THIS EVIDENCE REPORT IS OUTDATED AND IS NO LONGER VIEWED AS GUIDANCE FOR CURRENT MEDICAL PRACTICE. IT IS MAINTAINED FOR ARCHIVAL PURPOSES ONLY.

Evidence Report/Technology Assessment

Number 2, Supplement

Prepared for: Agency for Health Care Policy and Research

Department of Health and Human Services
2101 East Jefferson Street
Rockville, MD 20852
http://www.ahcpr.gov

Contract No. 290-97-0018

Prepared by:
Oregon Health Sciences University, Portland, OR

Nancy Carney, PhD, Principal Investigator
Hugo du Coudray, PhD
Cynthia Davis-O'Reilly, BS
Melanie Zimmer-Gembeck, PhD
N. Clay Mann, PhD
Kathryn Pyle Krages, AMLS, MA
Mark Helfand, MD, EPC Director

AHCPR Publication No. 00-E001

September 1999

The Agency for Health Care Policy and Research (AHCPR), through its Evidence-based Practice Centers (EPCs), sponsors the development of evidence reports and technology assessments to assist public- and private-sector organizations in their efforts to improve the quality of health care in the United States. The reports and assessments provide organizations with comprehensive, science-based information on common, costly medical conditions and new health care technologies. The EPCs systematically review the relevant scientific literature on topics assigned to them by AHCPR and conduct additional analyses when appropriate prior to developing their reports and assessments.

To bring the broadest range of experts into the development of evidence reports and health technology assessments, AHCPR encourages the EPCs to form partnerships and enter into collaborations with other medical and research organizations. The EPCs work with these partner organizations to ensure that the evidence reports and technology assessments they produce will become building blocks for health care quality improvement projects throughout the nation. The reports undergo peer review prior to their release.

AHCPR expects that the EPC evidence reports and technology assessments will inform individual health plans, providers, and purchasers as well as the health care system as a whole by providing important information to help improve health care quality.

We welcome written comments on this evidence report. They may be sent to: Director, Center for Practice and Technology Assessment, Agency for Health Care Policy and Research, 6010 Executive Blvd., Suite 300, Rockville, MD 20852.

Douglas B. Kamerow, MD	John M. Eisenberg, MD
Director, Center for Practice	Administrator
and Technology Assessment	Agency for Health Care Policy and Research
Agency for Health Care Policy and Research

The authors of this report are responsible for its content. Statements in the report should not be construed as endorsement by the Agency for Health Care Policy and Research or the U.S. Department of Health and Human Services of a particular drug, device, test, treatment, or other clinical service.

We thank the Brain Injury Support Group of Portland, Randall Chesnut, MD, and Mark Ylvisaker, PhD, for their help in preparing this report.

Objectives. The goal was to conduct a systematic review of the literature about child and adolescent traumatic brain injury (TBI) oriented around key research questions and to create a tool that would be used in future evidence-based investigations about recovery from TBI in this population. A matrix was prepared to organize research according to the developmental dimensions and age categories addressed by studies. With the assistance of technical experts, key questions were formulated about (1) the effectiveness of early, intensive rehabilitation; (2) referral of children with TBI to special education; (3) the effectiveness of special education for children with TBI; (4) the effect of developmental phase on prediction and outcome; and (5) the effect of support for families. Patient populations, interventions, and outcome measures were defined, and literature was compiled and categorized from seven databases -- medical, educational, and psychological. The strongest studies that contained data about the key questions were reviewed, and an evidence table template was created for use in future comprehensive studies. In the evidence report, methods are proposed for conducting research and evaluating data about children and adolescents with TBI that can incorporate and account for individual differences and the influence of growth and change on the recovery process.

Search Strategy. A search was conducted of MEDLINE (1976-1998), CINAHL (1982-1998), HealthSTAR 1995-1998), PsychINFO (1982-1998), ERIC (1996-1998), Current Contents (1998), and the Cochrane Library, supplemented by reference lists of review articles, book chapter bibliographies, and the advice of peers.

Results. The initial search strategy yielded 1,464 abstracts of potentially relevant studies. All studies about child or adolescent TBI -- from acute management through long-term rehabilitation -- were categorized by study design, deficit, intervention, outcome, and predictors, and they were included in the bibliography. Three-hundred fifty-six articles identified from electronic databases and manual searches relevant to one of the five research questions were retrieved and read. Studies containing data were evaluated by a member of the research team. One study was found for question 1; 15 studies were found for question 2; 8 studies were found for question 3; 61 studies were found for question 4; and 3 studies were found for question 5 (88 total). The remaining 268 articles described programs or interventions without providing patient or student data.

No randomized controlled trials and very few comparative studies were found that addressed the key research questions. One study suggests that the early introduction of physiatry during the acute care phase of treatment aids in detecting musculoskeletal trauma that may otherwise be missed. No literature was found that accurately documents rates of referral for children with TBI to special services. Although the available single-subject studies suggest beneficial effects of special school programs, methodologic flaws in the one comparative study render its findings inconclusive. A large body of literature documents the utility of patients' developmental information in predicting deficits and outcomes. Correlational studies associate family support with better family functioning.

In general, studies have not been conducted with designs capable of providing evidence on the effectiveness of interventions for children and adolescents with TBI. The published literature for this topic is primarily exploratory. It provides descriptions of programs that are widely accepted, including logical approaches to treatment that have not been validated either through experimental design or in carefully controlled observational studies. The clinical experience represented in the published literature that has guided the design of intervention programs should generate the important hypotheses for controlled studies.

This document is in the public domain and may be used and reprinted without permission.

Carney N, du Coudray H, Davis-O'Reilly C, et al. Rehabilitation for traumatic brain injury in children and adolescents. Evidence report no. 2, supplement (Contract 290-97-0018 to Oregon Health Sciences University). Rockville, MD: Agency for Health Care Policy and Research. September 1999.

The estimated incidence of traumatic brain injury (TBI) doubles between the ages of 5 and 14 years and peaks for both males and females during adolescence and early adulthood to approximately 250 per 100,000. Children and adolescents are more likely than adults to survive following TBI. Because the lives of most survivors of moderate to severe TBI involve chronic, life-long disabilities with varying degrees of dependence, the cost in individual suffering, family burden, and financial burden to society is greater for those who have more years to live.

Limitations in bathing, dressing, and walking are observed in between 50 percent and 90 percent of children with TBI with multiple functional deficits, depending upon and directly proportional to the number of functional deficits. For children with four or more functional deficits, 75 percent have impairments in self-feeding, cognition, and behavior; there also may be impairments in speech (67 percent), vision (29 percent), and hearing (16 percent).

In the 18^th Annual Report to Congress on the Implementation of the Individuals with Disabilities Education Act, the number of children receiving services because of TBI for the 1994 to 1995 school year was 7,188 students aged 6 to 21 years. The gap between this number and reports of incidence of TBI among children and adolescents suggests that many children with TBI may be misidentified or unidentified. The concern is that the problems of these children may go unrecognized, or they may be treated with methods that were developed for other pathologies but are inappropriate to the special needs of children with TBI.

Although many children with TBI may transition to inpatient rehabilitation and some to long-term care facilities, the goal, when possible, is return to school. Laws requiring schools to provide for the special education needs of students define schools as the best place for ongoing rehabilitation of most children with TBI. Ideally, a child identified with TBI would be evaluated for special needs and provided with an individualized education program (IEP) designed to meet those needs. However, the content and quality of the program would depend on the resources available in the school and varies across States and regions.

Characteristics such as social inappropriateness, lack of awareness, and decreased control of attention, memory, and strategic thinking may result in difficulties when integrating a child with TBI into mainstream educational settings. Some States provide training for public school teachers that is focused on the special needs of children with TBI. Some programs serve both children who live at home and those who require residential treatment in a specialized educational/neurorehabilitative setting. Choice of model (mainstream vs. separate) may be dictated by the severity of deficits and functional capabilities of the child; it also may be influenced by the availability of resources within the community, family choice, or the local or regional philosophy of inclusive vs. segregated education of students with disabilities.

In 1997 the Evidence-based Practice Center (EPC) at Oregon Health Sciences University (OHSU) contracted with the Agency for Health Care Policy and Research (AHCPR) to produce an evidence report on rehabilitation from TBI in adults. At the conclusion of the project, AHCPR requested that the OHSU EPC conduct a survey of the literature regarding child and adolescent TBI rehabilitation. Specific objectives for the project were to:

The main goal was to create a template for a comprehensive, systematic review of existing literature. A secondary goal was to describe research projects capable of closing information gaps revealed by a survey of the literature.

Children are naturally changing and developing both before and after they are injured, including while they are receiving rehabilitative interventions. Therefore, in order to examine the effects of rehabilitation, information is required on normal child development. Longitudinal studies that compare development of injured and uninjured children also are needed. To be useful the studies must assess capabilities of children at different ages and determine individual variation in developmental outcomes of children with different injuries and in different social environments.

The best organizing principle for reviewing the literature on child and adolescent TBI comes from the modern study of human development and, in particular, the metatheoretical approach known as Life-Span Developmental Psychology. Simply stated, any research question about child and adolescent TBI must be oriented to the relevant developmental category and age group. Also, subsets of information within a research question must address developmental phases. For example, when describing the sample of a particular study, age at injury, age at evaluation, and time since injury, as well as the developmental implications of those age-related landmarks, all should be identified.

Two panels of experts, one local and one national, worked with the research team to identify the following key questions in the rehabilitation and survivor phases of recovery from TBI for children and adolescents. For each question, they provided a rationale for asking the question, as well as definitions, target populations, and outcome measures. The questions are:

Literature was searched using MEDLINE (1976-1998), CINAHL (1982-1998), HealthSTAR (1995-1998), PsychINFO (1982-1998), ERIC (1996-1998), the Cochrane Library, and Current Contents (1998), and 1,464 potential references were identified. Abstracts of these references were reviewed independently by two members of the research team, who applied predefined, broad eligibility criteria. The process resulted in 376 studies about TBI in children or adolescents. These studies were coded and categorized according to study design, deficit, intervention, outcomes, and predictors and placed in a bibliography in both written and electronic form for use in future investigations. The studies were read a second time, and 236 were identified that were relevant to one or more of the five key questions. An additional 120 articles were added from reference lists, book chapter bibliographies, and the advice of peers. A total of 356 full-text articles were retrieved and read, and those studies containing data relevant to one of the key questions were identified. One study was found for question 1, 15 for question 2, 8 for question 3, 61 for question 4, and 3 for question 5 (88 total). The remaining 268 articles described programs or interventions without providing patient or student data.

There were no randomized controlled trials and no comparative studies that investigated the efficacy of early, intensive rehabilitation for children or adolescents. Inferences about this intervention for children have been drawn from studies with adult samples. One prospective, uncontrolled observational study and two retrospective studies were reviewed for indirect information about the effects of the intervention. Of these, one study suggests that early, thorough physical and occupational therapy evaluations that include bone scans may serve to identify otherwise undetected musculoskeletal trauma and heterotopic ossification, indirectly arguing for early physiatry intervention. Authors suggested that the difficulties in communication unique to TBI warrant special methods for detecting physical trauma in people with TBI.

This question has two parts: (1) How many children with TBI receive special education services? and (2) Are the programs and services they receive delivered by people who understand and are able to manage TBI in children? Only the first part of this question could be addressed, since no studies were found on special education programs delivered by personnel who have been trained in caring for and educating students with TBI.

Three retrospective studies and one cross-sectional State-wide study suggest that between 9 percent and 38 percent of students with identified brain injury receive referral to special education. However, since no evaluation of the students with TBI who did not receive special education was provided, it is not known whether these students needed special services, or how many were functioning well without services. Therefore, it is not possible to determine whether these reported referral rates indicate adequate referral, under-referral, or over-referral. The important question is whether the child with TBI who needs special services receives them. The answer to that question depends on being able to measure independently the need for or potential benefit from special education and then determining the proportion of children who could benefit and are actually referred. These data were not found in the published literature.

One nonrandomized comparative study, one small case series, one survey, and five case studies provide limited data about the effects of special education programs for children with TBI, with varied results. No significant treatment effect was found in the comparative study; however, the comparison group performed significantly better than the treatment group at pretest on neuropsychological and intelligence tests and on adaptive and behavioral measures, suggesting that they were not as impaired as the treatment group from the beginning. In the case series, there was significant improvement from pre- to posttreatment on one of nine laboratory-based neuropsychological tests. In the five case studies, all patients showed improvement on measures taken during intervention when compared with those taken at pretreatment. However, because these studies are uncontrolled, the effect cannot be generalized to a larger population of children.

Sixty-one studies reporting data related to predictability of deficits based on developmental issues in child and adolescent TBI were found: 51 were prospective; 4 were population-based; 13 were multicenter; and 12 studies evaluated patients for 3 or more years. Because of the diversity of topics in this question and the large number of citations found, a system was developed for rating the overall quality of each article on an ordinal scale; the articles with the highest ratings were selected for review. Criteria for rating quality were (1) number of developmental categories included in the study, (2) study length, (3) design, (4) setting, (5) sample selection method, (6) age range in sample at time of measurement, (7) span of developmental stages covered in age range, (8) comparison method, (9) specification of injury severity, (10) specification of location of injury, (11) span of developmental stages covered by range of age at injury, (12) time from injury to assessment, (13) followup, and (14) analysis methods.

Seven studies that had the highest methodological scores using this system were reviewed. One cross-sectional/longitudinal evaluation of language acquisition demonstrates a predictable pattern of delays and deficits in language acquisition for children up to the age of 3 years when compared with uninjured children. Two additional cross-sectional studies establish the base rate measures of brain growth at each stage of development that are necessary to detect the developmental effects of injury. Two comparative studies revealed the presence of subtle, hidden deficits in cases of apparently normal performance in pediatric TBI with focal brain damage. Two studies used the analytic method of growth modeling and the use of growth trajectories in their research. By analyzing individual growth curves, researchers were able to control for differences in the ages of the children. They discovered systematic, nonlinear changes in growth that were strongly related to injury variables.

There were no randomized controlled trials that compared the effects of support to families with no support. One trial using random assignment evaluated the differential effects of two forms of support to families, and two prospective studies contained indirect evidence about the effects of provision of support. Results of the trial suggest that an intervention for parents of children with brain injury may be more effective in reducing the burden of illness if it focuses on the needs of the parents as opposed to the needs of the child. One prospective observational study found a significant and direct correlation between the presence of social support and measures of family functioning at 3 years postinjury; a second suggested that the presence of case management may serve to reduce parents' financial problems associated with their children's TBI.

The reciprocal effects of family functioning on outcomes for the child with TBI were not addressed. A number of studies have demonstrated a relationship between higher family functioning and better outcomes for the injured child and bolster the argument for provision of support to families as an intervention for the child.

In general, studies have not been conducted with designs capable of providing evidence for effectiveness of interventions for children and adolescents with TBI. Because the focus of this project was effectiveness, many studies were excluded because they did not provide experimental evidence that could be used to guide practice. The published literature for this topic is primarily exploratory. It provides descriptions of programs that are widely accepted, including logical approaches to treatment that have not been validated either through experimental design or in carefully controlled observational studies. The clinical experience represented in the published literature that has guided the design of intervention programs should generate important hypotheses for controlled studies.

Investigations of what might work to rehabilitate children with TBI may benefit from the literature in other related fields. Future research could be guided by themes that have emerged across many disability groups. Although TBI has its unique features, it shares many characteristics with other disabilities. The task is to identify the shared characteristics, and include what has been learned in other fields when designing interventions. One example is social skills training. Certain models for social skills training and cognitive rehabilitation have been shown to be ineffective with people who have other, similar disabilities, yet these models are being used in TBI rehabilitation. At the very least, the failure of these interventions in other fields should call into question their effectiveness with TBI. Similarly, it is important to pay attention to and systematically test successful approaches in other fields.

Three gaps in the literature about child and adolescent TBI define priorities for future research.

Finally, the field of Life-Span Developmental Psychology provides a technology for designing studies and analyzing data that accounts for individual variation, which is important for the evaluation of recovery from TBI for both adults and children. The approaches suggested here will result in complex research designs and data sets in which children with and without TBI are followed for extended periods of time with multiple times of assessment. In addition, the children -- and possibly their families, teachers, and/or friends -- would be asked to complete multiple assessments at each time of measurement. The focus becomes predicting individual growth curves from antecedent variables such as age at injury, initial status, environmental factors (e.g., family functioning), and intervention techniques. Hence, the focus is on the prediction of growth curves and trajectories in performance among children following TBI compared with control children and/or children with TBI participating in alternative interventions. To maximize these data, several statistical techniques are available, including growth curve modeling, cluster analysis, and time-series analysis.