A systematic review of hospital experiences of people with intellectual disability

Internationally, hospital and primary health care systems have come under increasing strain as a result of ageing populations and a rise in chronic disease [1]. Australia sits around the middle of OECD countries in terms of the number of hospital episodes per 1,000 population [2]. Initiatives to reduce primary health and hospital inefficiencies have led to the identification of large health inequalities, in particular for Aboriginal and Torres Strait Islanders, people with mental illness and those living in remote areas of Australia [2]. Missing from these health care initiatives are people with intellectual disability - another group who similarly experience large health and health care inequities [3]. Intellectual disability has its onset before the age of 18 years, is characterised by lifelong limitations in IQ (70 or less) and adaptive functioning and accounts for 1% to 3% of the world’s population [3]. In developed countries, people with intellectual disability (a) have higher mortality rates and shorter life expectancy than the general population, (b) have greater health care needs, (c) engage in fewer preventative health screenings, and (d) have conditions that often go undiagnosed or are mismanaged [4]-[7]. They are also high users of hospital services. Recent international data demonstrate that people with intellectual disability use hospital services, particularly emergency departments (ED), more often than other people, especially for conditions more appropriately treated elsewhere (such as out-patient or primary care clinics), and their stays are longer [8]-[10].

In Australia, there have been few attempts to compare the experiences and outcomes of hospitalisations among people with intellectual disability to those of the general population. There has, however, been some acknowledgement of their experience of health inequalities in the National Disability Strategy that is driving large scale reform of the interface between mainstream and specialist disability services and support, and has resulted in a National Disability Insurance Scheme to provide a sustainable individualised funding model to deliver disability services [11]. This strategy was the impetus for a recent state government commissioned report, which provides the most detailed analysis of hospital usage for people with intellectual disability in Australia [12]. Linking disability services and health department data, hospital separations^a were determined for disability clients, predominantly intellectual disability, physical disability, and acquired brain injury, each year from 2005-2010. This analysis revealed their over-representation in the hospital data, with some service users having as many as 8.7 times that of the general population. The last year of data indicated they accounted for 11% to 17% of high end hospital separations and ED presentations, yet accounted for around 68% of all bed days, 44% to 67% of separations, and around 65% of hospital costs [12].

It would seem, then, that people with intellectual disability are high frequency and costly users of hospital services. Unfortunately, there is mounting evidence that they are at particular safety risk in acute hospital settings [13] and have poor experiences, in some cases pointing to institutional discrimination [7],[14],[15]. Research into the hospital experiences of people with intellectual disability has been driven in the UK by a peak body report describing the disturbingly poor hospital treatment of six adults who died in hospitals in England and Wales, arguably as a result of neglect, discrimination, and mismanagement [16]. Despite government effort [17] to adopt 10 recommendations from an independent inquiry [18], there has been recent evidence of continued neglect and discrimination, with dire outcomes for hospital patients with intellectual disability [15]. Most recently, a UK population-based confidential inquiry revealed that avoidable deaths (preventable or amenable through good quality care) were more common among people with intellectual disability than the general population: 37% vs. 13% of avoidable deaths [19]. The contributors were failures in health care provision that resulted in delays in diagnosis and treatment.

The UK case studies [15] and population study reflect experiences in other countries. Iacono and Davis [14], for example, provided reports by family and paid carers of people with intellectual and/or physical disabilities in Australia, which pointed to negative attitudes by hospital staff, delays in diagnostic evaluations and treatment, and reliance on family and paid carers for both advocacy and care during hospital encounters. Others have similarly explored the experience of hospital encounters for people with intellectual disability, including in Australia [20], England ([21], Scotland [22], Canada [23], and Northern Ireland [24]. Furthermore, a submission to inform a New South Wales (NSW) state health plan by a peak body indicated that the poor experiences and outcomes documented for people with intellectual disability are similarly experienced by those with other forms of disability [25]. Mounting evidence in both the grey and published literature suggests continued and serious problems during hospitalisations for people with intellectual and other types of disability and those who support them.

This review was conducted in Australia where it is particularly timely to consider the research evidence about how hospitals may meet the needs of people with disabilities. Recently, the government called for reasonable adjustments by mainstream services, including health care, to ensure equality for people with disability [11]. There is also a need to understand the nature and degree of adjustment required of hospital staff, and how these adjustments may contribute to hospital quality and safety performance [26]. In Australia, policy directives to address the needs of people with disability in hospitals have been evident only in one state [27]. These directives include reduced reliance on family and paid disability staff, and adjustments to pre- admission and discharge plans. Unfortunately, the NSW policy directive fails to compel services to make changes [25]. Similarly, other policy and practice documents have failed to address the needs of people with disability, more broadly, during hospital encounters [26].

A number of reviews have explored underlying contributors to poor hospital experiences by people with intellectual disability [13],[28]-[30]. These reviews have similarly revealed that some people with intellectual disability are fearful of hospital encounters, there is reliance on carers during the entirety of stays, and problem attitudes and limited knowledge of hospital staff, sometimes with dire outcomes. Recommendations to ameliorate problems identified in studies reviewed by Backer, Chapman and Mitchell [13] reflect those from the UK inquiry [18]: that is, developing liaison models, especially through a disability liaison nurse, supporting carers, and improving communication. Mencap's [15] concern of little progress since these recommendations were published has been borne out in the literature, with little evidence, for example, of adopting the frequently recommended Learning Disability Liaison Nurse strategy [29].

Backer et al. [13] argued for further research into hospital access and experiences for people with intellectual disability, but there is little to indicate the value of or specific need for continued research. Previous reviews have focused on summarising findings of largely qualitative research, with little critique of their quality. Phillips [29], for example, noted the use of a critical appraisal tool, but reported methodological limitations of studies reviewed rather than providing quality scores. Bradbury-Jones et al. [30] used a dichotomised rating of weak or strong, but provided little in the way of rating criteria or reliability. Also, there has been no attempt to determine the extent to which studies have contributed specific and new information about the nature and points of hospital encounters that may present particular challenges in meeting the needs of patients with disability. Hospital encounters, or points of encounter refer to specific points on the patient’s journey through a hospitalisation, such as admission, diagnostic testing, placement on a ward, and discharge. Recommendations for adequate assessment of needs and discharge planning [13] fail to take into account the many points of encounter with staff filling varied roles, or of patient requirements as they engage in the journey from presentation to and exit from hospitals.

Prior to undertaking further research into hospital experiences, in general, a critical quality review and analysis of the points of hospital encounters are needed. Such a review promises insights into barriers and enablers of reasonable adjustments by hospital services across the patient journey. It would also provide direction for further research to ensure quality hospital care for people with intellectual disability and others made vulnerable by reliance on others for care and advocacy.

Searches were conducted within Medline, CINAHL, EMBASE, Sociological Abstracts and PsycINFO, with the limiters English-language, primary peer-reviewed research articles, published between 1990 and 2013, and research with original data. Search terms are presented in Table 1. Inclusion criteria were that most of the research population had an intellectual disability (arbitrarily set at 70%), and the main topic related directly to one or more of the specific aims. Retrieved abstracts were scanned manually and potentially suitable papers further assessed against the inclusion criteria. Potential papers were assessed by three authors (TI, CU& PF) and a fourth (CB) was consulted if consensus could not be reached. Corresponding authors were contacted for papers with insufficient details to enable a decision about inclusion: these inquiries were usually about the participant groups. Reference and forward citation searches were performed on included articles and relevant literature reviews.

Tools to evaluate the studies were developed from Downs and Black [31], with additions from the McMaster guidelines for the qualitative studies [32], to provide overall quality scores from a possible score of 11^b. The first author completed quality appraisals with two other authors appraising two each for reliability (4/17 studies). Item-item agreement was 80%-100% (mean =90%); overall scores differed by one point for one of the four studies with agreement for the other three.

The data extracted were participant characteristics, including demographic details, points of hospital encounter and staff-patient interactions; these are presented in Additional file 1. Results of audits of Learning Disability Liaison Nurse (LDLN) services from two mixed methods studies [22],[33] have not been reported in Additional file 1; rather only details of the qualitative components. The qualitative components/ studies were then analysed thematically for patterns of findings and gaps in the research identified. This process involved listing themes reported in each study and conducting a content analysis for grouping according to over-arching and sub-themes (completed initially by TI and independently reviewed and confirmed by CB).

A total of 614 abstracts were retrieved from the database searches and a further 94 articles identified through citation, reference, hand and general web searches. Of these, 608 articles were excluded and the remaining 100 were obtained and assessed for eligibility. The search identified 16 studies that met the inclusion criteria.

This review extends that of Backer et al. [13], whose focus included, but was not limited to, hospital experiences of people with intellectual disability. In addition to their 7 studies that met our selection criteria, an additional 10 were identified, 8 published since their search limit of March 2008. The search strategy used for the current study also yielded 8 more studies that met inclusion criteria than included by Bradbury-Jones et al., perhaps reflecting the use of five versus two databases and less restrictive search terms, despite both reviews having a focus on acute hospital care. In terms of quality, in the current review, the highest scores (7-9) were assigned to recent studies only [20],[40], suggesting an overall improvement over time.

Themes that emerged from the studies indicate little additional insight into the nature of the problems experienced by people with intellectual disability to those in the previous reviews [30]. They continue to experience fear in relation to hospital encounters. Further, they continue to rely on paid or family carers to provide constant care so that basic needs are met, and for advocacy to ensure they receive appropriate assessments and treatments. Negative attitudes from nurses, as well as other hospital staff continue to be evident, although there remains limited information on interactions with varied staff. While researchers have repeatedly made recommendations for training of staff [14],[38], in one of the most recent studies reviewed, hospital staff themselves identified their lack of knowledge and need for education about intellectual disability [33]. Worryingly, results by Ali, Scior, Ratti, Strydom, King and Hassiotis [41] suggest that people with intellectual disability from certain cultural backgrounds may be particularly at risk of discrimination. Their participants with intellectual disability suggested that their experience of lack of care was no different to that of other patients, a potential indictment on the hospital system in general.

There was also evidence supporting Mencap's [15] concern that published recommendations to support hospitals in caring for people with intellectual disabilities have not been implemented. Phillips [29], for example, located only one study evaluating LDLN services. These nurses provide specialist knowledge in intellectual disability within general hospital settings. Evaluations suggest the valuable role they play in promoting and supporting effective co-ordination of care from admission through to discharge, and in assisting hospital staff to make reasonable adjustments to meet patient needs [22],[33]. The implementation of this or a similar service no doubt demonstrates a hospital’s commitment to care, also demonstrated through relevant policies and procedures, suggested by Webber, Bowers and Bigby [20] to contribute to quality care. Sustained initiatives, such as LDLN services, and policies that lead to effective strategies may rely on understanding hospital systems and resourcing. There is limited evidence of capacity to provide the types of accommodations sought in policy [15],[25], and these factors appear to be missing from both health and disability service systems.

Over the previous two decades, evidence of poor quality hospital care for people with intellectual disability has been mounting. Most of this evidence is qualitative in nature and of variable quality. Despite problems with the evidence base, and arguments for more research into hospital experiences and access to health care by people with intellectual disability, we have argued for a more expanded research focus. There is a need to explore specific points of encounter along the patient journey, and also extend understanding of the patient experience to other vulnerable groups, such as people with TBI. Such research, conducted in conjunction with exploration of hospital systems and processes, offers the potential to ensure quality of care for people with diverse needs. A further potential outcome of this expanded focus is to provide direct resource benefits to hospitals by identifying strategies that can be adopted readily, and reduce unnecessary and repeat presentations, and associated costs.

^aHospital episodes that end with discharge, transfer to another service, or death.

^bCopies of the appraisal tools are included in Additional file 2.

TI contributed to the initial concept and design, and determination of inclusion criteria, evaluated each study to determine if it met the inclusion criteria, summarised the articles, conducted quality appraisals, drafted the initial manuscript and conducted follow-up revisions; CB contributed to the initial concept and design, and determination of inclusion criteria, evaluated articles when there was disagreement between other authors, and contributed to the initial drafts of the paper and follow-up revisions; CU contributed to the initial concept and design, and determination of inclusion criteria, selected and modified the appraisal tools, evaluated articles to determine if they met the inclusion criteria, conducted quality appraisals, and contributed to revisions of the manuscript; JD contributed to the initial concept and design, and determination of inclusion criteria, evaluated articles to determine if they met the inclusion criteria, and contributed to the drafting of the manuscript; PF conducted the literature searches, obtained the articles, participated in evaluating the articles for inclusion, assisted with summarising the articles, and conducted quality appraisals. All authors read and approved the final manuscript.