Title

Validation of Postinduction Curie Scores in High-Risk Neuroblastoma: A Children's Oncology Group and SIOPEN Group Report on SIOPEN/HR-NBL1

Authors

Gregory A. Yanik, Department of Pediatrics and Communicable Diseases, University of Michigan Medical Center, Ann Arbor, Michigan gyanik@umich.edu.
Marguerite T. Parisi, Department of Radiology, University of Washington School of Medicine/Seattle Children's Hospital, Seattle, Washington.
Arlene Naranjo, Children's Oncology Group Statistics and Data Center, University of Florida, Gainesville, Florida.
Helen Nadel, Department of Radiology, BC Children's Hospital, Vancouver, British Columbia, Canada.
Michael J. Gelfand, Department of Radiology, Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio.
Julie R. Park, Department of Pediatrics, University of Washington School of Medicine/Seattle Children's Hospital, Seattle, Washington.
Ruth L. Ladenstein, Department of Pediatrics, St. Anna Children's Hospital, Vienna, Austria.
Ulrike Poetschger, Department of Statistics, St. Anna Children's Cancer Research Institute, Vienna, Austria.
Ariane Boubaker, Institute of Radiology, Clinique de La Source, Lausanne, Switzerland.
Dominique Valteau-Couanet, Department of Pediatric and Adolescent Oncology, Gustave Roussy Institute, Universite Paris-Sud, Villejuif, France.
Bieke Lambert, Radiology and Nuclear Medicine, Ghent University, Ghent, Belgium.
Maria-Rita Castellani, Nuclear Medicine Division, Istituto Nazionale Tumori di Milano, Milan, Italy.
Zvi Bar-Sever, Schneider Children's Medical Center of Israel, Petah-Tivka, Israel.
Aurore Oudoux, Centre Oscar-Lambret, Lille, France.
Anna Kaminska, Children's Memorial Health Institute, Warsaw, Poland.
Susan G. Kreissman, Department of Pediatrics, Duke University Medical Center, Durham, North Carolina.

Abstract

A semiquantitative I-metaiodobenzylguanidine (I-MIBG) scoring method (the Curie score, or CS) was previously examined in the Children's Oncology Group (COG) high-risk neuroblastoma trial, COG A3973, with a postinduction CS of more than 2 being associated with poor event-free survival (EFS). The validation of the CS in an independent dataset, International Society of Paediatric Oncology European Neuroblastoma/High-Risk Neuroblastoma 1 (SIOPEN/HR-NBL1), is now reported. A retrospective analysis of I-MIBG scans obtained from patients who had been prospectively enrolled in SIOPEN/HR-NBL1 was performed. All patients exhibited I-MIBG-avid, International Neuroblastoma Staging System stage 4 neuroblastoma. I-MIBG scans were evaluated at 2 time points, diagnosis ( = 345) and postinduction ( = 330), before consolidation myeloablative therapy. Scans of 10 anatomic regions were evaluated, with each region being scored 0-3 on the basis of disease extent and a cumulative CS generated. Cut points for outcome analysis were identified by Youden methodology. CSs from patients enrolled in COG A3973 were used for comparison. The optimal cut point for CS at diagnosis was 12 in SIOPEN/HR-NBL1, with a significant outcome difference by CS noted (5-y EFS, 43.0% ± 5.7% [CS ≤ 12] vs. 21.4% ± 3.6% [CS > 12], < 0.0001). The optimal CS cut point after induction was 2 in SIOPEN/HR-NBL1, with a postinduction CS of more than 2 being associated with an inferior outcome (5-y EFS, 39.2% ± 4.7% [CS ≤ 2] vs. 16.4% ± 4.2% [CS > 2], < 0.0001). The postinduction CS maintained independent statistical significance in Cox models when adjusted for the covariates of age and gene copy number. The prognostic significance of postinduction CSs has now been validated in an independent cohort of patients (SIOPEN/HR-NBL1), with a postinduction CS of more than 2 being associated with an inferior outcome in 2 independent large, cooperative group trials.

Publication Title

Journal of nuclear medicine : official publication, Society of Nuclear Medicine

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