Effect of family-centered care interventions on well-being of caregivers of children with cerebral palsy: a systematic review

Protocol registration

This systematic review was conducted in accordance with the preferred reporting items for systematic reviews and meta-analyses 2020 (PRISMA) guidelines.²⁰ The systematic review protocol was prospectively registered with PROSPERO (No. CRD42021233854) and can be accessed at https://www.crd.york.ac.uk/prospero/display_record.php?ID=CRD42021233854.

Search strategy

Seven databases - Cochrane, Pubmed, Scopus, CINAHL Plus, EMBASE, Web of Science, and ProQuest - were searched from inception to 30^th September 2022. An additional search was performed from the year 2022 to April 2024 to ensure there are no missed research article during the process where the manuscript was under review. A systematic search strategy (dataset 1 in Extended data) was used employing the PICO format using filters - Human and English.²¹ Furthermore, we examined the reference list of the included articles to identify any relevant articles for this review.

Eligibility criteria

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.¹⁸ The last component ‘General information’ 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 function and well-being of children with CP were also noted to provide a comprehensive understanding of the interventions but were not necessary for inclusion. Studies that only reported infant outcomes without caregiver outcomes were excluded. All non-human studies and those not in the English language were excluded.

Data screening and extraction

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’s d was calculated.²² Other methods were also used to calculate effect size from odds ratio and median values.^23,24 The corresponding authors were contacted for any missing or unclear information.

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.

Quality assessment

To assess the quality of the included studies, two reviewers (DP and RA) independently scored the risk of bias using the Cochrane ‘Risk of Bias 2’ (RoB 2) tool for randomized trials.²⁵ 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,²⁶ 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.

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.

Study characteristics

All the studies are randomized controlled trials, of which thirteen studies involved children with CP,^6,27–38 two studies involved infants at high risk of CP,^39,40 and lastly, one study involved both children diagnosed or at high risk of CP. ⁴¹ Four studies were located in Australia,^30,33,39,40 two in Iran,^6,35 and one each in the United Kingdom,³⁴ Tanzania,²⁷ Netherlands,⁴¹ Norway,³² Brazil,²⁹ Canada,³¹ Denmark,²⁸ Turkey,³⁷ Thailand,³⁶ and Bangladesh.³⁸ In five studies^{30,33,35,37,38,32} 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=251, for the parents and children with CP. Table 1⁵⁸ 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, self-efficacy, social capital score 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.

ROB and Quality Assessment

According to the Cochrane ROB 2.0 tool for RCTs, six RCTs had low ROB,^{6,29,30,32,36,40} five RCTs had some concerns,^{28,35,37–39} and five had a high ROB^{27,31,33,34,41} (Figure 2). 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’s awareness of intervention and its influence on outcome was rated as ‘probably no’ to avoid categorizing as high-risk of bias on this specific basis except for three articles which had mentioned as outcome assessor were blinded. Table 2⁵⁷ 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.

Figure 2. Risk of bias of included studies.

Risk of bias was low (green) in six studies,^{6,29,30,32,36,40} unclear (yellow) in five studies,^{28,35,37–39} and high (red) in five studies.^{27,31,33,34,41}

Figure 3 demonstrates the overall effect of FCC interventions on caregiver well-being using the traffic light system FCC interventions are effective (green) in improving caregivers’ satisfaction with attainment of child and caregiver goals, improving self-efficacy and improving their social capital score. 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.

Figure 3. Overall effect of FCC interventions using the traffic light system.

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).

Lastly, FCC interventions are largely ineffective (red) in improving caregiver’s mental health except for one study which showed a good effect (green), personal outcomes such as empowerment, parenting skills, caregiver burden, perception of family centeredness (except information provision about the child) and reduce caregiver assistance in daily activities.

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,^31,32 whereas the remaining studies involved multiple FCC components in their intervention.^{6,27–30,33–41} 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.

‘Information provision’ and ‘Enabling and partnership’

Five studies utilized these FCC components in their interventions.^{27–29,39,41} The results of these papers are also published in two secondary studies from the same sample.^42,43 Two studies, Morgan et al. (2016)³⁹ and Hielkema et al. (2020)⁴¹ involved infants at very high risk of CP ranging from 3 to 9 months corrected age. The intervention in one study³⁹ followed the GAME principles (Goals, Activity and Motor Enrichment) whereas the other study⁴¹ followed COPCA principles (Coping with and Caring for infants with special needs). Both studies^39,41 involved active caregiver learning through education and training for the caregivers to become independent in identifying infant’s movements and providing opportunities for motor task practice. Discussions and home programs directed towards a parent-identified goal were given.³⁹ However, the control groups in both studies^39,41 had some family involvement such as parental advice on positioning, handling, feeding, and developmental simulation.³⁹

Improvement was seen in caregiver satisfaction with attainment of goal at 12 months on the COPM (d=0.68).³⁹ Surprisingly, both the studies did not show any improvement in parents’ mental health as measured on Depression Anxiety and Stress Scale (DASS-21)³⁹ and Nijmeegse Ouderlijke Stress Index questionnaire, short version (NOSI-K)⁴¹ 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.⁴¹ 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).³⁹ 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.⁴¹

Three studies involved children diagnosed with CP ranging from age 1-12 years.^27–29All three studies involved caregiver education to facilitate a specific function- improve nutrition and feeding skills,²⁷ become informed about the child’s treatment for gait impairments,²⁸ and facilitate the child’s motor tasks respectively.²⁹ Two studies had active parent involvement via multiple modes of delivering education,²⁷and caregiver participation in goal setting and practice of functional activities.²⁹ In contrast, the study by Fonvig et al. (2020) only provided the information passively by mailing the instrumented gait-analysis report.²⁸ The control group in one study received general health education for parents,²⁷ whereas another study involved multidisciplinary health professionals collaboration.²⁸

The study by Mlinda et al. (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).²⁷ The study by Fonvig et al. (2020) showed no improvement in any domain of the MPOC-20.²⁸ The study by Saquetto et al. (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.²⁹ These interventions improved child’s mood (d=0.62),²⁷ gross motor function (η²=0.145, large effect),²⁸and self-care skills (Effect size=2.18, large effect).²⁹

‘Information provision’ and ‘Respectful and supportive care’

Only one study by Whittingham et al. (2022)³⁰ 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’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.

Enabling and partnership

Two studies were found in this category.^31,32 The results of a paper by Myrhaug et al. (2018) were also published in one secondary study from the same sample.⁴⁴ The two studies focussed on children diagnosed with CP utilized collaborative goal setting but differed in the level of parent involvement in therapy. Law et al. (2011)³¹ involved the parents to identify constraints affecting their child’s performance. Conversely, in the study by Myrhaug et al. in 2018,³² parents were not involved in therapy during the conductive education (CE) courses, while the control group participated in conventional practice with parental involvement.

In the study by Law et al. (2011),³¹ 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.³¹ In the study by Myrhaug et al. (2018),³² a large effect was seen on receiving more ‘specific information about their child’ 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’s quality of life post-intervention as compared to the control group^31,32,44

‘Enabling and partnership’ and ‘Respectful and supportive care’

Two studies were found in this category.^33,34 The results of a paper were also published in one secondary study from the same sample.⁴⁵ Both studies provided support to the parents by targeting their needs- needs identified using the family needs scale,³⁴ and positive parenting for child’s behavioural problems.³³ The study by Weindling et al. (2007) involved joint decision-making between the family support worker and the caregiver to target the needs.³⁴ The study by Whittingham et al. (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.³³ 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.³³

The study by Weindling et al. (2007)³⁴ did not show improvement in stress (measured on Parent Stress Index) whereas the SSTP+ ACT group in the study by Whittingham et al. (2014)³³ 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=–12.0) (but not post-intervention)³⁴ and change in parenting style (reduced overreactivity (d=1.1) and verbosity (d=0.93)).³³ The SSTP + ACT group³³ 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,³³ improvements were seen in child behaviour problems and emotional symptoms.³³

‘Information provision’, ‘Enabling and partnership’, and ‘Respectful and supportive care’

Five studies were included in this category.^{6,35,37,38,40} Four studies involved children diagnosed with CP, in the age group of 1-16 years,^6,35,37,38 from all GMFCS levels and one study included infants with high-risk CP (age 3 to 10 months).⁴⁰

In the study by Kahjoogh et al. (2019),⁶ goal setting was done for all caregivers using the COPM (one goal for themselves and two goals for their child) prior to randomization. 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’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. The intervention showed a significant and large effect on overall COPM performance and satisfaction scores (η²_p=0.41 and 0.38 respectively), on individual mother-related performance and satisfaction scores (η²_p=0.25 and 0.33 respectively), and on child-related performance and satisfaction scores (η²_p=0.35 and 0.41 respectively). Also, a large and significant increase was seen in the caregiver’s self-efficacy (η²_p=0.7) measured using the Sherer general self-efficacy scale.

In a study by Panahi et al. (2022)³⁵ the intervention consisted of orientation of the caregiver about their child’s disease, identifying the problems, collaborative discussion on goal setting, sensitising the caregivers regarding ongoing care behaviours for their child, and providing counselling and self-care advice. The intervention showed a significant improvement in the mental health score of the caregivers in the general health questionnaire (d=0.77) at 8 weeks post-intervention.

Another study by Berberoglu et al. (2024)³⁷ provided a structured supportive approach based on the theory of comfort where the caregivers were trained on how to deal with their child’s needs. The content of the training was based on the themes of physical, environmental, sociocultural, and psychospiritual aspects. The caregiver was provided with a booklet, video and an audio-recorded training session. The comfort behavioural checklist and the quality of life scale for children both showed significant improvements in scores at two months post-intervention, (d=1.27) and (d=0.35) respectively. The parents’ self-efficacy scale also had a large effect (d=0.98).

A study by AI Imam et al. (2022)³⁸ had a unique approach to address caregivers’ financial needs. They provided caregivers with a livelihood program which consisted of selecting a livelihood activity based on caregivers’ interest, providing the necessary commodities at no cost and knowledge and skill development for the particular activity. This was in combination with community-based rehabilitation sessions which consisted of collaborative goal setting, active participation of the caregivers and supporting the caregiver to set up an enriched environment to promote the child’s self-generated movements. The physical functioning domain of the health-related quality of life, the GMFM scores, and the monthly family income of caregivers did not show any improvement post-intervention. However, the caregiver social capital score showed an improvement with a significantly large effect (d=0.95).

A study by Benfer et al. (2024)⁴⁰ provided a peer-delivered intervention, ‘Learning through everyday activities with parents-CP’ (LEAP-CP), consisting of parented identified goals setting, capacity building of caregivers, parent education and environment enrichment. The control group received health advice (HA) on key family practices. Both LEAP-CP and HA were delivered by highly skilled community disability workers (CDW). The intervention did not show statistically significant improvements in child-related outcomes, including scores of the Pediatric Evaluation of Disability Inventory- Computer Adaptive Test (PEDI-CAT), Peabody Developmental Motor Scales (PDMS), Bayley Scales of Infant Development (BSID), Hammersmith Infant Neurological Examination (HINE), Canadian Occupational Performance Measure (COPM), Near Vision Detection (NVDS) and Home Observation for Measurement of the Environment (HOME). Similarly, there were no statistically significant improvements in the caregiver-related outcome, the Depression Anxiety and Stress Scale (DASS).

‘Enabling and partnership’ and ‘Coordinated and comprehensive care’

Only one article was identified under this category. The study by Palee et al. (2022)³⁶ involved goal-directed therapy which included team conference with the physiatrist, physical therapist, child and caregivers where one specific measurable achievable, relevant and timed SMART goals were discussed and set in collaboration with the team along with the entire family. Both the intervention and control groups received 50 minutes of physiotherapy sessions along with a home program to be performed by the caregiver. The GMFM showed a large effect in the total score (d=1.31), and the CPQOL scores also showed a large effect (d=1.30), However, the caregiver burden did not show any statistically significant improvement.

‘Information provision’ and ‘Enabling and partnership’

The interventions targeting infants at high risk of CP^39,41 allowed caregivers to become informed and actively participate in promoting their infant’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.¹⁰ 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 et al. (2020)⁴¹ had a longer intervention duration of one year, no difference was seen in infant outcomes. The lack of improvement in mental health 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.¹⁰ The study by Morgan et al. (2016)³⁹ clarifies that their intervention protocol was not designed to target parent’s mental health. Therefore, the lack of change only indicated that parents did remained stable during the course of the study. The influence on caregiver outcomes cannot be relied on as NOSI-K and ITQOL are inappropriate outcome measures for children less than 2 years. NOSI-K measures the parent’s general stress about their child’s life, their expectations, which may not change in the short term. 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 et al. (2018)²⁷ 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 et al. (2020)²⁸ as the intervention was delivered passively by mail, with no additional help to understand or translate the information to care.

Overall, no improvement was seen in caregiver stress^39,41 except on a non-standardized outcome measure.²⁷ 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.^39,41

‘Information provision’ and ‘Respectful and supportive care’

The intervention by Whittingham et al. (2022)³⁰ modified parent behaviour. The lack of effect on mental health should be considered with caution as parents in both the groups had normal mental health at baseline. Therefore, the authors did not expect any change in the results. 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 ‘Information provision’ and ‘Respectful and supportive care’ improved caregiver well-being.

Enabling and partnership

The two studies incorporated collaborative goal setting with the parents.^31,32 In the study by Law et al. (2011)³¹ 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 et al. (2018),³² no effect was seen on caregiver’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.⁴⁶ 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’s condition and development during therapy sessions.

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.

‘Enabling and partnership’ and ‘Respectful and supportive care’

The improvement in family needs in the study by Weindling et al. (2007) study³⁴ 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’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.⁴⁷ The family support workers helped in making the decisions, whereas in the study by Whittingham et al. (2014),³³ 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’ physical and psychological health, and advancement in child behaviour was associated with a better ability to handle stress and higher self-perception.⁷

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.³³

‘Information provision’, ‘Enabling and partnership’, and ‘Respectful and supportive care’

In the study by Kahjoog et al. (2019), the specific goals chosen by the caregivers for themselves and the child were addressed by the therapists in the intervention group.⁶ 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’ feeling of mastery over a caregiving situation and higher self-esteem predicts better psychological health.⁷ 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.

Similarly, the comfort and quality of life of children with CP, as well as parents’ self-efficacy, were enhanced by the supportive approach used in the Berberoglu et al. (2024)³⁷ study. This may be associated with the comprehensive approach to the child’s requirements that the theory of comfort adopts, together with the provision of caregiver-specific training sessions.⁴⁸

The research conducted by Panahi et al. (2022),³⁵ showed a noteworthy enhancement in the mental health score of caregivers. Given that mothers of children with CP have a significant responsibility in managing both daily tasks and caring for their children, which can result in burnout, the continuous care model employed in this study is a practical and easily accessible approach that has the potential to improve the mental well-being of mothers.⁴⁹ In contrast, the LEAP-CP programme in the Benfer et al. (2024) study found no evidence of a substantial overall benefit for either infants or caregivers when compared to the dose-matched HA programme. This may be explained by the fact that the HA control group included a few FCC components, such as receiving parent support advice from the highly skilled CDWs which was shared by both the control and intervention groups.

The implementation of an integrated community-based rehabilitation programme, in conjunction with a livelihood assistance initiative in the SUPPORT CP study by AI Imam et al. (2022) demonstrated a favourable enhancement in the health-related quality of life for children and the social capital of their families. However, it did not yield significant improvements in the child’s gross motor function or the monthly family income. This statement aligns with the concept of integrating the physical rehabilitation of children with the cognitive and economic empowerment of families to attain long-term sustainability.⁵⁰ The intervention arm had the lowest baseline values for monthly family income, which may have diluted the impact on the improvement of monthly family income. Furthermore, not all livelihood ventures can promise a substantial profit within a short period of time.

The overall results of the five studies in this domain suggest that interventions utilising “information provision,” “enabling and partnership,” and “respectful and supportive care” may have some impact on the comfort and quality of life of children with CP, as well as improving the caregivers’ social capital, self-efficacy, and overall mental health.

‘Enabling and partnership’ and ‘Co-ordinated and comprehensive care’

Goal-directed therapy in the study by Palee et al. (2022),³⁶ had better results when compared to the control group. Children’s improved quality of life may be due to improvements in their gross motor performance.⁵¹ The caregiving burden, however, did not indicate any significant change. This may be explained by the fact that caregiving burden is affected by variety of tasks and stresses while providing care for children with disabilities.⁵² It is noteworthy that there was a reduction in caregiver burden in both the intervention and control groups. It is likely that the intervention group’s benefit was lessened by the control group’s requirement to bring their child to physiotherapy appointments and receive advice on home exercise programs.

Few studies involved two to four healthcare professionals working as a team in either intervention or control groups such as physical and occupational therapists^27,39 or neuro-paediatrician, paediatric orthopaedic surgeon, physiotherapist, and biomechanist.²⁸ This brings to light the established importance of multidisciplinary care for children with CP. The study by Weindling et al. (2007)³⁴ which includes family support workers (FSWs) in addition to physiotherapists cannot be considered as multidisciplinary care, as FSWs were parents of children with CP, not health professionals.

Overall, all the studies incorporated the Enabling and partnership component,^6,27–41 thirteen had Information provision,^{6,27–30,39,41} eight had a Respectful and supportive care component.^{6,30,33–35,37,38,40} and one study had coordinated and comprehensive care as the component.³⁶ 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,³³ vs. passively mailing the information report to the caregivers.²⁸ Another example would be involving caregivers in goal setting,³² vs. involving them in the therapy activities as well.⁶ Therefore, the mode of delivering the intervention may also influence the intervention effects.

Research has reported the negative effects of caregiving on carers’ physical health such as fatigue, poor sleep, and musculoskeletal pain.⁵³ However, it is interesting to observe that none of the studies explored the effect of FCC interventions on caregivers’ physical health. This represents a significant gap in the literature and highlights the need for future research to explore this important area. The lack of effect on caregiver mental health in most of the studies may be because caregivers with CP have high burden and multiple sources of stress apart from caregiving responsibilities such as balancing family and work, and financial burdens.¹⁰ Any significant change in mental health may require comprehensive targeted interventions.

Conventional therapy cannot be carried out without involving the parents or family. The control group in multiple studies^{6,27,32,39,41} also had some degree of parent involvement which could not be excluded as they were a part of the ‘usual care’. 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’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.

Strengths and limitations

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.

Clinical implications

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’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’s condition, development, and handling during therapy visits to ensure a continuum of updates.

Future recommendations

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’ 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.

Underlying data

No data are associated with this article.

Extended data

Figshare: Search Strategy for “Effect of family centered care interventions on well-being of caregivers of children with cerebral palsy: a systematic review”. Data file 1: Search Strategy. https://doi.org/10.6084/m9.figshare.25718685.v1.⁵⁴

Figshare: Intervention details using TIDieR checklist for “Effect of family centered care interventions on well-being of caregivers of children with cerebral palsy: a systematic review”. Data file 3: Intervention details using TIDieR checklist. https://doi.org/10.6084/m9.figshare.25719021.v1.⁵⁵

Figshare: Description of outcome measures and results of intervention effectiveness for “Effect of family centered care interventions on well-being of caregivers of children with cerebral palsy: a systematic review”. Data file 4: Description of outcome measures and results of intervention effectiveness. https://doi.org/10.6084/m9.figshare.25718907.v1.⁵⁶

Figshare: Critical appraisal ‘CASP checklist’. https://doi.org/10.6084/m9.figshare.25718739.v1.⁵⁷

Figshare: Study characteristic. https://doi.org/10.6084/m9.figshare.25718730.v1.⁵⁸

Reporting guidelines

Figshare: PRISMA 2020 checklist for “Effect of family centered care interventions on well-being of caregivers of children with cerebral palsy: a systematic review”. Data file 2: PRISMA 2020 checklist. https://doi.org/10.6084/m9.figshare.25719117.v1.⁵⁹

Data are available under the terms of the Creative Commons Attribution 4.0 International license (CC-BY 4.0).