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Predictive Value of the Pretreatment Extent of Disease System in Hepatoblastoma: Results From the International Society of Pediatric Oncology Liver Tumor Study Group SIOPEL-1 Study

From the Pediatric Surgical Center of Amsterdam, Emma Children’s Hospital Academic Medical Center, Vrije Universiteit Medical Center; Departments of Radiology and Clinical Epidemiology and Biostatistics, Academic Medical Center, Amsterdam, the Netherlands; Department of Pediatric Surgical Unit, Universitäts-Kinderklinik, Inselspital, Bern, Switzerland; Department of Pediatrics, Universita di Padova, Padova, Italy; Northern and Yorkshire Clinical Trial and Research Unit, University of Leeds, Leeds; Department of Surgery, Royal Hospital for Sick Children, Edinburgh, United Kingdom; Department of Pediatric Surgery and Liver Transplantation, Université Catholique de Louvain, Clinique Saint-Luc, Brussels, Belgium; and Department of Pediatric Surgery, Medical University of Gdansk, Gdansk, Poland

Address reprint requests to Daniël C. Aronson, MD, PhD, Pediatric Surgical Center of Amsterdam, Emma Children's Hospital AMC, PO Box 22700, 1100 DE Amsterdam, the Netherlands; e-mail: d.c.aronson{at}amc.uva.nl

PURPOSE: Preoperative staging (pretreatment extent of disease [PRETEXT]) was developed for the first prospective liver tumor study by the International Society of Pediatric Oncology (SIOPEL-1 study; preoperative chemotherapy and delayed surgery). Study aims were to analyze the accuracy and interobserver agreement of PRETEXT and to compare the predictive impact of three currently used staging systems.

PATIENTS AND METHODS: Hepatoblastoma (HB) patients younger than 16 years who underwent surgical resection (128 of 154 patients) were analyzed. The centrally reviewed preoperative staging was compared with postoperative pathology (accuracy) in 91 patients (81%), and the local center staging was compared with the central review (interobserver agreement) in 97 patients (86%), using the agreement beyond change method (weighted ). The predictive values of the three staging systems were compared in 110 patients (97%) using survival curves and Cox proportional hazard ratio estimates.

RESULTS: Preoperative PRETEXT staging compared with pathology was correct in 51%, overstaged in 37%, and understaged in 12% of patients (weighted = 0.44; 95% CI, 0.26 to 0.62). The weighted value of the interobserver agreement was 0.76 (95% CI, 0.64 to 0.88). The Children's Cancer Study Group/Pediatric Oncology Group–based staging system showed no predictive value for survival (P = .516), but the tumor-node-metastasis–based system and PRETEXT system showed good predictive values (P = .0021 and P = .0006, respectively). PRETEXT seemed to be superior in the statistical fit.

CONCLUSION: PRETEXT has moderate accuracy with a tendency to overstage patients, shows good interobserver agreement (reproducibility), shows superior predictive value for survival, offers the opportunity to monitor the effect of preoperative therapy, and can also be applied in patients who have not had operations. For comparability reasons, we recommend that all HB patients included in trials also be staged according to PRETEXT.

Supported by the Swiss Cancer League, the Bernese Cancer League, and the Liver Tumour Parents Group, United Kingdom.

Both D.C.A. and J.M.S. contributed equally to this article.

Authors' disclosures of potential conflicts of interest are found at the end of this article.

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• Predictive Value of Staging Systems in Hepatoblastoma
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  JCO 2007 25: 737 [Full Text]

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