Eric J Gratias1, Jeffrey S Dome1, Lawrence J Jennings1, Yueh-Yun Chi1, Jing Tian1, James Anderson1, Paul Grundy1, Elizabeth A Mullen1, James I Geller1, Conrad V Fernandez1, Elizabeth J Perlman2. 1. Eric J. Gratias, University of Tennessee College of Medicine-Chattanooga, Chattanooga, TN; Jeffrey S. Dome, Children's National Health System, Washington, DC; Lawrence J. Jennings and Elizabeth J. Perlman, Northwestern University, Chicago, IL; Yueh-Yun Chi and Jing Tian, University of Florida, Gainesville, FL; James Anderson, University of Nebraska Medical Center, Omaha, NE; Paul Grundy, University of Alberta, Edmonton, Alberta; Conrad V. Fernandez, Dalhousie University and the IWK Health Centre, Halifax, Nova Scotia, Canada; Elizabeth A. Mullen, Boston Children's Hospital and Dana-Farber Cancer Institute, Boston, MA; and James I. Geller, University of Cincinnati, Cincinnati, OH. 2. Eric J. Gratias, University of Tennessee College of Medicine-Chattanooga, Chattanooga, TN; Jeffrey S. Dome, Children's National Health System, Washington, DC; Lawrence J. Jennings and Elizabeth J. Perlman, Northwestern University, Chicago, IL; Yueh-Yun Chi and Jing Tian, University of Florida, Gainesville, FL; James Anderson, University of Nebraska Medical Center, Omaha, NE; Paul Grundy, University of Alberta, Edmonton, Alberta; Conrad V. Fernandez, Dalhousie University and the IWK Health Centre, Halifax, Nova Scotia, Canada; Elizabeth A. Mullen, Boston Children's Hospital and Dana-Farber Cancer Institute, Boston, MA; and James I. Geller, University of Cincinnati, Cincinnati, OH. eperlman@luriechildrens.org.
Abstract
PURPOSE: The goal of this study was to analyze the association of copy number gain of 1q in favorable-histology Wilms tumors (FHWTs) with event-free survival (EFS) and overall survival (OS) within each tumor stage and with 1p and 16q copy number loss and/or loss of heterozygosity. METHODS: Unilateral FHWTs from 1,114 patients enrolled in National Wilms Tumor Study-5 that were informative for 1p and 16q microsatellite markers (previously determined) and informative for 1q gain, 1p loss, and 16q loss using multiplex ligation-dependent probe amplification were analyzed. RESULTS: Eight-year EFS was 86% (95% CI, 84% to 88%) for the entire cohort. Of 1,114 patients, 317 tumors (28%) displayed 1q gain. Eight-year EFS was 77% for those with 1q gain and 90% for those lacking 1q gain (P < .001). Eight-year OS was 88% for those with 1q gain and 96% for those lacking 1q gain (P < .001). Within each disease stage, 1q gain was associated with inferior EFS (stage I, 85% v 95%; P = .0052; stage II, 81% v 87%; P = .0775; stage III, 79% v 89%; P = .01; stage IV, 64% v 91%; P = .001). OS was significantly inferior in patients with stage I (P < .0015) and stage IV disease (P = .011). With multivariable analysis, 1q gain was associated with an increased relative risk of relapse of 2.4 (P < .001), whereas 1p loss was not, despite significance on univariable analysis. CONCLUSION: Gain of 1q is associated with inferior survival in unilateral FHWTs and may be used to guide risk stratification in future studies.
PURPOSE: The goal of this study was to analyze the association of copy number gain of 1q in favorable-histology Wilms tumors (FHWTs) with event-free survival (EFS) and overall survival (OS) within each tumor stage and with 1p and 16q copy number loss and/or loss of heterozygosity. METHODS: Unilateral FHWTs from 1,114 patients enrolled in National Wilms Tumor Study-5 that were informative for 1p and 16q microsatellite markers (previously determined) and informative for 1q gain, 1p loss, and 16q loss using multiplex ligation-dependent probe amplification were analyzed. RESULTS: Eight-year EFS was 86% (95% CI, 84% to 88%) for the entire cohort. Of 1,114 patients, 317 tumors (28%) displayed 1q gain. Eight-year EFS was 77% for those with 1q gain and 90% for those lacking 1q gain (P < .001). Eight-year OS was 88% for those with 1q gain and 96% for those lacking 1q gain (P < .001). Within each disease stage, 1q gain was associated with inferior EFS (stage I, 85% v 95%; P = .0052; stage II, 81% v 87%; P = .0775; stage III, 79% v 89%; P = .01; stage IV, 64% v 91%; P = .001). OS was significantly inferior in patients with stage I (P < .0015) and stage IV disease (P = .011). With multivariable analysis, 1q gain was associated with an increased relative risk of relapse of 2.4 (P < .001), whereas 1p loss was not, despite significance on univariable analysis. CONCLUSION: Gain of 1q is associated with inferior survival in unilateral FHWTs and may be used to guide risk stratification in future studies.
Authors: Conrad V Fernandez; Elizabeth J Perlman; Elizabeth A Mullen; Yueh-Yun Chi; Thomas E Hamilton; Kenneth W Gow; Fernando A Ferrer; Douglas C Barnhart; Peter F Ehrlich; Geetika Khanna; John A Kalapurakal; Tina Bocking; Vicky Huff; Jing Tian; James I Geller; Paul E Grundy; James R Anderson; Jeffrey S Dome; Robert C Shamberger Journal: Ann Surg Date: 2017-04 Impact factor: 12.969
Authors: S Hing; Y J Lu; B Summersgill; L King-Underwood; J Nicholson; P Grundy; R Grundy; M Gessler; J Shipley; K Pritchard-Jones Journal: Am J Pathol Date: 2001-02 Impact factor: 4.307
Authors: C Mackintosh; J L Ordóñez; D J García-Domínguez; V Sevillano; A Llombart-Bosch; K Szuhai; K Scotlandi; M Alberghini; R Sciot; F Sinnaeve; P C W Hogendoorn; P Picci; S Knuutila; U Dirksen; M Debiec-Rychter; K-L Schaefer; E de Álava Journal: Oncogene Date: 2011-08-08 Impact factor: 9.867
Authors: John-Paul Kilday; Biswaroop Mitra; Caroline Domerg; Jennifer Ward; Felipe Andreiuolo; Teresa Osteso-Ibanez; Audrey Mauguen; Pascale Varlet; Marie-Cecile Le Deley; James Lowe; David W Ellison; Richard J Gilbertson; Beth Coyle; Jacques Grill; Richard G Grundy Journal: Clin Cancer Res Date: 2012-02-14 Impact factor: 12.531
Authors: Richard D Williams; Sandra N Hing; Braden T Greer; Craig C Whiteford; Jun S Wei; Rachael Natrajan; Anna Kelsey; Simon Rogers; Colin Campbell; Kathy Pritchard-Jones; Javed Khan Journal: Genes Chromosomes Cancer Date: 2004-09 Impact factor: 5.006
Authors: Chiang-Ching Huang; Samantha Gadd; Norman Breslow; Colleen Cutcliffe; Simone T Sredni; Irene B Helenowski; Jeffrey S Dome; Paul E Grundy; Daniel M Green; Michael K Fritsch; Elizabeth J Perlman Journal: Clin Cancer Res Date: 2009-02-10 Impact factor: 12.531
Authors: Ken C Lo; Changxing Ma; Brian N Bundy; Scott L Pomeroy; Charles G Eberhart; John K Cowell Journal: Clin Cancer Res Date: 2007-12-01 Impact factor: 12.531
Authors: R Natrajan; S E Little; N Sodha; J S Reis-Filho; A Mackay; K Fenwick; A Ashworth; E J Perlman; J S Dome; P E Grundy; K Pritchard-Jones; C Jones Journal: J Pathol Date: 2007-01 Impact factor: 7.996
Authors: David B Dix; Nita L Seibel; Yueh-Yun Chi; Geetika Khanna; Eric Gratias; James R Anderson; Elizabeth A Mullen; James I Geller; John A Kalapurakal; Arnold C Paulino; Elizabeth J Perlman; Peter F Ehrlich; Marcio Malogolowkin; Julie M Gastier-Foster; Elizabeth Wagner; Paul E Grundy; Conrad V Fernandez; Jeffrey S Dome Journal: J Clin Oncol Date: 2018-04-16 Impact factor: 44.544
Authors: Kelly Klega; Alma Imamovic-Tuco; Gavin Ha; Andrea N Clapp; Stephanie Meyer; Abigail Ward; Catherine Clinton; Anwesha Nag; Eliezer Van Allen; Elizabeth Mullen; Steven G DuBois; Katherine Janeway; Matthew Meyerson; Aaron R Thorner; Brian D Crompton Journal: JCO Precis Oncol Date: 2018-07-05
Authors: David B Dix; Conrad V Fernandez; Yueh-Yun Chi; Elizabeth A Mullen; James I Geller; Eric J Gratias; Geetika Khanna; John A Kalapurakal; Elizabeth J Perlman; Nita L Seibel; Peter F Ehrlich; Marcio Malogolowkin; James Anderson; Julie Gastier-Foster; Robert C Shamberger; Yeonil Kim; Paul E Grundy; Jeffrey S Dome Journal: J Clin Oncol Date: 2019-08-26 Impact factor: 44.544
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: Samantha Gadd; Vicki Huff; Andrew D Skol; Lindsay A Renfro; Conrad V Fernandez; Elizabeth A Mullen; Corbin D Jones; Katherine A Hoadley; Kai Lee Yap; Nilsa C Ramirez; Sheena Aris; Quy H Phung; Elizabeth J Perlman Journal: Cell Rep Med Date: 2022-05-25
Authors: Conrad V Fernandez; Elizabeth A Mullen; Yueh-Yun Chi; Peter F Ehrlich; Elizabeth J Perlman; John A Kalapurakal; Geetika Khanna; Arnold C Paulino; Thomas E Hamilton; Kenneth W Gow; Zelig Tochner; Fredric A Hoffer; Janice S Withycombe; Robert C Shamberger; Yeonil Kim; James I Geller; James R Anderson; Paul E Grundy; Jeffrey S Dome Journal: J Clin Oncol Date: 2017-12-06 Impact factor: 44.544
Authors: Ryan Thibodeau; Abtin Jafroodifar; Marlon Coelho; Hsin Kwung Li; Lorenzo Gitto; Daniel J Zaccarini; Mary McGrath Journal: Radiol Case Rep Date: 2021-06-08