PROTOCOL: Personal assistance for children and adolescents (0-18) with both physical and intellectual impairments

Definition of impairments

The International Classification of Impairments, Activities, and Participation (ICIDH-2) refers to impairment as loss or abnormalities at the level of body, body part or organ. People may have difficulty performing particular activities as a result of impairments, and a person's participation in education, social life, work, and other areas may be limited as a result of interactions among impairments, activities, and environment (WHO 2003). Though the ICIDH-2 refers to adults, except with reference to studies using specific definitions of other terms, this review follows the classification in ICIDH-2, which does not include the terms disability or handicap.

This review will include children and adolescents with both physical impairments and intellectual impairments, which include learning impairments (e.g., Down's syndrome, global developmental delay, or pervasive developmental disorder), learning disability (also called ‘intellectual disability’ or ‘mental retardation’) and acquired brain injuries. Intellectual impairments and physical impairments affect activities and participation differently. Therefore, young people with intellectual impairments only or physical impairments only will be considered in other Cochrane and Campbell reviews, as will working-age adults and older adults.

Prevalence of impairments

Around the world, about six hundred million people have impairments (UN 1990), most of whom live in the developing world. Previous reviews have identified inconsistencies in the measurement of impairments and activity limitations (UN 1990) and cross-national estimates of impairments and activity limitations in childhood and adolescence are even more variable than national estimates.

The prevalence of intellectual impairments is difficult to quantify due to problems in diagnosis and classification. Most children and adolescents with intellectual impairments have mild to moderate impairments. Relatively few have ‘profound’ impairments (DSM-IV R). Over five million (10.6%) American children and adolescents experience a limitation in learning ability; respectively, about 4.0% and 3.2% have moderate and severe limitations (Hogan 1997).

Gross rates of impairments in the United States (U.S.) have increased substantially in recent decades as a result of an aging population that is living longer and, more recently, as a result of higher reported levels of impairments among children and young adults (Kaye 1996). Recent increases in the prevalence of impairments among children and adolescents may be a result of changes in medical care. For example, very low birthweight babies are more likely than ever to survive (Alberman 1991; Allen 1993; Doyle 1995). These children are at high risk of both physical impairments and intellectual impairments (Middle 1996; Williamson 1983; Wilson-Costello 2005).

Over six million (12.3%) American children and adolescents experience some type of functional limitation (as defined by (Hogan 1997)). Of the four million American children and adolescents who experience one serious functional limitation, about half experience one or more other functional limitations. About one million American children and adolescents experience serious limitations in two or more areas (Hogan 1997). Impairments are more prevalent in boys than in girls (Newacheck 2004) and more prevalent in low-income families than in high-income families (Newacheck 2004; UN 1990).

Of non-institutionalised children and adolescents aged 5-17, 1.3% experience limitations in mobility and about .2% experience a moderate or severe limitation in mobility. Respectively, .9% and .5% experience limitations in self care; 5.5% and 1.2% experience limitations in communication (Hogan 1997). About 3.2% of American children attend special schools or classes (Wenger 1995).

As far as possible, this review uses internationally accepted definitions of impairments and refers to impacts that are likely to occur across cultures. However, many epidemiological studies have been conducted in the United States and Western Europe. Readers should consider the applicability of epidemiological data to other settings.

Consequences

A discourse of disability ethics has evolved to discuss concepts of independence, defined not as people with disabilities “doing everything” for themselves, but as having maximum control over how help is provided (Morris 2001). Proponents of the social model of disability regard activity restrictions as caused by societal and structural barriers and stress the need for their removal (Abberley 1987; Oliver 1990). In addition to structural and environmental changes (e.g., making buildings accessible), the social model emphasises changes in public attitudes towards impairments to encourage increased participation and improved self-esteem.

Participation in age-appropriate activities may be limited for children and adolescents with impairments when physical, social and attitudinal environments restrict their involvement (Hammal 2004; Mihaylov 2004). Impairments in children are related to societal limitation, and different types of impairments contribute uniquely to societal limitations (Hogan et al., 1997). Impairments may affect the quality of life, health, development, and family functioning of children and adolescents (Lavigne 1993; Neely-Barnes 2004; Pit-Ten 2002; Varni 2005). Limited participation in activities may have negative impacts on the mental and physical functioning of children and adolescents and may lead to emotional and behavioural problems.

Children and adolescents with intellectual and physical impairments are at increased risk for mental health problems; however, most children and adolescents who might benefit from psychological services do not receive them (Witt 2001). Comorbid problems can also impact carers. For example, challenging behaviour often occurs in the context of learning impairments and mental health problems (Moss 2000). Parents and siblings of children with impairments are at risk for psychological problems (Rossiter 2001; Sharpe 2002; Thyen 1998).

Total healthcare costs for children and adolescents with impairments may be four times greater than for those without impairments (Newacheck 2004). However, children and adolescents with impairments require varying degrees of support. For example, in one large study, ‘those in the upper decile of the expenditure distribution accounted for 65% of all charges’. Costs born by families of children and adolescents with the most severe impairments were similarly high; out-of-pocket healthcare expenses (i.e. those not reimbursed by insurance or a health service) for the top 10% of the distribution accounted for 85% of all out of pocket expenses (Newacheck 2004).

All children and adolescents require some level of care. However, children and adolescents with extensive impairments may require many hours of assistance every day in addition to the normal assistance required by their peers. Consequently, family members (notably mothers) may withdraw from work and social life. Caring for a child with severe physical impairments can put great financial and emotional stress on parents and families (Neely-Barnes 2004; Witt 2001).

In the U.S., parents of children with impairments are less likely to have attended college and more likely to be single and poor (Hogan 1997). The stress of caring for children with impairments may have negative impacts on parents, but some problems associated with impairments may increase the risk of impairment. For example, poor children are less likely than their peers to receive preventive healthcare (Newacheck 1988) and depressed mothers are less likely than other mothers to use injury prevention strategies like car seats and electrical plug covers (McLennan 2000). Furthermore, some evidence suggests that the development of intellectual impairments is affected by sociodemographic factors (Resnick 1998). Low birthweight black babies are more likely than white babies to survive (Morse 2006), which might affect the relative prevalence of impairments in different racial groups; however, ‘controlling for socioeconomic differences and family structure, black children are no more at risk of functional limitation than whites’ (Hogan 1997). Since the causes and consequences of impairments are often difficult to disentangle, retrospective studies are difficult to interpret.

Interventions

Increased participation (inclusion in activities of daily life) may have positive effects on the social functioning, development, mental and physical health of children and adolescents.

There are many ways to increase participation by children and adolescents with impairments. For example, school schedules may be designed such that children and adolescents with impairments can engage in age-appropriate activities with their peers. Clinicians and policymakers can work together to influence policy, discourse, and planning and to apply the social model in support of children and their families (Colver 2005). However, broad interventions may not be sufficient to meet all needs. People with severe impairments require interventions tailored to their unique impairments, lifestyles, living arrangements, etc. Assistive devices, skills training, physical therapy, education, and human support help young people control their lives appropriately and engage in normal activities.

Personal assistance

Previous studies

Compared to other interventions, personal assistance may have unique benefits and potential drawbacks. Assistants may help children achieve more, but having a personal assistant could be stigmatising. Parents of children with impairments might be relieved to have assistants help care for their children, but assistants might interfere with family life and with users’ need for privacy, or with parents’ own needs to see themselves as adequate carers for their children. Compared to other ways of compensating for particular activity limitations, personal assistance may create unnecessary dependencies for some people.

Even if personal assistance is clearly preferred over other services by working adults with physical impairments, groups that are underrepresented in the public discourse about the rights of people with impairments (e.g., children, people with intellectual impairments, and people in rural areas) may prefer other services, particularly since these groups may be more susceptible to abuse and less able to manage employees. Direct payments for personal assistance may not be ideal for children and families who have difficulty finding an assistant, administering services, negotiating or giving instructions (Pijl 2000).

While many personal assistants are managed by users or their representatives, the nature of personal assistance can make it difficult to separate the roles that individuals play in supporting people with impairments. For example, Askheim identified one mother of a child with intellectual impairments in Norway who acted both as the manager of her child's payments and as a full-time personal assistant (Askheim 2003). Policies that permit different care arrangements may have substantially different impacts.

As the personal assistance movement gained strength, Ratzka noted that ‘there has been surprisingly little in the way of policy evaluation. The work that has been done in this area is restricted to gathering descriptive statistics on number of hours provided by one type of service, number of consumers, staff, and expenditures’ (Ratzka 1986). Some research now suggests that personal assistance may meet otherwise unmet needs of people with impairments. Shortly after its introduction, a survey of direct payment recipients in the UK found that 40% had a need for additional hours of personal service while 80% of people receiving other services had a similar need (Zarb 1994). However, traditional reviews have failed to locate many evaluation studies and have not offered a definitive account of international research on personal assistance. A recent report by the Swedish National Board of Health and Welfare (Socialstyrelsen) highlighted the need for a sensitive and exhaustive search for trials and a systematic synthesis of existing studies (Socialstyrelsen 2005).

Types of studies

Randomised controlled trials, quasi-randomised controlled trials and nonrandomised controlled studies of personal assistance compared to other forms of support or to ‘no-intervention’ (which may include unpaid care) in which participants were prospectively assigned to study groups and in which control group outcomes were measured concurrently with intervention group outcomes.

Types of participants

Children and adolescents (0-18) living in the community who require assistance to perform tasks of daily living (bathing, eating, getting around, etc.) and participate in normal activities due to permanent physical and intellectual impairments.

With the exception of young people living in student accommodation (e.g. residential schools), young people living outside their own homes (e.g., in private or public institutions for people with impairments) will be excluded.

Children and adolescents with physical impairments only and intellectual impairments only will be excluded because these impairments affect activities and participation differently.

Types of interventions

Personal assistance is paid individualised human support that is designed to promote participation of people with permanent impairments. In consultation with experts and the reference group, the reviewers sought to determine what minimal amount of assistance would could be offered and still follow the personal assistance model for this population. For inclusion in this review, personal assistance must have been delivered for at least 20 hours per week.

Comparisons might include, either singly or in combination, family care, institutionalisation, on-demand services, escort services, and other alternatives to personal assistance. ‘No-treatment’ and ‘waiting list’ groups will be included even if other services received are no described. These will be treated as separate comparisons.

Studies examining different forms of personal assistance (e.g., assistance organised by users compared to assistance organised by others) will be included in the review, though these comparisons will be discussed separately as the outcomes from such studies would not indicate the effectiveness of personal assistance relative to other interventions.

Types of outcome measures

Primary outcomes will include:

1) Global quality of life, both (a) generic measures (e.g., the Pediatric Quality of Life Inventory; Varni 2005) and (b) specific measures designed for people with particular impairments. Though well-validated measures for the general population will be considered, a review of global health measures found that ‘very few measures have been validated specifically for cognitively impaired respondents’ (Riemsma 2001) or for people with both physical and intellectual impairments.

2) User satisfaction. Direct reports will be preferred, though proxies might be used if users are unable to communicate.

3) Participation, including social activities, ability to participate in spontaneous activities, time outside the home, and mobility.

Secondary outcomes will include:

1) Unmet needs, particularly the inability to perform activities of daily living.

2) Developmental outcomes, including cognitive milestones, acquisition of skills, and school attendance.

3) Health outcomes, including direct measures of muscle strength, disease, injuries, abuse or pain and indirect measures such as nutrition, emergency room visits or need for hospitalisation or institutionalisation.

4) Psychiatric outcomes, including self-harm, pica (eating non-food substances), and outwardly directed challenging behaviour. Measures might include items from the externalising scale of the Behavior Problem Inventory (Sturmey 1993).

5) Impact on others, including parental (maternal) employment, satisfaction, and quality of family life. For example, measures might include the Short-Form Health Survey (SF-36; (Ware 1992) or General Health Questionnaire (GHQ; (Counsell 1994).

6) Direct and indirect costs, both immediate and long-term.

Outcome intervals

To account for normal development and the changing impacts of impairments, outcomes will be grouped by length of follow-up (e.g., 1-3 years, 4-6 years, 6+ years).

The organisation of services is often a complicated task and new users or their representatives must train personal assistants. Outcomes measured during the first year of receiving personal assistance will be considered apart from outcomes measured after one or more years to account for this adjustment period, which may not be representative of personal assistance as a whole.

I. Electronic search

Databases will be searched for published and unpublished studies. All electronic searches will be limited to research reported since 1980 because scoping for this project, including a review of relevant laws and policy documents and contacts with international experts, found that widespread personal assistance programmes began in the mid 1990s. Experts have noted that personal assistance was available in some form before the introduction of programmes in the 1990s, but they and the reviewers believe it is extremely unlikely that any relevant trials were conducted before 1980.

No language restrictions will be imposed on any results from any search attempts, although most databases will be searched in English. Latin American and Caribbean Health Sciences Literature (LILACs) will be searched using Spanish and Portuguese terms and Scandinavian databases will be searched in appropriate languages.

No filters based on methodology will be applied because test searches indicated that such filters might eliminate relevant studies.

The authors worked with a reference group of users, clinicians, policymakers, and analysts (Jackson 2005) to develop this protocol and search strategy. The group recommended a highly sensitive search (one that will likely to capture all relevant reports) rather than a more specific one (a search that would identify fewer irrelevant papers).

The following databases will be searched electronically:

II. Personal communications

Appropriate government departments, non-governmental organisations, non-profit groups, advocacy groups, user groups, and experts in the field will be contacted and listed in an appendix to the review. These approaches and any replies will be documented by the authors. Additionally, impairment-oriented email lists (list-servs) will be sent a letter requesting assistance in locating studies.

The reviewers will contact authors of all included and excluded studies to request details of ongoing and unpublished studies.

III. Reference lists

Reference lists from previous reviews and from all included and excluded studies will be searched. Relevant websites, including those maintained by users, governments, other agencies, and academics will be searched.

Trial selection strategy

From the resultant list of articles, outcome evaluations about people with impairments will be identified through electronic and hand searches. Two authors (EMW and PM) will check titles for relevance. When a title appears potentially relevant, both authors will examine the abstract. If one author feels an abstract might be relevant, the full article will be obtained. Two authors will examine the remaining papers to determine eligibility. Study authors will be contacted if further information could resolve initial disagreements about inclusion. Remaining disagreements will be discussed with the third reviewer (JD). If a consensus cannot be reached, the Coordinating Editor of the CDPLPG will be consulted. A flowchart of the process of trial selection will be made in accordance with the QUORUM statement (Moher 1999).

Data management

Data synthesis

Subgroup analyses

Large numbers of subgroups may lead to misleading conclusions and are best kept to a minimum (Counsell 1994; Oxman 1992; Yusuf 1991). If possible, this review will include separate effect estimates for the following subgroups:

1) Organisation of services

Personal assistance organised by users or their representatives (e.g., through direct payment schemes) will be considered apart from personal assistance organised and managed by others (e.g., social workers or government agencies).

2) Acquisition of impairment

Separate effects will be reported for children and adolescents who had impairments from birth, who have long-standing impairments, and who recently acquired impairments.

3) Amount of assistance

The number of hours of assistance received per week is related to user needs, which are determined by social context, the availability of other services, severity of impairments, etc. Separate effect estimates will be reported for users receiving different levels of assistance (e.g., 20-50 hours, 51-80 hours, more than 80 hours).

Assessment of bias

Sensitivity analyses will investigate the influence of lower quality studies (i.e., those rated C and D on allocation concealment) on the results of the review.

To investigate the possibility of bias, including publication bias, funnel plots will be drawn (Deeks 2005; Egger 1997; Sterne 2001). In the event of asymmetry, the reviewers will seek input from methodologists, including the Cochrane and Campbell Collaboration Methods Groups, on appropriate analyses.

Graphs

When meta-analyses are performed, data will be entered into RevMan in such a way that the area to the left of the line of no effect indicates a favourable outcome for personal assistance.