David O Walterhouse1, Donald A Barkauskas1, David Hall1, Andrea Ferrari1, Gian Luca De Salvo1, Ewa Koscielniak1, Michael C G Stevens1, Hélène Martelli1, Guido Seitz1, David A Rodeberg1, Margarett Shnorhavorian1, Roshni Dasgupta1, John C Breneman1, James R Anderson1, Christophe Bergeron1, Gianni Bisogno1, William H Meyer1, Douglas S Hawkins1, Veronique Minard-Colin1. 1. David O. Walterhouse, Ann & Robert H. Lurie Children's Hospital of Chicago, Chicago, IL; Donald A. Barkauskas, University of Southern California, Los Angeles; David Hall, Children's Oncology Group, Monrovia, CA; Andrea Ferrari, Fondazione Istituto di Ricovero e Cura a Carattere Scientifico Istituto Nazionale Tumori, Milan; Gian Luca De Salvo, Istituto Oncologico Veneto Istituto di Ricovero e Cura a Carattere Scientifico, Padua; Gianni Bisogno, University of Padua, Padova, Italy; Ewa Koscielniak, Olgahospital, Stuttgart; Guido Seitz, University Hospital, Marburg, Germany; Michael C.G. Stevens, Royal Hospital for Children, Bristol, United Kingdom; Hélène Martelli, Assistance Publique-Hôpitaux de Paris, Hopital Bicetre, Le Kremlin-Bicetre; Christophe Bergeron, Centre Léon Bérard, Lyon; Veronique Minard-Colin, Gustave Roussy, Villejuif, France; David A. Rodeberg, East Carolina University, Greenville, NC; Margarett Shnorhavorian and Douglas S. Hawkins, Seattle Children's Hospital, University of Washington; Douglas S. Hawkins, Fred Hutchinson Cancer Research Center, University of Washington, Seattle, WA; Roshni Dasgupta and John C. Breneman, Cincinnati Children's Hospital Medical Center, Cincinnati, OH; James R. Anderson, Merck Research Laboratories, North Wales, PA; and William H. Meyer, University of Oklahoma School of Medicine, Oklahoma City, OK.
Abstract
PURPOSE: Treatment recommendations for localized paratesticular rhabdomyosarcoma (PT RMS) differ in North America and Europe. We conducted a pooled analysis to identify demographic features and treatment choices that affect outcome. PATIENTS AND METHODS: We retrospectively analyzed the effect of nine demographic variables and four treatment choices on event-free survival (EFS) and overall survival (OS) from 12 studies conducted by five cooperative groups. RESULTS: Eight hundred forty-two patients with localized PT RMS who enrolled from 1988 to 2013 were included. Patients age ≥ 10 years were more likely than younger patients to have tumors that were > 5 cm, enlarged nodes (N1), or pathologically involved nodes ( P ≤ .05 each). With a median follow-up of 7.5 years, Kaplan-Meier estimates for 5-year EFS and OS were 87.7% and 94.8%, respectively. Of demographic variables, cooperative group, era of enrollment, age category, tumor size, Intergroup Rhabdomyosarcoma Study group, and T stage affected EFS ( P ≤ .05 each). Surgical assessment of regional nodes, which was performed in 23.5% of patients-usually in those age ≥ 10 years or with suspicious or N1 nodes-was the only treatment variable associated with EFS by univariable and multivariable analyses ( P ≤ .05 each) in patients age ≥ 1 year. A variable selection procedure on a proportional hazards regression model selected era of enrollment, age, tumor size, and surgical assessment of regional nodes as significant ( P ≤ .05 each) in the EFS model, and era of enrollment, age, tumor size, and histology ( P ≤ .05 each) in the OS model. CONCLUSION: Localized PT RMS has a favorable prognosis. Age ≥ 10 years at diagnosis and tumor size larger than 5 cm are unfavorable prognostic features. Surgical assessment of regional nodes is important in patients age ≥ 10 years and in those with N1 nodes as it affects EFS.
PURPOSE: Treatment recommendations for localized paratesticular rhabdomyosarcoma (PT RMS) differ in North America and Europe. We conducted a pooled analysis to identify demographic features and treatment choices that affect outcome. PATIENTS AND METHODS: We retrospectively analyzed the effect of nine demographic variables and four treatment choices on event-free survival (EFS) and overall survival (OS) from 12 studies conducted by five cooperative groups. RESULTS: Eight hundred forty-two patients with localized PT RMS who enrolled from 1988 to 2013 were included. Patients age ≥ 10 years were more likely than younger patients to have tumors that were > 5 cm, enlarged nodes (N1), or pathologically involved nodes ( P ≤ .05 each). With a median follow-up of 7.5 years, Kaplan-Meier estimates for 5-year EFS and OS were 87.7% and 94.8%, respectively. Of demographic variables, cooperative group, era of enrollment, age category, tumor size, Intergroup Rhabdomyosarcoma Study group, and T stage affected EFS ( P ≤ .05 each). Surgical assessment of regional nodes, which was performed in 23.5% of patients-usually in those age ≥ 10 years or with suspicious or N1 nodes-was the only treatment variable associated with EFS by univariable and multivariable analyses ( P ≤ .05 each) in patients age ≥ 1 year. A variable selection procedure on a proportional hazards regression model selected era of enrollment, age, tumor size, and surgical assessment of regional nodes as significant ( P ≤ .05 each) in the EFS model, and era of enrollment, age, tumor size, and histology ( P ≤ .05 each) in the OS model. CONCLUSION: Localized PT RMS has a favorable prognosis. Age ≥ 10 years at diagnosis and tumor size larger than 5 cm are unfavorable prognostic features. Surgical assessment of regional nodes is important in patients age ≥ 10 years and in those with N1 nodes as it affects EFS.
Authors: Odile Oberlin; Annie Rey; José Sanchez de Toledo; Hélène Martelli; Meriel E M Jenney; Marcelo Scopinaro; Christophe Bergeron; Johannes H M Merks; Nathalie Bouvet; Caroline Ellershaw; Anna Kelsey; David Spooner; Michael C G Stevens Journal: J Clin Oncol Date: 2012-06-04 Impact factor: 44.544
Authors: Michael C G Stevens; Annie Rey; Nathalie Bouvet; Caroline Ellershaw; Françoise Flamant; Jean Louis Habrand; H Basil Marsden; Helene Martelli; Jose Sanchez de Toledo; Richard D Spicer; David Spooner; Marie Jose Terrier-Lacombe; Adrian van Unnik; Odile Oberlin Journal: J Clin Oncol Date: 2005-02-22 Impact factor: 44.544
Authors: R Beverly Raney; David O Walterhouse; Jane L Meza; Richard J Andrassy; John C Breneman; William M Crist; Harold M Maurer; William H Meyer; David M Parham; James R Anderson Journal: J Clin Oncol Date: 2011-02-28 Impact factor: 44.544
Authors: Timothy Rogers; Veronique Minard-Colin; Nathalie Cozic; Meriel Jenney; Johannes H M Merks; Soledad Gallego; Christine Devalck; Mark N Gaze; Anna Kelsey; Odile Oberlin; Mike Stevens; Richard D Spicer; Christophe Bergeron; Helene Martelli Journal: Pediatr Blood Cancer Date: 2017-02-16 Impact factor: 3.167
Authors: E S Wiener; W Lawrence; D Hays; T E Lobe; R Andrassy; S Donaldson; W Crist; W Newton; J Johnson; E Gehan Journal: J Pediatr Surg Date: 1994-02 Impact factor: 2.545
Authors: Guido Seitz; Jörg Fuchs; Peter Martus; Thomas Klingebiel; Ivo Leuschner; Andreas Schuck; Tobias M Dantonello; Ewa Koscielniak Journal: Ann Surg Date: 2016-12 Impact factor: 12.969
Authors: Andrea Ferrari; G Bisogno; M Casanova; I B Brecht; R Alaggio; G Cecchetto; M Provenzi; E Koscielniak; J Treuner; M Carli Journal: Pediatr Blood Cancer Date: 2004-02 Impact factor: 3.167
Authors: Jacquelyn N Crane; Wei Xue; Amira Qumseya; Zhengya Gao; Carola A S Arndt; Sarah S Donaldson; Douglas J Harrison; Douglas S Hawkins; Corinne M Linardic; Leo Mascarenhas; William H Meyer; David A Rodeberg; Erin R Rudzinski; Barry L Shulkin; David O Walterhouse; Rajkumar Venkatramani; Aaron R Weiss Journal: Pediatr Blood Cancer Date: 2022-03-06 Impact factor: 3.838
Authors: Timothy N Rogers; Guido Seitz; Jörg Fuchs; Helene Martelli; Roshni Dasgupta; Jonathan C Routh; Douglas S Hawkins; Ewa Koscielniak; Gianni Bisogno; David A Rodeberg Journal: Pediatr Blood Cancer Date: 2021-02-01 Impact factor: 3.838
Authors: Jonathan C Routh; Roshni Dasgupta; Yueh-Yun Chi; Margarett Shnorhavorian; Jing Tian; David O Walterhouse; John Breneman; Suzanne L Wolden; Carola A Arndt; Douglas S Hawkins; David A Rodeberg Journal: Int J Cancer Date: 2020-07-11 Impact factor: 7.396