<?xml version="1.0" encoding="UTF-8"?><!DOCTYPE article PUBLIC "-//NLM//DTD JATS (Z39.96) Journal Publishing DTD v1.2 20190208//EN" "http://jats.nlm.nih.gov/publishing/1.2/JATS-journalpublishing1.dtd"><article xmlns:mml="http://www.w3.org/1998/Math/MathML" xmlns:xlink="http://www.w3.org/1999/xlink" article-type="systematic-review" dtd-version="1.2" xml:lang="en">
    <front>
        <journal-meta>
            <journal-id journal-id-type="pmc">F1000Research</journal-id>
            <journal-title-group>
                <journal-title>F1000Research</journal-title>
            </journal-title-group>
            <issn pub-type="epub">2046-1402</issn>
            <publisher>
                <publisher-name>F1000 Research Limited</publisher-name>
                <publisher-loc>London, UK</publisher-loc>
            </publisher>
        </journal-meta>
        <article-meta>
            <article-id pub-id-type="doi">10.12688/f1000research.133314.1</article-id>
            <article-categories>
                <subj-group subj-group-type="heading">
                    <subject>Systematic Review</subject>
                </subj-group>
                <subj-group>
                    <subject>Articles</subject>
                </subj-group>
            </article-categories>
            <title-group>
                <article-title>Effect of family-centered care interventions on well-being of caregivers of children with cerebral palsy: a systematic review</article-title>
                <fn-group content-type="pub-status">
                    <fn>
                        <p>[version 1; peer review: 1 approved, 1 approved with reservations]</p>
                    </fn>
                </fn-group>
            </title-group>
            <contrib-group>
                <contrib contrib-type="author" corresp="no">
                    <name>
                        <surname>Poojari</surname>
                        <given-names>Deepalaxmi Paresh</given-names>
                    </name>
                    <role content-type="http://credit.niso.org/">Conceptualization</role>
                    <role content-type="http://credit.niso.org/">Data Curation</role>
                    <role content-type="http://credit.niso.org/">Formal Analysis</role>
                    <role content-type="http://credit.niso.org/">Funding Acquisition</role>
                    <role content-type="http://credit.niso.org/">Methodology</role>
                    <role content-type="http://credit.niso.org/">Resources</role>
                    <role content-type="http://credit.niso.org/">Supervision</role>
                    <role content-type="http://credit.niso.org/">Writing &#x2013; Original Draft Preparation</role>
                    <role content-type="http://credit.niso.org/">Writing &#x2013; Review &amp; Editing</role>
                    <uri content-type="orcid">https://orcid.org/0000-0003-0035-8478</uri>
                    <xref ref-type="aff" rid="a1">1</xref>
                </contrib>
                <contrib contrib-type="author" corresp="yes">
                    <name>
                        <surname>Umakanth</surname>
                        <given-names>Shashikiran</given-names>
                    </name>
                    <role content-type="http://credit.niso.org/">Conceptualization</role>
                    <role content-type="http://credit.niso.org/">Formal Analysis</role>
                    <role content-type="http://credit.niso.org/">Methodology</role>
                    <role content-type="http://credit.niso.org/">Project Administration</role>
                    <role content-type="http://credit.niso.org/">Resources</role>
                    <role content-type="http://credit.niso.org/">Supervision</role>
                    <role content-type="http://credit.niso.org/">Writing &#x2013; Review &amp; Editing</role>
                    <uri content-type="orcid">https://orcid.org/0000-0001-5210-7457</uri>
                    <xref ref-type="corresp" rid="c1">a</xref>
                    <xref ref-type="aff" rid="a2">2</xref>
                </contrib>
                <contrib contrib-type="author" corresp="no">
                    <name>
                        <surname>Maiya</surname>
                        <given-names>G. Arun</given-names>
                    </name>
                    <role content-type="http://credit.niso.org/">Conceptualization</role>
                    <role content-type="http://credit.niso.org/">Data Curation</role>
                    <role content-type="http://credit.niso.org/">Project Administration</role>
                    <role content-type="http://credit.niso.org/">Resources</role>
                    <role content-type="http://credit.niso.org/">Supervision</role>
                    <role content-type="http://credit.niso.org/">Writing &#x2013; Review &amp; Editing</role>
                    <xref ref-type="aff" rid="a1">1</xref>
                </contrib>
                <contrib contrib-type="author" corresp="no">
                    <name>
                        <surname>Rao</surname>
                        <given-names>Bhamini Krishna</given-names>
                    </name>
                    <role content-type="http://credit.niso.org/">Conceptualization</role>
                    <role content-type="http://credit.niso.org/">Formal Analysis</role>
                    <role content-type="http://credit.niso.org/">Project Administration</role>
                    <role content-type="http://credit.niso.org/">Resources</role>
                    <role content-type="http://credit.niso.org/">Supervision</role>
                    <role content-type="http://credit.niso.org/">Writing &#x2013; Review &amp; Editing</role>
                    <uri content-type="orcid">https://orcid.org/0000-0002-2708-0245</uri>
                    <xref ref-type="aff" rid="a1">1</xref>
                </contrib>
                <contrib contrib-type="author" corresp="no">
                    <name>
                        <surname>Khurana</surname>
                        <given-names>Sonia</given-names>
                    </name>
                    <role content-type="http://credit.niso.org/">Conceptualization</role>
                    <role content-type="http://credit.niso.org/">Data Curation</role>
                    <role content-type="http://credit.niso.org/">Formal Analysis</role>
                    <role content-type="http://credit.niso.org/">Investigation</role>
                    <role content-type="http://credit.niso.org/">Methodology</role>
                    <role content-type="http://credit.niso.org/">Supervision</role>
                    <role content-type="http://credit.niso.org/">Writing &#x2013; Original Draft Preparation</role>
                    <role content-type="http://credit.niso.org/">Writing &#x2013; Review &amp; Editing</role>
                    <xref ref-type="aff" rid="a3">3</xref>
                </contrib>
                <contrib contrib-type="author" corresp="no">
                    <name>
                        <surname>Kumaran D</surname>
                        <given-names>Senthil</given-names>
                    </name>
                    <role content-type="http://credit.niso.org/">Conceptualization</role>
                    <role content-type="http://credit.niso.org/">Data Curation</role>
                    <role content-type="http://credit.niso.org/">Formal Analysis</role>
                    <role content-type="http://credit.niso.org/">Methodology</role>
                    <role content-type="http://credit.niso.org/">Supervision</role>
                    <role content-type="http://credit.niso.org/">Writing &#x2013; Review &amp; Editing</role>
                    <uri content-type="orcid">https://orcid.org/0000-0001-6491-2584</uri>
                    <xref ref-type="aff" rid="a1">1</xref>
                </contrib>
                <contrib contrib-type="author" corresp="no">
                    <name>
                        <surname>Attal</surname>
                        <given-names>Radhika</given-names>
                    </name>
                    <role content-type="http://credit.niso.org/">Data Curation</role>
                    <role content-type="http://credit.niso.org/">Formal Analysis</role>
                    <role content-type="http://credit.niso.org/">Investigation</role>
                    <role content-type="http://credit.niso.org/">Methodology</role>
                    <role content-type="http://credit.niso.org/">Writing &#x2013; Original Draft Preparation</role>
                    <role content-type="http://credit.niso.org/">Writing &#x2013; Review &amp; Editing</role>
                    <xref ref-type="aff" rid="a1">1</xref>
                </contrib>
                <contrib contrib-type="author" corresp="no">
                    <name>
                        <surname>Brien</surname>
                        <given-names>Marie</given-names>
                    </name>
                    <role content-type="http://credit.niso.org/">Formal Analysis</role>
                    <role content-type="http://credit.niso.org/">Investigation</role>
                    <role content-type="http://credit.niso.org/">Methodology</role>
                    <role content-type="http://credit.niso.org/">Supervision</role>
                    <role content-type="http://credit.niso.org/">Writing &#x2013; Original Draft Preparation</role>
                    <role content-type="http://credit.niso.org/">Writing &#x2013; Review &amp; Editing</role>
                    <xref ref-type="aff" rid="a4">4</xref>
                </contrib>
                <aff id="a1">
                    <label>1</label>Department of Physiotherapy, Manipal College of Health Professions, Manipal Academy of Higher Education, Manipal, Karnataka, 576104, India</aff>
                <aff id="a2">
                    <label>2</label>Department of Medicine, Dr TMA Pai Hospital, Udupi, Manipal Academy of Higher Education, Manipal, Karnataka, 576101, India</aff>
                <aff id="a3">
                    <label>3</label>Department of Physical therapy, College of Health Sciences, Old Dominion University, Norfolk, Virginia, USA</aff>
                <aff id="a4">
                    <label>4</label>Enabling Inclusion Program, Amar Seva Sangam, Ayikudi, Tamil Nadu, India</aff>
            </contrib-group>
            <author-notes>
                <corresp id="c1">
                    <label>a</label>
                    <email xlink:href="mailto:shashikiran.u@manipal.edu">shashikiran.u@manipal.edu</email>
                </corresp>
                <fn fn-type="conflict">
                    <p>No competing interests were disclosed.</p>
                </fn>
            </author-notes>
            <pub-date pub-type="epub">
                <day>6</day>
                <month>7</month>
                <year>2023</year>
            </pub-date>
            <pub-date pub-type="collection">
                <year>2023</year>
            </pub-date>
            <volume>12</volume>
            <elocation-id>790</elocation-id>
            <history>
                <date date-type="accepted">
                    <day>20</day>
                    <month>4</month>
                    <year>2023</year>
                </date>
            </history>
            <permissions>
                <copyright-statement>Copyright: &#x00a9; 2023 Poojari DP et al.</copyright-statement>
                <copyright-year>2023</copyright-year>
                <license xlink:href="https://creativecommons.org/licenses/by/4.0/">
                    <license-p>This is an open access article distributed under the terms of the Creative Commons Attribution Licence, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.</license-p>
                </license>
            </permissions>
            <self-uri content-type="pdf" xlink:href="https://f1000research.com/articles/12-790/pdf"/>
            <abstract>
                <p>
                    <bold>Background</bold>: Caring for a child with long-term functional limitations can have a negative impact on the physical and psychological well-being of the caregiver. Family-centered care (FCC) interventions have the potential to empower caregivers and contribute to their well-being. This systematic review aimed to synthesize existing evidence on the effectiveness of FCC interventions in improving the well-being of caregivers of children with cerebral palsy (CP), and identify the key components of such interventions that are most commonly practiced and deemed effective.</p>
                <p>
                    <bold>Methods</bold>: This review systematically searched seven databases for randomized controlled trials that evaluated the effectiveness of any FCC intervention on the well-being of caregivers of children with or at risk of CP. We used the Cochrane RoB 2.0 tool to assess risk of bias and Critical Appraisal Skills Programme (CASP) checklist for critical appraisal. Due to high heterogeneity of studies, narrative synthesis was used to summarize the data.</p>
                <p>
                    <bold>Results</bold>: The review consists of 11 studies which were categorized into five sections based on the components of FCC intervention provided in each individual study: 1. Information provision, and Enabling and partnership (n= 5); 2. Information provision, and Respectful and supportive care (n= 1); 3. Enabling and partnership (n= 2); 4. Enabling and partnership, and Respectful and supportive care (n= 2); 5. Information provision, Enabling and partnership and Respectful and supportive care (n= 1). Risk of bias was low in four studies, unclear in two studies, and high in five studies.</p>
                <p>
                    <bold>Conclusion</bold>: FCC interventions were found to be effective in improving caregivers&#x2019; satisfaction with attainment of child and caregiver goals. Evidence from multiple studies does not strongly support the effectiveness of FCC interventions on caregiver&#x2019;s mental health, parenting and personal outcomes. Limited evidence precludes a conclusion on the effectiveness of the components of FCC on well-being of caregivers of children with CP.</p>
            </abstract>
            <kwd-group kwd-group-type="author">
                <kwd>Developmental disability</kwd>
                <kwd>family participation</kwd>
                <kwd>Parent well-being</kwd>
                <kwd>Physical health</kwd>
                <kwd>Mental health</kwd>
                <kwd>Parent-professional partnership</kwd>
                <kwd>Parent education</kwd>
                <kwd>Collaborative care</kwd>
            </kwd-group>
            <funding-group>
                <funding-statement>The author(s) declared that no grants were involved in supporting this work.</funding-statement>
            </funding-group>
        </article-meta>
    </front>
    <body>
        <sec id="sec1" sec-type="intro">
            <title>Introduction</title>
            <p>Family-centered care (FCC) approach holds fundamental importance to professional practice that conveys dignity and respect to families, information provision for informed decision-making, consideration of the family&#x2019;s preferences and priorities, and collaborative partnerships between the provider and family.
                <sup>
                    <xref ref-type="bibr" rid="ref1">1</xref>
                </sup> This approach may enable caregiver access to various healthcare services through education and counseling,
                <sup>
                    <xref ref-type="bibr" rid="ref2">2</xref>
                </sup> support groups,
                <sup>
                    <xref ref-type="bibr" rid="ref3">3</xref>
                </sup> information about their child&#x2019;s condition,
                <sup>
                    <xref ref-type="bibr" rid="ref4">4</xref>
                </sup> skill training,
                <sup>
                    <xref ref-type="bibr" rid="ref5">5</xref>
                </sup> involvement in setting goals for their children,
                <sup>
                    <xref ref-type="bibr" rid="ref6">6</xref>
                </sup> or establishing a strong caregiver-professional partnership.</p>
            <p>Children with cerebral palsy (CP) may have a range of impairments that limit their daily activities such as mobility, self-care, communication, and participation, requiring special care services.
                <sup>
                    <xref ref-type="bibr" rid="ref7">7</xref>
                </sup>
                <sup>,</sup>
                <sup>
                    <xref ref-type="bibr" rid="ref8">8</xref>
                </sup> Apart from their daily duties, caregivers take up multiple roles such as handling the medical, rehabilitation, and financial services, in an attempt to provide the best care for their child. Hence, caring for a child with CP demands adjustment in the caregiver&#x2019;s lifestyle based on the child&#x2019;s needs and impacts the caregiver&#x2019;s personal, family, social, and financial well-being.
                <sup>
                    <xref ref-type="bibr" rid="ref9">9</xref>
                </sup>
                <sup>,</sup>
                <sup>
                    <xref ref-type="bibr" rid="ref10">10</xref>
                </sup>
                <sup>,</sup>
                <sup>
                    <xref ref-type="bibr" rid="ref11">11</xref>
                </sup> Caring for a child with long-term functional limitations may affect the physical as well as the psychological well-being of the caregiver.
                <sup>
                    <xref ref-type="bibr" rid="ref12">12</xref>
                </sup>
                <sup>,</sup>
                <sup>
                    <xref ref-type="bibr" rid="ref13">13</xref>
                </sup> Parents nurturing a child with CP often experience isolation, anxiety, and depression.
                <sup>
                    <xref ref-type="bibr" rid="ref14">14</xref>
                </sup>
                <sup>,</sup>
                <sup>
                    <xref ref-type="bibr" rid="ref15">15</xref>
                </sup> Therefore, meeting the informational, resources, emotional, social, and monetary needs of the caregiver would be crucial to reduce their burden.
                <sup>
                    <xref ref-type="bibr" rid="ref16">16</xref>
                </sup>
                <sup>,</sup>
                <sup>
                    <xref ref-type="bibr" rid="ref17">17</xref>
                </sup> Moreover, since children with CP require long-term multidisciplinary care, providing a continuum of care through a family-centered approach may be able to reduce the caregiver burden, enhance their capacities and empower them to care for their children.
                <sup>
                    <xref ref-type="bibr" rid="ref1">1</xref>
                </sup>
                <sup>,</sup>
                <sup>
                    <xref ref-type="bibr" rid="ref5">5</xref>
                </sup> This will help improve health consequences for both children and their caregivers as well as facilitate their active participation in the community.
                <sup>
                    <xref ref-type="bibr" rid="ref18">18</xref>
                </sup>
            </p>
            <p>A review of systematic reviews on family-centered care interventions by Park 
                <italic toggle="yes">et al.</italic> (2018),
                <sup>
                    <xref ref-type="bibr" rid="ref4">4</xref>
                </sup> provided evidence of the benefits of family-centered care interventions on patients, families and healthcare professionals. However, this study pertained to varied patient populations. A systematic review of family-centered care for children with special healthcare needs by Kuhlthau 
                <italic toggle="yes">et al.</italic> (2011) also found positive effects on health, family function and impact, satisfaction, and communication.
                <sup>
                    <xref ref-type="bibr" rid="ref2">2</xref>
                </sup> However, there is a dearth of literature assessing the effectiveness of family-centered care interventions on the well-being of caregivers of children with CP. A comprehensive synthesis of the effectiveness of FCC is essential to provide reliable evidence to practitioners, researchers, and policymakers for the development of strategies for the implementation of care, and hence pave the way for the effective delivery of services to the CP community. Hence, the primary objective of this review was to synthesize evidence on the effectiveness of family-centered care interventions on the well-being of caregivers of children with CP. Realizing the importance of families as a resource in care delivery, it is crucial to identify the best way of empowering them, meeting their needs, and incorporating their participation in therapy. Therefore, our secondary objective was to identify the components of family-centered intervention that are commonly practiced and deemed to be most effective for caregiver well-being.</p>
        </sec>
        <sec id="sec2" sec-type="methods">
            <title>Methods</title>
            <sec id="sec3">
                <title>Protocol registration</title>
                <p>This systematic review was conducted in accordance with the preferred reporting items for systematic reviews and meta-analyses 2020 (PRISMA) guidelines.
                    <sup>
                        <xref ref-type="bibr" rid="ref19">19</xref>
                    </sup> The systematic review protocol was prospectively registered with PROSPERO (No. CRD42021233854) and can be accessed at 
                    <ext-link ext-link-type="uri" xlink:href="https://www.crd.york.ac.uk/prospero/display_record.php?ID=CRD42021233854">https://www.crd.york.ac.uk/prospero/display_record.php?ID=CRD42021233854</ext-link>.</p>
            </sec>
            <sec id="sec4">
                <title>Search strategy</title>
                <p>Seven databases - Cochrane, Pubmed, Scopus, CINAHL Plus, EMBASE, Web of Science, and ProQuest - were searched from inception to 30
                    <sup>th</sup> September 2022. A systematic search strategy (dataset 1 in Extended data) was used employing the PICO format using filters - Human and English.
                    <sup>
                        <xref ref-type="bibr" rid="ref20">20</xref>
                    </sup> Furthermore, we examined the reference list of the included articles to identify any relevant articles for this review.</p>
            </sec>
            <sec id="sec5">
                <title>Eligibility criteria</title>
                <p>Randomized controlled trials (RCTs) that assessed the effectiveness of the Family-centered approach on caregiver well-being, and conformed to our inclusion criteria were included. The population was limited to primary caregivers of age above 18 years, providing care for children with or at risk of CP at any severity (any level of GMFCS) and up to 18 years. However, studies that included caregivers who are not the primary caregiver of the child, with a diagnosed psychiatric illness, or have children with any other physical disability were excluded. Articles with children with multiple disabilities were excluded if authors failed to provide information and sub-group analysis for children with CP and their caregivers within two weeks of email request. Any intervention which is family driven or has the core components of family-centered care were included: Respectful and supportive care - Social or peer support groups; Information provision - Information sharing, caregiver education through direct education employing online presentation or guiding manual or web-based education, caregiver skill training, caregiver instructions; Co-ordinated and comprehensive care - Interdisciplinary communication, multidisciplinary approach or rehabilitation; Enabling and partnership - collaborative relationship with the caregivers, collaborative goal setting, joint/shared decision making, activity selection, ongoing evaluation, parent-professional partnership, parental advocacy.
                    <sup>
                        <xref ref-type="bibr" rid="ref18">18</xref>
                    </sup> The last component &#x2018;General information&#x2019; was not included as a part of FCC in this review as it often forms a part of usual care. Studies comparing the intervention to standard practice such as regular care advised by the paediatrician or other health professionals applied in any setting were included. Studies that reported any well-being outcomes related to caregivers such as quality of life, physical health and fitness, psychological health, satisfaction, family empowerment, adaptation, burden, and level of knowledge were primarily included. Secondary outcomes evaluating the health and well-being of children with CP were also noted but were not necessary for inclusion. All non-human studies and those not in the English language were excluded.</p>
            </sec>
            <sec id="sec6">
                <title>Data screening and extraction</title>
                <p>Data screening and selection were done using Rayyan software (alternative to Covidence or DistillerSR). Two reviewers (DP and RA) independently performed title and abstract, and full-text screening on Rayyan software. Any discord between the two reviewers was settled by consensus. If disagreement persisted, it was settled by team discussions with other researchers in this review (SK, SKD). Independent double data extraction was performed by two reviewers (DP and RA) using a data collection form prepared on Microsoft Word, and discrepancies were handled via discussions. The following data were extracted from each study: Basic study details- author, setting, study design, year of publication; sample size, eligibility criteria; Characteristics of caregivers - age, sex, education, occupation, type of family, socio-demographic details; Characteristics of children with CP - age, sex, type of CP, GMFCS level, MACS level; Intervention details using TIDieR checklist; Results of outcomes - outcome measures, time points, statistical analysis methods such as measures of mean, median, SD, interquartile range, confidence interval, effect size, p-value, and missing information. If effect size was not reported, wherever possible Cohen&#x2019;s d was calculated.
                    <sup>
                        <xref ref-type="bibr" rid="ref21">21</xref>
                    </sup> Other methods were also used to calculate effect size from odds ratio and median values.
                    <sup>
                        <xref ref-type="bibr" rid="ref22">22</xref>
                    </sup>
                    <sup>,</sup>
                    <sup>
                        <xref ref-type="bibr" rid="ref23">23</xref>
                    </sup> The corresponding authors were contacted for any missing or unclear information.</p>
                <p>As there is a large disparity amongst the included studies in type and dose of intervention, and outcome measures, conducting a meta-analysis is not valid. Therefore, narrative synthesis was chosen to answer the objectives of this review. We classified the studies based on the components of family-centered care (as discussed under eligibility criteria) reflected in their interventions. An intervention may include more than one FCC component. Therefore, for the purpose of synthesis, studies with similar combinations of FCC domains will be combined, compared, and contrasted.</p>
            </sec>
            <sec id="sec7">
                <title>Quality assessment</title>
                <p>To assess the quality of the included studies, two reviewers (DP and RA) independently scored the risk of bias using the Cochrane &#x2018;Risk of Bias 2&#x2019; (RoB 2) tool for randomized trials.
                    <sup>
                        <xref ref-type="bibr" rid="ref24">24</xref>
                    </sup> Disagreements between reviewers were resolved through discussion or expert advice from a third reviewer (SK or SKD). Authors of studies were directly contacted if the target information was unreported or unclear. The studies were summarized as having low risk, some concern, or high risk of bias. For critically appraising the RCTs, CASP Randomised Controlled Trial Standard Checklist
                    <sup>
                        <xref ref-type="bibr" rid="ref25">25</xref>
                    </sup> was scored independently by two reviewers (DP and RA). Any disagreements were solved via discussions or by involving a third reviewer (SK or SKD). No scoring system was used as recommended by the CASP checklist developers.</p>
                <p>A traffic light system was used to categorize the effectiveness of different outcome domains across the studies to summarize the effectiveness of FCC interventions on caregiver well-being and infant outcomes. Moderate to large effect sizes in a low/some concern risk of bias study were coded green. Small effect sizes in a low/some concerns risk of bias study, or moderate and large effect sizes in a high risk of bias study were coded yellow. No or negative effect was indicated via red colour. The green, yellow and red colour coding indicate advice for implementing the intervention in clinical practice as- effective, use with caution, and not effective respectively.</p>
            </sec>
        </sec>
        <sec id="sec8" sec-type="results">
            <title>Results</title>
            <p>The database search yielded 1,544 studies, and an additional 12 articles were found through a secondary search. After removing 28 duplicates, 1,528 studies were screened for title and abstract eligibility, with 1,414 studies getting excluded. 114 articles underwent full-text examination, of which 99 articles were excluded for various reasons reported below. Out of the 15 articles included, n=7 studies were published as a single paper, and n=4 studies were published as eight papers. Therefore, 11 unique articles are included in this synthesis. The PRISMA Flow chart in 
                <xref ref-type="fig" rid="f1">Figure 1</xref> depicts the results of the search process, and the PRISMA 2020 checklist is provided in data set 2 in Extended data.</p>
            <fig fig-type="figure" id="f1" orientation="portrait" position="float">
                <label>Figure 1. </label>
                <caption>
                    <title>PRISMA 2020 flow diagram.</title>
                    <p>11 unique articles are included in this synthesis.</p>
                </caption>
                <graphic id="gr1" orientation="portrait" position="float" xlink:href="https://f1000research-files.f1000.com/manuscripts/146293/3fc20874-c5ba-4d93-86be-92f106ed7e12_figure1.gif"/>
            </fig>
            <sec id="sec9">
                <title>Study characteristics</title>
                <p>All the studies are randomized controlled trials, of which nine studies involved children with CP,
                    <sup>
                        <xref ref-type="bibr" rid="ref6">6</xref>
                    </sup>
                    <sup>,</sup>
                    <sup>
                        <xref ref-type="bibr" rid="ref26">26</xref>
                    </sup>
                    <sup>&#x2013;</sup>
                    <sup>
                        <xref ref-type="bibr" rid="ref33">33</xref>
                    </sup> one study involved infants at high risk of CP,
                    <sup>
                        <xref ref-type="bibr" rid="ref34">34</xref>
                    </sup> and lastly, one study involved both children diagnosed or at high risk of CP.
                    <sup>
                        <xref ref-type="bibr" rid="ref35">35</xref>
                    </sup> Three studies were located in Australia,
                    <sup>
                        <xref ref-type="bibr" rid="ref29">29</xref>
                    </sup>
                    <sup>,</sup>
                    <sup>
                        <xref ref-type="bibr" rid="ref32">32</xref>
                    </sup>
                    <sup>,</sup>
                    <sup>
                        <xref ref-type="bibr" rid="ref34">34</xref>
                    </sup> and one each in the United Kingdom,
                    <sup>
                        <xref ref-type="bibr" rid="ref33">33</xref>
                    </sup> Tanzania,
                    <sup>
                        <xref ref-type="bibr" rid="ref26">26</xref>
                    </sup> Netherlands,
                    <sup>
                        <xref ref-type="bibr" rid="ref35">35</xref>
                    </sup> Norway,
                    <sup>
                        <xref ref-type="bibr" rid="ref31">31</xref>
                    </sup> Brazil,
                    <sup>
                        <xref ref-type="bibr" rid="ref28">28</xref>
                    </sup> Iran,
                    <sup>
                        <xref ref-type="bibr" rid="ref6">6</xref>
                    </sup> Canada,
                    <sup>
                        <xref ref-type="bibr" rid="ref30">30</xref>
                    </sup> and Denmark.
                    <sup>
                        <xref ref-type="bibr" rid="ref27">27</xref>
                    </sup> In two studies,
                    <sup>
                        <xref ref-type="bibr" rid="ref29">29</xref>
                    </sup>
                    <sup>,</sup>
                    <sup>
                        <xref ref-type="bibr" rid="ref32">32</xref>
                    </sup> the interventions directly targeted the caregivers while in the remaining studies, the interventions targeted the caregiver indirectly by focussing on improvement in child-related outcomes. The sample size in these studies varied from n= 21 to n= 118, for the parents and children with CP. 
                    <xref ref-type="table" rid="T1">Table 1</xref> provides an overview of the characteristics of each study, while the TIDieR checklist (Extended data, dataset 3) details the interventions used in each trial. There was a lot of heterogeneity in the focus of interventions. Various caregiver well-being outcomes such as family needs, mental health, empowerment, parenting, satisfaction, quality of life, perception of family centeredness, or caregiver assistance, and child outcomes such as feeding, behaviour, motor or function were included. Dataset 4 in Extended data summarizes the description of outcome measures and intervention effectiveness.</p>
                <table-wrap id="T1" orientation="portrait" position="float">
                    <label>Table 1. </label>
                    <caption>
                        <title>Characteristics of the included studies.</title>
                    </caption>
                    <table content-type="article-table" frame="hsides">
                        <thead>
                            <tr>
                                <th align="left" colspan="1" rowspan="1" valign="top">Author, year, Country</th>
                                <th align="left" colspan="1" rowspan="1" valign="top">Sample Size</th>
                                <th align="left" colspan="1" rowspan="1" valign="top">Participants (Children with CP) Age in years Mean (SD) or median (IQR) Sex in %</th>
                                <th align="left" colspan="1" rowspan="1" valign="top">Participants (Caregivers) Age in years Mean (SD) or median (IQR)</th>
                                <th align="left" colspan="2" rowspan="1" valign="top">Intervention group</th>
                                <th align="left" colspan="1" rowspan="1" valign="top">Control group</th>
                            </tr>
                        </thead>
                        <tbody>
                            <tr>
                                <td align="left" colspan="1" rowspan="1" valign="top">Weindling 
                                    <italic toggle="yes">et al.</italic>, 2007
                                    <sup>
                                        <xref ref-type="bibr" rid="ref33">33</xref>
                                    </sup>
                                    <break/>United Kingdom</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">N=88
                                    <break/>Intervention 1: 31
                                    <break/>Intervention 2: 28
                                    <break/>Control: 29</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Children with spastic CP &lt; 4 years
                                    <break/>FSWG: 21.2 &#x00b1; 9.2 months, 59% male
                                    <break/>PAG: 19.3 &#x00b1; 8.7 months, 57% males
                                    <break/>Control: 18.9 &#x00b1; 8.7 months, 68% males</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Caregivers of children with CP
                                    <break/>Maternal age: 30.9 &#x00b1; 0.2 years
                                    <break/>Paternal age 34.4 &#x00b1; 6.6 years</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Intervention 1: Family support worker group (FSWG): 
                                    <break/>Standard physiotherapy + Family support worker to discuss family needs and provide support</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Intervention 2: Physiotherapy assistant group (PAG): Extra Physiotherapy along with standard physiotherapy to increase dose of intervention</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Standard Physiotherapy (mainly NDT)</td>
                            </tr>
                            <tr>
                                <td align="left" colspan="1" rowspan="1" valign="top">Mlinda 
                                    <italic toggle="yes">et al.</italic>, 2018
                                    <sup>
                                        <xref ref-type="bibr" rid="ref26">26</xref>
                                    </sup>
                                    <break/>Tanzania</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">N= 118
                                    <break/>Intervention: 69
                                    <break/>Control: 49</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Children &lt; 5 years with moderate-to-severe CP
                                    <break/>Intervention: 28.5 (12.3) months, 46.0% males
                                    <break/>Control: 28.9 (13.0) months, 48.9% males</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Caregivers of children with CP
                                    <break/>Intervention: 30.3 &#x00b1; 5.2 years
                                    <break/>Control: 31.5 &#x00b1; 5.34 years</td>
                                <td align="left" colspan="2" rowspan="1" valign="top">Nutrition education and training on feeding and positioning skills</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Usual Care</td>
                            </tr>
                            <tr>
                                <td align="left" colspan="1" rowspan="1" valign="top">Whittingham 
                                    <italic toggle="yes">et al.</italic>, 2014
                                    <sup>
                                        <xref ref-type="bibr" rid="ref32">32</xref>
                                    </sup>
                                    <break/>Secondary study: Whittingham 
                                    <italic toggle="yes">et al.</italic>, 2016
                                    <sup>
                                        <xref ref-type="bibr" rid="ref39">39</xref>
                                    </sup>
                                    <break/>Australia</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">N= 67
                                    <break/>Intervention 1: 22
                                    <break/>Intervention 2: 23
                                    <break/>Control: 22</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Children with CP, 2&#x2013; 12 years of age, GMFCS level I-V
                                    <break/>SSTP: 5.45 &#x00b1; 3.16 years, 59.1 % boys
                                    <break/>SSTP + ACT: 5.52 &#x00b1; 3.17 years, 73.9 % boys
                                    <break/>Waitlist: 4.96 &#x00b1; 2.95 years, 59.1% boys</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Parents of children with CP who self-identified the need for a parenting intervention
                                    <break/>SSTP: 38.67 (5.55) years
                                    <break/>SSTP + ACT: 37.88 (9.39) years
                                    <break/>Waitlist: 39.65 (6.09) years</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Intervention 1: Stepping Stones Triple P (SSTP)
                                    <break/>Parenting Interventions for targeting behavioural and emotional problems in childhood</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Intervention 2: Stepping Stones Triple P and Acceptance and Commitment Therapy (SSTP + ACT)
                                    <break/>ACT: Cognitive behavioural therapy for improving psychological flexibility to handle behavioural problems</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Waitlist
                                    <break/>After postintervention assessment, SSTP was offered.</td>
                            </tr>
                            <tr>
                                <td align="left" colspan="1" rowspan="1" valign="top">Whittingham 
                                    <italic toggle="yes">et al.</italic>, 2022
                                    <sup>
                                        <xref ref-type="bibr" rid="ref29">29</xref>
                                    </sup>
                                    <break/>Australia</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">N= 67
                                    <break/>Intervention: 37
                                    <break/>Control: 30</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Child with CP, between 2-10 years
                                    <break/>Intervention: 5 years 8 months &#x00b1; 2.36, 59% males
                                    <break/>Waitlist: 5 years 6 months &#x00b1; 2.60, 87% males</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Parents of children with CP
                                    <break/>Age not reported</td>
                                <td align="left" colspan="2" rowspan="1" valign="top">Immediate Parenting Acceptance and Commitment Therapy (PACT) intervention via an online course to improve psychological flexibility to parent this population</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Waitlist control</td>
                            </tr>
                            <tr>
                                <td align="left" colspan="1" rowspan="1" valign="top">Hielkema 
                                    <italic toggle="yes">et al.</italic>, 2020
                                    <sup>
                                        <xref ref-type="bibr" rid="ref35">35</xref>
                                    </sup>
                                    <break/>Secondary study: Hielkema 
                                    <italic toggle="yes">et al.</italic>, 2020
                                    <sup>
                                        <xref ref-type="bibr" rid="ref37">37</xref>
                                    </sup>
                                    <break/>Netherlands</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">N=43
                                    <break/>Intervention: 23
                                    <break/>Control: 20</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Infants at very high-risk of CP before 9 months CA
                                    <break/>Intervention: 1.4 (0.7&#x2013;2.8) months, 65% males
                                    <break/>Control: 2.5 (1.8&#x2013;4.7) months, 55% males</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Parents of infants at high risk of CP
                                    <break/>Intervention: 29 (27&#x2013;35) years
                                    <break/>Control: 31 (29&#x2013;35) years</td>
                                <td align="left" colspan="2" rowspan="1" valign="top">Coping with and caring for infants with special needs (COPCA) intervention involving parent coaching and adaptive infant motor training</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Typical infant physiotherapy (TIP)
                                    <break/>NDT with a functional approach</td>
                            </tr>
                            <tr>
                                <td align="left" colspan="1" rowspan="1" valign="top">Saquetto 
                                    <italic toggle="yes">et al.</italic>, 2018
                                    <sup>
                                        <xref ref-type="bibr" rid="ref28">28</xref>
                                    </sup>
                                    <break/>Brazil</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">N=60
                                    <break/>Intervention: 29
                                    <break/>Control: 31</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Children with CP between 1&#x2013;12 years of age.
                                    <break/>Intervention: 4.66 &#x00b1; 2.78 years, 41.4% boys
                                    <break/>Control: 4.52 &#x00b1; 2.71, 71% boys</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Full-time caregiver of child with CP
                                    <break/>Intervention: 33.38 &#x00b1; 9.6 years
                                    <break/>Control: 34.42&#x00b1;10.92 years</td>
                                <td align="left" colspan="2" rowspan="1" valign="top">Educational programme for primary caregivers to create opportunities for their infants to practice motor control via everyday activities along with conventional rehabilitation</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Conventional rehabilitation</td>
                            </tr>
                            <tr>
                                <td align="left" colspan="1" rowspan="1" valign="top">Myrhaug 
                                    <italic toggle="yes">et al.</italic>, 2018
                                    <sup>
                                        <xref ref-type="bibr" rid="ref31">31</xref>
                                    </sup>
                                    <break/>Secondary study: Myrhaug 
                                    <italic toggle="yes">et al.</italic>, 2019
                                    <sup>
                                        <xref ref-type="bibr" rid="ref38">38</xref>
                                    </sup>
                                    <break/>Norway</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">N= 21
                                    <break/>Intervention: 11
                                    <break/>Control: 10</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Children with CP between 3&#x2013;6 years. All types and functional levels of CP who are eligible for CE courses
                                    <break/>Intervention: 4 (3&#x2013;4.5) years, 45% male
                                    <break/>Control: 4 (3&#x2013;4) years,
                                    <break/>70% males</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Parents of children with CP
                                    <break/>Age not reported</td>
                                <td align="left" colspan="2" rowspan="1" valign="top">Conductive education (CE) followed by conventional practice</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Waiting list: Conventional practice (Functional training)</td>
                            </tr>
                            <tr>
                                <td align="left" colspan="1" rowspan="1" valign="top">Kahjoogh 
                                    <italic toggle="yes">et al.</italic>, 2019
                                    <sup>
                                        <xref ref-type="bibr" rid="ref6">6</xref>
                                    </sup>
                                    <break/>Iran</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">N=30
                                    <break/>Intervention: 15
                                    <break/>Control: 15</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Children with CP, aged 5&#x2013;11 years with learning capacity
                                    <break/>GMFCS level I-V
                                    <break/>Intervention: 6.64 &#x00b1; 0.97, 46.7% boys
                                    <break/>Control: Age 7.56 &#x00b1; 1.59, 66.7% boys</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Mothers between 25 and 50 years
                                    <break/>Intervention: 34.69 &#x00b1; 4.29 years
                                    <break/>Control: 38.22 &#x00b1; 5.98 years</td>
                                <td align="left" colspan="2" rowspan="1" valign="top">Occupational Performance Coaching (OPC) providing emotional support, information and a structured problem solving process that helped achieve goals along with standard occupational therapy services</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Standard occupational therapy services (mainly NDT)</td>
                            </tr>
                            <tr>
                                <td align="left" colspan="1" rowspan="1" valign="top">Morgan 
                                    <italic toggle="yes">et al.</italic>, 2016
                                    <sup>
                                        <xref ref-type="bibr" rid="ref34">34</xref>
                                    </sup>
                                    <break/>Australia</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">N=30
                                    <break/>Intervention: 15
                                    <break/>Control: 15</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Infants aged between 3 and 6 months CA with a diagnosis of CP or at high-risk of CP
                                    <break/>Intervention: 15.73 &#x00b1; 4.76 weeks, 53% boys
                                    <break/>Control: 20.07 &#x00b1; 5.08 weeks, 60% boys</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Mothers of children with CP
                                    <break/>Intervention: 33.73 (4.73) years
                                    <break/>Control: 31.07 &#x00b1; 7.11 years</td>
                                <td align="left" colspan="2" rowspan="1" valign="top">Goals, Activity and Motor Enrichment (GAME) intervention involving task practice using motor learning strategies delivered along with parent education and involvement.</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Standard care</td>
                            </tr>
                            <tr>
                                <td align="left" colspan="1" rowspan="1" valign="top">Law 
                                    <italic toggle="yes">et al.</italic>, 2011
                                    <sup>
                                        <xref ref-type="bibr" rid="ref30">30</xref>
                                    </sup>
                                    <break/>Canada</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">N=91
                                    <break/>Intervention: 67
                                    <break/>Control: 79</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Children between 12 months and 5 years 11 months of age, diagnosed with CP, and all GMFCS levels.
                                    <break/>Intervention: 3.92 &#x00b1; 1.42 years, 51% males
                                    <break/>Control: 3.53 &#x00b1; 1.43 years, 70% males</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Parents of children with CP
                                    <break/>Age not reported</td>
                                <td align="left" colspan="2" rowspan="1" valign="top">Context-Focused Approach that involved parents to change constraints in task or environment that hinder child&#x2019;s performance</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Child-Focused Approach: contemporary interventions such as ROM, weight-bearing, etc</td>
                            </tr>
                            <tr>
                                <td align="left" colspan="1" rowspan="1" valign="top">Fonvig 
                                    <italic toggle="yes">et al.</italic>, 2020
                                    <sup>
                                        <xref ref-type="bibr" rid="ref27">27</xref>
                                    </sup>
                                    <break/>Secondary study: Rasmussen et al., 2019
                                    <sup>
                                        <xref ref-type="bibr" rid="ref36">36</xref>
                                    </sup>
                                    <break/>Denmark</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">N=60
                                    <break/>Intervention: 30
                                    <break/>Control: 30</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Children with spastic CP, between 5-8 years, GMFCS level I-II
                                    <break/>Median age: 6 years and 10 months
                                    <break/>Intervention: Median 6y 6m (2y 8m), 70% boys
                                    <break/>Control: Median 6y 11m (1y 10 m), 60% boys</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Parents of children with CP
                                    <break/>Age not reported</td>
                                <td align="left" colspan="2" rowspan="1" valign="top">Individually tailored interdisciplinary intervention based on recommendations from clinical examination as well as an IGA report. Family involvement in planning treatment.</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Standard care: Individually tailored interdisciplinary intervention based on clinical examinations without an IGA report.</td>
                            </tr>
                        </tbody>
                    </table>
                    <table-wrap-foot>
                        <p>
                            <bold>Note.</bold> ACT: Acceptance and Commitment Therapy; CA: Corrected age; CE: Conductive education; COPCA: Coping with and Caring for infants with special needs; CP: Cerebral Palsy; FSWG: Family support worker; GAME: Goals, Activity and Motor Enrichment; GMFCS: Gross Motor Function Classification System; IQR: Inter-quartile range; NDT: neurodevelopmental therapy; OPC: Occupational Performance Coaching; PACT: Parenting Acceptance and Commitment Therapy; PAG: physiotherapy assistant group; SSTP: Stepping Stones Triple P; TIP: Typical infant physiotherapy;</p>
                    </table-wrap-foot>
                </table-wrap>
            </sec>
            <sec id="sec10">
                <title>ROB and Quality Assessment</title>
                <p>According to the Cochrane ROB 2.0 tool for RCTs, four RCTs had low ROB,
                    <sup>
                        <xref ref-type="bibr" rid="ref6">6</xref>
                    </sup>
                    <sup>,</sup>
                    <sup>
                        <xref ref-type="bibr" rid="ref28">28</xref>
                    </sup>
                    <sup>,</sup>
                    <sup>
                        <xref ref-type="bibr" rid="ref29">29</xref>
                    </sup>
                    <sup>,</sup>
                    <sup>
                        <xref ref-type="bibr" rid="ref31">31</xref>
                    </sup> two RCTs had some concerns,
                    <sup>
                        <xref ref-type="bibr" rid="ref27">27</xref>
                    </sup>
                    <sup>,</sup>
                    <sup>
                        <xref ref-type="bibr" rid="ref34">34</xref>
                    </sup> and five had a high ROB
                    <sup>
                        <xref ref-type="bibr" rid="ref26">26</xref>
                    </sup>
                    <sup>,</sup>
                    <sup>
                        <xref ref-type="bibr" rid="ref30">30</xref>
                    </sup>
                    <sup>,</sup>
                    <sup>
                        <xref ref-type="bibr" rid="ref32">32</xref>
                    </sup>
                    <sup>,</sup>
                    <sup>
                        <xref ref-type="bibr" rid="ref33">33</xref>
                    </sup>
                    <sup>,</sup>
                    <sup>
                        <xref ref-type="bibr" rid="ref35">35</xref>
                    </sup> (
                    <xref ref-type="fig" rid="f2">Figure 2</xref>). As our review focussed on caregiver well-being, the majority of caregiver outcomes were patient-reported. Moreover, given the nature of the intervention, the caregivers could not have been blinded to the intervention, and by default that influenced the ROB grading. Therefore, the risk of bias domain assessing outcome assessor&#x2019;s awareness of intervention and its influence on outcome was rated as &#x2018;probably no&#x2019; to avoid categorizing as high-risk of bias on this specific basis. 
                    <xref ref-type="table" rid="T2">Table 2</xref> represents the results of the CASP Randomised Controlled Trial Standard Checklist. The majority of the studies showed limitations in allocation concealment, investigator and participant blinding, and reporting adverse effects and costs of intervention.</p>
                <fig fig-type="figure" id="f2" orientation="portrait" position="float">
                    <label>Figure 2. </label>
                    <caption>
                        <title>Risk of bias of included studies.</title>
                        <p>Risk of bias was low (green) in four studies,
                            <sup>
                                <xref ref-type="bibr" rid="ref6">6</xref>
                            </sup>
                            <sup>,</sup>
                            <sup>
                                <xref ref-type="bibr" rid="ref28">28</xref>
                            </sup>
                            <sup>,</sup>
                            <sup>
                                <xref ref-type="bibr" rid="ref29">29</xref>
                            </sup>
                            <sup>,</sup>
                            <sup>
                                <xref ref-type="bibr" rid="ref31">31</xref>
                            </sup> unclear (yellow) in two studies,
                            <sup>
                                <xref ref-type="bibr" rid="ref27">27</xref>
                            </sup>
                            <sup>,</sup>
                            <sup>
                                <xref ref-type="bibr" rid="ref34">34</xref>
                            </sup> and high (red) in five studies.
                            <sup>
                                <xref ref-type="bibr" rid="ref26">26</xref>
                            </sup>
                            <sup>,</sup>
                            <sup>
                                <xref ref-type="bibr" rid="ref30">30</xref>
                            </sup>
                            <sup>,</sup>
                            <sup>
                                <xref ref-type="bibr" rid="ref32">32</xref>
                            </sup>
                            <sup>,</sup>
                            <sup>
                                <xref ref-type="bibr" rid="ref33">33</xref>
                            </sup>
                            <sup>,</sup>
                            <sup>
                                <xref ref-type="bibr" rid="ref35">35</xref>
                            </sup>
                        </p>
                    </caption>
                    <graphic id="gr2" orientation="portrait" position="float" xlink:href="https://f1000research-files.f1000.com/manuscripts/146293/3fc20874-c5ba-4d93-86be-92f106ed7e12_figure2.gif"/>
                </fig>
                <table-wrap id="T2" orientation="portrait" position="float">
                    <label>Table 2. </label>
                    <caption>
                        <title>The results of the critical appraisal using CASP checklist.</title>
                    </caption>
                    <table content-type="article-table" frame="hsides">
                        <thead>
                            <tr>
                                <th align="left" colspan="1" rowspan="2" valign="top">Author</th>
                                <th align="left" colspan="1" rowspan="2" valign="top">Item 1</th>
                                <th align="left" colspan="1" rowspan="2" valign="top">Item 2</th>
                                <th align="left" colspan="1" rowspan="2" valign="top">Item 3</th>
                                <th align="left" colspan="3" rowspan="1" valign="top">Item 4</th>
                                <th align="left" colspan="1" rowspan="2" valign="top">Item 5</th>
                                <th align="left" colspan="1" rowspan="2" valign="top">Item 6</th>
                                <th align="left" colspan="1" rowspan="2" valign="top">Item 7</th>
                                <th align="left" colspan="1" rowspan="2" valign="top">Item 8</th>
                                <th align="left" colspan="1" rowspan="2" valign="top">Item 9</th>
                                <th align="left" colspan="1" rowspan="2" valign="top">Item 10</th>
                                <th align="left" colspan="1" rowspan="2" valign="top">Item 11</th>
                            </tr>
                            <tr>
                                <th align="left" colspan="1" rowspan="1" valign="top">4A</th>
                                <th align="left" colspan="1" rowspan="1" valign="top">4B</th>
                                <th align="left" colspan="1" rowspan="1" valign="top">4C</th>
                            </tr>
                        </thead>
                        <tbody>
                            <tr>
                                <td align="left" colspan="1" rowspan="1" valign="top">Weindling 
                                    <italic toggle="yes">et al.</italic>, 2007
                                    <sup>
                                        <xref ref-type="bibr" rid="ref33">33</xref>
                                    </sup>
                                </td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">N</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">N</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">C</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">C</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">C</td>
                            </tr>
                            <tr>
                                <td align="left" colspan="1" rowspan="1" valign="top">Mlinda 
                                    <italic toggle="yes">et al.,</italic> 2018
                                    <sup>
                                        <xref ref-type="bibr" rid="ref26">26</xref>
                                    </sup>
                                </td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">C</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">N</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">N</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">N</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">C</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">C</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                            </tr>
                            <tr>
                                <td align="left" colspan="1" rowspan="1" valign="top">Whittingham 
                                    <italic toggle="yes">et al.</italic>, 2014
                                    <sup>
                                        <xref ref-type="bibr" rid="ref32">32</xref>
                                    </sup>
                                </td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">N</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">N</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">N</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">C</td>
                            </tr>
                            <tr>
                                <td align="left" colspan="1" rowspan="1" valign="top">Whittingham 
                                    <italic toggle="yes">et al.</italic>, 2022
                                    <sup>
                                        <xref ref-type="bibr" rid="ref29">29</xref>
                                    </sup>
                                </td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">N</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">N</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">C</td>
                            </tr>
                            <tr>
                                <td align="left" colspan="1" rowspan="1" valign="top">Hielkema 
                                    <italic toggle="yes">et al.,</italic> 2020
                                    <sup>
                                        <xref ref-type="bibr" rid="ref35">35</xref>
                                    </sup>
                                </td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">N</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">N</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">N</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">C</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">C</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">C</td>
                            </tr>
                            <tr>
                                <td align="left" colspan="1" rowspan="1" valign="top">Saquetto 
                                    <italic toggle="yes">et al.</italic>, 2018
                                    <sup>
                                        <xref ref-type="bibr" rid="ref28">28</xref>
                                    </sup>
                                </td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">N</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">N</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                            </tr>
                            <tr>
                                <td align="left" colspan="1" rowspan="1" valign="top">Myrhaug 
                                    <italic toggle="yes">et al.</italic>, 2018
                                    <sup>
                                        <xref ref-type="bibr" rid="ref31">31</xref>
                                    </sup>
                                </td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">N</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">N</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">C</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">C</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">N</td>
                            </tr>
                            <tr>
                                <td align="left" colspan="1" rowspan="1" valign="top">Kahjoogh 
                                    <italic toggle="yes">et al.</italic>, 2019
                                    <sup>
                                        <xref ref-type="bibr" rid="ref6">6</xref>
                                    </sup>
                                </td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">N</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">N</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">C</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">C</td>
                            </tr>
                            <tr>
                                <td align="left" colspan="1" rowspan="1" valign="top">Morgan 
                                    <italic toggle="yes">et al.,</italic> 2016
                                    <sup>
                                        <xref ref-type="bibr" rid="ref34">34</xref>
                                    </sup>
                                </td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">N</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">N</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">C</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                            </tr>
                            <tr>
                                <td align="left" colspan="1" rowspan="1" valign="top">Law 
                                    <italic toggle="yes">et al.,</italic> 2011
                                    <sup>
                                        <xref ref-type="bibr" rid="ref30">30</xref>
                                    </sup>
                                </td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">C</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">N</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">N</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">C</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">N</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">C</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">C</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">C</td>
                            </tr>
                            <tr>
                                <td align="left" colspan="1" rowspan="1" valign="top">Fonvig. 
                                    <italic toggle="yes">et al.,</italic> 2020
                                    <sup>
                                        <xref ref-type="bibr" rid="ref27">27</xref>
                                    </sup>
                                </td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">N</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">N</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">Y</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">C</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">C</td>
                                <td align="left" colspan="1" rowspan="1" valign="top">N</td>
                            </tr>
                        </tbody>
                    </table>
                    <table-wrap-foot>
                        <p>Note: Item 1: Clear research question; Item 2: Random assignment of participants to intervention; Item 3: Accounting all participants at conclusion; Item 4: Blinding; 4A: Participant blinding; 4B: Investigator blinding; 4C: Assessor blinding; Item 5: Groups similar at baseline; Item 6: Equal treatment for each study group; Item 7: Intervention effects reported comprehensively; Item 8: Reported precision of the estimate of the intervention effect; Item 9: Benefits of the experimental intervention outweigh the harms and costs; Item 10: Results applicable to your local population; Item 11: Experimental intervention provide greater value to the people in our care than any of the existing interventions; CASP: Critical Appraisal Skills Programme; Y: yes; C: Can't tell; N: No.</p>
                    </table-wrap-foot>
                </table-wrap>
                <p>
                    <xref ref-type="fig" rid="f3">Figure 3</xref> demonstrates the overall effect of FCC interventions on caregiver well-being using the traffic light system. FCC interventions are effective (green) in improving caregivers&#x2019; satisfaction with attainment of child and caregiver goals. Evidence from a single study indicates that FCC interventions should be used with caution (yellow) to improve family needs and feeding skills. There is inconclusive evidence on the quality of life. Lastly, FCC interventions are not effective (red) to improve caregiver&#x2019;s mental health, personal outcomes such as empowerment, parenting skills, perception of family centeredness (except information provision about the child), and reduce caregiver assistance in daily activities.</p>
                <fig fig-type="figure" id="f3" orientation="portrait" position="float">
                    <label>Figure 3. </label>
                    <caption>
                        <title>Overall effect of FCC interventions using the traffic light system.</title>
                        <p>Green: moderate to large effect sizes in a low/some concern risk of bias study. Yellow: small effect sizes in a low/some concerns risk of bias study, or moderate and large effect sizes in a high risk of bias study. Red: no or negative effect. *Green on only one domain of Measure of Processes of Care scale (information about child).</p>
                    </caption>
                    <graphic id="gr3" orientation="portrait" position="float" xlink:href="https://f1000research-files.f1000.com/manuscripts/146293/3fc20874-c5ba-4d93-86be-92f106ed7e12_figure3.gif"/>
                </fig>
                <p>Since the studies include different components of FCC in their interventions, the following section classifies the results based on the core components of family-centered care reflected in the studies. Only two studies incorporated only one FCC component- Enabling and partnership,
                    <sup>
                        <xref ref-type="bibr" rid="ref30">30</xref>
                    </sup>
                    <sup>,</sup>
                    <sup>
                        <xref ref-type="bibr" rid="ref31">31</xref>
                    </sup> whereas the remaining studies involved multiple FCC components in their intervention.
                    <sup>
                        <xref ref-type="bibr" rid="ref26">26</xref>
                    </sup>
                    <sup>&#x2013;</sup>
                    <sup>
                        <xref ref-type="bibr" rid="ref29">29</xref>
                    </sup>
                    <sup>,</sup>
                    <sup>
                        <xref ref-type="bibr" rid="ref32">32</xref>
                    </sup>
                    <sup>&#x2013;</sup>
                    <sup>
                        <xref ref-type="bibr" rid="ref35">35</xref>
                    </sup> The interventions targeted different needs of the caregiver such as social or informational support. Various modes of delivering the intervention such as online or offline platforms, actively through discussions, or passively through mailing child-related information reports were utilized.</p>
            </sec>
            <sec id="sec11">
                <title>&#x2018;Information provision&#x2019; and &#x2018;Enabling and partnership&#x2019;</title>
                <p>Five studies utilized these FCC components in their interventions.
                    <sup>
                        <xref ref-type="bibr" rid="ref26">26</xref>
                    </sup>
                    <sup>&#x2013;</sup>
                    <sup>
                        <xref ref-type="bibr" rid="ref28">28</xref>
                    </sup>
                    <sup>,</sup>
                    <sup>
                        <xref ref-type="bibr" rid="ref34">34</xref>
                    </sup>
                    <sup>,</sup>
                    <sup>
                        <xref ref-type="bibr" rid="ref35">35</xref>
                    </sup> The results of these papers are also published in two secondary studies from the same sample.
                    <sup>
                        <xref ref-type="bibr" rid="ref36">36</xref>
                    </sup>
                    <sup>,</sup>
                    <sup>
                        <xref ref-type="bibr" rid="ref37">37</xref>
                    </sup> Two studies, Morgan 
                    <italic toggle="yes">et al.</italic> (2016)
                    <sup>
                        <xref ref-type="bibr" rid="ref34">34</xref>
                    </sup> and Hielkema 
                    <italic toggle="yes">et al.</italic> (2020)
                    <sup>
                        <xref ref-type="bibr" rid="ref35">35</xref>
                    </sup> involved infants at very high risk of CP ranging from 3 to 9 months corrected age. The intervention in one study
                    <sup>
                        <xref ref-type="bibr" rid="ref34">34</xref>
                    </sup> followed the GAME principles (Goals, Activity and Motor Enrichment) whereas the other study
                    <sup>
                        <xref ref-type="bibr" rid="ref35">35</xref>
                    </sup> followed COPCA principles (Coping with and Caring for infants with special needs). Both studies
                    <sup>
                        <xref ref-type="bibr" rid="ref34">34</xref>
                    </sup>
                    <sup>,</sup>
                    <sup>
                        <xref ref-type="bibr" rid="ref35">35</xref>
                    </sup> involved active caregiver learning through education and training for the caregivers to become independent in identifying infant&#x2019;s movements and providing opportunities for motor task practice. Discussions and home programs directed towards a parent-identified goal were given.
                    <sup>
                        <xref ref-type="bibr" rid="ref34">34</xref>
                    </sup> However, the control groups in both studies
                    <sup>
                        <xref ref-type="bibr" rid="ref34">34</xref>
                    </sup>
                    <sup>,</sup>
                    <sup>
                        <xref ref-type="bibr" rid="ref35">35</xref>
                    </sup> had some family involvement such as parental advice on positioning, handling, feeding, and developmental simulation.
                    <sup>
                        <xref ref-type="bibr" rid="ref34">34</xref>
                    </sup>
                </p>
                <p>Improvement was seen in caregiver satisfaction with attainment of goal at 12 months on the COPM (d=0.68).
                    <sup>
                        <xref ref-type="bibr" rid="ref34">34</xref>
                    </sup> Surprisingly, both the studies did not show any improvement in parents&#x2019; mental health as measured on Depression Anxiety and Stress Scale (DASS-21)
                    <sup>
                        <xref ref-type="bibr" rid="ref34">34</xref>
                    </sup> and Nijmeegse Ouderlijke Stress Index questionnaire, short version (NOSI-K)
                    <sup>
                        <xref ref-type="bibr" rid="ref35">35</xref>
                    </sup> respectively. However, in the study by Hielkema et al. (2020), significant improvement was seen in caregivers' quality of life (d=0.46) and Infant and Toddler Quality of Life Questionnaire-parent concepts (ITQOL) (impact emotional: d=0.55, impact time: d=0.68) post-intervention.
                    <sup>
                        <xref ref-type="bibr" rid="ref35">35</xref>
                    </sup> No difference was seen in family empowerment and coping mechanisms. With respect to child outcomes, one study showed significant improvement in motor skills at 16 weeks and 12 months on PDMS-2 (d=0.09 and 0.31 respectively), GMFM (d=0.20), cognitive skills at 12 months on BSID-III (d=0.42), and on COPM performance at 16-weeks (d=0.25).
                    <sup>
                        <xref ref-type="bibr" rid="ref34">34</xref>
                    </sup> In contrast, the other study found no significant difference in infant motor, cognitive, behaviour, function outcomes, and quality of life (except general health perceptions d=0.62) as compared to the control group.
                    <sup>
                        <xref ref-type="bibr" rid="ref35">35</xref>
                    </sup>
                </p>
                <p>Three studies involved children diagnosed with CP ranging from age 1-12 years.
                    <sup>
                        <xref ref-type="bibr" rid="ref26">26</xref>
                    </sup>
                    <sup>&#x2013;</sup>
                    <sup>
                        <xref ref-type="bibr" rid="ref28">28</xref>
                    </sup> All three studies involved caregiver education to facilitate a specific function- improve nutrition and feeding skills,
                    <sup>
                        <xref ref-type="bibr" rid="ref26">26</xref>
                    </sup> become informed about the child&#x2019;s treatment for gait impairments,
                    <sup>
                        <xref ref-type="bibr" rid="ref27">27</xref>
                    </sup> and facilitate the child&#x2019;s motor tasks respectively.
                    <sup>
                        <xref ref-type="bibr" rid="ref28">28</xref>
                    </sup> Two studies had active parent involvement via multiple modes of delivering education,
                    <sup>
                        <xref ref-type="bibr" rid="ref26">26</xref>
                    </sup> and caregiver participation in goal setting and practice of functional activities.
                    <sup>
                        <xref ref-type="bibr" rid="ref28">28</xref>
                    </sup> In contrast, the study by Fonvig 
                    <italic toggle="yes">et al.</italic> (2020) only provided the information passively by mailing the instrumented gait-analysis report.
                    <sup>
                        <xref ref-type="bibr" rid="ref27">27</xref>
                    </sup> The control group in one study received general health education for parents,
                    <sup>
                        <xref ref-type="bibr" rid="ref26">26</xref>
                    </sup> whereas another study involved multidisciplinary health professionals collaboration.
                    <sup>
                        <xref ref-type="bibr" rid="ref27">27</xref>
                    </sup>
                </p>
                <p>The study by Mlinda 
                    <italic toggle="yes">et al.</italic> (2018) showed improvement in caregiver feeding skills such as positioning, feeding speed, and feeding support with effect size 0.92, 0.91, 0.69 respectively, and stress (effect size=0.5).
                    <sup>
                        <xref ref-type="bibr" rid="ref26">26</xref>
                    </sup> The study by Fonvig 
                    <italic toggle="yes">et al.</italic> (2020) showed no improvement in any domain of the MPOC-20.
                    <sup>
                        <xref ref-type="bibr" rid="ref27">27</xref>
                    </sup> The study by Saquetto 
                    <italic toggle="yes">et al.</italic> (2018) showed a large effect in the caregiver assistance required for self-care (ES=5.11) and mobility functions (ES=7.37) on the Pediatric Evaluation of Disability Inventory (PEDI) post-intervention.
                    <sup>
                        <xref ref-type="bibr" rid="ref28">28</xref>
                    </sup> These interventions improved child&#x2019;s mood (d=0.62),
                    <sup>
                        <xref ref-type="bibr" rid="ref26">26</xref>
                    </sup> gross motor function (
                    <bold>&#x03b7;</bold>
                    <sup>
                        <bold>2</bold>
                    </sup>=0.145, large effect),
                    <sup>
                        <xref ref-type="bibr" rid="ref27">27</xref>
                    </sup> and self-care skills (Effect size=2.18, large effect).
                    <sup>
                        <xref ref-type="bibr" rid="ref28">28</xref>
                    </sup>
                </p>
            </sec>
            <sec id="sec12">
                <title>&#x2018;Information provision&#x2019; and &#x2018;Respectful and supportive care&#x2019;</title>
                <p>Only one study by Whittingham 
                    <italic toggle="yes">et al.</italic> (2022)
                    <sup>
                        <xref ref-type="bibr" rid="ref29">29</xref>
                    </sup> was included in this category. In this study, an online/telehealth intervention to support the caregivers in positive parenting was delivered using online presentations, activities, and discussion. The intervention showed significant improvement at post-intervention (10 weeks) in non-intrusiveness (d=0.14) and child involvement (d=0.19) on the Emotional Availability Scale, in child involvement (d=0.28) as seen by the Emotional Availability Self Report, and in mindfulness during parenting (d=0.17) using the Interpersonal Mindfulness in Parenting Scale. Parents also reported improvement in acceptance of the child&#x2019;s CP diagnosis (d=0.64), seeking support (d=0.08), maintaining social connections (d=0.45), and meaningful living (d=0.47) post-intervention and at six months follow up. However, no intervention effect was seen on parent mental health and well-being as measured using the standard outcome measures- the Depression Anxiety and Stress Scale (DASS), the Personal Wellbeing Index (PWI), or the Acceptance and Action Questionnaire (AAQ). With respect to child outcomes, the intervention significantly improved the quality of life in domains of social well-being and acceptance (d=0.08) and participation and physical health subscale (d=0.31) on the Cerebral Palsy Quality of Life scale (CPQOL). However, no intervention effect was seen on child behaviour and adjustment.</p>
            </sec>
            <sec id="sec13">
                <title>Enabling and partnership</title>
                <p>Two studies were found in this category.
                    <sup>
                        <xref ref-type="bibr" rid="ref30">30</xref>
                    </sup>
                    <sup>,</sup>
                    <sup>
                        <xref ref-type="bibr" rid="ref31">31</xref>
                    </sup> The results of a paper by Myrhaug 
                    <italic toggle="yes">et al.</italic> (2018) were also published in one secondary study from the same sample.
                    <sup>
                        <xref ref-type="bibr" rid="ref38">38</xref>
                    </sup> The two studies focussed on children diagnosed with CP utilized collaborative goal setting but differed in the level of parent involvement in therapy. Law 
                    <italic toggle="yes">et al.</italic> (2011)
                    <sup>
                        <xref ref-type="bibr" rid="ref30">30</xref>
                    </sup> involved the parents to identify constraints affecting their child's performance. Conversely, in the study by Myrhaug 
                    <italic toggle="yes">et al.</italic> in 2018,
                    <sup>
                        <xref ref-type="bibr" rid="ref31">31</xref>
                    </sup> parents were not involved in therapy during the conductive education (CE) courses, while the control group participated in conventional practice with parental involvement.</p>
                <p>In the study by Law 
                    <italic toggle="yes">et al.</italic> (2011),
                    <sup>
                        <xref ref-type="bibr" rid="ref30">30</xref>
                    </sup> the intervention group showed a small intervention effect on the PEDI Caregiver assistance-mobility sub-scale at 9 months follow-up but no improvement was seen in family empowerment.
                    <sup>
                        <xref ref-type="bibr" rid="ref30">30</xref>
                    </sup> In the study by Myrhaug 
                    <italic toggle="yes">et al.</italic> (2018),
                    <sup>
                        <xref ref-type="bibr" rid="ref31">31</xref>
                    </sup> a large effect was seen on receiving more &#x2018;specific information about their child&#x2019; on the Measure of Processes of Care scale (MPOC-20) at follow-up (d=1.47). No difference was seen in their global quality of life. With respect to the child outcomes, both the studies did not show any difference in gross motor function, functional skills, preschool participation, and children&#x2019;s quality of life post-intervention as compared to the control group
                    <sup>
                        <xref ref-type="bibr" rid="ref30">30</xref>
                    </sup>
                    <sup>,</sup>
                    <sup>
                        <xref ref-type="bibr" rid="ref31">31</xref>
                    </sup>
                    <sup>,</sup>
                    <sup>
                        <xref ref-type="bibr" rid="ref38">38</xref>
                    </sup>
                </p>
            </sec>
            <sec id="sec14">
                <title>&#x2018;Enabling and partnership&#x2019; and &#x2018;Respectful and supportive care&#x2019;</title>
                <p>Two studies were found in this category.
                    <sup>
                        <xref ref-type="bibr" rid="ref32">32</xref>
                    </sup>
                    <sup>,</sup>
                    <sup>
                        <xref ref-type="bibr" rid="ref33">33</xref>
                    </sup> The results of a paper were also published in one secondary study from the same sample.
                    <sup>
                        <xref ref-type="bibr" rid="ref39">39</xref>
                    </sup> Both studies provided support to the parents by targeting their needs- needs identified using the family needs scale,
                    <sup>
                        <xref ref-type="bibr" rid="ref33">33</xref>
                    </sup> and positive parenting for child&#x2019;s behavioural problems.
                    <sup>
                        <xref ref-type="bibr" rid="ref32">32</xref>
                    </sup> The study by Weindling 
                    <italic toggle="yes">et al.</italic> (2007) involved joint decision-making between the family support worker and the caregiver to target the needs.
                    <sup>
                        <xref ref-type="bibr" rid="ref33">33</xref>
                    </sup> The study by Whittingham 
                    <italic toggle="yes">et al.</italic> (2014) involved Stepping Stones Triple P (SSPT-only) intervention that involved a partnership between the therapist and parent for collaborative goal setting, discussions, and various strategies for positive parenting.
                    <sup>
                        <xref ref-type="bibr" rid="ref32">32</xref>
                    </sup> Another intervention group, SSPT + ACT group (Acceptance and Commitment Therapy) provided additional support to the parents to build their psychological flexibility via goal setting and various exercises.
                    <sup>
                        <xref ref-type="bibr" rid="ref32">32</xref>
                    </sup>
                </p>
                <p>The study by Weindling 
                    <italic toggle="yes">et al.</italic> (2007)
                    <sup>
                        <xref ref-type="bibr" rid="ref33">33</xref>
                    </sup> did not show improvement in stress (measured on Parent Stress Index) whereas the SSTP+ ACT group in the study by Whittingham 
                    <italic toggle="yes">et al.</italic> (2014)
                    <sup>
                        <xref ref-type="bibr" rid="ref32">32</xref>
                    </sup> showed a medium effect in depression (d=0.74) and stress (d=0.79) measured on the Depression Anxiety Stress subscales. Both the studies met caregiver needs as seen by a significant reduction in the Family Needs Scale (FNS) at 18 months follow up (p = 0.001, effect size=&#x2013;12.0) (but not post-intervention)
                    <sup>
                        <xref ref-type="bibr" rid="ref33">33</xref>
                    </sup> and change in parenting style (reduced overreactivity (d=1.1) and verbosity (d=0.93)).
                    <sup>
                        <xref ref-type="bibr" rid="ref32">32</xref>
                    </sup> The SSTP + ACT group
                    <sup>
                        <xref ref-type="bibr" rid="ref32">32</xref>
                    </sup> showed improvements in child behaviour and emotional problems (ECBI problem d=1.32, ECBI intensity d=0.79, SDQ emotions d=0.16), child hyperactivity (d=0.21), child functional performance in the mobility domain (d=0.03), child quality of life in functioning (d=0.51) and social domains (d=0.64). In the SSTP-only group,
                    <sup>
                        <xref ref-type="bibr" rid="ref32">32</xref>
                    </sup> improvements were seen in child behaviour problems and emotional symptoms.
                    <sup>
                        <xref ref-type="bibr" rid="ref32">32</xref>
                    </sup>
                </p>
            </sec>
            <sec id="sec15">
                <title>&#x2018;Information provision&#x2019;, &#x2018;Enabling and partnership&#x2019;, and &#x2018;Respectful and supportive care&#x2019;</title>
                <p>Only one study was included in this category.
                    <sup>
                        <xref ref-type="bibr" rid="ref6">6</xref>
                    </sup> The study by Kahjoogh 
                    <italic toggle="yes">et al.</italic> (2019)
                    <sup>
                        <xref ref-type="bibr" rid="ref6">6</xref>
                    </sup> involved children diagnosed with CP, in the age group of 5-11 years, from all GMFCS levels. Goal setting was done for all caregivers using the COPM (one goal for themselves and two goals for their child) prior to randomization. The caregivers were coached based on the principles of Occupational performance coaching (OPC), from understanding the current scenario, planning actions, analysing performance and problem-solving to achieve the goal. Emotional support was also provided by intentionally listening to the caregivers and providing guidance and encouragement. Information was imparted in consideration of the parent&#x2019;s experiences as per the principles of OPC. The intervention was given once per week, for 10 weeks or till the goal was achieved. The control group that received conventional therapy, mainly NDT, reported having parent training to move and position their children at home.</p>
                <p>The intervention showed a significant and large effect on overall COPM performance and satisfaction scores (&#x03b7;
                    <sup>2</sup> 
                    <sub>p</sub>=0.41 and 0.38 respectively), on individual mother-related performance and satisfaction scores (&#x03b7;
                    <sup>2</sup> 
                    <sub>p</sub> =0.25 and 0.33 respectively), and on child-related (&#x03b7;
                    <sup>2</sup> 
                    <sub>p</sub>=0.35 and 0.41 respectively) performance and satisfaction scores. Also, a large and significant increase was seen in the caregiver&#x2019;s self-efficacy (&#x03b7;
                    <sup>2</sup> 
                    <sub>p</sub> =0.7) measured using the Sherer general self-efficacy scale.</p>
            </sec>
        </sec>
        <sec id="sec16" sec-type="discussion">
            <title>Discussion</title>
            <p>This systematic review aimed to identify the effectiveness of family-centered interventions on the well-being of caregivers of children with CP. We identified 11 unique randomized controlled trials
                <sup>
                    <xref ref-type="bibr" rid="ref6">6</xref>
                </sup>
                <sup>,</sup>
                <sup>
                    <xref ref-type="bibr" rid="ref26">26</xref>
                </sup>
                <sup>&#x2013;</sup>
                <sup>
                    <xref ref-type="bibr" rid="ref35">35</xref>
                </sup> to address our objectives. Additionally, we stratified the studies according to the various components of family-centered care in their interventions to identify the impact of these components on the caregiver well-being outcomes. Our review also analysed the effect of FCC interventions on child-related outcomes. We found that family centered care interventions are effective to improve caregivers&#x2019; satisfaction with attainment of child and caregiver goals. However, evidence from multiple studies does not strongly support the effectiveness in improving caregiver&#x2019;s mental health, personal, and parenting skills. Limited evidence in other caregiver outcomes suggests caution in effectiveness of FCC interventions in addressing family needs, and improving feeding skills and quality of life. These results emphasize the need for more interventional studies which are aimed directly at caregivers&#x2019; well-being. The following sections will discuss the studies based on the FCC components in their intervention.</p>
            <sec id="sec17">
                <title>&#x2018;Information provision&#x2019; and &#x2018;Enabling and partnership&#x2019;</title>
                <p>The interventions targeting infants at high risk of CP
                    <sup>
                        <xref ref-type="bibr" rid="ref34">34</xref>
                    </sup>
                    <sup>,</sup>
                    <sup>
                        <xref ref-type="bibr" rid="ref35">35</xref>
                    </sup> allowed caregivers to become informed and actively participate in promoting their infant&#x2019;s development during daily activities, showing improvement in caregiver satisfaction with goals. The lack of improvement in stress may be because caregivers with CP have a high burden and multiple sources of stress apart from the caregiving responsibilities such as balancing family and work, and financial burdens.
                    <sup>
                        <xref ref-type="bibr" rid="ref10">10</xref>
                    </sup> Moreover, it proved to be successful for infant outcomes in spite of having a greater severely affected population. On the contrary, even though the study by Hielkema 
                    <italic toggle="yes">et al.</italic> (2020)
                    <sup>
                        <xref ref-type="bibr" rid="ref35">35</xref>
                    </sup> had a longer intervention duration of one year, no difference was seen in infant outcomes. The influence on caregiver outcomes cannot be relied on as NOSI-K and ITQOL are inappropriate outcome measures for this age group. Moreover, a very small sample size and caregiver dropouts introduced a selection bias underpowering our ability to rely on these findings. With respect to studies on children with CP, all outcomes mentioned in the study by Mlinda 
                    <italic toggle="yes">et al.</italic> (2018)
                    <sup>
                        <xref ref-type="bibr" rid="ref26">26</xref>
                    </sup> were binary, non-standard and no clear information was given about them, therefore the results should be interpreted with caution. No improvement in perception of family-centered care was observed in the study by Fonvig 
                    <italic toggle="yes">et al.</italic> (2020)
                    <sup>
                        <xref ref-type="bibr" rid="ref27">27</xref>
                    </sup> as the intervention was delivered passively by mail, with no additional help to understand or translate the information to care.</p>
                <p>Overall, no improvement was seen in caregiver stress
                    <sup>
                        <xref ref-type="bibr" rid="ref34">34</xref>
                    </sup>
                    <sup>,</sup>
                    <sup>
                        <xref ref-type="bibr" rid="ref35">35</xref>
                    </sup> except on a non-standardized outcome measure.
                    <sup>
                        <xref ref-type="bibr" rid="ref26">26</xref>
                    </sup> As only one study assessed the effect on quality of life, family empowerment, perception of FCC, caregiver assistance in function, and satisfaction, there is limited evidence to draw a definite conclusion. There is conflicting evidence on the effectiveness of FCC intervention for motor and cognitive outcomes of high-risk infants.
                    <sup>
                        <xref ref-type="bibr" rid="ref34">34</xref>
                    </sup>
                    <sup>,</sup>
                    <sup>
                        <xref ref-type="bibr" rid="ref35">35</xref>
                    </sup>
                </p>
            </sec>
            <sec id="sec18">
                <title>&#x2018;Information provision&#x2019; and &#x2018;Respectful and supportive care&#x2019;</title>
                <p>The intervention by Whittingham 
                    <italic toggle="yes">et al.</italic> (2022)
                    <sup>
                        <xref ref-type="bibr" rid="ref29">29</xref>
                    </sup> modified parent behaviour. The lack of effect on mental health should be considered with caution as parents had normal mental health at baseline. Little improvement was observed in child behaviour problems. Therefore, additional interventions targeting the child behaviour may be necessary in conjunction with parent-focussed interventions. However, as only one study is present in this domain, we cannot conclude if interventions utilizing &#x2018;Information provision&#x2019; and &#x2018;Respectful and supportive care&#x2019; improved caregiver well-being.</p>
            </sec>
            <sec id="sec19">
                <title>Enabling and partnership</title>
                <p>The two studies incorporated collaborative goal setting with the parents.
                    <sup>
                        <xref ref-type="bibr" rid="ref30">30</xref>
                    </sup>
                    <sup>,</sup>
                    <sup>
                        <xref ref-type="bibr" rid="ref31">31</xref>
                    </sup> In the study by Law 
                    <italic toggle="yes">et al.</italic> (2011)
                    <sup>
                        <xref ref-type="bibr" rid="ref30">30</xref>
                    </sup> both intervention and control groups showed improvement in child outcomes and family empowerment, indicating no additional benefits of context focussed FCC intervention. Conversely, in the study by Myrhaug 
                    <italic toggle="yes">et al.</italic> (2018),
                    <sup>
                        <xref ref-type="bibr" rid="ref31">31</xref>
                    </sup> no effect was seen on caregiver&#x2019;s quality of life and child outcomes. The low sample size, with 50% children belonging to higher disability (GMFCS levels IV and V), and large amounts of conventional therapy in the control group may have diluted the effects. A review on conductive education found inconclusive evidence of its effectiveness due to a lack of quality studies.
                    <sup>
                        <xref ref-type="bibr" rid="ref40">40</xref>
                    </sup> However, positive findings on provision of information for parents on MPOC could be attributed to the availability of many opportunities for informally meeting with the conductor to discuss the child during the conductive education program. Hence, this highlights the power of providing continuous information to the parents about the child&#x2019;s condition and development during therapy sessions.</p>
                <p>Overall, due to varied outcomes, limited evidence exists to draw definitive conclusions on the effectiveness of FCC interventions on caregiver assistance in function activities, family empowerment, perception of FCC, and quality of life. Moreover, no improvement was observed on any child outcomes.</p>
            </sec>
            <sec id="sec20">
                <title>&#x2018;Enabling and partnership&#x2019; and &#x2018;Respectful and supportive care&#x2019;</title>
                <p>The improvement in family needs in the study by Weindling 
                    <italic toggle="yes">et al.</italic> (2007) study
                    <sup>
                        <xref ref-type="bibr" rid="ref33">33</xref>
                    </sup> must be accepted with caution due to reduced sample size at 18 months follow-up, especially due to withdrawal of participants with a higher Family Needs score from the intervention group. The family support workers were parents of children with CP who underwent a short training course by a psychologist. They could have identified the caregiver&#x2019;s needs, however professional support or actively identifying support resources may be required by the parents to meet their enormous and specific needs and actually reduce their stress as seen in a study exploring social support for caregivers of children with chronic diseases.
                    <sup>
                        <xref ref-type="bibr" rid="ref41">41</xref>
                    </sup> The family support workers helped in making the decisions, whereas in the study by Whittingham 
                    <italic toggle="yes">et al.</italic> (2014),
                    <sup>
                        <xref ref-type="bibr" rid="ref32">32</xref>
                    </sup> more active support is given to the caregivers such as the practice of beneficial strategies. The improvement in child behaviour and emotional problems may have resulted in reduced depression and stress. Child behavioural problems to be an important predictor for caregivers&#x2019; physical and psychological health, and advancement in child behaviour was associated with a better ability to handle stress and higher self-perception.
                    <sup>
                        <xref ref-type="bibr" rid="ref7">7</xref>
                    </sup>
                </p>
                <p>Overall, both studies provided support to caregivers, but evidence on the effectiveness of FCC components in reducing stress and depression is conflicting. Only one study showed improvement in child behaviour outcomes and quality of life.
                    <sup>
                        <xref ref-type="bibr" rid="ref32">32</xref>
                    </sup>
                </p>
            </sec>
            <sec id="sec21">
                <title>&#x2018;Information provision&#x2019;, &#x2018;Enabling and partnership&#x2019;, and &#x2018;Respectful and supportive care&#x2019;</title>
                <p>In the study by Kahjoog 
                    <italic toggle="yes">et al.</italic> (2019), the specific goals chosen by the caregivers for themselves and the child were addressed by the therapists in the intervention group.
                    <sup>
                        <xref ref-type="bibr" rid="ref6">6</xref>
                    </sup> The intervention process worked in collaboration with the caregivers at all stages-goal setting, analysing performance, and problem-solving to identify treatment solutions. This active involvement of the caregivers in goals chosen by them may have motivated them to work on the goals, and hence, explains the improvement in self-efficacy and COPM scores. Caregivers&#x2019; feeling of mastery over a caregiving situation and higher self-esteem predicts better psychological health.
                    <sup>
                        <xref ref-type="bibr" rid="ref7">7</xref>
                    </sup> However, the effect on children's outcomes is unknown. We witness parent involvement in the control group again highlighting their active role in conventional therapy.</p>
                <p>As only one study
                    <sup>
                        <xref ref-type="bibr" rid="ref6">6</xref>
                    </sup> was included in this domain, we cannot conclude if interventions utilizing &#x2018;Information provision&#x2019;, &#x2018;Enabling and partnership&#x2019;, and &#x2018;Respectful and supportive care&#x2019; improved caregiver well-being.</p>
                <p>No studies were found that compared the FCC component- &#x2018;Co-ordinated and comprehensive care&#x2019; to a control group. The family support worker in addition to physiotherapists in the study by Weindling 
                    <italic toggle="yes">et al.</italic> (2007)
                    <sup>
                        <xref ref-type="bibr" rid="ref33">33</xref>
                    </sup> cannot be considered as multidisciplinary care as they are parents of children with CP, not health professionals. It is interesting to observe that five studies involved two to four healthcare professionals working as a team in either intervention or control groups such as physical and occupational therapists
                    <sup>
                        <xref ref-type="bibr" rid="ref26">26</xref>
                    </sup>
                    <sup>,</sup>
                    <sup>
                        <xref ref-type="bibr" rid="ref34">34</xref>
                    </sup> or neuro-paediatrician, paediatric orthopaedic surgeon, physiotherapist, and a biomechanist.
                    <sup>
                        <xref ref-type="bibr" rid="ref27">27</xref>
                    </sup> This brings to light the established importance of multidisciplinary care for children with CP.</p>
                <p>Overall, ten studies incorporated the Enabling and partnership component,
                    <sup>
                        <xref ref-type="bibr" rid="ref6">6</xref>
                    </sup>
                    <sup>,</sup>
                    <sup>
                        <xref ref-type="bibr" rid="ref26">26</xref>
                    </sup>
                    <sup>&#x2013;</sup>
                    <sup>
                        <xref ref-type="bibr" rid="ref28">28</xref>
                    </sup>
                    <sup>,</sup>
                    <sup>
                        <xref ref-type="bibr" rid="ref30">30</xref>
                    </sup>
                    <sup>&#x2013;</sup>
                    <sup>
                        <xref ref-type="bibr" rid="ref35">35</xref>
                    </sup> seven had Information provision,
                    <sup>
                        <xref ref-type="bibr" rid="ref6">6</xref>
                    </sup>
                    <sup>,</sup>
                    <sup>
                        <xref ref-type="bibr" rid="ref26">26</xref>
                    </sup>
                    <sup>&#x2013;</sup>
                    <sup>
                        <xref ref-type="bibr" rid="ref29">29</xref>
                    </sup>
                    <sup>,</sup>
                    <sup>
                        <xref ref-type="bibr" rid="ref34">34</xref>
                    </sup>
                    <sup>,</sup>
                    <sup>
                        <xref ref-type="bibr" rid="ref35">35</xref>
                    </sup> and four had a Respectful and supportive care component.
                    <sup>
                        <xref ref-type="bibr" rid="ref6">6</xref>
                    </sup>
                    <sup>,</sup>
                    <sup>
                        <xref ref-type="bibr" rid="ref29">29</xref>
                    </sup>
                    <sup>,</sup>
                    <sup>
                        <xref ref-type="bibr" rid="ref32">32</xref>
                    </sup>
                    <sup>,</sup>
                    <sup>
                        <xref ref-type="bibr" rid="ref33">33</xref>
                    </sup> To address our second objective, Enabling and partnership was the most practiced FCC component. Most family-centered interventions aimed to establish a collaborative relationship with parents, involving them in goal-setting, and capacity-building. These interventions sought to empower the parents to provide independent care for their children, particularly in terms of functional activities at home. However, due to the limited studies and absence of a meta-analysis, we cannot identify which combined FCC components would be most effective for caregiver well-being.</p>
                <p>It is interesting to note that even though multiple studies utilized the same FCC component, the delivery of intervention varied. For example, utilizing information materials, discussions, and practical exercises to educate,
                    <sup>
                        <xref ref-type="bibr" rid="ref32">32</xref>
                    </sup> vs. passively mailing the information report to the caregivers.
                    <sup>
                        <xref ref-type="bibr" rid="ref27">27</xref>
                    </sup> Another example would be involving caregivers in goal setting,
                    <sup>
                        <xref ref-type="bibr" rid="ref31">31</xref>
                    </sup> vs. involving them in the therapy activities as well.
                    <sup>
                        <xref ref-type="bibr" rid="ref6">6</xref>
                    </sup> Therefore, the mode of delivering the intervention may also influence the intervention effects.</p>
                <p>Research has reported the negative effects of caregiving on carers&#x2019; physical health such as fatigue, poor sleep, and musculoskeletal pain.
                    <sup>
                        <xref ref-type="bibr" rid="ref42">42</xref>
                    </sup> However, it is interesting to observe that none of the studies explored the effect of FCC interventions on caregivers&#x2019; physical health. This represents a significant gap in the literature and highlights the need for future research to explore this important area.</p>
                <p>Conventional therapy cannot be carried out without involving the parents or family. The control group in multiple studies
                    <sup>
                        <xref ref-type="bibr" rid="ref6">6</xref>
                    </sup>
                    <sup>,</sup>
                    <sup>
                        <xref ref-type="bibr" rid="ref26">26</xref>
                    </sup>
                    <sup>,</sup>
                    <sup>
                        <xref ref-type="bibr" rid="ref31">31</xref>
                    </sup>
                    <sup>,</sup>
                    <sup>
                        <xref ref-type="bibr" rid="ref34">34</xref>
                    </sup>
                    <sup>,</sup>
                    <sup>
                        <xref ref-type="bibr" rid="ref35">35</xref>
                    </sup> also had some degree of parent involvement which could not be excluded as they were a part of the &#x2018;usual care&#x2019;. Parent involvement varied from education to involvement in therapy sessions or practice of functional activities at home as a home program. We realize that as the primary support system and an essential part of a child&#x2019;s environment, it is natural for the family to participate in therapy. Moreover, as children with CP require intensive therapy and substantial practice, conventional therapy may have increased parent involvement.</p>
            </sec>
            <sec id="sec22">
                <title>Strengths and limitations</title>
                <p>This review has several strengths and limitations that should be considered. One strength is that we included all studies that reported caregiver outcomes, regardless of whether they also reported infant outcomes. However, studies that only reported infant outcomes without caregiver outcomes were excluded. Therefore, this systematic review does not provide a complete picture of the effect of FCC interventions on infant outcomes. However, this was not our objective and was clearly stated in our eligibility criteria. A meta-analysis could not be conducted due to the heterogeneity of the included studies, and thus we cannot provide a definite summary of the effectiveness of FCC interventions on caregiver well-being. Moreover, as caregiver well-being was our primary outcome, most of the outcomes were participant reported. This introduces some amount of bias as blinding of caregivers is not possible in such cases. Additionally, studies that were not randomized controlled trials, and those published in languages other than English were excluded, which may have resulted in relevant information being missed. However, to the best of our knowledge, only one study was excluded on the basis of language.</p>
            </sec>
            <sec id="sec23">
                <title>Clinical implications</title>
                <p>Core components of FCC that include active interventions are more effective for caregiver well-being than passive interventions. Therefore, healthcare professionals should consider designing interventions that involve active parent engagement. Additionally, providing continuous information to the parents about the child&#x2019;s condition and development during the therapy sessions is a useful way to deliver information. Thus, health professionals should prioritize educating the parents on their child&#x2019;s condition, development, and handling during therapy visits to ensure a continuum of updates.</p>
            </sec>
            <sec id="sec24">
                <title>Future recommendations</title>
                <p>Future research should aim to conduct high-quality RCTs with larger sample sizes to better identify the effectiveness of FCC interventions. Future RCTs assessing the effectiveness of FCC interventions need to explore the interventional elements in the control group and clarify the extent of parent involvement. Studies should explore how best they can standardize the control group in an RCT to truly identify the benefits of family-centered interventions. Studies can also explore the effectiveness of different modes of parent participation in family centered care interventions. Further, larger sample sizes are required considering the higher dropout seen in multiple studies. Moreover, future studies should investigate the effect of FCC interventions on caregivers&#x2019; physical health and perform a cost analysis to identify the financial burden of these interventions. RCTs should carefully select appropriate outcome measures designed for age and diagnosis, and limit the outcome measures to include the most essential ones to avoid study burden and potential dropouts.</p>
            </sec>
        </sec>
        <sec id="sec25" sec-type="conclusion">
            <title>Conclusion</title>
            <p>Despite the many challenges faced by caregivers of children with CP, there are limited FCC interventions that are directly focussed on their well-being. The eleven reviewed studies vary greatly in sample size, interventions focus, dose, theoretical basis, and outcomes, making it difficult to draw concrete conclusions on the effectiveness of FCC interventions on caregiver well-being. However, it can be inferred that FCC interventions are effective in improving caregivers&#x2019; satisfaction with attainment of child and caregiver goals. Evidence from multiple studies does not strongly support the effectiveness in improving caregiver&#x2019;s mental health, personal, and parenting skills. They should be used with caution in addressing family needs, and improving feeding skills and quality of life. Limited evidence and overlap of FCC core components in individual studies precludes a conclusion on the effectiveness of distinct FCC components on the well-being of caregivers of children with CP. However, it is clear that active engagement interventions are more effective for caregiver well-being compared to passive interventions. Establishing active partnerships with caregivers are best to address their needs and priorities.</p>
        </sec>
    </body>
    <back>
        <sec id="sec28" sec-type="data-availability">
            <title>Data availability</title>
            <sec id="sec29">
                <title>Underlying data</title>
                <p>No data are associated with this article.</p>
            </sec>
            <sec id="sec30">
                <title>Extended data</title>
                <p>Figshare: Search Strategy for &#x201c;Effect of family centered care interventions on well-being of caregivers of children with cerebral palsy: a systematic review&#x201d;. Data file 1: Search Strategy. 
                    <ext-link ext-link-type="uri" xlink:href="https://doi.org/10.6084/m9.figshare.22344664.v1">https://doi.org/10.6084/m9.figshare.22344664.v1</ext-link>.
                    <sup>

                        <xref ref-type="bibr" rid="ref43">43</xref>
</sup>
                </p>
                <p>Figshare: Intervention details using TIDieR checklist for &#x201c;Effect of family centered care interventions on well-being of caregivers of children with cerebral palsy: a systematic review&#x201d;. Data file 3: Intervention details using TIDieR checklist. 
                    <ext-link ext-link-type="uri" xlink:href="https://doi.org/10.6084/m9.figshare.22344670.v1">https://doi.org/10.6084/m9.figshare.22344670.v1</ext-link>.
                    <sup>

                        <xref ref-type="bibr" rid="ref44">44</xref>
</sup>
                </p>
                <p>Figshare: Description of outcome measures and results of intervention effectiveness for &#x201c;Effect of family centered care interventions on well-being of caregivers of children with cerebral palsy: a systematic review&#x201d;. Data file 4: Description of outcome measures and results of intervention effectiveness. 
                    <ext-link ext-link-type="uri" xlink:href="https://doi.org/10.6084/m9.figshare.22344667.v1">https://doi.org/10.6084/m9.figshare.22344667.v1</ext-link>.
                    <sup>

                        <xref ref-type="bibr" rid="ref45">45</xref>
</sup>
                </p>
            </sec>
            <sec id="sec31">
                <title>Reporting guidelines</title>
                <p>Figshare: PRISMA 2020 checklist for &#x201c;Effect of family centered care interventions on well-being of caregivers of children with cerebral palsy: a systematic review&#x201d;. Data file 2: PRISMA 2020 checklist. 
                    <ext-link ext-link-type="uri" xlink:href="https://doi.org/10.6084/m9.figshare.22344724.v1">https://doi.org/10.6084/m9.figshare.22344724.v1</ext-link>.
                    <sup>

                        <xref ref-type="bibr" rid="ref46">46</xref>
</sup>
                </p>
                <p>Data are available under the terms of the 
                    <ext-link ext-link-type="uri" xlink:href="https://creativecommons.org/licenses/by/4.0/">Creative Commons Attribution 4.0 International license</ext-link> (CC-BY 4.0).</p>
            </sec>
        </sec>
        <ack>
            <title>Acknowledgement</title>
            <p>We acknowledge the effort of all authors of the RCTs included in our review. We thank Mrs. Savni Apte, PhD student at Manipal Academy of Higher Education, with her permission for helping us formulate the search strategy for this review.</p>
        </ack>
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    <sub-article article-type="reviewer-report" id="report248360">
        <front-stub>
            <article-id pub-id-type="doi">10.5256/f1000research.146293.r248360</article-id>
            <title-group>
                <article-title>Reviewer response for version 1</article-title>
            </title-group>
            <contrib-group>
                <contrib contrib-type="author">
                    <name>
                        <surname>Karthikbabu</surname>
                        <given-names>Suruliraj</given-names>
                    </name>
                    <xref ref-type="aff" rid="r248360a1">1</xref>
                    <role>Referee</role>
                    <uri content-type="orcid">https://orcid.org/0000-0002-7513-0606</uri>
                </contrib>
                <aff id="r248360a1">
                    <label>1</label>KMCH College of Physiotherapy, Dr NGP Research and Educational Trust, Coimbatore, Tamil Nadu, India</aff>
            </contrib-group>
            <author-notes>
                <fn fn-type="conflict">
                    <p>
                        <bold>Competing interests: </bold>No competing interests were disclosed.</p>
                </fn>
            </author-notes>
            <pub-date pub-type="epub">
                <day>25</day>
                <month>3</month>
                <year>2024</year>
            </pub-date>
            <permissions>
                <copyright-statement>Copyright: &#x00a9; 2024 Karthikbabu S</copyright-statement>
                <copyright-year>2024</copyright-year>
                <license xlink:href="https://creativecommons.org/licenses/by/4.0/">
                    <license-p>This is an open access peer review report distributed under the terms of the Creative Commons Attribution Licence, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.</license-p>
                </license>
            </permissions>
            <related-article ext-link-type="doi" id="relatedArticleReport248360" related-article-type="peer-reviewed-article" xlink:href="10.12688/f1000research.133314.1"/>
            <custom-meta-group>
                <custom-meta>
                    <meta-name>recommendation</meta-name>
                    <meta-value>approve</meta-value>
                </custom-meta>
            </custom-meta-group>
        </front-stub>
        <body>
            <p>Journal: F1000 
                <list list-type="bullet">
                    <list-item>
                        <p>I must appreciate the authors for conducting this important systematic review.</p>
                    </list-item>
                    <list-item>
                        <p>Overall, the paper is well written.</p>
                    </list-item>
                    <list-item>
                        <p>I have a few queries and thoughts and please find my review comments below.&#x00a0;&#x00a0;&#x00a0;</p>
                    </list-item>
                    <list-item>
                        <p>The authors conducted a systematic review of family-centered care (FCC) interventions on the well-being of caregivers of children with cerebral palsy. They concluded that FCC interventions were effective in improving caregivers&#x2019; satisfaction with the attainment of child and caregiver goals, but not for the caregiver&#x2019;s mental health, parenting and personal outcomes.</p>
                    </list-item>
                    <list-item>
                        <p>In the introduction, the authors stated how caring for a child with long-term functional disability affects both the physical and psychological well-being of the caregiver, warranting a systematic review to understand the benefits of FCC on multiple variables.</p>
                    </list-item>
                    <list-item>
                        <p>The authors stated that there is a dearth of literature assessing the FCC on the well-being of caregivers of children with CP. Instead, the authors can elaborate on a diversity of such interventions limiting healthcare professions to choose a specific FCC.</p>
                    </list-item>
                    <list-item>
                        <p>Search strategies are appropriate and were conducted in seven databases between inception and September 2022. The authors may check if there are any new papers published during the last 18 months. I believe the authors can perform it by the &#x201c;year&#x201d; filter and the search-strategy thread.</p>
                    </list-item>
                    <list-item>
                        <p>Eligibility criteria, data screening and extraction, and quality assessment sections are well explained.</p>
                    </list-item>
                    <list-item>
                        <p>&#x00a0;&#x201c;Risk of Bias 2&#x201d;, CASP checklist and traffic light system are useful and so are the tables and figures.&#x00a0;</p>
                    </list-item>
                    <list-item>
                        <p>Since caregiver-reported measures are taken in many studies, assessor bias is unavoidable.</p>
                    </list-item>
                    <list-item>
                        <p>Please share your thoughts on caregivers' burden on looking after younger versus older children with CP. Few studies included infants with CP.&#x00a0;&#x00a0;&#x00a0;</p>
                    </list-item>
                    <list-item>
                        <p>Five studies are reporting &#x201c;information provision&#x2019; and &#x2018;enabling and partnership&#x2019;, providing limited evidence on the said parameters.</p>
                    </list-item>
                    <list-item>
                        <p>The caregiver well-being domains such as &#x201c;Information provision&#x2019; and &#x2018;Respectful and supportive care&#x2019;; Enabling and partnership; &#x2018;Enabling and partnership&#x2019; and &#x2018;Respectful and supportive care&#x2019; and &#x2018;Information provision&#x2019;, &#x2018;Enabling and partnership&#x2019;, and &#x2018;Respectful and supportive care&#x2019; require more future studies to examine the true benefits of FCC.</p>
                    </list-item>
                </list> Best wishes.</p>
            <p> The reviewer</p>
            <p>Are the rationale for, and objectives of, the Systematic Review clearly stated?</p>
            <p>Yes</p>
            <p>Is the statistical analysis and its interpretation appropriate?</p>
            <p>Yes</p>
            <p>Are sufficient details of the methods and analysis provided to allow replication by others?</p>
            <p>Yes</p>
            <p>Are the conclusions drawn adequately supported by the results presented in the review?</p>
            <p>Yes</p>
            <p>Reviewer Expertise:</p>
            <p>neurorehabilitation</p>
            <p>I confirm that I have read this submission and believe that I have an appropriate level of expertise to confirm that it is of an acceptable scientific standard.</p>
        </body>
        <sub-article article-type="response" id="comment11490-248360">
            <front-stub>
                <contrib-group>
                    <contrib contrib-type="author">
                        <name>
                            <surname>Poojari</surname>
                            <given-names>Deepalaxmi Paresh</given-names>
                        </name>
                        <aff>Physiotherapy, Manipal Academy of Higher Education, Manipal, Karnataka, India</aff>
                    </contrib>
                </contrib-group>
                <author-notes>
                    <fn fn-type="conflict">
                        <p>
                            <bold>Competing interests: </bold>The authors declare that there is no conflict of interest.</p>
                    </fn>
                </author-notes>
                <pub-date pub-type="epub">
                    <day>29</day>
                    <month>4</month>
                    <year>2024</year>
                </pub-date>
            </front-stub>
            <body>
                <p>
                    <bold>Response to Reviewers</bold>
                </p>
                <p> </p>
                <p> </p>
                <p> 
                    <bold>Reviewer one - Dr&#x00a0;Tanya Tripathi</bold>
                </p>
                <p> </p>
                <p> Comment (C)1. "Firstly, consider tightening the results section to focus specifically on the primary and secondary objectives of the study, which are assessing caregiver well-being and identifying effective intervention components.&#x00a0;"</p>
                <p> </p>
                <p> Response(R)1.Thank you for the suggestion. We have modified our objective statement to state that outcomes for children with CP will also be included, as it is our secondary objective. We have clubbed "Effect on caregiver well-being" and "Components of FCC" as our primary objective and "Effect on children" as a secondary objective. The same has been clarified in our eligibility criteria and limitations. We have highlighted the same in yellow.</p>
                <p> </p>
                <p> </p>
                <p> C2."It would be beneficial to include outcome measures and results in Table 1 for each included study."&#x00a0;</p>
                <p> </p>
                <p> R2. We value your suggestion. However, we will not be able to incorporate outcome measures in Table 1, as there are huge data sets&#x00a0;with multiple outcome measures in each study. Please note the outcome measures and results are presented in Data File 4.&#x00a0;</p>
                <p> </p>
                <p> C3. "Secondly, provide further explanation for the lack of effect on mental health outcomes, considering that parents had normal mental health at baseline. This clarification will help readers better understand the results."</p>
                <p> </p>
                <p> R3. Thank you for your suggestion. We have incorporated the changes highlighted in green in the manuscript.</p>
                <p> </p>
                <p> C4. "Lastly, either remove information on child outcomes from the results section, as it was not part of the study's objectives or reword the objectives to include the intention to report on child outcomes. This will enhance the clarity of the study's goals and findings."</p>
                <p> </p>
                <p> R4.&#x00a0;We appreciate your comment and we have clarified it in the objectives.&#x00a0;</p>
                <p> </p>
                <p> </p>
                <p> 
                    <bold>Reviewer two - Dr&#x00a0;Suruliraj Karthikbabu</bold>
                </p>
                <p> Comment (C)1. The authors conducted a systematic review of family-centered care (FCC) interventions on the well-being of caregivers of children with cerebral palsy. They concluded that FCC interventions were effective in improving caregivers&#x2019; satisfaction with the attainment of child and caregiver goals, but not for the caregiver&#x2019;s mental health, parenting and personal outcomes.</p>
                <p> </p>
                <p> Response (R)1.Thank you for highlighting the important results of the study.</p>
                <p> </p>
                <p> C2.&#x00a0;In the introduction, the authors stated how caring for a child with long-term functional disability affects both the physical and psychological well-being of the caregiver, warranting a systematic review to understand the benefits of FCC on multiple variables. The authors stated that there is a dearth of literature assessing the FCC on the well-being of caregivers of children with CP. Instead, the authors can elaborate on a diversity of such interventions limiting healthcare professions to choose a specific FCC.</p>
                <p> R2. Thank you for your suggestion. We have added the following statement in the introduction:</p>
                <p> ''There is also a scarcity of RCTs which have implemented the Universal model of FCC considering the core components of FCC and there is a need for evidence to understand the specific core components of FCC."</p>
                <p> </p>
                <p> C3.&#x00a0;Search strategies are appropriate and were conducted in seven databases between inception and September 2022. The authors may check if there are any new papers published during the last 18 months. I believe the authors can perform it by the &#x201c;year&#x201d; filter and the search-strategy thread.</p>
                <p> </p>
                <p> R3.&#x00a0;Thank you for suggesting this. We have completed an additional review between September 2022 and April 2024 and have included five new articles in the review. All the data sets have been modified accordingly and are highlighted in yellow and the reference DOI has been provided.</p>
                <p> </p>
                <p> C4.&#x00a0;Eligibility criteria, data screening and extraction, and quality assessment sections are well explained.</p>
                <p> R4. Thank you for this acknowledgement.</p>
                <p> </p>
                <p> C5&#x00a0;&#x201c;Risk of Bias 2&#x201d;, CASP checklist and traffic light system are useful and so are the tables and figures.&#x00a0;</p>
                <p> R5.&#x00a0;Thank you, we appreciate this recognition of our work.</p>
                <p> </p>
                <p> C6.&#x00a0;Since caregiver-reported measures are taken in many studies, assessor bias is unavoidable.</p>
                <p> R6.&#x00a0;Yes, we do agree with this.</p>
                <p> </p>
                <p> C7.Please share your thoughts on caregivers' burden on looking after younger versus older children with CP</p>
                <p> R7.&#x00a0; In the present study, children with CP were between the ages of 3 months and 16 years. Information needs may have differed for caregivers of younger children, children who had been recently diagnosed with or at risk of CP, or children above 16 years of age who might have gotten used to the entire care system. The craving for child-specific information may be greater in caregivers of younger children than in older children.</p>
                <p> </p>
                <p> C8. Five studies are reporting &#x201c;information provision&#x2019; and &#x2018;enabling and partnership&#x2019;, providing limited evidence on the said parameters.</p>
                <p> R8.We agree. Thank you for highlighting this.&#x00a0; Although there are five studies under this domain, the overall effect on caregivers' outcome on standardized outcome was not seen and the child outcome showed conflicting evidence.</p>
                <p> </p>
                <p> C9.The caregiver well-being domains such as &#x201c;Information provision&#x2019; and &#x2018;Respectful and supportive care&#x2019;; Enabling and partnership; &#x2018;Enabling and partnership&#x2019; and &#x2018;Respectful and supportive care&#x2019; and &#x2018;Information provision&#x2019;, &#x2018;Enabling and partnership&#x2019;, and &#x2018;Respectful and supportive care&#x2019; require more future studies to examine the true benefits of FCC.</p>
                <p> R9.&#x00a0;Indeed, the studies had varied ways of providing intervention and sample sizes were not large enough and with high dropouts, thereby hindering conclusive evidence. Overall, five studies fell into the category of high risk. Hence, in order to examine the true benefits of FCC, it is important to have high-quality randomised controlled trials with care in the control groups not overlapping with the intervention groups in terms of the components of FCC received.</p>
            </body>
        </sub-article>
    </sub-article>
    <sub-article article-type="reviewer-report" id="report187453">
        <front-stub>
            <article-id pub-id-type="doi">10.5256/f1000research.146293.r187453</article-id>
            <title-group>
                <article-title>Reviewer response for version 1</article-title>
            </title-group>
            <contrib-group>
                <contrib contrib-type="author">
                    <name>
                        <surname>Tripathi</surname>
                        <given-names>Tanya</given-names>
                    </name>
                    <xref ref-type="aff" rid="r187453a1">1</xref>
                    <role>Referee</role>
                </contrib>
                <aff id="r187453a1">
                    <label>1</label>Virginia Commonwealth University, The Ohio State University, Columbus, OH, USA</aff>
            </contrib-group>
            <author-notes>
                <fn fn-type="conflict">
                    <p>
                        <bold>Competing interests: </bold>No competing interests were disclosed.</p>
                </fn>
            </author-notes>
            <pub-date pub-type="epub">
                <day>31</day>
                <month>8</month>
                <year>2023</year>
            </pub-date>
            <permissions>
                <copyright-statement>Copyright: &#x00a9; 2023 Tripathi T</copyright-statement>
                <copyright-year>2023</copyright-year>
                <license xlink:href="https://creativecommons.org/licenses/by/4.0/">
                    <license-p>This is an open access peer review report distributed under the terms of the Creative Commons Attribution Licence, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.</license-p>
                </license>
            </permissions>
            <related-article ext-link-type="doi" id="relatedArticleReport187453" related-article-type="peer-reviewed-article" xlink:href="10.12688/f1000research.133314.1"/>
            <custom-meta-group>
                <custom-meta>
                    <meta-name>recommendation</meta-name>
                    <meta-value>approve-with-reservations</meta-value>
                </custom-meta>
            </custom-meta-group>
        </front-stub>
        <body>
            <p>This systematic review aimed to assess the effectiveness of family-centered care (FCC) interventions in improving the well-being of caregivers of children with cerebral palsy. The review included 11 studies, which were grouped into five sections based on the components of FCC intervention provided in each study. The review highlights the potential benefits of FCC interventions for caregivers but also underscores the need for further research in this area. Overall, the review is promising, and the methods are scientifically sound. Below are a few suggestions for the authors to improve their reporting of results.</p>
            <p> </p>
            <p> Firstly, consider tightening the results section to focus specifically on the primary and secondary objectives of the study, which are assessing caregiver well-being and identifying effective intervention components. It would be beneficial to include outcomes measures and results in Table 1 for each included study.</p>
            <p> </p>
            <p> Secondly, provide further explanation for the lack of effect on mental health outcomes, considering that parents had normal mental health at baseline. This clarification will help readers better understand the results.</p>
            <p> </p>
            <p> Lastly, either remove information on child outcomes from the results section, as it was not part of the study's objectives, or reword the objectives to include the intention to report on child outcomes. This will enhance the clarity of the study's goals and findings.</p>
            <p>Are the rationale for, and objectives of, the Systematic Review clearly stated?</p>
            <p>Yes</p>
            <p>Is the statistical analysis and its interpretation appropriate?</p>
            <p>Not applicable</p>
            <p>Are sufficient details of the methods and analysis provided to allow replication by others?</p>
            <p>Yes</p>
            <p>Are the conclusions drawn adequately supported by the results presented in the review?</p>
            <p>Yes</p>
            <p>Reviewer Expertise:</p>
            <p>Physiotherapy and early intervention</p>
            <p>I confirm that I have read this submission and believe that I have an appropriate level of expertise to confirm that it is of an acceptable scientific standard, however I have significant reservations, as outlined above.</p>
        </body>
        <sub-article article-type="response" id="comment11490-187453">
            <front-stub>
                <contrib-group>
                    <contrib contrib-type="author">
                        <name>
                            <surname>Poojari</surname>
                            <given-names>Deepalaxmi Paresh</given-names>
                        </name>
                        <aff>Physiotherapy, Manipal Academy of Higher Education, Manipal, Karnataka, India</aff>
                    </contrib>
                </contrib-group>
                <author-notes>
                    <fn fn-type="conflict">
                        <p>
                            <bold>Competing interests: </bold>The authors declare that there is no conflict of interest.</p>
                    </fn>
                </author-notes>
                <pub-date pub-type="epub">
                    <day>29</day>
                    <month>4</month>
                    <year>2024</year>
                </pub-date>
            </front-stub>
            <body>
                <p>
                    <bold>Response to Reviewers</bold>
                </p>
                <p> </p>
                <p> </p>
                <p> 
                    <bold>Reviewer one - Dr&#x00a0;Tanya Tripathi</bold>
                </p>
                <p> </p>
                <p> Comment (C)1. "Firstly, consider tightening the results section to focus specifically on the primary and secondary objectives of the study, which are assessing caregiver well-being and identifying effective intervention components.&#x00a0;"</p>
                <p> </p>
                <p> Response(R)1.Thank you for the suggestion. We have modified our objective statement to state that outcomes for children with CP will also be included, as it is our secondary objective. We have clubbed "Effect on caregiver well-being" and "Components of FCC" as our primary objective and "Effect on children" as a secondary objective. The same has been clarified in our eligibility criteria and limitations. We have highlighted the same in yellow.</p>
                <p> </p>
                <p> </p>
                <p> C2."It would be beneficial to include outcome measures and results in Table 1 for each included study."&#x00a0;</p>
                <p> </p>
                <p> R2. We value your suggestion. However, we will not be able to incorporate outcome measures in Table 1, as there are huge data sets&#x00a0;with multiple outcome measures in each study. Please note the outcome measures and results are presented in Data File 4.&#x00a0;</p>
                <p> </p>
                <p> C3. "Secondly, provide further explanation for the lack of effect on mental health outcomes, considering that parents had normal mental health at baseline. This clarification will help readers better understand the results."</p>
                <p> </p>
                <p> R3. Thank you for your suggestion. We have incorporated the changes highlighted in green in the manuscript.</p>
                <p> </p>
                <p> C4. "Lastly, either remove information on child outcomes from the results section, as it was not part of the study's objectives or reword the objectives to include the intention to report on child outcomes. This will enhance the clarity of the study's goals and findings."</p>
                <p> </p>
                <p> R4.&#x00a0;We appreciate your comment and we have clarified it in the objectives.&#x00a0;</p>
                <p> </p>
                <p> </p>
                <p> 
                    <bold>Reviewer two - Dr&#x00a0;Suruliraj Karthikbabu</bold>
                </p>
                <p> Comment (C)1. The authors conducted a systematic review of family-centered care (FCC) interventions on the well-being of caregivers of children with cerebral palsy. They concluded that FCC interventions were effective in improving caregivers&#x2019; satisfaction with the attainment of child and caregiver goals, but not for the caregiver&#x2019;s mental health, parenting and personal outcomes.</p>
                <p> </p>
                <p> Response (R)1.Thank you for highlighting the important results of the study.</p>
                <p> </p>
                <p> C2.&#x00a0;In the introduction, the authors stated how caring for a child with long-term functional disability affects both the physical and psychological well-being of the caregiver, warranting a systematic review to understand the benefits of FCC on multiple variables. The authors stated that there is a dearth of literature assessing the FCC on the well-being of caregivers of children with CP. Instead, the authors can elaborate on a diversity of such interventions limiting healthcare professions to choose a specific FCC.</p>
                <p> R2. Thank you for your suggestion. We have added the following statement in the introduction:</p>
                <p> ''There is also a scarcity of RCTs which have implemented the Universal model of FCC considering the core components of FCC and there is a need for evidence to understand the specific core components of FCC."</p>
                <p> </p>
                <p> C3.&#x00a0;Search strategies are appropriate and were conducted in seven databases between inception and September 2022. The authors may check if there are any new papers published during the last 18 months. I believe the authors can perform it by the &#x201c;year&#x201d; filter and the search-strategy thread.</p>
                <p> </p>
                <p> R3.&#x00a0;Thank you for suggesting this. We have completed an additional review between September 2022 and April 2024 and have included five new articles in the review. All the data sets have been modified accordingly and are highlighted in yellow and the reference DOI has been provided.</p>
                <p> </p>
                <p> C4.&#x00a0;Eligibility criteria, data screening and extraction, and quality assessment sections are well explained.</p>
                <p> R4. Thank you for this acknowledgement.</p>
                <p> </p>
                <p> C5&#x00a0;&#x201c;Risk of Bias 2&#x201d;, CASP checklist and traffic light system are useful and so are the tables and figures.&#x00a0;</p>
                <p> R5.&#x00a0;Thank you, we appreciate this recognition of our work.</p>
                <p> </p>
                <p> C6.&#x00a0;Since caregiver-reported measures are taken in many studies, assessor bias is unavoidable.</p>
                <p> R6.&#x00a0;Yes, we do agree with this.</p>
                <p> </p>
                <p> C7.Please share your thoughts on caregivers' burden on looking after younger versus older children with CP</p>
                <p> R7.&#x00a0; In the present study, children with CP were between the ages of 3 months and 16 years. Information needs may have differed for caregivers of younger children, children who had been recently diagnosed with or at risk of CP, or children above 16 years of age who might have gotten used to the entire care system. The craving for child-specific information may be greater in caregivers of younger children than in older children.</p>
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                <p> C8. Five studies are reporting &#x201c;information provision&#x2019; and &#x2018;enabling and partnership&#x2019;, providing limited evidence on the said parameters.</p>
                <p> R8.We agree. Thank you for highlighting this.&#x00a0; Although there are five studies under this domain, the overall effect on caregivers' outcome on standardized outcome was not seen and the child outcome showed conflicting evidence.</p>
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                <p> C9.The caregiver well-being domains such as &#x201c;Information provision&#x2019; and &#x2018;Respectful and supportive care&#x2019;; Enabling and partnership; &#x2018;Enabling and partnership&#x2019; and &#x2018;Respectful and supportive care&#x2019; and &#x2018;Information provision&#x2019;, &#x2018;Enabling and partnership&#x2019;, and &#x2018;Respectful and supportive care&#x2019; require more future studies to examine the true benefits of FCC.</p>
                <p> R9.&#x00a0;Indeed, the studies had varied ways of providing intervention and sample sizes were not large enough and with high dropouts, thereby hindering conclusive evidence. Overall, five studies fell into the category of high risk. Hence, in order to examine the true benefits of FCC, it is important to have high-quality randomised controlled trials with care in the control groups not overlapping with the intervention groups in terms of the components of FCC received.</p>
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