Najat C Daw1, Yueh-Yun Chi2, Yeonil Kim3, Elizabeth A Mullen4, John A Kalapurakal5, Jing Tian6, Geetika Khanna7, James I Geller8, Elizabeth J Perlman9, Peter F Ehrlich10, Anne B Warwick11, Paul E Grundy12, Conrad V Fernandez13, Jeffrey S Dome14. 1. Division of Pediatrics, The University of Texas MD Anderson Cancer Center, 1515 Holcombe Blvd Unit 87, Houston, TX 77030-4009, USA. Electronic address: ndaw@mdanderson.org. 2. Department of Biostatistics, University of Florida, 6011 NW 1st Place Suite 10, Gainesville, FL 32607, USA. Electronic address: ychi@cog.ufl.edu. 3. Department of Biostatistics, University of Florida, 6011 NW 1st Place Suite 10, Gainesville, FL 32607, USA. 4. Department of Pediatric Hematology/Oncology, Dana-Farber/Harvard Cancer Center, Dana Farber Cancer Institute, 450 Brookline Ave. Office#: SW360A, Boston, MA 02215-5450, USA. Electronic address: elizabeth_mullen@dfci.harvard.edu. 5. Department of Radiation Oncology, Ann and Robert H. Lurie Children's Hospital of Chicago, Northwestern Memorial Hospital, 25i East Huron Street Suite#L178, Chicago, IL 60611, USA. Electronic address: j-kalapurakal@northwestern.edu. 6. Children's Oncology Group Data Center, Statistics Data Center, 6011 NW 1st Place Suite 10, Gainesville, FL 32607, USA. Electronic address: jtian@cog.ufl.edu. 7. Mallinckrodt Institute of Radiology, Washington University School of Medicine, 510 S. Kingshighway, Box 8131, St. Louis, MO 63110, USA. Electronic address: khannag@wustl.edu. 8. Division of Oncology, Cincinnati Children's Hospital Medical Center, University of Cincinnati, 3333 Burnet Avenue MLC 7015, Cincinnati, OH 45229, USA. Electronic address: james.geller@cchmc.org. 9. Department of Pathology, Ann and Robert H. Lurie Children's Hospital of Chicago, Northwestern University Feinberg School of Medicine, 225 East Chicago Avenue, Chicago, IL 60611, USA. Electronic address: eperlman@luriechildrens.org. 10. Department of Pediatric Surgery, University of Michigan, 4-945 C.S. Mott Children's Hospital SPC 4211, 1500 E. Medical Center Drive, Ann Arbor, MI 48109-4211, USA. Electronic address: pehrlich@med.umich.edu. 11. Department of Pediatrics, F. Edward Hébert School of Medicine, Uniformed Services University, 4301 Jones Bridge Road, Bethesda, MD 20814, USA. Electronic address: anne.warwick@usuhs.edu. 12. Department of Pediatrics, University of Alberta Hospital, WMC 4E2.28, 8440-112 Street, Edmonton, Alberta T6G 2B7, Canada. Electronic address: paul.e.grundy@ahs.ca. 13. Departments of Pediatrics and Bioethics, IWK Health Centre, Dalhousie University and the IWK Health Centre, 5850 University Avenue, Halifax, Nova Scotia B3K 6R8, Canada. Electronic address: conrad.fernandez@iwk.nshealth.ca. 14. Division of Oncology, Children's National Medical Center, Center for Cancer and Blood Disorders, George Washington University School of Medicine and Health Sciences, 111 Michigan Avenue NW, Washington, DC 20010, USA. Electronic address: jdome@childrensnational.org.
Abstract
BACKGROUND: In the fifth National Wilms Tumor Study (NWTS-5), the 4-year event-free survival (EFS) and overall survival (OS) estimates for 29 patients with stage I focal (n = 10) or diffuse (n = 19) anaplastic Wilms' tumour (AWT) treated with vincristine and dactinomycin without flank radiation were 69.5% and 82.6%, respectively. The Children's Oncology Group AREN0321 study evaluated whether adding doxorubicin and flank radiation improves survival for these patients. PATIENTS AND METHODS: Tumour histology and stage were confirmed by real-time central pathology, surgery and radiology review. The patients received 25 weeks of vincristine, dactinomycin and doxorubicin (cumulative dose 150 mg/m2) with flank radiation (1080 cGy). We retrospectively analysed outcomes of all patients with stage I AWT enrolled in NWTSs 1-5 and AREN0321 with respect to treatment regimens. RESULTS: Eighteen patients with stage I AWT (8 focal and 10 diffuse) were enrolled on AREN0321. With a median follow-up of 4.6 years, the 4-year EFS and OS were 100%. One patient with diffuse AWT had pulmonary relapse 4.12 years after diagnosis. In the 112 patients with stage I AWT treated in NWTSs 1-5 and AREN0321, the EFS was significantly improved with doxorubicin treatment (p = 0.01; 4-year EFS: 97.2% [95% confidence interval {CI}: 91.3-100] vs. 77.5% [95% CI: 67.6-87.4]) but not by flank radiation (p = 0.15). CONCLUSIONS: Treatment of stage I AWT with vincristine, dactinomycin, doxorubicin and flank radiation in AREN0321 yielded excellent survival outcomes. Retrospective analysis of AREN0321 and NWTS patients suggests that doxorubicin had a greater contribution to the excellent outcomes than radiation.
BACKGROUND: In the fifth National Wilms Tumor Study (NWTS-5), the 4-year event-free survival (EFS) and overall survival (OS) estimates for 29 patients with stage I focal (n = 10) or diffuse (n = 19) anaplastic Wilms' tumour (AWT) treated with vincristine and dactinomycin without flank radiation were 69.5% and 82.6%, respectively. The Children's Oncology Group AREN0321 study evaluated whether adding doxorubicin and flank radiation improves survival for these patients. PATIENTS AND METHODS: Tumour histology and stage were confirmed by real-time central pathology, surgery and radiology review. The patients received 25 weeks of vincristine, dactinomycin and doxorubicin (cumulative dose 150 mg/m2) with flank radiation (1080 cGy). We retrospectively analysed outcomes of all patients with stage I AWT enrolled in NWTSs 1-5 and AREN0321 with respect to treatment regimens. RESULTS: Eighteen patients with stage I AWT (8 focal and 10 diffuse) were enrolled on AREN0321. With a median follow-up of 4.6 years, the 4-year EFS and OS were 100%. One patient with diffuse AWT had pulmonary relapse 4.12 years after diagnosis. In the 112 patients with stage I AWT treated in NWTSs 1-5 and AREN0321, the EFS was significantly improved with doxorubicin treatment (p = 0.01; 4-year EFS: 97.2% [95% confidence interval {CI}: 91.3-100] vs. 77.5% [95% CI: 67.6-87.4]) but not by flank radiation (p = 0.15). CONCLUSIONS: Treatment of stage I AWT with vincristine, dactinomycin, doxorubicin and flank radiation in AREN0321 yielded excellent survival outcomes. Retrospective analysis of AREN0321 and NWTS patients suggests that doxorubicin had a greater contribution to the excellent outcomes than radiation.
Authors: John A Kalapurakal; Jeffrey S Dome; Elizabeth J Perlman; Marcio Malogolowkin; Gerald M Haase; Paul Grundy; Max J Coppes Journal: Lancet Oncol Date: 2004-01 Impact factor: 41.316
Authors: Ariadne H A G Ooms; Samantha Gadd; Daniela S Gerhard; Malcolm A Smith; Jaime M Guidry Auvil; Daoud Meerzaman; Qing-Rong Chen; Chih Hao Hsu; Chunhua Yan; Cu Nguyen; Ying Hu; Yussanne Ma; Zusheng Zong; Andrew J Mungall; Richard A Moore; Marco A Marra; Vicki Huff; Jeffrey S Dome; Yueh-Yun Chi; Jing Tian; James I Geller; Charles G Mullighan; Jing Ma; David A Wheeler; Oliver A Hampton; Amy L Walz; Marry M van den Heuvel-Eibrink; Ronald R de Krijger; Nicole Ross; Julie M Gastier-Foster; Elizabeth J Perlman Journal: Clin Cancer Res Date: 2016-10-04 Impact factor: 12.531
Authors: Jeffrey S Dome; Cecilia A Cotton; Elizabeth J Perlman; Norman E Breslow; John A Kalapurakal; Michael L Ritchey; Paul E Grundy; Marcio Malogolowkin; J Bruce Beckwith; Robert C Shamberger; Gerald M Haase; Max J Coppes; Peter Coccia; Morris Kletzel; Robert M Weetman; Milton Donaldson; Roger M Macklis; Daniel M Green Journal: J Clin Oncol Date: 2006-05-20 Impact factor: 44.544
Authors: D Ashley Hill; Torin D Shear; Tiebin Liu; Catherine A Billups; Pratima K Singh; Jeffrey S Dome Journal: Cancer Date: 2003-05-01 Impact factor: 6.860
Authors: G J D'Angio; N Breslow; J B Beckwith; A Evans; H Baum; A deLorimier; D Fernbach; E Hrabovsky; B Jones; P Kelalis Journal: Cancer Date: 1989-07-15 Impact factor: 6.860
Authors: John A Kalapurakal; Sierra M Li; Norman E Breslow; J Bruce Beckwith; Roger Macklis; Patrick R M Thomas; Guilio J D'Angio; Tae Kim; Alfred de Lorimier; Panayotis Kelalis; Steven Shochat; Michael Ritchey; Gerald Haase; Ellen Hrabovsky; H Biemann Otherson; Paul Grundy; Daniel M Green Journal: Int J Radiat Oncol Biol Phys Date: 2003-10-01 Impact factor: 7.038
Authors: Mariana Maschietto; Richard D Williams; Tasnim Chagtai; Sergey D Popov; Neil J Sebire; Gordan Vujanic; Elizabeth Perlman; James R Anderson; Paul Grundy; Jeffrey S Dome; Kathy Pritchard-Jones Journal: PLoS One Date: 2014-10-14 Impact factor: 3.240
Authors: Jeffrey S Dome; Elizabeth A Mullen; David B Dix; Eric J Gratias; Peter F Ehrlich; Najat C Daw; James I Geller; Murali Chintagumpala; Geetika Khanna; John A Kalapurakal; Lindsay A Renfro; Elizabeth J Perlman; Paul E Grundy; Conrad V Fernandez Journal: J Natl Compr Canc Netw Date: 2021-08-01 Impact factor: 11.908
Authors: M E Madeleine van der Perk; Nicholas G Cost; Annelies M E Bos; Robert Brannigan; Tanzina Chowdhury; Andrew M Davidoff; Najat C Daw; Jeffrey S Dome; Peter Ehrlich; Norbert Graf; James Geller; John Kalapurakal; Kathleen Kieran; Marcus Malek; Mary F McAleer; Elizabeth Mullen; Luke Pater; Angela Polanco; Rodrigo Romao; Amanda F Saltzman; Amy L Walz; Andrew D Woods; Marry M van den Heuvel-Eibrink; Conrad V Fernandez Journal: Int J Cancer Date: 2022-05-11 Impact factor: 7.316