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Feasibility of Conducting Long-Term Follow-Up of Children and Infants Treated for CNS Tumors on the Same Cooperative Group Clinical Trial Protocol

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Abstract

Given the barriers to conducting long-term assessment of neurocognitive and psychosocial functioning of those treated in infancy for central nervous system (CNS) tumors, a multi-site feasibility study was conducted. The primary objective was to demonstrate that it is feasible to identify, locate and assess the functioning of children treated on the same protocol 10-years post-treatment. Six sites obtained institutional approval, identified and recruited subjects, and obtained comprehensive neurocognitive and psychosocial data. All feasibility objectives were met. Barriers to participation included length of time for Institutional Review Board submission and review, clinical demands, limited eligible participants at individual institutions, difficulty locating long-term subjects and stipend/reimbursement concerns. Results indicate that long-term studies are feasible and essential given the need to address long-term issues of children treated at a young age for CNS tumors, especially as they relate to later academic and vocational planning, but require significant coordination and commitment of cooperative group and institutional resources.

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Acknowledgments

The investigators are grateful to the National Children’s Cancer Society for its support. We are also grateful to the children and families who agreed to participate, the Children’s Oncology Group Behavioral Science, Late Effects, and CNS committees, especially Paul Fisher, MD, and Danielle Logan for her editorial expertise.

Conflict of interest

Drs. Jennifer Hoag, Mary Jo Kupst, Marie-Eve Briere, Donald Mabbott, T. David Elkin, Jill Isenberg, Suzanne Holm, Cheryl Ambler, and Douglas R. Strother declare that they have no conflict of interest. Dr. Christine L. Trask has a family member that receives salary from Sanofi. She also has stocks in Pfizer and Sanofi.

Human and Animal Rights and Informed Consent

All procedures followed were in accordance with the ethical standards of the responsible committee on human experimentation (institutional and national) and with the Helsinki Declaration of 1975, as revised in 2000. Informed consent was obtained from all patients for being included in the study.

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Correspondence to Jennifer Hoag.

Appendix

Appendix

Measures

Each study participant completed a standardized battery of neurocognitive and psychosocial measures to examine their current level of functioning. All are widely used and considered reliable and valid within the context of research.

Wechsler Abbreviated Scale of Intelligence (WASI; Wechsler, 1999 )

The WASI is a nationally standardized measure of intelligence that can be administered to individuals as young as six years of age. Scores include a verbal, performance, and full scale IQ with a mean of 100 and standard deviation of 15.

Wechsler Intelligence Scale for Children-4th Edition, Processing Speed Index (WISC-IV PSI; Wechsler, 2003 )

The PSI measures speed, accuracy, visual-motor coordination and visual scanning. Standard scores have a mean of 100 and a standard deviation of 15.

Wide Range Achievement Test 4, Reading and Arithmetic Composites (WRAT4; Wilkinson & Robertson, 2006 )

The WRAT4 is a measure of fundamental academic skills, including general reading and math ability. Composite scores have a mean of 100 and a standard deviation of 15.

Children’s Memory Scale (CMS; Cohen, 1997 )

The CMS is a comprehensive measure of memory across the domains of verbal, visual and attention/concentration. The General Memory Index is a measure of global memory skills. Scores have a mean of 100 and a standard deviation of 15.

Conners’ Continuous Performance Test II (CPT II; Conners & MHS Staff, 2002 )

The CPT II is a computerized measure of sustained and selective attention and response prevention. The Overall Index has a T-score mean of 50, with a standard deviation of 10.

Behavior Rating Inventory of Executive Function (BRIEF; Gioia, Andrews, & Peter, 2003 )

The BRIEF is a parent-reported measure of executive functioning in children. Scores include a behavioral regulation index, which measures self-control; metacognition index, which measures problem-solving skills; and global composite of executive functioning. The BRIEF has a T-score mean of 50, with a standard deviation of 10.

Behavior Assessment System for Children-Second Edition (BASC-2; Reynolds & Kamphaus, 2005 )

The BASC-2 is a parent-reported measure of internalizing and externalizing behavior problems. The T-score mean on the BASC-2 is 50, with a standard deviation of 10.

Pediatric Quality of Life Inventory (PedsQL; Varni, Seid, & Rode, 1999 )

The health-related quality of life component of the study described in this paper was carried out using the PedsQL™, developed by Dr. James W. Varni. The current study utilized the self-reported and parent-reported Generic Core Scales, which measure physical and psychosocial (i.e., emotional, social, school) functioning. Scores range from 0–100 with higher scores indicating better quality of life (Varni, Seid, & Kurtin, 2001; Varni et al., 2002; Varni, Burwinkle, Seid, & Skarr, 2003).

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Hoag, J., Kupst, M.J., Briere, ME. et al. Feasibility of Conducting Long-Term Follow-Up of Children and Infants Treated for CNS Tumors on the Same Cooperative Group Clinical Trial Protocol. J Clin Psychol Med Settings 21, 136–143 (2014). https://doi.org/10.1007/s10880-014-9391-6

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