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The Pediatric Toronto Extremity Salvage Score (pTESS)

Validation of a Self-reported Functional Outcomes Tool for Children with Extremity Tumors

Piscione, Janine BA, BScPT, MSc; Barden, Wendy PScPT, MSc; Barry, Janie MSc; Malkin, Alexandra MSc; Roy, Trisha MD, PhD; Sueyoshi, Tyki BSc; Mazil, Karen BN; Salomon, Steven MSc; Dandachli, Firas MD, MSc; Griffin, Anthony MSc; Saint-Yves, Hugo MSc; Giuliano, Pina; Gupta, Abha MD, MSc; Ferguson, Peter MD, MSc; Scheinemann, Katrin MD; Ghert, Michelle MD; Turcotte, Robert E. MD; Lafay-Cousin, Lucie MD, MSc; Werier, Joel MD; Strahlendorf, Caron MB, BCh; Isler, Marc MD; Mottard, Sophie MD; Afzal, Samina MBBS; Anderson, Megan E. MD; Hopyan, Sevan MD, PhD

Clinical Orthopaedics and Related Research®: September 2019 - Volume 477 - Issue 9 - p 2127–2141
doi: 10.1097/CORR.0000000000000756
CLINICAL RESEARCH
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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.

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

S. Hopyan, 686 Bay Street, 16-9713, Toronto, Ontario, M5G 0A4 Canada, Email: sevan.hopyan@sickkids.ca

One of the authors (PF) received personal fees from Stryker (Kalamazoo, MI, USA), outside the submitted work. One of the authors (MG) received personal fees from Wright Medical Group, Inc (Memphis, TN, USA), grants from the Canadian Institutes of Health Research, and grants from the Canadian Cancer Society Research Institute, outside the submitted work. One of the authors (MEA) is on the Editorial Board of Clinical Orthopaedics and Related Research®. This study was funded by the Garron Family Cancer Centre, Hospital for Sick Children.

All ICMJE Conflict of Interest Forms for authors and Clinical Orthopaedics and Related Research® editors and board members are on file with the publication and can be viewed on request.

Each author certifies that his or her institution approved the human protocol for this investigation and that all investigations were conducted in conformity with ethical principles of research.

This work was performed at The Hospital for Sick Children, Toronto, Canada; Hôpital Maisonneuve-Rosemont et Université de Montréal, Montréal, Canada; Boston Children’s Hospital, Jimmy Fund Clinic/Dana Farber Cancer Institute and Harvard Medical School, Boston, MA, USA; BC Children’s Hospital and University of British Columbia, Vancouver, Canada; Children’s Hospital of Eastern Ontario and University of Ottawa, Ottawa, Canada; Alberta Children’s Hospital and University of Calgary, Calgary, Canada; McGill University Health Centre and McGill University, Montréal, Canada; Mt Sinai Hospital and University of Toronto, Toronto, Canada; McMaster University Health Sciences Centre, Juravinski Cancer Centre and McMaster University, Hamilton, Canada; and IWK Health Centre and Dalhousie University, Halifax, Canada.

Received October 04, 2018

Accepted March 12, 2019

Online date: April 27, 2019

© 2019 Lippincott Williams & Wilkins LWW
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