Cerebral palsy (CP) is not just a physical disability

Motor impairments are the clinical hallmark of CP; however, other impairments are common.1 Further, the optimal development of children with CP—psychological, social, emotional and cognitive development—is at risk. One in four children with CP have a behavioural disorder1 compared to 1 in 10 typically developing children.2 Seven in 10 preschool children with CP show significant delay in social milestones relative to community norms with continued social difficulties at school age.3–5 One in five preschool children with CP show a significant delay in self-care skills.5 In the long term, focusing on adults with CP without intellectual impairment, one in five have not completed high school, 4 in 10 are not living independently, one in two are not competitively employed and two in three are not in a long-term relationship.6 Families want support and solutions; however, they receive little support in optimising their children's psychological, social, emotional and cognitive development or in optimising child quality of life. Increases in parental responsiveness are a key factor in predicting child developmental benefits of early intervention,7 suggesting that parents themselves are pivotal to optimising child development.

Impact of CP on the whole family

CP impacts on the entire family in a manner that is long-term, complex and multifactorial. The birth of a child with CP may precipitate complex care responsibilities, financial hardship, limits to occupational attainments, relationship distress, grief and social isolation, all of which have further flow-on effects for the parents, child and family.8 In particular, mothers of children with CP experience greater parental stress than mothers of typically developing children.9 They are four times more likely to have elevated anxiety symptoms and five times more likely to have moderate depressive symptoms.10 ,11 In a cross-sectional study of 818 families of children (8–12 years) with CP living across Europe, parents were five times more likely to be experiencing very high stress levels than the general population. Interestingly, the magnitude of parenting stress was not associated with severity of motor impairment of the child;12 rather, parenting stress was associated with child behavioural and emotional problems.13–16 Parents commonly experience an ongoing grieving process, with grief intensifying during transitions and major child and family life events.17 Parents of children with CP parent in an emotional context that may include significant stress, ongoing grief, a loss of typical parenting rewards (eg, seeing your child walk for the first time), anxiety about the future and a lack of understanding from the broader community.

The developmental abilities of children impact on the quality of parental caregiving that the child is able to elicit within day-to-day interactions.18 Children with neurodevelopmental disabilities, who most benefit from optimal parental support, are unable to effectively elicit optimal parental support. Parents of children with CP need to be proactive, skilled and conscious in their moment by moment parenting choices to provide their children with optimal developmental support. Parenting a child with CP, and optimising their development, involves forward thinking, scaffolding abilities, a commitment to supervision, patience, compassion, behavioural management skills and effort above and beyond the optimal parenting of typically developing children. Further, all of that requires a strong and loving emotional bond and parental psychological resilience.

Parental responsiveness fuels development

The quality of the parent–child relationship is important for all children, regardless of individual characteristics and impacts greatly on diverse child outcomes, including developmental, emotional, cognitive, behavioural, relational, educational and quality of life outcomes.19–22 A meta-analysis demonstrated that parent–child relationships with high parental responsiveness—prompt, child-directed, contingent and appropriate parenting—are associated with improved cognitive, emotional, behavioural and social outcomes.23 Children who have experienced a warm and emotionally available parent–child bond are more likely to be socially competent and emotionally well-adjusted, establishing stable relationships in later life.24 Crucially, language, cognitive reasoning and preacademic skills are learnt through the day-to-day interactions of the parent–child relationship. In a longitudinal observational study with 42 families, the quality of everyday parent–child interactions in the first 3 years of life was found to predict 61% of the variance in rates of vocabulary growth and use, as well as 59% of the variance in a child's Standford Binet IQ score at age three.25 During everyday relationship-driven encounters, parents provide hour after hour of environmental enrichment: rich, quality experience in verbal skills, cognitive reasoning and playful exploration. Parental responsiveness can be conceptualised as nature's dose-control system for environmental enrichment,26 enabling the child to obtain ‘just right’ stimulation for learning and development organically in day-to-day life via parent–child interaction and the flow-on effects of such interaction. Nowhere is this more important than in the parent–child relationships of children with developmental delays or disabilities. These relationships may be built and enhanced through the targeted assistance of various psychological approaches.

Acceptance and Commitment Therapy (ACT): a promising approach

ACT, said as the word ‘act’ is now an accepted part of the broad cognitive–behavioural therapy (CBT) approach. ACT incorporates: mindfulness or deliberate non-judgemental attention to moment to moment experience, experiential acceptance or ongoing non-judgemental contact with psychological events such as difficult emotions, cognitions and memories and vital living or engagement in meaningful, values-driven activities.27 The goal of ACT is to increase psychological flexibility: the ability to persist or change one's behaviour, with full awareness of the situational context and one's own present-moment experience, in the service of chosen values. ACT and related psychological therapies have a growing evidence base for a diverse range of issues, for example, occupational stress, depression, anxiety and smoking cessation.28–31 Meta-analyses, examining the efficacy of ACT and related psychological therapies with varying dosages (30.0±29.8 hours in30), and targeting a variety of issues, have identified moderate effect sizes ranging from 0.5 to 0.75.28–31 Psychological flexibility, the clinical target of ACT, predicts levels of parental psychological symptoms, experienced parenting burden and symptoms of grief in parents of children with CP,17 as well as parental psychological symptoms, maternal bonding and responsiveness in mothers of infants born preterm.32

Face-to-face parenting intervention incorporating ACT is beneficial

A parenting intervention incorporating ACT was shown to be efficacious within a three-group RCT.33–35 Sixty-seven parents of children (mean age 5.3) with CP participated in the trial. Parents were randomly assigned to: wait-list control, the parenting intervention Stepping Stones Triple P or to a combined Stepping Stones Triple P and ACT group. Families receiving Stepping Stones Triple P alone, compared to wait-list, showed improvements in child behaviour and emotional symptoms. Families receiving the combined Stepping Stones Triple P and ACT intervention, compared to wait-list control, showed improvements in child behaviour, child hyperactivity, child functional mobility performance, child quality of life, parenting style and parental adjustment. During the randomised controlled trial (RCT), face-to-face delivery was identified as a barrier to effective translation requiring an innovative solution to capitalise on the potential benefits from the ACT-based approach.

Online therapy is effective and translatable

A meta-analysis of web-based psychological therapy indicates that web-based delivery is effective with an average effect size across intervention types and measures of 0.53 consistent with a medium effect and with greater effect sizes of 0.83 for cognitive–behavioural interventions.36 Further, 14 RCTs were identified that directly compared web-based psychological therapy with traditional face-to-face psychological therapy and no significant differences between the two treatment modalities could be found. Online delivery modalities may be an ideal vehicle for low-cost and sustainable knowledge translation directly to parents in all situations, but specifically those families with children with disabilities who may require extra time and assistance, therefore reducing the capacity to access traditional forms of support.

If found effective, parenting acceptance and commitment therapy (PACT) could be easily and rapidly disseminated population-wide. Potentially, families of children with CP in different countries could access immediate evidence-based psychological support, from first receiving diagnosis onwards, to promoting parent and child flourishing and to harnessing existing strengths within the parent–child relationship to foster long-term developmental gains for the child. PACT is flexible and family-centred, leveraging the understanding, skills and the day-to-day interactions that already exist within the family system for the benefit of both child and family and placing the parents as the central experts in their own child's behaviour.

Aim

Our aim is to demonstrate the efficacy of an innovative, translatable-by-design online parenting support package: PACT, said as the word ‘PACT’ for families of young children (2–10 years of age) with CP. PACT will be delivered in the form of an online course for parents: PARENT101: Parenting with Purpose.

Hypotheses

We predict that participating in the PACT course, PARENT101: Parenting with Purpose, will be associated with improvements in the parent–child relationship and in parent and child adjustment and quality of life. Following baseline assessment, outcomes will be assessed postintervention after the final review session and at 6 months postintervention (retention of treatment effects).

Design

The study is a RCT following Consolidated Standards of Reporting Trials (CONSORT) guidelines. Parents of children with CP will be randomly allocated to one of two groups:

1. Online PACT in the form of the online course: PARENT101: Parenting with Purpose

2. Wait-list control

Follow-up will occur at 6 months postintervention (retention). The wait-list condition will be offered the intervention after completion of the review assessment.

Recruitment

We aim to recruit 66 parents of children (2–10 years old) diagnosed with CP. Families of children with CP will be recruited through the Queensland Cerebral Palsy and Rehabilitation Research Centre, hospitals around Australia, the Australian Cerebral Palsy Register and word of mouth. Since PACT uses online delivery, there are no impediments to participants being recruited from across Australia including in regional areas, making this project highly feasible.

Exclusion criteria: Since PACT uses integrated web-based delivery, parents are required to have: (1) reliable internet access at home (eg, ADSL) and be committed to maintaining internet access for the duration of the study; (2) a mobile phone for receiving text messages that they are asked to check regularly throughout the study; (3) an email address for receiving emails that they are asked to check several times a week throughout the study; and (4) access to Skype.

Sample size: To have sufficient power to detect an effect size of 0.40 (power 0.8), a total sample size of 52 is required. If a conservative retention rate of 80% is assumed, this leads to a total of 66 families to be recruited.

The PACT intervention

PACT includes ACT techniques already established in an RCT as effective with families of children with CP, including mindfulness, experiential acceptance and cognitive defusion.33–35 It includes some techniques from the established parenting literature but focuses on strategies to enhance the parent–child relationship and promote parental emotional responsiveness. An online course was created using the edX platform, an open source course management system created by founding partners Harvard and MIT (http://www.edx.org/). The edX platform is used to host Massive Open Online Courses and allows for the online delivery of a variety of educational courses that can be accessed from anywhere that has internet availability.

Within the edX framework, the PACT programme has been developed to give it the look and accessibility of a course and has been named PARENT101: Parenting with Purpose. PARENT101 incorporates: virtual written text, video presentations, screen-based interactive activities, journal activities, reflections, guided experiential and mindfulness exercises and participation in a moderated discussion board. In addition, research participants will be offered Skype sessions with a therapist as well as reminder text messages and emails. PARENT101 is presented as three modules plus a review session 4 weeks after the final module. Modules are scheduled to be completed each fortnight, with a 4-week break after that to allow for practice of the materials from the course. Overall, the course lasts for 10 weeks (6 weeks for modules plus review 4 weeks later) in total, with parents able to move through each module's content with some flexibility. Access to PARENT101 will be restricted to study participants until efficacy is established.

Study procedure

After obtaining ethical approval, a recruitment strategy was developed to maximise opportunity for interested parents to participate. This involves recruiting through a variety of public hospitals, CP-related organisations and potentially social media sites relevant to parents of children with CP. In hospitals, relevant clinicians have agreed to develop recruitment protocols that introduce the study details and provide mechanisms to register interest in the study. In other settings, flyers/promotional letters will be disseminated with contact details of the researchers for interested participants.

Following registration of interest by individuals, they will contacted by researchers to assess eligibility for participation. After provision of written informed consent, parents will be asked to complete all baseline measures, including recording a parent–child interaction for observation, before being randomly allocated to either intervention or control. Randomisation will be applied using a computer-generated block randomisation. The initial randomisation sequence will be managed by an individual outside of the study. Following recruitment, the research manager will determine each participant's status by opening an opaque envelope. Although the researchers will not be involved in the initial creation of the randomisation sequence, owing to the study design they will not be blind to participants' condition status. Families allocated to the PACT condition will then receive access to the PARENT101 course for immediate enrolment and the control condition participants will be advised of their timelines. In the interests of equity and retaining participants, the control condition participants will be offered access to the course after completion of the postreview assessment.

Assessment will be conducted at baseline, postreview (10 weeks after course initiation) and at 6 months postintervention follow-up. All written assessment measures will be completed by parents. Parents will also have the opportunity to provide course evaluation feedback after each module and at the end of the review session.

Measures

A Family Background Questionnaire will be developed, tailoring existing standardised measures to assess general demographic variables such as SES and parental education and family factors specific to the CP context.

Gross Motor Function Classification System is a parent rating measure using a five-level system to classify children into their age-specific gross motor ability.37 It is valid and reliable and frequently used to classify functional abilities of children with physical disability.

Emotional Availability Scales (EAS; Z Biringen, J Robinson, R Emde. Emotional Availability (EA) Scales. 3rd edn. Unpublished manual, 199838) The EAS is the primary outcome measure. Parents will be asked to video record a 20 min naturalistic observation of the parent–child relationship in the home. Families will be able to send their recordings through a secure File Transfer Protocol) connection. It will be scored by an independent rater, blind to the intervention condition, using the EAS. The independent rater will be trained in the EAS. The EAS is a dyadic measure, that is, it measures the quality of the relationship itself across six scales: parental sensitivity, parental structuring, parental non-intrusiveness, parental non-hostility, child responsiveness and child involvement. The EAS also generates a global relationship quality rating. The scale has high inter-rater reliability for parental responsiveness (0.96), involving (0.87), sensitivity (0.93) and structuring (0.76).

Interpersonal Mindfulness in Parenting (IMP39) scale is a 10-item measure of a parent's ability to maintain present-centred attention and emotional awareness during parent–child interactions. Parents respond on a five-point Likert scale to a series of statements. The Intramuscular-P scale produces four subscales: present-centred attention in parenting, present-centred emotional awareness in parenting, non-reactivity/low-reactivity in parenting and non-judgemental acceptance in parenting. The IMP scale has adequate concurrent and discriminant validity.40

Emotional Availability-Self Report (EA-SR41) is a 32-item parent-report measure of emotional availability within the parent–child relationship. It has excellent reliability and validity. The EA-SR produces five subscales: mutual attunement, affect quality, capacity to involve the parent, intrusiveness and hostility.

Acceptance and Action Questionnaire (AAQ-742). This 7-item version of the Acceptance and Action Questionnaire measures experiential avoidance, that is, attempts to control the form, frequency or situational sensitivity of private events such as memories, cognitions and emotions, particularly when doing so causes harm. It produces a single total scale and can be scored either so that high scores reflect high experiential acceptance or so that high scores reflect high experiential avoidance. A series of statements is rated on a seven-point scale. The AAQ has satisfactory internal consistency (α=0.79).

Depression Anxiety Stress Scale (DASS-2143) is a 21-item questionnaire that assesses symptoms of depression, anxiety and stress in adults. Respondents rate items on a four-point Likert scale reflecting how much the statement applied to them in the past week. The DASS produces three subscales each with good internal consistency: the depression (α=.91), anxiety (α=.84) and stress (α=.90) scales. The DASS also has good discriminant and concurrent validity (Brown, Chorpita, Korotitsch, and Barlow, 1997; Lovibond and Lovibond, 1995).

Personal Well-being Index: (PWI;44) is an eight-item measure of personal well-being in adults. Respondents rate their degree of satisfaction with various domains of living on a 10-point Likert scale. The responses are summed to create an average score representing subjective well-being. Respondents can also be asked to rate their life as a whole. The PWI has good validity and internal consistency (α>0.70;44 ,45).

Strengths and Difficulties Questionnaire (SDQ46) is a 25-item parent-report measure of child behaviour and adjustment in which the frequency of behaviours is rated on a three-point Likert scale. The SDQ produces five subscales: emotional symptoms, conduct problems, inattention/hyperactivity, peer problems and prosocial behaviour (range 0–10). It produces a total difficulties score (range 0–40) that has been found to have adequate internal reliability (α=.76) and test–retest reliability (r=.86) as well as discriminant and concurrent validity.47 The SDQ is widely used in CP research.16 ,48

CP QOL-Child49 is a 66-item parent-report measure of child quality of life that is specifically developed for use in children with CP. It measures quality of life across several aspects of the child's life: physical well-being, social well-being, emotional well-being, school, service access and social acceptance. Higher scores on each scale reflect a higher quality of life in that domain. It has good concurrent validity, internal consistency (α=0.76–0.89) and test–retest reliability (r=0.80–0.90).

What do parents think of PACT?

A key aspect of the study is ongoing parental evaluation of the course and its suitability for their situation. Client satisfaction with PARENT101 content and format will be measured using questions embedded into the course after completion of each module and review. The questions will ask parents to rate various aspects of PARENT101 on 10-point Likert scales as well as give qualitative feedback. This feedback will be examined in the light of unintended adverse effects and appropriate steps will be taken if any are noted.

Data management

Questionnaire data will be collected online through Qualtrics and uploaded into SPSS. Observational data will be entered manually. All data will be stored securely on the university network.

Statistical analysis

Analysis will follow standard methods for randomised controlled trials using comparisons between the two groups. Initially, the two groups will be assessed to determine if they differ on key variables at baseline and covariates will be used to control for this in postintervention analyses if necessary. The protocol of intention-to-treat analysis will be followed in order to minimise inflation of treatment effects consistent with CONSORT guidelines. The experimental unit will be the family represented by parent-reported data. Attrition analysis will be conducted, although strategies will be used in order to minimise missing data and attrition. Data will be analysed using SPSS. The hypotheses relating to intervention efficacy will be tested using general linear models, specifically via analysis of variance or analysis of covariance. Where continuous data exhibit substantial skewness not overcome by transformation, non-parametric methods will be used for simple comparisons.

Ethics and dissemination

This trial has been registered with the Australian New Zealand Clinical Trials Registry (ACTRN12616000351415). Study results will be disseminated through publication in scientific journals and presentation at relevant conferences as well as directly to research participants. If efficacious, PARENT101 has the potential to be widely disseminated.