Piotr Czauderna1, Beate Haeberle2, Eiso Hiyama3, Arun Rangaswami4, Mark Krailo5, Rudolf Maibach6, Eugenia Rinaldi7, Yurong Feng5, Daniel Aronson8, Marcio Malogolowkin9, Kenichi Yoshimura10, Ivo Leuschner11, Dolores Lopez-Terrada12, Tomoro Hishiki13, Giorgio Perilongo14, Dietrich von Schweinitz2, Irene Schmid15, Kenichiro Watanabe16, Marisa Derosa7, Rebecka Meyers17. 1. Department of Surgery and Urology for Children and Adolescents, Medical University of Gdansk, Poland. Electronic address: czauderna.p@gmail.com. 2. Department of Paediatric Surgery, University of Munich, Germany. 3. Department of Pediatric Surgery, Hiroshima University, Japan. 4. Department of Pediatric Hematology Oncology, Stanford University, USA. 5. Children's Oncology Group, USA. 6. IBCSG, Bern, Switzerland. 7. CINECA, Bologna, Italy. 8. Queen Elisabeth's Central Hospital University of Malawi, Blantyre, Malawi. 9. Medical College of Wisconsin, USA. 10. Innovative Clinical Research Center (iCREK), Kanazawa University Hospital, Japan. 11. Institute of Pathology, University of Kiel, Germany. 12. Department of Pathology & Immunology, Baylor College of Medicine, Houston, TX, USA. 13. Department of Pediatric Surgery, Chiba University Hospital, Japan. 14. Department of Pediatrics, University of Padova, Italy. 15. Department of Pediatric Oncology and Hematology, University of Munich, Germany. 16. Department of Pediatrics, Kyoto University, Japan. 17. Department of Surgery, University of Utah, Salt Lake City, USA.
Abstract
INTRODUCTION: Contemporary state-of-the-art management of cancer is increasingly defined by individualized treatment strategies. For very rare tumors, like hepatoblastoma, the development of biologic markers, and the identification of reliable prognostic risk factors for tailoring treatment, remains very challenging. The Children's Hepatic tumors International Collaboration (CHIC) is a novel international response to this challenge. METHODS: Four multicenter trial groups in the world, who have performed prospective controlled studies of hepatoblastoma over the past two decades (COG; SIOPEL; GPOH; and JPLT), joined forces to form the CHIC consortium. With the support of the data management group CINECA, CHIC developed a centralized online platform where data from eight completed hepatoblastoma trials were merged to form a database of 1605 hepatoblastoma cases treated between 1988 and 2008. The resulting dataset is described and the relationships between selected patient and tumor characteristics, and risk for adverse disease outcome (event-free survival; EFS) are examined. RESULTS: Significantly increased risk for EFS-event was noted for advanced PRETEXT group, macrovascular venous or portal involvement, contiguous extrahepatic disease, primary tumor multifocality and tumor rupture at enrollment. Higher age (≥ 8 years), low AFP (<100 ng/ml) and metastatic disease were associated with the worst outcome. CONCLUSION: We have identified novel prognostic factors for hepatoblastoma, as well as confirmed established factors, that will be used to develop a future common global risk stratification system. The mechanics of developing the globally accessible web-based portal, building and refining the database, and performing this first statistical analysis has laid the foundation for future collaborative efforts. This is an important step for refining of the risk based grouping and approach to future treatment stratification, thus we think our collaboration offers a template for others to follow in the study of rare tumors and diseases.
INTRODUCTION: Contemporary state-of-the-art management of cancer is increasingly defined by individualized treatment strategies. For very rare tumors, like hepatoblastoma, the development of biologic markers, and the identification of reliable prognostic risk factors for tailoring treatment, remains very challenging. The Children's Hepatic tumors International Collaboration (CHIC) is a novel international response to this challenge. METHODS: Four multicenter trial groups in the world, who have performed prospective controlled studies of hepatoblastoma over the past two decades (COG; SIOPEL; GPOH; and JPLT), joined forces to form the CHIC consortium. With the support of the data management group CINECA, CHIC developed a centralized online platform where data from eight completed hepatoblastoma trials were merged to form a database of 1605 hepatoblastoma cases treated between 1988 and 2008. The resulting dataset is described and the relationships between selected patient and tumor characteristics, and risk for adverse disease outcome (event-free survival; EFS) are examined. RESULTS: Significantly increased risk for EFS-event was noted for advanced PRETEXT group, macrovascular venous or portal involvement, contiguous extrahepatic disease, primary tumor multifocality and tumor rupture at enrollment. Higher age (≥ 8 years), low AFP (<100 ng/ml) and metastatic disease were associated with the worst outcome. CONCLUSION: We have identified novel prognostic factors for hepatoblastoma, as well as confirmed established factors, that will be used to develop a future common global risk stratification system. The mechanics of developing the globally accessible web-based portal, building and refining the database, and performing this first statistical analysis has laid the foundation for future collaborative efforts. This is an important step for refining of the risk based grouping and approach to future treatment stratification, thus we think our collaboration offers a template for others to follow in the study of rare tumors and diseases.
Authors: Daniël C Aronson; J Marco Schnater; Chris R Staalman; Gerrit J Weverling; Jack Plaschkes; Giorgio Perilongo; Julia Brown; Angela Phillips; Jean-Bernard Otte; Piotr Czauderna; Gordon MacKinlay; Anton Vos Journal: J Clin Oncol Date: 2005-02-20 Impact factor: 44.544
Authors: Andrea Ferrari; Gianni Bisogno; Gian Luca De Salvo; Paolo Indolfi; Giorgio Perilongo; Giovanni Cecchetto Journal: Eur J Cancer Date: 2006-10-16 Impact factor: 9.162
Authors: J A Ortega; E C Douglass; J H Feusner; M Reynolds; J J Quinn; M J Finegold; J E Haas; D R King; W Liu-Mares; M G Sensel; M D Krailo Journal: J Clin Oncol Date: 2000-07 Impact factor: 44.544
Authors: József Zsíros; Rudolf Maibach; Elizabeth Shafford; Laurence Brugieres; Penelope Brock; Piotr Czauderna; Derek Roebuck; Margaret Childs; Arthur Zimmermann; Veronique Laithier; Jean-Bernard Otte; Beatriz de Camargo; Gordon MacKinlay; Marcelo Scopinaro; Daniel Aronson; Jack Plaschkes; Giorgio Perilongo Journal: J Clin Oncol Date: 2010-04-20 Impact factor: 44.544
Authors: Giorgio Perilongo; Rudolf Maibach; Elisabeth Shafford; Laurence Brugieres; Penelope Brock; Bruce Morland; Beatriz de Camargo; Jozsef Zsiros; Derek Roebuck; Arthur Zimmermann; Daniel Aronson; Margaret Childs; Eva Widing; Veronique Laithier; Jack Plaschkes; Jon Pritchard; Marcello Scopinaro; Gordon MacKinlay; Piotr Czauderna Journal: N Engl J Med Date: 2009-10-22 Impact factor: 91.245
Authors: Howard M Katzenstein; Kay W Chang; Mark Krailo; Zhengjia Chen; Milton J Finegold; Jon Rowland; Marleta Reynolds; Alberto Pappo; Wendy B London; Marcio Malogolowkin Journal: Cancer Date: 2009-12-15 Impact factor: 6.860
Authors: Jörg Fuchs; Jana Rydzynski; Dietrich Von Schweinitz; Udo Bode; Hartmut Hecker; Peter Weinel; Dietrich Bürger; Dieter Harms; Rudolf Erttmann; Karl Oldhafer; Hermann Mildenberger Journal: Cancer Date: 2002-07-01 Impact factor: 6.860
Authors: Rebecka L Meyers; Jon R Rowland; Mark Krailo; Zhengjia Chen; Howard M Katzenstein; Marcio H Malogolowkin Journal: Pediatr Blood Cancer Date: 2009-12 Impact factor: 3.167
Authors: Alexander J Towbin; Rebecka L Meyers; Helen Woodley; Osamu Miyazaki; Christopher B Weldon; Bruce Morland; Eiso Hiyama; Piotr Czauderna; Derek J Roebuck; Greg M Tiao Journal: Pediatr Radiol Date: 2018-02-09
Authors: C Matthew Hawkins; Alexander J Towbin; Derek J Roebuck; Eric J Monroe; Anne E Gill; Avnesh S Thakor; Richard B Towbin; Anne Marie Cahill; Matthew P Lungren Journal: Pediatr Radiol Date: 2018-02-02