Jean-François Brasme1, Martin Chalumeau2, Odile Oberlin2, Dominique Valteau-Couanet2, Nathalie Gaspar2. 1. Jean-François Brasme, Martin Chalumeau, INSERM U953; Jean-François Brasme, Martin Chalumeau, Hôpital Necker-Enfants Malades, Assistance Publique-Hôpitaux de Paris; Jean-François Brasme, Martin Chalumeau, Université Paris Descartes, Paris; Jean-François Brasme, Odile Oberlin, Dominique Valteau-Couanet, Nathalie Gaspar, Institut Gustave Roussy, Villejuif; Jean-François Brasme, Odile Oberlin, Dominique Valteau-Couanet, Nathalie Gaspar, Université Paris Sud, Le Kremlin Bicêtre, France. jean-francois.brasme@inserm.fr. 2. Jean-François Brasme, Martin Chalumeau, INSERM U953; Jean-François Brasme, Martin Chalumeau, Hôpital Necker-Enfants Malades, Assistance Publique-Hôpitaux de Paris; Jean-François Brasme, Martin Chalumeau, Université Paris Descartes, Paris; Jean-François Brasme, Odile Oberlin, Dominique Valteau-Couanet, Nathalie Gaspar, Institut Gustave Roussy, Villejuif; Jean-François Brasme, Odile Oberlin, Dominique Valteau-Couanet, Nathalie Gaspar, Université Paris Sud, Le Kremlin Bicêtre, France.
Abstract
PURPOSE: The time to diagnosis (TtD) of Ewing tumors is one of the longest among pediatric tumors. Its precise consequences, however, have not been studied well. We analyzed the distribution of TtD for Ewing tumors in children and adolescents and its association with clinical features, tumor characteristics, surgical outcome, and long-term survival. PATIENTS AND METHODS: We analyzed prospectively collected data from two multicenter clinical trials of patients younger than 21 years old who had Ewing bone tumors treated in France between 1988 and 2000. Clinical and tumoral features, TtD, and outcome associations were studied by univariable and multivariable analyses. RESULTS: The median TtD for the 436 patients was 70 days (interquartile range, 27 to 146 days), with no significant decrease during the study period (P > .2). The factors associated with long TtD were older age and some tumor sites (pelvis, extremities of limbs). Increased tumor volume and decreased histologic response to chemotherapy were associated with long TtD on univariable analysis (P < .05) but not after adjustment. Presence of a nerve or spinal-cord compression at diagnosis, presence or site of metastasis, surgical treatment, mutilating surgery, complete resection, or survival were not associated with TtD. CONCLUSION: TtD of Ewing tumors was long, especially for adolescents and for certain tumor sites, and did not improve over time. But TtD was not associated with metastasis, surgical outcome, or survival. These findings could be used to comfort parents at diagnosis and in expert testimony produced for malpractice claims.
PURPOSE: The time to diagnosis (TtD) of Ewing tumors is one of the longest among pediatric tumors. Its precise consequences, however, have not been studied well. We analyzed the distribution of TtD for Ewing tumors in children and adolescents and its association with clinical features, tumor characteristics, surgical outcome, and long-term survival. PATIENTS AND METHODS: We analyzed prospectively collected data from two multicenter clinical trials of patients younger than 21 years old who had Ewing bone tumors treated in France between 1988 and 2000. Clinical and tumoral features, TtD, and outcome associations were studied by univariable and multivariable analyses. RESULTS: The median TtD for the 436 patients was 70 days (interquartile range, 27 to 146 days), with no significant decrease during the study period (P > .2). The factors associated with long TtD were older age and some tumor sites (pelvis, extremities of limbs). Increased tumor volume and decreased histologic response to chemotherapy were associated with long TtD on univariable analysis (P < .05) but not after adjustment. Presence of a nerve or spinal-cord compression at diagnosis, presence or site of metastasis, surgical treatment, mutilating surgery, complete resection, or survival were not associated with TtD. CONCLUSION: TtD of Ewing tumors was long, especially for adolescents and for certain tumor sites, and did not improve over time. But TtD was not associated with metastasis, surgical outcome, or survival. These findings could be used to comfort parents at diagnosis and in expert testimony produced for malpractice claims.
Authors: Nathaniel D Anderson; Richard de Borja; Matthew D Young; Fabio Fuligni; Andrej Rosic; Nicola D Roberts; Simon Hajjar; Mehdi Layeghifard; Ana Novokmet; Paul E Kowalski; Matthew Anaka; Scott Davidson; Mehdi Zarrei; Badr Id Said; L Christine Schreiner; Remi Marchand; Joseph Sitter; Nalan Gokgoz; Ledia Brunga; Garrett T Graham; Anthony Fullam; Nischalan Pillay; Jeffrey A Toretsky; Akihiko Yoshida; Tatsuhiro Shibata; Markus Metzler; Gino R Somers; Stephen W Scherer; Adrienne M Flanagan; Peter J Campbell; Joshua D Schiffman; Mary Shago; Ludmil B Alexandrov; Jay S Wunder; Irene L Andrulis; David Malkin; Sam Behjati; Adam Shlien Journal: Science Date: 2018-08-31 Impact factor: 47.728
Authors: R D Neal; P Tharmanathan; B France; N U Din; S Cotton; J Fallon-Ferguson; W Hamilton; A Hendry; M Hendry; R Lewis; U Macleod; E D Mitchell; M Pickett; T Rai; K Shaw; N Stuart; M L Tørring; C Wilkinson; B Williams; N Williams; J Emery Journal: Br J Cancer Date: 2015-03-31 Impact factor: 7.640
Authors: Song Lee Jin; Seung Min Hahn; Hyo Sun Kim; Yoon Jung Shin; Sun Hee Kim; Yoon Sun Lee; Chuhl Joo Lyu; Jung Woo Han Journal: Yonsei Med J Date: 2016-05 Impact factor: 2.759
Authors: Elise Launay; Jérémie F Cohen; Patrick M Bossuyt; Pierre Buekens; Jonathan Deeks; Timothy Dye; Richard Feltbower; Andrea Ferrari; Michael Kramer; Mariska Leeflang; David Moher; Karel G Moons; Erik von Elm; Philippe Ravaud; Martin Chalumeau Journal: BMC Med Date: 2016-09-27 Impact factor: 8.775