Statistical Framework in Support of a Revised Children's Oncology Group Neuroblastoma Risk Classification System - PubMed (original) (raw)

Statistical Framework in Support of a Revised Children's Oncology Group Neuroblastoma Risk Classification System

Arlene Naranjo et al. JCO Clin Cancer Inform. 2018 Dec.

Abstract

Purpose: The International Neuroblastoma Risk Group (INRG) Staging System (INRGSS) was developed through international consensus to provide a presurgical staging system that uses clinical and imaging data at diagnosis. A revised Children's Oncology Group (COG) neuroblastoma (NB) risk classification system is needed to incorporate the INRGSS and within the context of modern therapy. Herein, we provide statistical support for the clinical validity of a revised COG risk classification system.

Patients and methods: Nine factors were tested for potential statistical and clinical significance in 4,569 patients diagnosed with NB who were enrolled in the COG biology/banking study ANBL00B1 (2006-2016). Recursive partitioning was performed to create a survival-tree regression (STR) analysis of event-free survival (EFS), generating a split by selecting the strongest prognostic factor among those that were statistically significant. The least absolute shrinkage and selection operator (LASSO) was applied to obtain the most parsimonious model for EFS. COG patients were risk classified using STR, LASSO, and per the 2009 INRG classification (generated using an STR analysis of INRG data). Results were descriptively compared among the three classification approaches.

Results: The 3-year EFS and overall survival (± SE) were 72.9% ± 0.9% and 84.5% ± 0.7%, respectively (N = 4,569). In each approach, the most statistically and clinically significant factors were diagnostic category (eg, NB, ganglioneuroblastoma), INRGSS, MYCN status, International Neuroblastoma Pathology Classification, ploidy, and 1p/11q status. The results of the STR analysis were more concordant with those of the INRG classification system than with LASSO, although both methods showed moderate agreement with the INRG system.

Conclusion: These analyses provide a framework to develop a new COG risk classification incorporating the INRGSS. There is statistical evidence to support the clinical validity of each of the three classifications: STR, LASSO, and INRG.

PubMed Disclaimer

Conflict of interest statement

Arlene Naranjo

No relationship to disclose

Meredith S. Irwin

No relationship to disclose

Michael D. Hogarty

No relationship to disclose

Susan L. Cohn

Stock and Other Ownership Interests: United Therapeutics, United Therapeutics (I), Varian Medical Systems (I), Vermillion, Resmed (I), Merck, Merck (I), Stryker, Stryker (I), Amgen, Amgen (I), Pfizer, Pfizer (I), Abbvie, Jazz Pharmaceuticals, Eli Lilly, Sanofi, Varex Imaging, Vermillion

Research Funding: United Therapeutics (Inst), Merck (Inst)

Julie R. Park

Honoraria: Bristol-Myers Squibb

Travel, Accommodations, Expenses: Roche

Wendy B. London

No relationship to disclose

Figures

Fig 1.

Fig 1.

Relationship between the INSS and the INRGSS. IDRF, image-defined risk factor; INRGSS, International Neuroblastoma Risk Group Staging System; INSS, International Neuroblastoma Staging System; mIBG, metaiodobenzylguanidine.

Fig 2.

Fig 2.

Distribution of patients by INSS stage 1 through 3 and IDRF status into INRGSS L1 and L2 (n = 2,192 patients with INRGSS L1 or L2 tumors). IDRF, image-defined risk factor; INRGSS, International Neuroblastoma Risk Group Staging System; INSS, International Neuroblastoma Staging System.

Fig 3.

Fig 3.

EFS survival-tree regression of 4,569 patients enrolled on the Children’s Oncology Group NB biology and banking study ANBL00B1 between August 18, 2006, and June 30, 2016. Quoted values are 3-year EFS and OS rates. Diff, differentiated; EFS, event-free survival; GNB, ganglioneuroblastoma; GNBI, ganglioneuroblastoma, intermixed; Ind, indeterminate; INRG, International Neuroblastoma Risk Group; MKI, mitosis-karyorrhexis index; NB, neuroblastoma; OS, overall survival; Undiff, undifferentiated.

Similar articles

Cited by

References

    1. Howlader N, Noone AM, Krapcho M, et al., editors. SEER Cancer Statistics Review, 1975-2009 (Vintage 2009 Populations) Bethesda, MD: National Cancer Institute; 2012. http://seer.cancer. gov/csr/1975_2009_pops09/
    1. Gurney JG, Ross JA, Wall DA, et al. Infant cancer in the U.S.: Histology-specific incidence and trends, 1973 to 1992. J Pediatr Hematol Oncol. 1997;19:428–432. - PubMed
    1. Monclair T, Brodeur GM, Ambros PF, et al. The International Neuroblastoma Risk Group (INRG) staging system: An INRG Task Force report. J Clin Oncol. 2009;27:298–303. - PMC - PubMed
    1. Brodeur GM, Seeger RC, Barrett A, et al. International criteria for diagnosis, staging, and response to treatment in patients with neuroblastoma. J Clin Oncol. 1988;6:1874–1881. - PubMed
    1. Brodeur GM, Pritchard J, Berthold F, et al. Revisions of the international criteria for neuroblastoma diagnosis, staging, and response to treatment. J Clin Oncol. 1993;11:1466–1477. - PubMed

Publication types

MeSH terms

Substances

LinkOut - more resources