Janine Piscione1, Wendy Barden, Janie Barry, Alexandra Malkin, Trisha Roy, Tyki Sueyoshi, Karen Mazil, Steven Salomon, Firas Dandachli, Anthony Griffin, Hugo Saint-Yves, Pina Giuliano, Abha Gupta, Peter Ferguson, Katrin Scheinemann, Michelle Ghert, Robert E Turcotte, Lucie Lafay-Cousin, Joel Werier, Caron Strahlendorf, Marc Isler, Sophie Mottard, Samina Afzal, Megan E Anderson, Sevan Hopyan. 1. J. Piscione, W. Barden, Rehabilitation Department, Hospital for Sick Children, Toronto, Canada J. Barry, H. Saint-Yves, M. Isler, S. Mottard, Service d'orthopédie, Hôpital Maisonneuve-Rosemont et Université de Montréal, Montréal, Canada A. Malkin, T. Roy, S. Hopyan, Division of Orthopaedic Surgery, Hospital for Sick Children, Toronto, Canada T. Sueyoshi, C. Strahlendorf, Division of Hematology and Oncology, BC Children's Hospital and University of British Columbia, Vancouver, Canada K. Mazil, P. Giuliano, L. Lafay-Cousin, Department of Oncology, Alberta Children's Hospital and University of Calgary, Calgary, Canada S. Salomon, F. Dandachli, R. E. Turcotte, Divisions of Orthopaedic Surgery and Surgical Oncology, McGill University Health Centre and McGill University, Montréal, Canada A. Griffin, P. Ferguson, Division of Orthopaedic Surgery, Mt Sinai Hospital and University of Toronto, Toronto, Canada A. Gupta, Division of Haematology/Oncology, Hospital for Sick Children, Toronto, Canada K. Scheinemann, Division of Hematology/Oncology, McMaster University Health Sciences Centre, Hamilton, Canada M. Ghert, Division of Orthopaedic Surgery, Juravinski Cancer Centre and McMaster University, Hamilton, Canada J. Werier, Division of Paediatric Orthopaedic Surgery, Children's Hospital of Eastern Ontario and University of Ottawa, Ottawa, Canada S. Afzal, Division of Hematology/Oncology, IWK Health Centre and Dalhousie University, Halifax, Canada M. E. Anderson, Orthopedic Center, Boston Children's Hospital, Jimmy Fund Clinic/Dana Farber Cancer Institute and Harvard Medical School, Boston, MA, USA S. Hopyan, Program in Developmental and Stem Cell Biology, Hospital for Sick Children, Departments of Surgery and Molecular Genetics, University of Toronto, Toronto, Canada.
Abstract
BACKGROUND: The physical function of children with sarcoma after surgery has not been studied explicitly. This paucity of research is partly because of the lack of a sufficiently sensitive pediatric functional measure. The goal of this study was to establish and validate a standardized measure of physical function in pediatric patients with extremity tumors. QUESTIONS/PURPOSES: (1) What is the best format and content for new upper- and lower-extremity measures of physical function in the pediatric population? (2) Do the new measures exhibit floor and/or ceiling effects, internal consistency, and test-retest reliability? (3) Are the new measures valid? METHODS: In Phase 1, interviews with 17 consecutive children and adolescents with bone tumors were conducted to modify the format and content of draft versions of the pediatric Toronto Extremity Salvage Score (pTESS). In Phase 2, the pTESS was formally translated into French. In Phase 3, 122 participants between 7 and 17.9 years old with malignant or benign-aggressive bone tumors completed the limb-specific measure on two occasions. Older adolescents also completed the adult TESS. Floor and ceiling effects, internal consistency, test-retest reliability, and validity were evaluated. RESULTS: Feedback from interviews resulted in the removal, addition, and modification of draft items, and the pTESS-Leg and pTESS-Arm questionnaires were finalized. Both versions exhibited no floor or ceiling effects and high internal consistency (α > 0.92). The test-retest reliability was excellent for the pTESS-Leg (intraclass correlation coefficient [ICC] = 0.94; 95% CI, 0.90-0.97) and good for the pTESS-Arm (ICC = 0.86; 95% CI, 0.61-0.96). Known-group validity (ability to discriminate between groups) was demonstrated by lower mean pTESS-Leg scores for participants using gait aids or braces (mean = 68; SD = 21) than for those who did not (mean = 87; SD = 11; p < 0.001). There was no significant difference between pTESS arm scores among respondents using a brace (n = 5; mean = 73; SD = 11) and those without (n = 22; mean = 83; SD = 19; p = 0.13). To evaluate construct validity, we tested a priori hypotheses. The duration since chemotherapy correlated moderately with higher pTESS-Leg scores (r = 0.4; p < 0.001) but not with pTESS-Arm scores (r = 0.1; p = 0.80), and the duration since tumor resection correlated moderately with higher pTESS-Leg scores (r = 0.4; p < 0.001) but not pTESS-Arm scores (r = 0.2; p = 0.4). Higher VAS scores (that is, it was harder to do things) antecorrelated with both pTESS versions (pTESS-Leg: r = -0.7; p < 0.001; pTESS-Arm: r = -0.8; p < 0.001). To assess criterion validity, we compared the pTESS with the current "gold standard" (adult TESS). Among adolescents, strong correlations were observed between the TESS and pTESS-Leg (r = 0.97, p < 0.001) and pTESS-Arm (r = 0.9, p = 0.007). CONCLUSIONS: Both pTESS versions exhibited no floor or ceiling effects and had high internal consistency. The pTESS-Leg demonstrated excellent reliability and validity, and the pTESS-Arm demonstrated good reliability and reasonable validity. The pTESS is recommended for cross-sectional evaluation of self-reported physical function in pediatric patients with bone tumors. LEVEL OF EVIDENCE: Level II, outcome measurement development.
BACKGROUND: The physical function of children with sarcoma after surgery has not been studied explicitly. This paucity of research is partly because of the lack of a sufficiently sensitive pediatric functional measure. The goal of this study was to establish and validate a standardized measure of physical function in pediatric patients with extremity tumors. QUESTIONS/PURPOSES: (1) What is the best format and content for new upper- and lower-extremity measures of physical function in the pediatric population? (2) Do the new measures exhibit floor and/or ceiling effects, internal consistency, and test-retest reliability? (3) Are the new measures valid? METHODS: In Phase 1, interviews with 17 consecutive children and adolescents with bone tumors were conducted to modify the format and content of draft versions of the pediatric Toronto Extremity Salvage Score (pTESS). In Phase 2, the pTESS was formally translated into French. In Phase 3, 122 participants between 7 and 17.9 years old with malignant or benign-aggressive bone tumors completed the limb-specific measure on two occasions. Older adolescents also completed the adult TESS. Floor and ceiling effects, internal consistency, test-retest reliability, and validity were evaluated. RESULTS: Feedback from interviews resulted in the removal, addition, and modification of draft items, and the pTESS-Leg and pTESS-Arm questionnaires were finalized. Both versions exhibited no floor or ceiling effects and high internal consistency (α > 0.92). The test-retest reliability was excellent for the pTESS-Leg (intraclass correlation coefficient [ICC] = 0.94; 95% CI, 0.90-0.97) and good for the pTESS-Arm (ICC = 0.86; 95% CI, 0.61-0.96). Known-group validity (ability to discriminate between groups) was demonstrated by lower mean pTESS-Leg scores for participants using gait aids or braces (mean = 68; SD = 21) than for those who did not (mean = 87; SD = 11; p < 0.001). There was no significant difference between pTESS arm scores among respondents using a brace (n = 5; mean = 73; SD = 11) and those without (n = 22; mean = 83; SD = 19; p = 0.13). To evaluate construct validity, we tested a priori hypotheses. The duration since chemotherapy correlated moderately with higher pTESS-Leg scores (r = 0.4; p < 0.001) but not with pTESS-Arm scores (r = 0.1; p = 0.80), and the duration since tumor resection correlated moderately with higher pTESS-Leg scores (r = 0.4; p < 0.001) but not pTESS-Arm scores (r = 0.2; p = 0.4). Higher VAS scores (that is, it was harder to do things) antecorrelated with both pTESS versions (pTESS-Leg: r = -0.7; p < 0.001; pTESS-Arm: r = -0.8; p < 0.001). To assess criterion validity, we compared the pTESS with the current "gold standard" (adult TESS). Among adolescents, strong correlations were observed between the TESS and pTESS-Leg (r = 0.97, p < 0.001) and pTESS-Arm (r = 0.9, p = 0.007). CONCLUSIONS: Both pTESS versions exhibited no floor or ceiling effects and had high internal consistency. The pTESS-Leg demonstrated excellent reliability and validity, and the pTESS-Arm demonstrated good reliability and reasonable validity. The pTESS is recommended for cross-sectional evaluation of self-reported physical function in pediatric patients with bone tumors. LEVEL OF EVIDENCE: Level II, outcome measurement development.
Authors: Paul A Meyers; Cindy L Schwartz; Mark Krailo; Eugenie S Kleinerman; Donna Betcher; Mark L Bernstein; Ernest Conrad; William Ferguson; Mark Gebhardt; Allen M Goorin; Michael B Harris; John Healey; Andrew Huvos; Michael Link; Joseph Montebello; Helen Nadel; Michael Nieder; Judith Sato; Gene Siegal; Michael Weiner; Robert Wells; Lester Wold; Richard Womer; Holcombe Grier Journal: J Clin Oncol Date: 2005-03-20 Impact factor: 44.544
Authors: R Nagarajan; D R Clohisy; J P Neglia; Y Yasui; P A Mitby; C Sklar; J Z Finklestein; M Greenberg; G H Reaman; L Zeltzer; L L Robison Journal: Br J Cancer Date: 2004-11-29 Impact factor: 7.640