: Validation of the prognostic value of the SIOPEN mIBG skeletal scoring system in two independent stage 4, mIBG avid, high-risk neuroblastoma populations. Results: The semi-quantitative SIOPEN score evaluates skeletal meta-iodobenzylguanidine (mIBG) uptake on a 0-6 scale in 12 anatomical regions. Evaluable mIBG scans from 216 COG-A3973 and 341 SIOPEN/HR-NBL1 trial patients were reviewed pre- and post-induction chemotherapy. The prognostic value of skeletal scores for 5-year event free survival (5 yr.-EFS) was tested in the source and validation cohorts. At diagnosis, both cohorts showed a gradual non-linear increase in risk with cumulative scores. Several approaches were explored to test the relationship between score and EFS. Ultimately, a cutoff score of ≤3 was the most useful predictor across trials. A SIOPEN score ≤ 3 pre-induction was found in 15% SIOPEN patients and in 22% of COG patients and increased post-induction to 60% in SIOPEN patients and to 73% in COG patients. Baseline 5 yr.-EFS rates in the SIOPEN/HR-NBL1 cohort for scores ≤3 were 47% ± 7% versus 26% ± 3% for higher scores at diagnosis ( p < 0.007) and 36% ± 4% versus 14% ± 4% ( p < 0.001) for scores obtained post-induction. The COG-A3973 showed 5 yr.-EFS rates for scores ≤3 of 51% ± 7% versus 34% ± 4% for higher scores (p < 0.001) at diagnosis and 43% ± 5% versus 16% ± 6% ( p = 0.004) for post-induction scores. Hazard ratios (HR) significantly favoured patients with scores ≤3 after adjustment for age and MYCN-amplification. Optimal outcomes were recorded in patients who achieved complete skeletal response. Conclusions: Validation in two independent cohorts confirms the prognostic value of the SIOPEN skeletal score. In particular, patients with an absolute SIOPEN score > 3 after induction have very poor outcomes and should be considered for alternative therapeutic strategies.
- Subjects
NEUROBLASTOMA; MIBG (Chemical); PROGRESSION-free survival; SKELETON; PROGNOSIS; PATIENTS
- Publication
European Journal of Nuclear Medicine & Molecular Imaging, 2018, Vol 45, Issue 2, p292
- ISSN
1619-7070
- Publication type
Article
- DOI
10.1007/s00259-017-3829-7