A realist review of programs for siblings of children who have an intellectual/developmental disability

• Published date: 01 October 2023
• Author: Sandra Marquis,Renee O'Leary,Nikita K. Hayden,Jennifer Baumbusch
• Date: 01 October 2023
• DOI: http://doi.org/10.1111/fare.12789

RESEARCH

A realist review of programs for siblings of children

who have an intellectual/developmental disability

Sandra Marquis

1

|Renee O’Leary

2

|Nikita K. Hayden

3

|

Jennifer Baumbusch

1

1

School of Nursing, University of British

Columbia, Vancouver, Canada

2

Principal, 21st Century Literature Reviews,

Victoria, Canada

3

Centre for Educational Development,

Appraisal and Research, University of

Warwick, Coventry, United Kingdom

Correspondence

Sandra Marquis, School of Nursing,

University of British Columbia, T201-2211

Wesbrook Mall, Vancouver, BC Canada

V6T 2B5.

Email: sandra.marquis@ubc.ca

Funding information

Sandra Marquis is supported by a Michael

Smith Health Research British Columbia

trainee grant (RT-2020-0516). The funder had

no involvement in the collection, analysis, and

interpretation of data; in the writing of the

report; or in the decision to submit the article

for publication. Renee O’Leary, Nikita

K. Hayden, and Jennifer Baumbusch received

no funding support for this study.

Abstract

Objective: Our purpose is to answer the following question:

What mechanisms or components of programs, groups, or

interventions improve psychological and social aspects of

the lives of siblings of children who have intellectual/

developmental disabilities (IDD)? Secondly, using a realist

review format, we develop program theories and recom-

mendations that can guide current and future sibling

support programs.

Background: There is evidence that having a sibling with

IDD can affect the physical health, behavior, and mental

health of siblings who do not have IDD. As a result, sup-

port programs have been developed for these nondisabled

siblings.

Methods: We conducted a realist review of 31 studies

assessing support programs for siblings of children with

IDD. Gray literature was also included. Applying the real-

ist paradigm, we identified program contexts, mechanisms

that promote program effectiveness, and the outcomes of

the programs.

Results: We found that contextual information was often

lacking in the research and no studies examined the effec-

tiveness of individual program components. However, the

mechanisms of validation of feelings and experiences, time

with parents, and respite were observed as beneficial for

the siblings. From our analysis and using a realist review

paradigm, we developed four program theories that reflect

the siblings’experiences in the various programs.

Conclusion: The four program theories derived from this

realist review can be summarized as “I am not the only

one,”“I have needs too,”“I need my parents’attention,”

and “I need a break.”

Implications: Based upon the four theories, we offer rec-

ommendations to guide program development.

Received: 7 December 2021Revised: 22 January 2022Accepted: 5 September 2022

DOI: 10.1111/fare.12789

© 2022 National Council on Family Relations.

Family Relations. 2023;72:2083–2102. wileyonlinelibrary.com/journal/fare 2083

KEYWORDS

developmental disability, intellectual disability, sibling, support program

There is evidence that the number of children diagnosed with an intellectual/developmental dis-

ability (IDD) is increasing in developed countries (Houtrow et al., 2014; Marquis et al., 2018).

Using data from the U.S. National Health Interview Survey, Zablotsky et al. (2019) found that

from 2009 to 2011 and 2015 to 2017, there were significant increases in the prevalence of any

developmental disability and in the prevalence of Autism Spectrum Disorder and intellectual

disability. Many of these children with IDD have siblings. Several studies have noted that fami-

lies that include a child with IDD tend to have a greater number of children than comparison

families (M. M. Burke et al., 2011;Haetal.,2008; Seltzer et al., 2001).

In general, sibling relationships are some of the longest lasting and most influential relation-

ships in people’s lives (Buchanan & Rotkirch, 2021). There is a considerable body of research

examining the effects of having a sibling with an IDD upon the health, behavior, and mental

health of siblings who do not have an IDD (Marquis et al., 2019b). Often research has bee n pre-

mised on assumptions that effects are always negative (Hastings, 2016). However, evidence has

also been found that having a sibling with IDD can have positive effects (Macks &

Reeve, 2007). Other studies have found no significant impact upon nondisabled siblings

(Cuskelly & Gunn, 2006; Hastings & Petalas, 2014; Rossiter & Sharpe, 2001). Mounting evi-

dence demonstrates that any effects upon siblings without IDD are not solely a result of having

a disabled sibling. Instead, sibling outcomes result from complex interactions among social

determinants of health, the characteristics of the child with IDD, characteristics of the siblings

without IDD, and family factors (Kovshoff et al., 2017; Marquis et al., 2019b).

Many researchers have called for support for the well-being of siblings of children with IDD

(P. Burke & Montgomery, 2001; Garrido et al., 2020; Naylor & Prescott, 2004). Consequently,

a variety of policies, programs, and interventions have been developed. However, very little is

known about the effectiveness of these interventions. The few assessments that have been con-

ducted have focused on single programs that differ widely in service delivery, target group,and

expected outcomes (Evans et al., 2001; Kryzak & Jones, 2017; Roberts et al., 2015). Four sys-

tematic reviews (Claudel-Valentin et al., 2020; Hartling et al., 2014; McKenzie Smith

et al., 2018; Tudor & Lerner, 2015) concluded that interventions for siblings of children with

disabilities or chronic health conditions may be beneficial but that programs need to clearly

identify intended results, types of siblings who would benefit, and the most effective methods of

delivery.

To pinpoint which program components are efficacious for which groups of siblings entails

an examination of not only program outcomes but also how the programs bring about or fail to

deliver improvements in sibling well-being. In contrast to systematic reviews, realist reviews

provide a fuller picture of interventions because their purpose is to describe “What works, how,

why, for whom, to what extent and in what circumstances, in what respect and over what dura-

tion”(Wong et al., 2013, p. 17). Realist reviews are designed to address complexity and to ana-

lyze evidence of why and when interventions are effective. A systematic review does not address

these critical issues (Booth et al., 2020).

The foundational concept of a realist review is context plus mechanisms produce outcomes

(C +M=O; Pawson, 2018; Wong et al., 2013). Contexts are the situational influences on an

intervention (Greenhalgh et al., 2017c). Contexts can originate from multiple levels: the micro-

level (i.e., age), meso-level (i.e., family income), and macro-levels (i.e., government policies;

Emmel et al., 2018). Other contextual factors include the training and abilities of program staff

and the program setting (Lemire et al., 2020).

2084 FAMILY RELATIONS