Exploring the

perspectives of current
and future sibling
caregivers to a person
with a
neurodevelopmental
disorder – implications for
occupational therapy.
A scoping review.

PAPER WITHIN Occupational Therapy

AUTHOR: Kayleigh Tracy Tudor
SUPERVISOR: Frida Lygnegård
EXAMINER: Petra Wagman
JÖNKÖPING September 2023
Exploring the perspectives of current and future

sibling caregivers to a person with

neurodevelopmental disorder – implications for

occupational therapy.

A scoping review.

Author: Kayleigh Tracy Tudor

E-mail: kayleightracytudor@gmail.com

Acknowledgement

This review is part of the author’s postgraduate Master of Science with a Major in

Occupational Therapy at Jönköping University, Sweden. No funding was received for this

review. The completion of this thesis would not have been possible without the

encouragement of my supervisor, Frida Lygnegård and the continued support of examiner

Petra Wagman.

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Exploring the perspectives of current and future sibling caregivers

to a person with neurodevelopmental disorder – implications for

occupational therapy. A scoping review.

Abstract

Introduction: Numerous studies have examined caregiver burden and stress, yet

few represent the perspective from the sibling caregiver of a person with a

neurodevelopmental disorder within the context of occupational therapy

practice. Therefore, this scoping review provides a review of current evidence.

Aim: The aim of the study is to review current literature to find sibling caregiver

perspectives on their roles as caregivers to an individual with a neurodevelopmental

disorder and whether their perspectives can influence improvements to the

occupational therapy practice.

Method: A scoping review methodology was chosen to map out the available

literature and summarise the findings. The author conducted this review using

guidance from the Joanna Briggs Institute.

Results: The review resulted in 11 articles and reviewed the perspectives of 1181

sibling participants. The findings resulted in five main themes: (1) Need for

education and training opportunities in relation to caregiving; (2) Policy and

guideline changes in relation to sibling caregiver involvement; (3) Sibling caregiver

inclusion into the care pathway; (4) Need for improvements in service

coordination; and (5) Sibling caregivers' lack of knowledge in guardianship

matters.
 Conclusions: Both positive experiences and negative challenges were reported

from sibling experiences when accessing healthcare services. Ideas and

strategies were discussed by sibling caregivers in how to improve services.

Significance: This review identified practical strategies which can be used in

occupational therapy practice to support and improve services in relation to

working with sibling caregivers.

Keywords

Adults; Brothers; Intellectual Disability; Learning Disability; Occupational Therapy; Sisters.

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List of abbreviations

Activities of daily living – ADLs

Evidence-based practice – EBP

Neurodevelopmental disorder – NDD

Occupational therapy – OT

Occupational therapists’ – OTs

Exploring the perspectives of current and future sibling caregivers

to a person with neurodevelopmental disorder – implications for

occupational therapy. A scoping review.

Introduction

The World Health Organization (WHO) estimates that 349 million people worldwide are

dependent on caregivers for their care needs (WHO, 2017). Caregivers are defined as an

individual who cares for and/or assists a disabled, ill, or frail family member, spouse or friend

(Moghimi, 2007). Growing evidence indicates that the number of caregivers is formidably

increasing and the Center for Disease Control and Prevention (CDC) states that the effect on

caregivers’ quality of life is a public health issue (WHO, 2022; CDC, 2018). Estimating the

actual number of caregivers worldwide is difficult, although, the International Alliance of

Carer Organizations (IACO) estimates that there are already over 22 million caregivers across

Europe alone (IACO, 2021). The Europe Quality of Life Survey estimates that 20% of its

population are formal carers and as much as 80% of all long-term care is provided by

informal carers - between 10% to 25% of the population (European Commission, 2018).

Totsika, Hastings, and Vagenas (2017) estimate that in future years, social care providers will

increasingly rely upon informal caregivers further to provide care to people with intellectual

disabilities. As advances in healthcare have increased the life expectancy for adults with

neurodevelopmental disabilities (NDD), the possibility of these adults living longer than their

parents increases and so does the need for families to plan for long term care (Lindahl et al.

2019) and when parents are no longer able to manage the caregiving role, siblings become the

next generation to undertake the role (Heller and Kramer, 2009).

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However, the role of siblings as caregivers has not been well documented within existing

research (Namkung, Greenberg and Malick, 2017). Arnold, Heller, and Kramer (2012) state

how siblings are often overlooked by parents, health professionals and researchers as

knowledgeable advocates for their disabled siblings - yet are often the next generation of

caregivers. Scorgie (2017) concurs, affirming that within disability research, studies tend to

address the effects of caregiving from a parental view, mainly the mother’s perspective,

leaving other family members such as siblings, underrepresented. Moghimi (2007) states that

a patient’s rehabilitation may be impacted by the burden and stress experienced by a

caregiver and should be an area addressed by healthcare providers.

The American Diagnostic and Statistical Manual of Mental Disorders 5th Edition (DSM-5)

defines NDD as a group of conditions which typically manifest early with onset in the

developmental period. These are a group of conditions which a categorised by developmental

deficits which result in impairments in personal, social, academic or occupational

functioning. A range of conditions which may include for example, intellectual disabilities,

learning disabilities, autism, attention deficit hyperactivity disorder (ADHD), global

development delay, communication disorders, tic disorder, motor disorder such as

developmental coordination disorders and other unspecified disorders (American Psychiatric

Association, 2013).

People diagnosed with an NDD condition can present with a range of impairments which

differ in both type and severity, meaning the levels of support required to manage activities of

daily living (ADLs) can also vary (Bathje et al. 2018). Occupational Therapists (OTs) work

with people with NDDs and their families by using a person’s daily occupations and activities

to develop, recover or maintain skills needed for daily living needed through meeting specific

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goals. The American Occupational Therapy Practice Framework: Domain and Process

(Framework) states that ADLs are activities that are required for engagement in home,

community, financial and health matters (American Occupational Therapy Association,

2020).

Lindahl et al (2019) highlights challenges faced by both parental and sibling caregivers,

including that caregivers feel anxiety around the long-term care plans for their adult relatives

with an intellectual disability. The authors also highlighted that caregivers are likely to be

socially and economically disadvantaged and at a greater risk to develop serious health

issues. Moghimi (2007) highlights how occupation is central to a person’s identity and

competence and many caregivers usually have a variety of occupations fulfilling their lives –

with caregiver role often not being an occupation they have chosen. Also, that caregiver

burden is often due to the lack of caregiver training from healthcare professionals, as the

primary focus for the healthcare professional is the patient, not the caregiver.

The Royal College of Occupational Therapists (2021) have stated that further research is

needed to address how OTs can work more effectively with the family and carers of people

who access services. Therefore, it is important to consider the perspective of the sibling

caregiver, to understand the challenges faced within healthcare services to be able to improve

effective working between healthcare professionals and the sibling caregiver.

Theoretical background

As stated, the primary focus for the healthcare professional is foremost, the patient, not the

caregiver. Yet, occupational therapy (OT) theory is ideally placed to support both the patient

and the caregiver. Whilst traditional models, such as the biomedical model have focused on

physical factors to aid medical intervention to cure disease or disorder, OT models have

4
shifted their focus to occupational engagement as an intervention to aid recovery. Thus,

historically causing shifts between the biomedical model and holistic occupation-based

approaches in occupational theory (Wong, Fagan and Leland, 2018).

OT conceptual models can be used with all client groups and there are no specific

recommendations for certain models to be used with a person with an NDD or their caregiver.

Therefore, this paper will explore two occupation-focused models, the Model of Human

Occupation (MOHO), the Canadian Model of Occupational Performance and Engagement

(CMOP-E) and their applicability with supporting caregivers specifically.

MOHO assesses how people generate and modify their occupations within four components:

volition, habituation, occupational performance, and environment in a dynamic open-cycle

system of human actions. For example, people are motivated to perform occupations

(volition), repeat their performance over time (habituation) and consider their perception of

their ability to perform that occupation (occupational performance). This process of events is

performed with a social and physical context (environment) (Taylor, 2017).

The CMOP-E includes three main components: the person, occupation, and the environment.

The person is the core of the model, and is made up of four aspects, spirituality, affective,

cognitive, and physical. Occupation includes the domains of self-care, productivity, and

leisure, and these are performed in the context of the environment, which is depicted by the

domains of physical, social, cultural, and institutional environment. Within the CMOP-E

model, the person and the environment are connected through their occupations. The

interaction between these three components results in occupational engagement (Townsend

and Polatajko, 2013). When the three components work together, this results in function,

dysfunction occurs when there is a disruption to one of the three components (Wong and

Fisher, 2015).

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There is no current evidence applying the models in the context of directly supporting the

sibling caregiver as the primary focus within either model. As therapeutic models focus on

the ‘client’ specifically, caregivers generally fall into the category of the ‘environment’ for

example, in the CMOP-E - social, and in MOHO – people, which include caregivers as input

and feedback from the care recipient’s environment (Demers, 2022). MOHO primarily

focuses on the client, yet the CMOP-E provides guidelines for using the model with

organisations and communities, which could be explored further in context of the caregiver

support with organisations and communities (Wong and Fisher, 2015).

Restall and Egan (2021) state that this is a problem in relation to the terminology used by

therapists in relation to ‘client-centredness’. The authors state that ‘client-centredness’ has

been defined previously to include individuals, families, groups, communities, and

populations. Yet, the authors argue that therapists tend to only utilise models with the

perspective of the ‘client’ as the individual, not utilising models with larger groups of people.

The authors propose a shift in the therapist’s thinking, away from the single, client-centred

approach to occupational participation being the central aspect of the model, thus proposing

the caregiver can be both the care-recipient and the caregiver.

Aim

The aim of the study is to review current literature to find current and future caregiver

perspectives on their roles as caregivers to an individual with a NDD and whether their

perspectives of healthcare services can influence improvements to the OT practice.

Method

A scoping review methodology was undertaken. This type of methodology allowed the author

to search and summarise themes within the existing literature and highlight potential gaps

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across the current research, allowing for recommendations to be made for future practice

(Arksey and O’Malley, 2005). This study has utilised the proposed framework from the Joanna

Briggs Institute (JBI) scoping review methodology provided by Peters et al. (2020).

Eligibility Criteria

Inclusion criteria included (1) adult (aged 18+) current and future sibling caregivers, with an

adult brother or sister diagnosed with a neurodevelopmental disability; (2) Literature which

focuses primarily on experiences of adult siblings caring for people with neurodevelopmental

disorders, living with sibling caregivers or in social service supported homes; (3) OT-based

intervention/support or related interventions given by another healthcare professional; (4)

Literature published in English language from 2010 to 2021; (5) Literature with abstracts

available; and (6) The search included quantitative, qualitative, and mixed method study

designs.

Exclusion criteria includes articles that reference (1) child sibling interventions and

perspectives; (2) a sibling diagnosis that is not an NDD; (3) literature including interventions

with the mother/father or other relatives and their perspectives; (4) literature that focuses on

role identity of siblings within a family context; and (5) interventions from an educational or

medical perspective.

Search

Sources of literature included published and unpublished data available via electronic

databases, reference lists and grey literature. A systematic search of the literature was

conducted throughout March 2022. The author performed the search via five primary

electronic databases: CINAHL, Medline, AMED, PubMed and PsychINFO and two

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secondary databases: Cochrane Library and OT Seeker (Table 1). The author employed a

variety of search strings which were revised with the assistance of a university librarian.

Similar search strings were used across the databases, although were refined dependent on the

structure of the database's search parameters. Initially the search term “neurodevelopmental

disorders” was utilised to incorporate a broad spectrum of disorders, yet due to the

terminology used across literature internationally, the terms “intellectual disability” and

“learning disability” were used alongside “neurodevelopmental disorders” to increase the

search field and number of hits. Initial searches including the terms “occupational therapy or

OT” produced too narrow results and no relevant evidence. Therefore, this search term was

removed to broaden the results and articles were selected via their possible relevancy to OT.

The search strings also employed a variation of Boolean operators and truncations.

[Insert Table 1]

An electronic search of specific journals was conducted in four journals: The British Journal

of Occupational Therapy; Journal of Autism and Developmental Disorders; Research in

Developmental Disabilities and Intellectual and Developmental Disabilities. The author

limited the search to these journals as the author believed these may give the most relevant

information required to answer the research question. A similar search string was used during

the journal search, dependent on the search parameters of the journal (Table 2). Eventually,

the search string had to be reduced and generalised to “adult sibling or sibling AND

caregiving” (using truncations) as the search field was too narrow to produce relevant results.

[Insert Table 2]

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A scoping study method aims to identify all existing literature regardless of the study design

(Arksey and O’Malley, 2005). Therefore, the author performed a Google search to find

potentially relevant authorities, organisations, and key stakeholders relevant to the topic

which could provide further evidence related to the study categorised as grey literature. This

resulted in the author reviewing information from authorities such as the UK Department of

Health and Social care, National Institute for Healthcare and Excellence (NICE) clinical

guidelines and charities such as the Sibling and Leadership Network and Sibs, a UK-based

charity. The author also conducted searches of 3 search engines, Google Scholar, OAlster and

the Beilefeld Academic Search Engine (BASE).

Study selection

The concluding search of the electronic databases resulted in a total of 235 articles, the articles

were then exported to EndNote library and some articles were removed as duplicates. Articles

were then screened via title and abstract and all literature was screened against both the

inclusion and exclusion criteria when selecting literature to be included into the review. The

articles retrieved from the journal search were also screened via their title and abstracts and

compared against the inclusion and exclusion criteria. When completing the search of the grey

literature they author chose to use information from within the past 5 years as the quality and

production of grey literature can vary (Paez, 2017). A full-text assessment of 52 potentially

relevant combined articles and grey literature pieces were assessed for eligibility by the author.

From the full-text assessment, 11 pieces of literature were included in the final selection. The

full screening process is depicted in a PRISMA flow diagram (Figure 1).

[Insert Figure 1]

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Data charting

The final 11 studies were reviewed, and key information was extracted into a “Data charting”

table (Table 3). As suggested, for a scoping review methodology, a critical analysis of the

quality of the selected studies was not performed (Peters et al. 2020). Instead, an iterative

approach was used to produce a summary of extracted characteristic and demographic data

from the studies. The ‘data charting form’ was conducted independently by the author and

was not tested with an additional researcher.

[Insert Table 3]

Ethical considerations

Scoping reviews do not require ethical approval yet, Suri (2020) suggests critically reflecting

upon the position of primary researchers when conducting systematic reviews as the

secondary information received may be presented through the subjective lens of the primary

researchers. As in systematic reviews, scoping reviews review literature conducted by

primary researchers as secondary information. Suri (2020) discusses how researchers of

systematic reviews draw upon the philosophical traditions of consequentialism, deontology or

virtue ethics when making ethical decisions in relation to secondary research and denotes six

steps or guiding principles to ensure the researcher can reduce bias within their systematic or

scoping review.

As this review had a sole author, it is important to consider the position of the author (step 1)

to understand the author's epistemological perspective, as this demonstrates transparency and

limits bias within the review. Duncan (2011) and Suri (2020) both highlight the need for

understanding the author's epistemological perspective and how this may impact both

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research and practice. In this context, as the author of this paper, I acknowledge that my

perspective alines mostly from a post-positivistic paradigm in deontological ethics, as first

and foremost, I, as the author have personal experience as a sibling of a person with an NDD.

To ensure my own personal preunderstanding (step 2) or opinion does not transpire into the

literature selected for review, it is important as stated by Taylor (2007) that prior to the

search, I abide by a clear inclusion/exclusion criterion, to ensure that the internal and external

validity of the selected articles are upheld, enhancing the methodological quality. During the

search (step 3) it is important as the author to try to scope as much literature as possible,

including grey literature, to reduce the possibility of publication and search bias, yet as

literature is not peer-review it is important that the author clearly documents each stage of the

scoping review study, allowing steps to the study be replicated in future. When evaluating

and interpreting results (step 4), it is important that the author consider the quality and

relevancy of the evidence in respect to the purpose of the review. Again, abiding by a clear

inclusion/exclusion criterion will aid with this and when interpreting the data for thematic

analysis, the author can attempt to acknowledge potential bias by using a reflexive approach,

reflecting on thoughts and feelings about the selected literature and assumptions that may be

generated from the process (step 5). When recommending implications for practice, the

author must acknowledge the methodological considerations of the study and how aspects of

the study may impact the recommendations (step 6). For example, within this study,

acknowledging that the small, limited number of results from this scoping review cannot be

generalised to a larger population.

Results

The final 11 studies compromised of 8 qualitative, 2 quantitative and 1 mixed method study.

Most of the studies originated from the USA (n = 6) and other countries including, UK (n =

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2), Croatia (n = 1), Ireland (n = 1) and Canada (n = 1). There were a total of 1181

participants in the review, with 1181 (100%) being a sibling of a brother or sister diagnosed

with an NDD. 1180 (99.9%) of the participants were over 18 years old, in line with the

inclusion criteria, only 1 participant was aged 17 years old, and this study was included due

to its relevancy to answering the aim of the study.

To answer the aim accordingly, the author reviewed all 11 articles, read and re-read the

articles, kept reflective notes, assessed, and interpreted the data and extracted the key findings

from the articles into themes, relevant to answering the aim. The results highlighted that the

role of the sibling caregiver was not addressed within OT service in the literature. However,

the experiences of how well sibling caregivers are supported within healthcare services were

addressed and ideas and opinions of how services can be improved to support sibling

caregivers was found from the 11 articles. The data charting process and analysis resulted in

key findings in 5 areas: (1) Need for education and training opportunities in relation to

caregiving; (2) Policy and guideline changes in relation to sibling caregiver involvement; (3)

Sibling caregiver inclusion into the care pathway; (4) Need for improvements in service

coordination; and (5) Sibling caregivers' lack of knowledge in guardianship matters.

Need for education and training opportunities in relation to caregiving.

The most prominent theme to arise from the literature was the need for sibling caregivers to

access opportunities to engage in education and training specific to their caregiving role.

Many of the studies chosen for analysis discussed this topic in some context. Service-specific

needs included more education and training about the understanding of disability services,

roles of staff and more support for the sibling caregiver as they transition into the caring role

(Arnold et al. 2012; Rawson 2010). Some sibling caregivers highlighted activity specific

12
needs, such as time management issues with daily living tasks, knowledge about social

activities which can enhance the sibling relationship and advice for supporting their siblings

with typical self-care and daily life tasks (Burke et al. 2015; Davys et al. 2016; Mauldin and

Saxena 2018; Leane 2019).

Hinek and Milakovic (2019) reported sibling caregivers requiring other types of support, such

as challenges faced with communication, interaction and challenging behaviours of their

brother and sisters. The sibling caregivers suggested healthcare services could support further

by linking them to other typically developing sibling caregivers to share experiences. Rawson

(2010) also noted that sibling caregivers discussed concerns that their sibling’s cognitive

development changes as they age and similarly, Mauldin and Saxena (2018) highlight the

difference in sibling caregiver needs at different life stages, noting that task-based activities

increase as the sibling's age increases. One study had implemented a training program with

future sibling caregivers, in which future sibling caregivers attended four 2-hr training

sessions about sibling perspectives, identifying support needs, learning problem solving

skills, legal and financial planning and finally writing a letter of intent and connecting to

other sibling organisations. Outcomes from this study demonstrated that participants felt

more empowered, connected to others, felt improved communication with family, and had

more knowledge of adult disability services (Lee and Burke, 2021). Another study reported

that the sibling participants felt that their preferred way to access information is from online

website sources (Redquest et al. 2020).

Policy and guideline changes in relation to sibling caregiver involvement.

The second most prominent topic refers to how policies can be changed to express the ‘voice’

of sibling caregivers and how these caregivers are powerful advocates for their

13
brothers/sisters. Some of the articles referred to advocacy. Arnold et al. (2012) states that the

disability advocacy movement would benefit from involving sibling caregivers. Many sibling

caregivers stated their preference for learning more about advocacy, supporting their siblings

in self-advocacy and stated that having a greater involvement in advocacy matters may lead

to a positive outcome in current caregiving (Brady et al. 2019; Lee et al. 2019). In the sibling

training program conducted by Lee and Burke (2021) sibling participants of this study

reported feeling more empowered when handling advocacy matters.

Sibling caregiver inclusion into the care pathway.

Inclusion of sibling caregivers into the care pathway was a recurring theme in the data. Some

of the articles discussed how sibling caregivers should be included in their brother/sisters’

care planning from the start of service intervention or stated issues within current service

delivery. Several participants discussed how they felt as the sibling caregiver that they had

important information that related to the care needs of their brother/sister, or they were

already heavily involved with their brother/sister’s care planning (Arnold et al. 2012; Burke

et al. 2015; Rawson 2010). Yet, these sibling caregivers did not feel that they were either

included in the care planning of their brother/sister or were not included early enough

(Arnold et al. 2012). Also, some sibling participants discussed their concern with

transitioning into the caregiver role (Burke et al. 2015). Many sibling caregivers stated that

services had poor coordination and reported a distrust in services or a lack of confidence in

service providers (Arnold et al. 2012; Davys et al. 2016).

Need for improvements in service coordination.

Many sibling caregivers shared frustrations related to the need for improvements within the

system and how access to other services could support their needs. Two of the articles

14
revealed their frustrations with the ‘system’, needing more services to support their own

health and wellbeing and for information to be shared more freely, with support in

understanding bureaucracy and ultimately coordination of services (Arnold et al. 2012; Burke

et al. 2015).

Sibling caregivers' lack of knowledge in guardianship matters.

Three of the articles referred to sibling caregivers expressing their ‘lack of knowledge’, in

relation – predominately, to legalities about their role as a caregiver. Nearly all participants

in the study completed by Rawson (2010) reported a lack of knowledge about guardianship

and their rights. Brady et al. (2019) stated that 40% of participants reported they did not

understand guardianship, yet all participants reported that guardianship was necessary for

their brother/sister. Participants suggested that siblings could be involved in care planning as

early as 16 years of age (Rawson 2010).

Discussion

The aim of this study is to review current literature to find current and future sibling caregiver

perspectives on their roles as caregivers to an individual with a neurodevelopmental disorder

and whether their perspectives of healthcare services can influence improvements to the OT

practice.

Implications for research

In relation to research, the most prominent need arose around sibling caregivers feeling a

need for their role as a current or prospective sibling caregiver to be included specifically at

both a local and national policy level, suggesting that this may lead to their inclusion to their

brother or sister’s care pathway right from the start. Many national policies do not reflect the

15
role of the sibling caregiver. For example, in the UK, the Royal College of Occupational

Therapists (2022) highlights laws and policies which guide OT services and there is little

reflection on the role of the sibling caregiver. Concurrent with this, ‘The Care Act 2014’ UK

law states rights specifically for both young carers and parents, yet not siblings. Formalising

documentation to include siblings as caregivers, would ensure the sibling caregiver is

included in the planning process.

The Royal College of Occupational Therapists (2022) holds regular consultations to discuss

aspects of policy and changes needed. OTs may find it beneficial to involve themselves with

local meetings, such as these to ensure they can influence policies relevant to sibling

caregivers. However, OT services are also instructed to meet certain expectations set out by

healthcare authorities. Often, OTs report problems in meeting recommendations relayed from

research and using evidence-based practice (EBP). Practical barriers such as lack of time,

little support from their employment and a need for more training prevented using EBP in

practice (Reep, 2018). Therefore, the author recommends that from this review, OTs consider

the most significant strategies which could be implemented into service in their country,

whilst acknowledging the barriers in their practice.

Implications for practice

In relation to practice, the results have highlighted the most prominent views of both current

and future sibling caregivers in relation to both their personal situations and sibling

perspectives of healthcare services in general. Therefore, this review finds some practical

strategies that can be taken into consideration within OT, to improve services.

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Relevant strategies from participants include prominently, providing access to educational

opportunities in relation to caregiving for their sibling. Many strategies were suggested by

participants of the studies, these included sibling-specific training days, group work

scenarios, sibling-specific information books or websites and more information sessions

relating to their sibling’s condition and how this may change over time. One study included a

sibling training program, tested with a small sample of 18 sibling participants and resulted in

positive feedback from the sibling participants (Lee and Burke, 2021). Further research is

needed to demonstrate whether this type of sibling training program could be supported by

OTs as the study by Request et al. (2020) reported that sibling participants would prefer to

receive information via online sources, Therefore, how to disperse this information most

effectively would need to be considered.

Some participants discussed daily living specific issues of their own, for example, time-

management as a barrier to their own performance in supporting their sibling with an NDD.

Some of the current sibling participants discussed requiring more assistance when supporting

the care of their sibling in their ADLs. This is possibly where OTs can work more effectively

in practice – by providing further support to sibling caregivers by arranging support days in

the form of training.

Including sibling caregivers into the care pathway was discussed as an important problem by

the sibling participants of the studies. Concerns arose around the feasibility of being able to

share information with healthcare professionals about their brother or sister, being included to

the care planning stages early enough and challenges around transitioning into the caregiver

role. Ensuring involvement in the OT process of siblings at a younger age in the care

17
planning process may provide more opportunities for siblings to learn the key elements

involved in care planning and their rights as caregivers. As the sibling participants reported

distrust and a lack of confidence in service providers, it may be one strategy to ensure that

future or prospective sibling caregivers of a person with an NDD, are invited to join care

planning meetings within a multi-disciplinary setting. Prospective sibling caregivers would

be able to discuss pertinent information regarding both their brother or sister, and themselves

in their future transition to the caregiver role. This may start to or continue to build trust

between the families and the healthcare services.

Transitioning into roles and role identity is one aspect which falls into the OT domain and is

one area where OTs are more able to support sibling caregivers. Termed ‘occupational

identity’, this refers to a person’s sense of self, from how they engage in their own

occupations (Hansson, et al. 2022). This includes work, social and leisure occupations.

Howes and Ellison (2022) describe how threats to participation in leisure and social activities

impacted and adapted some caregiver’s occupational participation to engage and meet the

demands of the caregiving role. The authors also state that some caregivers are required to

change their role at work or give up their role entirely to meet the demands of being a

caregiver, yet also that OTs have the knowledge and skills to support caregivers with their

occupational participation, which can have a positive impact on their occupational identify.

There are some aspects from the results, that sibling participants discussed - that don’t fall

into directly into the OT domain, these were identified as finding more information related to

guardianship matters, knowledge about advocacy and sibling participants concerns over their

own health and wellbeing. However, OTs being able to or continuing to signpost/refer to

18
relevant services would support service coordination further in this instance. In the UK the

Royal College of Occupational Therapists (2019) actively promotes OTs to signpost to local

community support services to enhance social participation.

Conclusions

Although all the participants in the studies were siblings who had brother/sister with an NDD,

there was no link found specifically between the role of siblings within OT service. As The

Royal College of Occupational Therapists (2021) stated that further research is needed to

address how OT’s can work more effectively with family and carers of people who access

OT services, this review demonstrates that more research is required in this area, both in

areas of research and practice. This review demonstrates some practical strategies which OTs

can take, which may help to improve OT services, from the sibling caregiver perspective.

Although, further empirical research is required to truly reflect how the sibling caregiver role

is experienced within the domain of OT.

Methodological considerations

There are methodological limitations within this review. The review had one sole author. As

suggested by Taylor (2007) to enhance the rigour and reduce bias within this study, the

author should have engaged at least one more reviewer of the data. To increase the reliability

of the results as recommended by Arksey and O’Malley (2005), the author has clearly

detailed the process and steps taken to complete this review, to ensure the process can be

repeated.

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Other limitations include that only a partial number of databases, journals and grey literature

sources were searched, in only English language, limiting the number of relevant articles

found. The search was limited to articles which included an abstract only, further hindering

the scope of the search and enhancing the possibility that some literature which may have

fitted the inclusion criteria was excluded.

Finally, most of the studies included in the review used convenience, snowball, or deliberate

sampling method types, the highest method of sampling used was convenience sampling and

then snowball sampling. This demonstrates that the primary researchers may have been

unable to attain a purposive sample which can lead to a biased and unrepresentative sample

within the scoping review. reducing the transferability of the review and the ability to

generalise the findings to a larger population (Taylor, 2007). Also, majority of the selected

studies reported recruiting their participants through Autism/sibling or disability networks

and organisations. Therefore, it is likely a high percentage of the siblings may already have

some form of support network in place and may not show a typical representation of the

sample. It is clear within the selected literature that there are a small group of researchers

who regularly engage in research about sibling caregiving and these authors crossover and

co-author other selected literature. Whilst this may impact the quality and content of the data

available, it also demonstrates the current limited available evidence and supports the

conclusion of the need for further research.

20
References
Articles included into the review are marked with an *

American Psychiatric Association (2013) Diagnostic and statistical manual of mental

disorders: DSM-5 (5th ed.) Washington, D.C.: American Psychiatric Publishing.

American Occupational Therapy Association. (2020). Occupational Therapy Practice

Framework: Domain and Process (4th ed.). American Journal of Occupational
Therapy 74(2) http://doi.org/10.5014/ajot.2020.74S200

Arksey, H., and O’Malley, L. (2005). Scoping studies: towards a methodological framework.
International Journal of Social Research Methodology. 8(1) 19-32.
http://doi.org/10.1080/1364557032000119616

*Arnold, C., Heller, T., and Kramer, J. (2012). Support Needs of Siblings of People with
Developmental Disabilities. Intellectual and Developmental Disabilities. 50(5) 373-
382 http://doi.org/10.1352/1934-9556-50.5.373

Bathje, M., Lannoyeb, M., Mercierc, A., and Panter, K. (2018). A Review of Occupation-
Based life Skills Interventions for Adults with Neurodevelopmental Disorders.
Occupational Therapy in Mental Health. 34(2), 165-180.
https://doi.org/10.1080/0164212X.2017.1360168

*Brady, A. M., Burke, M. M., Landon, T., and Oertle, K. (2019). Siblings of adults with intellectual
and developmental disabilities: Their knowledge and perspectives on guardianship and its
alternatives. Journal of Applied Research in Intellectual Disabilities, 32(5), 1078-1087.
https://doi.org/10.1111/jar.12597

*Burke, M. M., Fish, T., and Lawton, K. (2015). A comparative analysis of adult siblings'
perceptions toward caregiving. Intellect Dev Disabil, 53(2), 143-157.
https://doi.org/10.1352/1934-9556-53.2.143

21
Center for Disease Control and Prevention. (2018). Caregiving for family and friends – A
public health issue. https://www.cdc.gov/aging/caregiving/caregiver-brief.html

*Davys, D., Mitchell, D., and Haigh, C. (2016). Adult Siblings Consider the Future: Emergent
Themes. J Appl Res Intellect Disabil, 29(3), 220-230. https://doi.org/10.1111/jar.12172

Duncan, E. (2011). Foundations for Practice in Occupational Therapy. Churchill Livingstone

Elsevier.

Demers, L. (2022) Expanding Occupational Therapy Perspectives with Family Caregivers. Canadian
Journal of Occupational Therapy, 89(3), 223-237.
https://doi.org/10.1177/00084174221103952

European Commission, Directorate-General for Employment, Social Affairs, and Inclusion,

Zigante, V. (2018). Informal care in Europe: exploring formalisation, availability,
and quality. https://data.europa.eu/doi/10.2767/78836

Hansson, S., and Carlstedt, A., and Morville, A. (2022). Occupational identity in
occupational therapy: A concept analysis. Scandinavian Journal of Occupational
Therapy, 29(3), 198-209. https://doi.org/10.1080/11038128.2021.1948608

Heller, T., and Kramer, J. (2009). Involvement of adult siblings of persons with developmental
disabilities in future planning. Intelletucal and Developmental Disabilites, 47(3), 208-219.
https://doi.org/10.1352/1934-9556-47.3.208

*Hinek, S., and Milaković, A. T. (2019). Growing up with a brother diagnosed with autism spectrum
disorder: The siblings’ perspective. Hrvatska Revija Za Rehabilitacijska Istraživanja, 55(1),
1-12. https://doi.org/10.31299/hrri.55.1.1

Howes, M., and Ellison, D. (2022). Understanding the occupational identity of care-givers for people
with mental health problems. British Journal of Occupational Therapy, 85(4), 274-282.
https://doi.org/10.1177/03080226211018153

22
International Alliance of Carer Organizations. (2021). Global Carer Facts.
https://internationalcarers.org/carer-facts/global-carer-stats/

*Leane, M. (2019). Siblings caring for siblings with Intellectual Disabilities: Naming and negotiating
emotional tensions. Soc Sci Med, 230, 264-270.
https://doi.org/10.1016/j.socscimed.2019.04.022

*Lee, C. E., Burke, M., Arnold, C. K., and Owen, A. (2019). Correlates of current caregiving among
siblings of adults with intellectual and developmental disabilities. Journal of Applied
Research in Intellectual Disabilities, 32(6), 1490-1500. https://doi.org/10.1111/jar.12644

*Lee, C. E., and Burke, M. M. (2021). A Pilot Study of a Future Planning Program for Siblings of
People with Intellectual and Developmental Disabilities. Intellectual & Developmental
Disabilities, 59(1), 70-83. https://doi.org/10.1352/1934-9556-59.1.70

Lindahl, J., Stollon, N., Wu, K., Liang, A., Changolkar, S., Steinway, C., Trachtenberg, S., Coccia,
A., Devaney, M., and Jan, S. (2019). Domains of planning for future long-term care of adults
with intellectual and developmental disabilities: Parent and sibling perspectives. Journal of
Applied Research in Intellectual Disabilities. 32(5), 1103-115.
https://doi.org/http://dx.doi.org/10.1111/jar.12600

*Mauldin, L., and Saxena, M. (2018). Mutual exchange: Caregiving and life enhancement in siblings
of individuals with developmental disabilities. Journal of Family Issues, 39(8), 2232-2264.
https://doi.org/http://dx.doi.org/10.1177/0192513X17746452

Moghimi, C. (2007). Issues in caregiving: The role of occupational therapy in caregiver training.
Topics in Geriatric Rehabilitation, 23(3), 269-279.
https://doi.org/10.1097/01.TGR.0000284770.39958.79

Namkung, E., Greenberg, J., and Malick, M. (2017). Well-being of Sibling Caregivers:
Effects of Kinship, Relationship and Race. The Gerontologist. 57(4) 626-636
http://doi.org/10.1093/geront/gnw008

23
Paez, A. (2017). Gray literature: An important resource in systematic reviews. Journal of
Evidence-Based Medcine, 10(3), 233-240. https://doi.org/10.1111/jebm.12266

Page, MJ., McKenzie, J., Bossuyt, P., Boutron, I., Hoffmann, T., Mulrow, CD., and Moher,
D. (2020). The PRISMA 2020 statement: an updated guideline for reporting
systematic reviews. PRISMA. http://doi.org/10.31222/osf.io/v7gm2

Peters MDJ, Godfrey C, McInerney P, Munn Z, Tricco AC, Khalil, H. (2020). Chapter 11:
Scoping Reviews. In: Aromataris E, Munn Z (Editors). JBI Manual for Evidence
Synthesis, JBI. Available from https://synthesismanual.jbi.global

*Rawson, H. (2010). ‘I’m going to be here long after you’ve gone’ – sibling perspectives of the
future. British Journal of Learning Disabilities, 38(3), 225-231.
https://doi.org/10.1111/j.1468-3156.2009.00599.x

*Redquest, B. K., Tint, A., Ries, H., Goll, E., Rossi, B., and Lunsky, Y. (2020). Support needs of
Canadian adult siblings of brothers and sisters with intellectual/developmental disabilities.
Journal of Policy & Practice in Intellectual Disabilities, 17(3), 239-246.
https://doi.org/10.1111/jppi.12339

Reep, J. (2018). Developing the occupational therapy practice in a local community health
team working with adults with learning disabilities: An action research study.
[Doctoral dissertation, London South Bank University]. EThOS: Electronic Theses
Online Service. http://doi.org/10.18744/PUB.002756

Restall, G., and Egan, M. (2021). Collaborative Relationship-Focused Occupational Therapy:

Evolving Lexicon and Practice. Canadian Journal of Occupational Therapy, 88(3),
220-230. https://doi.org/10.1177/00084174211022889

Royal College of Occupational Therapists. (2022). Policy and legislation.

https://www.rcot.co.uk/practice-resources/policy-and-legislation

24
Royal College of Occupational Therapists. (2022). Professional standards for occupational therapy
practice, conduct and ethics. https://www.rcot.co.uk/publications/professional-standards-
occupational-therapy-practice-conduct-and-ethics

Royal College of Occupational Therapists. (2021). Identifying research priorities for

occupational therapy in the UK: What matters most to the people accessing and
delivering services? https://www.rcot.co.uk/identifying-research-priorities-
occupational-therapy-uk

Royal College of Occupational Therapists. (2019). Occupational therapy’s role in social

prescribing. https://www.rcot.co.uk/news/occupational-therapy-role-social-
prescribing

Scorgie, K. (2017). Broadening Family Perspectives: The Experiences of Fathers and

Siblings When a Child Has Chronic Illness or Disability. In Working with Families
for Inclusive Education (pp. 97–113). Emerald Publishing Limited.
https://doi.org/10.1108/S1479-363620170000010012

Suri, H. (2020). Ethical Consideration of Conducting Systematic Reviews in Educational

Research. In: Zawacki-Ritcher O., Kerres M., Bedenlier, S., Bond, M., and Buntin's
K. (eds) Systematic Reviews in Educational Research. Springer VS, Wiesbaden.
http://doi.org/10.1007/978-3-858-27602-7_3

Taylor, M. C. (2007). Evidence-based practice for occupational therapists. Blackwell

Science Ltd.

Taylor, R. R. (2017). Kielhofner’s Model of Human Occupation: Theory and Application. (5th
ed.). Philadelphia, PA: Lippincott Williams & Wilkins.

Totsika, V., Hastings, R., and Vagenas, D. (2017). Informal caregivers of people with an
intellectual disability in England: health, quality of life and impact of caring. Health
and Social Care in the Community. 25(3) 951-961. http://doi.org/10.1111/hsc.12393

25
Townsend, E., and Polatajko, H. J. (2013). Enabling occupation II: advancing an
occupational therapy vision for health, well-being & justice through occupation: 9th
Canadian occupational therapy guidelines. (2nd ed.) Canadian Association of
Occupational Therapists.

Wong, C., Fagan, B., and Leland, N. E. (2018). Occupational therapy practitioners’
perspectives on occupation-based interventions for clients with hip fracture. American
Journal of Occupational Therapy, 72(4), 72042005050.
https://doi.org/10.5014/ajot.2018.026492

Wong, SR., and Fisher, G. (2015). Comparing and Using Occupation-Focused Models.
Occupational Therapy in Health Care, 29(3), 297-315.
http://doi.org/10.3109/07380577.2015.1010130

World Health Organization. (2017). Evidence Profile: Caregiver Support

World Health Organization. (2022). ICD-11: International Classification of Diseases: (11th

revision). https://icd.who.int/

26
Table 1. Electronic database search
Primary Limiters Search terms Number Chosen by Duplicated Final number
Database of hits abstract removed of articles
included
CINAHL Abstract available neurodevelopmental disorders OR autis* OR intellectual disabilit* 52 24 4 4
Complete English language 2010- OR IDD OR learning disabilit* AND sibling* OR brother* OR
2021 sister* AND caregiv*
Age: all adult

AMED Abstract available neurodevelopmental disorders OR autis* OR intellectual disabilit* 2 2 1 1

English language 2010- OR IDD OR learning disabilit* AND sibling* OR brother* OR
2021 sister* AND caregiv*
PubMed Abstract available (neurodevelopmental disorders OR autis* OR intellectual disabilit* 67 28 12 3
English language 2010- OR IDD OR learning disabilit*) AND (sibling* OR brother* OR
2021 sister*) AND (caregiv*)
Age: Young adult 19-24
years, Adult: 19+ years,
Adult 19-44 years,
Middle Aged + Aged:
45+ years, Middle
Aged: 45-65 years,
Aged: 65+ years, 80 and
over: 80+ years
Medline Abstract available, neurodevelopmental disorders OR autis* OR intellectual disabilit* 56 28 25 2
English language, OR IDD OR learning disabilit* AND sibling* OR brother* OR
2010-2021, All Adult: sister* AND caregiv*
19+ years, Boolean
phrase

27
PsychINFO Abstract available, neurodevelopmental disorders OR autis* OR intellectual disabilit* 53 21 14 1
English language, OR IDD OR learning disabilit* AND sibling* OR brother* OR
2010-2021, Include: sister* AND caregiv*
Adulthood (18yrs and
older)
Secondary Limiters Search terms Number of Chosen by Duplicates
Database hits abstract removed
Cochrane 2010-2021 neurodevelopmental disorders OR autis* OR intellectual disabilit* 5 trials - - 0
library OR IDD OR learning disabilit* AND sibling* OR brother* OR
sister* AND caregiv*
OTseeker No limiters neurodevelopmental disorders AND sibling 0 - - 0
intellectual disabili* AND sibling 0
learning disabilit* AND sibling 0
autis* AND sibling 0

28
Table 2. Journal search
Journal Limits Search terms Number of hits Chosen by abstract Final number of
articles included
1. British Journal of 2010-2021 sibling AND caregiv* 29 0 0
Occupational Therapy
2. Journal of Autism and 2010-2021, English language adult sibling* AND caregiv* 286 7 0
Developmental
Disorders
3. Research in 2010-2021 adult sibling AND caregiving 39 6 0
Developmental
Disabilities
4. Intellectual and 2010-01-01 to 2021-12-31, adult siblings AND caregiv* 51 8 0
Developmental English language
Disabilities

29
Table 3. Data charting
Author Population Sample Recruitment Study design Ethical considerations Purpose
Year Method
Country
Arnold, C, 139 sibling Convenience Recruited Grounded theory Consent and ethics for study not Examines support needs of adult siblings of people
Heller, T and participants through an reported. with developmental disabilities
Kramer (2012) online sibling
US group and state-
wide sibling
conference.

Brady, A., 10 sibling Combination of Recruited Grounded theory Board ethics gained. Explore the knowledge and understanding of
Burke, M., participants (3 convenience and through guardianship for siblings
Landon, T., and current theoretical disability
Oertle, K. caregivers, 1 sampling organisations
(2019) past, 6 possible and social
US future media.
caregivers)

Burke, M., Fish, 42 sibling Not reported Personal Qualitative Board ethics gained. Participant Compare the perceptions of caregiving between
T., and Lawton, participants contacts and study consent gained. adult siblings who currently are caregivers and
K. (2015) (25 current social media those who are anticipated caregivers in the future
US caregivers and used to recruit
17 anticipated from three USA
caregivers. states through
sibling
organisations.

30
Davys, D., 15 sibling Not reported Participants Mixed method Board consent gained. Consent Exploring adult sibling’s perspectives on future care
Mitchell, D., and participants recruited study gained. planning.
Haigh, C (2016) through self- Anonymity reported.
UK help
organisations.

Hinek, S., and 6 sibling Deliberate Recruited via the Qualitative Oral consent gained, ethics for To explore the experiences of siblings living with a
Milakovic participants sampling Association for study study not reported brother with ASD and the perceptions of received
(2019) Autism Zagreb and needed support
Croatia

Leane, M (2019) 25 sibling Not reported Recruited Qualitative Board ethics gained. Written To explore perceptions of sibling relationships and
Ireland participants through study consent gained. Anonymity expectations and aspirations of their caregiving
Inclusion Ireland reported. roles in the future.
and 2 ASD
support groups.

Lee, C., Burke, 429 sibling Convenience Flyers sent to Quantitative Board ethics gained. Consent not To explore the bioecological correlates in current
M., Arnold, C., participants sampling local, parent and study reported. sibling caregiving to identify who is likely to
and Owen, A. national support become a caregiver and identified for support.
(2019) groups,
US agencies,
organisations,
and networks.

Lee., C., and 18 sibling Selective Targeted Mixed method Board ethics gained. Written To evaluate the outcomes and feasibility of the
Burke, M. participants (all snowball recruitment study consent gained. Sibling Training for Early Future Planning (STEP)
(2021) future planned sampling through local, program.
US caregivers) state-wide and

31
 community
organisations
and Sibling
Leadership
Network.

Mauldin, L., and 224 sibling Snowball Link to web Grounded theory Board ethics gained. Consent not Understanding life enhancement of adult sibling
Saxena, M participants sampling survey sent to reported. experiences of life with a brother/sister with
(2018) 245 developmental disabilities
US organisations
and agencies
across USA.

Rawson, H 13 sibling Not reported Siblings invited Qualitative Board ethics gained. Consent Exploring what concerns young adult siblings have
(2010) participants via letter to their study gained. Anonymity reported. for the future and what support do they feel they
UK home via their need?
brother/sisters
who attend a
special needs
residential
school.

Redquest, B., 260 sibling Not reported Secondary Quantitative Board ethics gained. To better understand experiences and challenges of
Tint, A., Ries, participants analysis of data. study sibling care across three ages (20-29, 30-49 and
H., Goll, E., 50yrs +)
Rossi, B., and
Lunsky, Y
(2020)
Canada

32
Figure 1: PRISMA 2020 flow diagram (Page, et al. 2020)