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Validation of the mIBG skeletal SIOPEN scoring method in two independent high-risk neuroblastoma populations : the SIOPEN/HR-NBL1 and COG-A3973 trials

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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.
Keywords
STAGE 4 NEUROBLASTOMA, STEM-CELL TRANSPLANTATION, CHILDRENS ONCOLOGY, GROUP, HIGH-DOSE CHEMOTHERAPY, INDUCTION CHEMOTHERAPY, MULTIVARIATE-ANALYSIS, RANDOMIZED-TRIAL, PHASE-3 TRIAL, ROC CURVES, AGE, High-risk neuroblastoma, MIBG, SIOPEN score

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Chicago
Ladenstein, Ruth, Bieke Lambert, Ulrike Potschger, Maria-Rita Castellani, Valerie Lewington, Zvi Bar-Sever, Aurore Oudoux, et al. 2018. “Validation of the mIBG Skeletal SIOPEN Scoring Method in Two Independent High-risk Neuroblastoma Populations : the SIOPEN/HR-NBL1 and COG-A3973 Trials.” European Journal of Nuclear Medicine and Molecular Imaging 45 (2): 292–305.
APA
Ladenstein, R., Lambert, B., Potschger, U., Castellani, M.-R., Lewington, V., Bar-Sever, Z., Oudoux, A., et al. (2018). Validation of the mIBG skeletal SIOPEN scoring method in two independent high-risk neuroblastoma populations : the SIOPEN/HR-NBL1 and COG-A3973 trials. EUROPEAN JOURNAL OF NUCLEAR MEDICINE AND MOLECULAR IMAGING, 45(2), 292–305.
Vancouver
1.
Ladenstein R, Lambert B, Potschger U, Castellani M-R, Lewington V, Bar-Sever Z, et al. Validation of the mIBG skeletal SIOPEN scoring method in two independent high-risk neuroblastoma populations : the SIOPEN/HR-NBL1 and COG-A3973 trials. EUROPEAN JOURNAL OF NUCLEAR MEDICINE AND MOLECULAR IMAGING. 2018;45(2):292–305.
MLA
Ladenstein, Ruth, Bieke Lambert, Ulrike Potschger, et al. “Validation of the mIBG Skeletal SIOPEN Scoring Method in Two Independent High-risk Neuroblastoma Populations : the SIOPEN/HR-NBL1 and COG-A3973 Trials.” EUROPEAN JOURNAL OF NUCLEAR MEDICINE AND MOLECULAR IMAGING 45.2 (2018): 292–305. Print.
@article{8552701,
  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 {\textlangle}= 3 was the most useful predictor across trials. A SIOPEN score {\textlangle}= 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 {\textlangle}= 3 were 47\% +/- 7\% versus 26\% +/- 3\% for higher scores at diagnosis (p {\textlangle} 0.007) and 36\% +/- 4\% versus 14\% +/- 4\% (p {\textlangle} 0.001) for scores obtained post-induction. The COG-A3973 showed 5 yr.-EFS rates for scores {\textlangle}= 3 of 51\% +/- 7\% versus 34\% +/- 4\% for higher scores (p {\textlangle} 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 {\textlangle}= 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 {\textrangle} 3 after induction have very poor outcomes and should be considered for alternative therapeutic strategies.},
  author       = {Ladenstein, Ruth and Lambert, Bieke and Potschger, Ulrike and Castellani, Maria-Rita and Lewington, Valerie and Bar-Sever, Zvi and Oudoux, Aurore and Sliwinska, Anna and Taborska, Katerina and Biassoni, Lorenzo and Yanik, Gregory A and Naranjo, Arlene and Parisi, Marguerite T and Shulkin, Barry L and Nadel, Helen and Gelfand, Michael J and Matthay, Katherine K and Park, Julie R and Kreissman, Susan G and Valteau-Couanet, Dominique and Boubaker, Ariane},
  issn         = {1619-7070},
  journal      = {EUROPEAN JOURNAL OF NUCLEAR MEDICINE AND MOLECULAR IMAGING},
  keyword      = {STAGE 4 NEUROBLASTOMA,STEM-CELL TRANSPLANTATION,CHILDRENS ONCOLOGY,GROUP,HIGH-DOSE CHEMOTHERAPY,INDUCTION CHEMOTHERAPY,MULTIVARIATE-ANALYSIS,RANDOMIZED-TRIAL,PHASE-3 TRIAL,ROC CURVES,AGE,High-risk neuroblastoma,MIBG,SIOPEN score},
  language     = {eng},
  number       = {2},
  pages        = {292--305},
  title        = {Validation of the mIBG skeletal SIOPEN scoring method in two independent high-risk neuroblastoma populations : the SIOPEN/HR-NBL1 and COG-A3973 trials},
  url          = {http://dx.doi.org/10.1007/s00259-017-3829-7},
  volume       = {45},
  year         = {2018},
}

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