The reported effects of the COVID-19 pandemic on people with intellectual disability and their carers: a scoping review

Abstract Background People with an intellectual disability are a vulnerable group during COVID-19 due to multi-morbidity, frailty, underlying conditions/health problems, social circumstances and limitations in understanding. This place them at greater risk of more severe outcomes from COVID-19. Objective To chart the evidence of the effects of the COVID-19 pandemic on people with intellectual disability and their carers reported in the research. Methods A scoping review method was used to review literature published across eight databases in 2020 and included manual searches of reference lists of included articles. Results Sixteen studies were selected for inclusion. The findings highlight that professionals, people with intellectual disability and their carers were keen to comply with pandemic related public health guidance. However, implementing infection prevention and control measures in certain contexts for people with intellectual disability was challenging. Thereby a rights-based, person-centred approach to care is essential and professionals need to extend their understanding and practice of collaborative working to include people with intellectual disability and their family/carers. The findings highlight that compliance with public health guidance and infection control measures can be difficult for people with intellectual disability to implement. Conclusions There is limited good quality robust research on COVID-19 and people with intellectual disability and/or their carers experiences.


Introduction
A pandemic is a worldwide epidemic in which a disease spreads easily and rapidly [1] and while rare they can be deadly. Pandemics occur when a virus exists and is easily spread from person to person in a population with little or no natural immunity [2]. On the 11th of March 2020, the WHO acknowledged the novel human coronavirus (COVID-19) as a pandemic and cases continued to rise globally. Throughout history, pandemics have occurred such as: Spanish flu (1918)(1919)(1920), Asian flu (1957)(1958)  . However, the present pandemic COVID-19 has already proved to be a formidable foe [2].
People with multi-morbidity, frailty and underlying conditions are at increased risk of COVID-19 [3]. Public health response focussed on minimizing human-to-human spread to decrease secondary infections and infection control measures such as evading close contact with people with respiratory symptoms, engaging in good hand hygiene, cough etiquette practices [4]. In addition Governments implemented a variety of actions, including closure of non-essential services, travel limitations, cocooning and self-isolation to minimize interactions. COVID-19 is a serious health hazard to people across the world as it spreads rapidly [5] and up to 11th March 2021 there has been, 117,160,237 cases and 2,598,892 deaths reported [6]. People with intellectual disability are a susceptible group due to their need for support services and personnel [7] and are at increased risk of serious illness associated with COVID-19 due to health problems, social circumstances and limitations in understanding [8]. Challenges include access to understandable information about the disease; minimising the risk of infection; the risk of supports ceasing and the risk of increased anxiety, distress and behaviours that challenge [9]. The rapid changing environment and support structures can be difficult to accept as changes can result in distress due to confusion and disruption in their daily lives. Furthermore, carers often depend on support services for assistance and respite and the change to service provision affects carers well-being.
Public health actions to avoid transmission can increase the risk of loneliness and social isolation, which are well-recognized risk factors for physical and mental illness [10]. Attaining an understanding of the experiences of people with intellectual disability and their carers during COVID-19 is vital as both infectious diseases and actions to deal with them (e.g. quarantine and self-isolation) pose unique threats to their health and well-being [11]. This knowledge is essential if clear resources and supports are to be identified to manage such crisis in the future and to support people with intellectual disability and/or their carers, as often their voices are notably absent from research and discussions. Thus, this paper aims to review the current literature and identify key aspects relevant to managing COVID-19 to guide responsive and appropriate services in the future.

Methods
A scoping review was selected as it allows for the evidence on a topic to be mapped, an examination of practice, policy and research, and the identification of gaps in evidence and policy [12]. Scoping reviews discover the nature and extend of the available evidence and plot the evidence by categorising the components of the literature, such as study design, sample, setting, intervention, underlying knowledge framework, aspects of relevance and findings. This creates a picture of the scope and significance of the literature and are used to determine literature on a specified area, recognise key thoughts, identify gaps in current literature and publicise outcomes [13]. The scoping review utilised Arksey and Malley [14] five-step process: (a) identifying the research question, (b) identifying relevant studies, (c) study choice, (d) plotting the data, and (e) arranging, summarizing, and communicating the outcomes. While these five steps are identified as a sequenced approach, it is in fact an interactive process where each step were revisited and progressed throughout the review. Results are expressed by way of a narrative in conjunction with tables and diagram illustrations [15].

Identification of the research question
To meet the aim of this review, the authors addressed the following questions: (a) What effects of the COVID-19 pandemic on people with intellectual disabilities and their carers are reported? (b) What responses have been directed towards people with intellectual disability and their carers? and (c) What recommendations have been made regarding people with intellectual disability and their carers?

Identification of relevant studies
To identify a breadth of literature a wide ranging set of keywords for search terms searching were adopted. Processes for searching integrated subject headings and Boolean operators were also used to expand and merge searches. Searches were completed using the Title OR Abstract and Keyword options, with individual searching of each search string and subsequent combination of all strings (Table 1). Inclusion and exclusion criteria were developed and applied (  Papers that do not addresses information pertaining to coronavirus. Papers where it is not possible to extract data focussing on persons with intellectual disability and/or their carers. Non-English language paper. Non-primary research such as reports, working papers/protocols, conference abstracts, government and non-governmental organisations documents, opinion papers, letters to the editor, discussion papers, correspondence papers

Study selection
Electronic database searches yielded 268 papers which were exported to Endnote and duplicates removed leaving 129 papers remaining. Following screening of title and abstracts guided by the inclusion and exclusion criteria 99 papers were excluded. 30 papers went forward to full text review and were assessed and agreed by the authors with 14 excluded and 16 included in the review. This review was limited to peer review research papers in 2020 in order to scope the evidence of the effects, responses and recommendations regarding COVID-19 and people with intellectual disability and their carers. All studies that met the inclusion criteria were included in this review as the authors wanted to capture all studies on COVID-19 within the intellectual disability population published in 2020. Furthermore, as critical appraisal and risk of bias are a choice in scoping reviews [16], the selection and reporting process followed [17] Preferred Reporting Items for Scoping Reviews (PRISMA-Sc-R) and PRISMA flow diagram [18] (Figure 1).

Plotting the data
This scoping review aims to map the existing literature in terms of volume, nature, characteristics, and sources of evidence [19]. Plotting the data involved charting and extracting summaries from each paper and details of the extracted data are included in Table 3. Of the 16 included papers four were electronic surveys, four descriptive qualitative studies, three descriptive quantitative studies, two observational studies, one case series, one clinical study and one quasi experimental design.
Arranging, summarizing, and communicating the outcomes The final stage summarizes and communicates the findings. This scoping review generated 16 papers across seven countries (United States n ¼ 5,   It is essential that healthcare organisations emphasise the collective approach and support for direct support staff to overcome the sense of lone responsible and been in it on one's own.
The small sample size in the study raises the question of whether the current findings can be generalised. (continued)

What effects of the COVID-19 pandemic on people with intellectual disabilities and their carers are reported?
This review highlights that people with an intellectual disability and their carers are especially vulnerable to the physical, mental, and social effects of the pandemic. Such challenges include higher rates of mortality; more severe health outcomes; substantial reduction in the availability and access to face-to-face services; minimizing the risk of infection; risk of increased mental illness, such as loneliness, agitation, anxiety, distress and in some cases increased challenging behaviour. From the perspective of people with intellectual disability there is evidence that there is a higher risk for those residing in residential group homes [24], of developing more severe outcomes from COVID-19 [31], higher co-morbidities [30,31] and much higher mortality [23,24], particularly among adults under 75 years of age [31] as compared to the general population. Diseases of the circulatory and respiratory system were high along with endocrine, nutritional and metabolic disorders [31]. The main COVID-19 symptoms included upper respiratory infection, diarrhoea, fatigue, fever and respiratory signs [28]. The most common underlying physical health conditions were diagnosis of epilepsy, dysphagia, dementia, hypertension, asthma and diabetes [29]. However [25] identifies that while those receiving intellectual disability services had a lower-case rate, they had a higher case-fatality rate of COVID-19. As a result of COVID-19, there was a loss of structure and daily routine [22] creating challenges around being stuck at home and unable to leave the house, finding activities and preventing boredom [27]. However, some benefits were reported such as, having more time together as a family, enjoyed the slower pace of life, gaining more sleep, learned to be more patient and communities coming together to support each other [27]. A reduction in service and societal demands has also led to some people with an intellectual disability being much more settled and content in their lives [33]. From a family perspective, parents greatest concern was their family's health, not getting COVID-19 and dealing with their child's behaviour problems [27]. Financial concerns, managing significant challenges related to their child's services decreasing or ceasing and feeling like they could not meet their child's needs at home impacted on their mental well-being [27]. Of note was that families caring for children with intellectual disability reported significantly greater anxiety, depression, defeat/entrapment and wish fulfilment [33] and their mental health was positively related to behavioural changes [20]. Additional family demands, a loss of routine, educational and therapeutic supports and the introduction of new stressors [22,27] all resulting in a decreased capacity to cope and increased isolation [35]. On the other hand, staff reported moderate levels of personal and work-related burnout and mild levels of anxiety and depression [26]. Staff working in independent living settings and individuals with challenging behaviour report highest level anxiety, and this was related to incidents with aggression [30,32]. Of note was that staff in home care felt least supported by their employer [26] and in residential care settings a drop in staff medication errors from pre-COVID-19 levels was noted [30]. Furthermore, from a staff perspective, they identified that Covid-19 had an impact across all domains of their life at an emotional level there is a fear of infection, a sense of frustration, disappointment and responsibility that can lead to a sense of been overwhelmed by emotions [21,35]. At a cognitive level staff were challenged by COVID-19 in terms of coping skills, reflection, problem-solving skills, attitudes and perseverance [35]. From a practical perspective, preventive measures-imposed limitation for people with intellectual disability and resulted in staff experiencing; time pressure, a lack of face-to-face contact and a demand to be able to use and access digital technologies [35]. Furthermore, from a professional perspective, there was a loss of connectedness with other professional and support staff [35]. These factors all increased the sense of isolation, exacerbated the sense of anxiety about COVID-19 and added to the difficulties in understanding and applying the new rules [21].
At a service level COVID-19 had quite an impact on service delivery resulting in many of them closing, reducing service access and/or going online and providing one to one home support [22,27]. Services reported dealing with a higher number of unplanned online contacts per day during COVID-19, indicating that people were considerably worried and experienced a high level of anxiety especially during the first weeks of the crisis [34]. Within some residential services, incident reports reduced overall as a result of lockdown measures, however incidents of aggression increased [30]. This review highlights a paucity of data on COVID-19 trends among the intellectual disability population, variations and limitations in recording processes utilised and little evidence of interventions utilised to support people with intellectual disability and their families [23,24,28,29,31].

What responses have been directed towards people with intellectual disability and their carers?
The COVID-19 pandemic has affected all aspects of the intellectual disability community and particularly posed an increased risk for individuals living in congregated care settings due to the challenges these types of residence present regarding physical distancing [24]. A COVID-19 unit was one clear measure taken to limit the spread of the virus [28]. This response was a collaboration between staff, service providers and infectologists. However, for those in the community there was generally a lack of accessible information on the virus and the containment measures leading to a demand for support addressed via online support [21,34]. As quarantine limited access to education, rehabilitation, training, or intervention and treatment services, behavioural regression were evident [20]. Leading to greater use of adaptive (problem-solving) coping strategies rather than maladaptive (wish fulfilment) coping strategies [35]. In addition, from a staff perspective greater compliance with health care protocols and more structured day routines, lead to a decrease in medication errors [30].
Evident with the findings of this review was that there is a paucity of data on COVID-19 trends among the intellectual disability population which exposes the reality that there is no adequate surveillance structure in place to monitor COVID-19, or other public health outcomes, among the intellectual disability population [31]. In the absence of such data there is a risk of a potential under-reporting of cases and related deaths and measures targeted at the general population should not be assumed to be sufficient for the intellectual disability population [29]. Thereby medical practitioners should be aware of the increased risk for individuals with intellectual disability, even mild symptomatology, and need to accurately code deaths of those who die from COVID-19 [24]. Adverse COVID-19 outcomes were associated with the level of skilled nursing care, where low levels of skilled care resulted in higher levels of adverse effects [25].
Response of external agencies such as schools and community teams to providing continued support, albeit remotely is mixed with some agencies attempting to provide continued support but others providing little if any assistance. Those that needed to access healthcare providers did so primarily through telemedicine [22]. Telehealth (both tele-education and telemedicine) were found to be helpful, and often there was a need to augment these with 1:1 videoconference session [22]. Other supports included outdoor conversations, social media, provision of supplies and contact with professionals [33]. Telehealth supported the delivery of care and education in a sustainable way, during and post restrictions [22]. Within the literature, the sources rated as most helpful were partner/spouse followed by professionals; however, carers of adults with mild challenging behaviour were the least supported [33]. Social isolation measures affect the functional and psychosocial well-being of people with intellectual disability and lockdown should be considered a potentially traumatic life stressor event [32]. Given that COVID-19 restrictions have greatly affected access to services for individuals with intellectual disabilities, [22] the provision and use of online support has been a key service development [34].

What recommendations have been made regarding people with intellectual disability and their carers?
The literature reviewed clearly highlights that healthcare professionals need to be aware that people with intellectual disability are a high-risk population [23,25] and should be aware of the potential for severe outcomes [31]. This risk is also further increased for individuals living in congregate care settings due to the challenges these types of residence present for physical distancing [24]. All family members, care workers, and people with intellectual disability need to take extra precautions to ensure their health and safety during the COVID-19 [24]. Service leaders to recognise the need for extra supports such as social stories to explain the wearing of PPE [25] and the use of technology to provide virtual visiting and telemedicine systems.
Given the paucity of data on COVID-19 regarding the intellectual disability population, there is a need for surveillance structure to be put in place to monitor COVID-19 and other health outcomes [31]. This has implications for policy and practice such as the provision of clear information from healthcare organizations and COVID-19 crisis teams [35]. Such an approach requires public health surveillance systems to include disability status as a basic demographic characteristic [24]. People with intellectual disability should be included on the list of groups who are more vulnerable to mortality associated with COVID-19 [23] and there is a need for more accessible information for people with an intellectual disability such as easy read and accessible websites with up-to-date information about COVID-19 [21]. Furthermore, there is a need for risk reduction strategies to address increased risk associated with COVID-19 for people with intellectual disability [23].
Given the unpredictable and uncertain future the consequences of the disruption to all services and day-to-day life and loss of structure and routine raises anxiety for people with intellectual disability. There is a clear need for improving psychological support and identifying psychological intervention with a set of activities that can be safely implemented [24]. There is a need to consider the impact of the COVID-19 experience on the mental health and overall well-being of people with intellectual disability [33] and thus explore the specific impact of COVID-19 on people with intellectual disability and reduce their disproportionate risk [24]. In addition, consideration needs to be given as to how people with intellectual disability can be supported to work or be educated from home given the potential for additional waves of the virus [21].
The important role of families and carers [24] needs to be recognized and policies regarding family/visitor access need to be addressed, as restrictions over an extended period have a range of negative impacts on the family [27]. Families concerns regarding a lack of educational and developmental progress and emotional concerns need to be considered [27]. For staff, a collective approach to support direct support staff to overcome the sense of lone responsibility is required [35]. Moreover, employers need to consider staff wellbeing, given the levels of personal and work-related burnout, and anxiety and depression [26] and research should focus on proactive strategies for improving staff well-being in the short term, given the current resurgence of COVID-19 [26].

Discussion
This review and the wider literature highlights that people with intellectual disability are at increased risk and vulnerable to infection, heightened if the person resides in a congregate setting [10,[36][37][38]. In addition, it is difficult to ensure adequate physical, or social, distance as they often depend on physical contact with a professional or career for their daily activities and care needs and find it difficult to understand the new restrictions [39,40]. The greater the severity of disability the greater the need for support and thus the greater the risk. These factors increase the possibility of transmission of infection leading to complications such as pneumonia, acute respiratory distress and organ dysfunction [41]. Consequently, people with intellectual disability are likely to experience more severe cases [31] and as a result have poorer outcomes [42] resulting in death [43]. Given the increased likelihood of infection and comorbidities among people with intellectual disability they are likely to suffer more serious clinical outcomes [40] and experience higher fatality rates [44,45] which is double that of the general population [31]. One could expect these greater risks given the pre-existing health disparities faced by people with intellectual disability prior to the pandemic, including poorer health outcomes [46], limited access to health care services [47], participation in fewer prevention and health promotion activities [48], increased risk for chronic health conditions [46] and earlier age of death when compared to the general population [49]. However, despite the increased risk and health disparities, only 2% of people with intellectual have been reported as having a COVID-19 test [50].
Of concern within this review is the lack of surveillance or data collection systems which are needed to assure the right types of health protection actions (prevention, treatment, and mitigation). Populationbased registries have an important role in determining the prevalence, aetiology, distribution, frequency and severity of a particular condition/disability in a national or international region. However, there is little if any population-based data on people with intellectual disability and there needs to be an identification of people with intellectual disability in a systematic manner. Without the capacity to identify people with intellectual disability in emergency management surveillance systems, they may be overlooked when interventions are planned and evaluated. While this review highlights that infection control measures in healthcare facilities have managed the pandemic well there is little known about infection control knowledge, attitudes, and practices among staff [51]. This is essential as care environments have special infection control needs as they act as both the individual's permanent home and place where medical care is provided [52]. Evident within this review is that although there has been an increase in COVID-19 publications in intellectual disability there is an absence of the voice of the person with intellectual disability in such publications [53]. This absence needs to be considered as we look to the long-term impact and implications of Covid-19 regarding public health guidance, isolation, cocooning, withdrawal of services, family stress on individual's psychological, mental and physical well-being. This absence may currently be addressed and newer publications may have this as their focus such as [54] or nested within a study. Furthermore, as this review only covered the year 2020, there is no information on vaccination and monitoring of vaccination uptake is required. It is important that the feelings, experiences, awareness and the unique concerns of people with intellectual disability and their families should be prioritised and not overlooked, forgotten or neglected [55,56]. Furthermore, research is needed on strategies of improving the health protection measures for people with intellectual disability during the outbreak [57] and identify resilience measures [58].
This review highlights the psychological and social consequences of the pandemic for people with intellectual disability who have stopped participating in their communities and have had their routines interrupted during lockdown/restrictions for a prolonged period [59,60,61] and access to education, health, vocation and mental health service disrupted [50,62]. In addition, many have found it difficult to understand COVID-19 and self-regulate their behaviour, creating a risk of behaviours that challenge which negatively affect the emotional well-being and quality-of-life of the person [63,64]. One could argue further segregation is occurring not just by the wider society or generic health services, but also in terms of service provision and this intensifies the sense of isolation. Thus, services and society need to strike a balance between humanity and restrictions, as for many, proximity to skilled caregivers and loved ones is required to bridge gaps in intellectual and communication abilities and to make day-to-day life fulfilling and manageable. This humanity and rights debate is further emphasised in healthcare decision-making, where there are strong ethical justifications for robust triage systems when capacity and resources to provide critical care to patients are overwhelmed [65]. However, there are also difficult ethical dilemmas and decisions as to which COVID-19 patients should go on life-saving treatment because of the shortage of medical equipment [66] and accounts of using disability as a factor in denying life-saving treatment are discriminatory [67]. This raises serious concerns regarding disability and human rights [68] and some reports highlight vulnerable populations, including people living with disabilities, being spoken about as if they were disposable or expendable [69].
This review highlights the essential and important role families play in the support, health and well-being of individuals with intellectual disability [70]. With public health guidelines emphasising maintaining a 2meter distance to fight the virus, families face growing challenges that may be impossible to meet [71], and it may be unreasonable to expect a person with intellectual disability in care to cope when separated from their family [72]. Families caring in their own home are experiencing higher levels of concerns and anxiety [33,73,74] which is fuelled by a loss of routine, educational and therapeutic supports and the introduction of new stressors [27]. These all result in a decreased capacity to cope and increased isolation for the family [35,75] and despite some proactive approaches to use alternative communication means to provide support services they may not be effective in filling the gap created by reduced face-to-face contact [76]. In addition, electronic service provision needs to be evaluated and while they have shown benefits and effectiveness, implementation and integration within health systems, and user satisfaction [77,78] concerns have also been raised regarding screen time and the long-term impact of service reduction [27]. While telehealth/electronic service can show positive outcomes [79], there are issues with access, accessibility and support for usage [80,81]. As we face into 2021 and with the advent of a vaccine the question for people with intellectual disability and their families is, will they be identified as essential and prioritised for vaccination? A key issue for families is that if they as a direct family carer fall ill, can the health system be inclusive of the needs of individuals with intellectual disability [82] and provide a substitute caregiver, who is aware of the needs of the person and can this be put in place until recovery of the usual caregiver [83].

Limitations
As the focus of this review was to map the evidence, no evaluation of the quality of evidence/quality appraisal were performed and this paper only provides an explanatory account of available information. This review only included papers published in English and this may have resulted in the exclusion of relevant papers. In addition, the review only covers the year 2020 and we acknowledge that the evidence regarding COVID-19 grows daily. However, within the review, the authors engaged with relevant papers post 2020 for inclusion within the discussion. Furthermore, this review has to be considered within the limitations identified within the studies that met this paper's inclusion criteria mainly the fact that (1) most data collection took place very early in the pandemic and further data is required to identify the long-term impact of Covid-19, (2) samples were small in size and not representative of the full nature of the intellectual disability population, (3) data collection utilised selfreporting measures and were administered mainly online where there may be limited issues with access, connectivity and literacy and (4) the data collection tools used were not validated, inferential analysis was not conducted and no pre-COVID-19 measures were available to compare against. These factors all affect the reliability, validity and generalizable of the studies in this review and further analysis as to whether these aspects are addressed in 2021 studies is warranted.

Conclusion
This review highlights that the peer-reviewed press is dominated by professional opinion pieces, editorials and letters to the editors presenting various personal as well as professional polemics, some of which are supported with reference to the literature, as well as anecdotal evidence. However, there is limited research about pandemics/epidemics and people with intellectual disability. Rarely is space provided for population studies or the rich perspectives of the experiences of the person with an intellectual disability. In saying that, the research that does exist clearly highlights that core to the management of pandemics and epidemics are 1. A rights-based, person-centred approach to care is essential as professional and organisational approaches, as well as ubiquitous strategies often fail or fall short; 2. Professionals need to extend their understanding and practice of collaborative working to include people with an intellectual disability and their family/cares, not just begin to include other professional colleagues; 3. High levels of concordance with public health guidance and 4. Rigorous implementation of quality infection control measures are essential prerequisites.

Ethical approval
No ethical approval was necessary for the conduct of this review.

Author contributions
All authors meet the criteria by making a substantial contribution to the conception, design, analysis and interpretation of literature. All entitled to authorship are listed and given permission for the final approval of the version to be submitted and no other authors were involved with this paper.

Disclosure statement
No potential conflict of interest was reported by the author(s).

Data availability statement
No data are available other than that reported in this review and available in original published papers used in this review.