Article Text
Abstract
Introduction Mothers of children with intellectual disability (ID) are often distressed because of intensive workloads and difficulties in communicating with their children. Given the interdependence between the psychosocial well-being of such dyads, interventions that promote parent–child relationships and mutual communication would be beneficial. Arts provide alternative avenues for expression and offer an imaginative and playful environment for discovering new communication strategies. Given the lack of studies on arts-based dyadic interventions, this study aims to examine the effectiveness of dyadic expressive arts-based intervention (EXAT) in improving the psychosocial outcomes of children with ID and their mothers and the mother–child relationships.
Methods and analysis This study will adopt a mixed-methods randomised controlled trial design, wherein 154 dyads of children with ID and their mothers will be randomised into either the dyadic EXAT group or the treatment-as-usual waitlist control group. Quantitative data will be collected at four time points: baseline (T0), postintervention (T1), 3-month postintervention (T2) and 6-month postintervention (T3). Qualitative data will be collected from a subset of 30 mothers in the intervention group at T1 and T3 to document their experiences and perceived changes after the intervention. Mixed-effects models and path analysis will be adopted to analyse the quantitative data, whereas thematic analysis will be applied to the qualitative data. Both sets of data will be triangulated for an integrated view of the effectiveness and mechanism of the intervention.
Ethics and dissemination Ethical approval has been obtained from the Human Research Ethics Committee of the University of Hong Kong (Ref. no.: EA200329). Written consent forms will be obtained from all recruited participants (mothers, children with ID and teachers/social workers) before data collection. The study findings will be disseminated in international conferences and peer-reviewed academic journals.
Trial registration number NCT05214859.
- Mother-child relationship
- Psychosocial well-being
- Expressive arts therapy
- Intellectual disability
- Randomised controlled trial
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- Mother-child relationship
- Psychosocial well-being
- Expressive arts therapy
- Intellectual disability
- Randomised controlled trial
STRENGTHS AND LIMITATIONS OF THIS STUDY
This randomised controlled trial will be the first to explore the effectiveness of dyadic expressive arts-based intervention (EXAT) in improving the psychosocial well-being of children with intellectual disability and their mothers and the mother–child relationships.
The qualitative component of the study will explore the experiences of participating in the dyadic EXAT and its mechanism in affecting the dyad’s psychosocial well-being and relationship.
The study will include children with a diagnosis of autism spectrum disorder to ensure a sufficient number of eligible participants.
The study will include all the children in the primary section of special schools with a relatively wide range of ages.
Introduction
Intellectual disability (ID) refers to a developmental disability that significantly limits intellectual functioning and adaptive behaviours, which impedes daily functioning.1 ID onset typically occurs before adulthood1 and affects 0.05%–1.55% of the global population.2 The Diagnostic and Statistical Manual of Mental Disorders, Fifth Edition (DSM-5) classifies ID with four different severity levels, ‘mild’, ‘moderate’, ‘severe’ and ‘profound’, to differentiate one’s capacity in adaptive functioning and life skills.3 With appropriate care and developmental training, individuals with mild or moderate level ID are capable of acquiring some life skills and living independently in familiar settings.4 The ID prevalence in Hong Kong is 1.0%–1.4%,5 affecting approximately 0.84% of the primary school-age population,6 and around 88% of primary school students with ID are diagnosed with mild or moderate ID.6 As caring for children with ID relatively requires more care, effort and resources, the parents of children with ID are prone to emotional distress and poor quality of life.
Parents of children with intellectual disabilities are more likely to report higher parental stress than average.7 8 Interactions between parents and children with different levels of ID are also less effective due to children’s lack of initiation, responsiveness and salient cues during communication and their behavioural problems.9–11 School-age children with ID also exhibit less secure and more disorganised attachment behaviour that may affect their relationship with adults.12 Parents may also be more likely to adopt negative parenting styles to manage their children, such as being unsupportive and controlling.13 14 Moreover, children with ID, particularly with a moderate level of ID, may also be comorbid with other developmental disorders, such as autism spectrum disorder (ASD), which may further induce stress and impair the quality of life of their parents.15 Despite facing the immense challenge, some parents can still find positivity in parenting their children with ID.16 Various supports, such as early screening with intervention and inclusive education,17 have been developed to promote the development of children with ID at different levels and to ameliorate the burden on the parents.
A recent local survey has demonstrated that more than 90% of caregivers of children with special educational needs, including intellectual disabilities, are mothers.18 This may be due to entrenched patriarchal beliefs in Hong Kong society, which expects mothers to be caregivers and homemakers and fathers to be breadwinners.19 Compared with other family caregivers, such as fathers and siblings, mothers may be at a higher risk of psychosocial difficulties because of the lack of social support, low income and being forced to be unemployed.20 21 Mothers are also known to devote enormous amounts of time and energy to caring for children with ID, which subjects them to intensive stress, marital issues, psychological disturbance, disrupted social lives, poor physical health and uncertainty about the future.22–26 Furthermore, the mothers of younger or school-age children with ID have reported experiencing more stress than the mothers of older children with ID, as younger children require higher levels of care. Mothers also often face dilemmas in decision-making, such as when choosing schools and rehabilitation programmes.25 26
Extensive interventions or services have been provided to support children with ID and their families, such as in the form of parenting skills training, psychoeducational support for parents and behavioural management programmes for children.27 28 Nonetheless, few interventions focus on enhancing communication, relationships and overall well-being for mother–child dyads. The provision of parent–child support for this population is crucial as the quality of the relationship and interactions between parents and the child with ID determines the development and mental health of the children.29 30 Positive parenting behaviour and parent–child relationship also help infuse resilience that may lower parental stress31 and may also help reduce the frequency of externalising behaviour problems in children with mild or borderline ID.32 Therefore, it is essential to promote positive interactions, mutual understanding and communication between mothers and their children.27 Support is also needed to improve the mother’s mental health and equip them with strategies to cope with the children’s behaviours.33
Need for arts
Arts can serve as an avenue for self-expression and communication, particularly for individuals who find verbal self-expression difficult,34 like children with ID. Previous studies on arts-based dyadic interventions for parents and their children with or without disabilities have also demonstrated an array of benefits, including promoting mothers’ mental health, decreasing child’s behavioural problems, nurturing secure attachment, expanding playfulness, and facilitating interconnection, communication, understanding, and synchrony within dyads.35–40 All these effects benefit children with ID and their mothers in terms of developing a positive relationship and promoting mutual understanding and communication. In addition to applying a single art modality approach, this study will employ an expressive arts-based intervention (EXAT) that uses different art modalities, such as music, drama, movement and visual arts, to attain therapeutic goals.41 It adheres to an intermodal approach, which posits that the experience of expressing oneself can be deepened and expanded by interchanging art modalities.42 The flexibility of the use of arts can also allow dyads to explore and experiment with different arts materials. A group dyadic setting will also be adopted as it provides room for cultivating mutual support and connections within and across dyads, which possibly create impacts on the parent–child relationships and empower the mothers.39 While there are existing studies on parent–child dyadic arts intervention in different populations, studies focusing on children with ID and their mothers and with more rigorous designs, such as those with larger samples or comparable control groups, are still limited. Including a treatment-as-usual control group in the study design helps reveal the treatment effects of EXAT on top of the generic routine and care of the dyads.
Research objectives
The primary objective of the study is to examine the effectiveness of the dyadic EXAT in improving psychosocial outcomes of children with ID and their mothers and the mother–child relationships compared with a treatment-as-usual waitlist control group across different time points. The sustainability of the effects will be examined at 3-month and 6-month postintervention follow-up time points. It is hypothesised that dyads in the EXAT group will experience improved psychosocial conditions and mother–child relationships, relative to the dyads in the control group. The second objective is to explore the mechanism by which the intervention acts on mother–child dyads; the study will investigate the relationships between psychosocial variables and intervention effectiveness, such as mediation or moderation effects. Qualitative interviews will be used to document the subjective experiences of and changes in mothers after participating in the intervention.
Methods and analysis
This study will employ a two-arm, mixed-methods, randomised controlled trial design. The intervention phase will last for 8 weeks. The outcome measures will be assessed at four time points: baseline (T0), 2-month after baseline (postintervention, T1), 5-month after baseline (3-month postintervention follow-up, T2) and 8-month after baseline (6-month postintervention follow-up, T3). This will help capture the immediate and sustained effects of the dyadic EXAT on mother–child relationships and psychosocial well-being. In addition to quantitative outcome measures, qualitative interviews with a subset of mothers (ie, 30 mothers) from the intervention group will be conducted at T1 and T3. The interviews will help document information regarding the experiences of participating in the dyadic EXAT and whether these experiences shape the caregiver experience, particularly the relationship between mothers and their children and the mothers’ coping strategies during caregiving. Adopting the mixed-methods design will help triangulate the findings and enhance our understanding of the effects of the dyadic EXAT on mother–child dyads. Figure 1 depicts the schedule of enrolment, allocation and assessments. Figure 2 shows the study flow based on the Consolidated Standards of Reporting Trials (CONSORT) flow diagram.
The schedule of enrolment, allocation and assessments. aEXAT: Dyadic Expressive arts-based group. bOnly a subset of mothers in the intervention group (ie, 30 mothers) will participate in a semi-structured in-depth interview for understanding their perceived changes and experiences after participating in the dyadic expressive arts-based group.
The study flow based on the Consolidated Standards of Reporting Trials (CONSORT) flow diagram.
Intervention
Dyads in the intervention group will receive eight 90 min weekly sessions. The sessions will be conducted at the children’s schools by either a registered EXAT therapist, an EXAT trainee or a mental professional trained in EXAT. Each session will be conducted in a small group of 3–4 dyads. The group facilitators will receive training in the standardised protocol and safety precautions. The principal investigator (PI; corresponding author) will provide on-site and/or off-site supervision to ensure treatment fidelity. Themes related to mother–child relationships will be covered. The content of the intervention was informed by the previous works on applying arts-based activities or interventions for individuals with ID or mother–child dyads in Hong Kong.36 43 44 Each session will comprise four stages, which will involve both within dyads or across dyads arts activity. All the dyads will work together as a group during greetings and sharing, whereas dyads will mostly work individually during the warm-up and core art(s) activities. Table 1 depicts the themes and objectives of each session. Some selected examples of arts activities are also listed. Table 2 describes the stages of each session.
Themes and objectives of each session
Structure of each session
The dyads in the treatment-as-usual control group will continue their normal routines and care. They will be invited to join the intervention group on completion of all assessments.
Randomisation and blinding
Eligible dyads will be allocated to either the dyadic EXAT group or the treatment-as-usual waitlist control group on a 1:1 basis based on a list of non-repeating computer-generated random numbers. The list will be generated by a staff who will not be involved in the trial. The designated staff of each school will be notified of the allocation results. A research assistant will be responsible for the randomisation and case allocation procedures.
Given the nature of the trial, it will not be feasible to blind the participants (dyads and teachers/social workers) and therapists to the group allocation results. No unblinding procedures are predetermined for revealing the participants’ allocation during the trial. However, the data analyst will be blinded. A research assistant will recode the group information in the dataset before passing it on to the data analyst for data analysis.
Sample size
The sample size was estimated using the analysis software G*power V.3.1.45 A moderate effect size (Cohen’s d=0.5) was expected based on a previously unpublished local study conducted in 2017 (Li IMY, unpublished thesis, the effectiveness of an attachment-based expressive arts therapy parenting programme for parents with special needs children, 2017). Assuming an attrition rate of 20%, a total sample of 154 dyads will be needed (ie, 77 dyads per arm) to reach 80% statistical power and attain a moderate effect size (Cohen’s d=0.5) in the proposed two-arm randomised controlled trial with four measurement time points at a 5% level of statistical significance.
Participants
The recruitment of participants will be carried out at special schools for children with ID in Hong Kong. The study has been started on 9 July 2022, and is estimated to be ended on 31 December 2025. This study will collaborate with the Hong Chi Association, a major non-governmental organisation (NGO) in Hong Kong that provides special education services for students with different grades of ID. This study will also aim to collaborate with special schools run by other NGOs. Social workers and teachers at the interested schools will assist with the screening procedures and refer potential participants to the research team. The eligibility of mother–child dyads will be determined using the following inclusion and exclusion criteria.
Inclusion criteria
The child is 6–12 years old (primary school student).
The child is diagnosed with mild to moderate ID, IQ score ranges from 35 to 69 (based on the assessment conducted by certified clinicians).
By the judgement of the health/school professional staff, the child is capable of responding to assessments and participating in group activities.
The dyad is willing and able to give consent for participation.
Exclusion criteria
The child is diagnosed with attention-deficit/hyperactivity disorder.
The dyad is currently participating in other behavioural or pharmacological trial(s).
Either the mother or child has other contraindications or severe comorbidities that may impede participation (eg, severe physical disabilities).
Outcome measures
The outcome measures of this trial will comprise (A) psychosocial condition of mothers; (B) psychosocial condition of children; (C) mothers’ perceptions of parent–child relationships and (D) demographics and clinical profiles of the dyads. The mothers will complete a self-administered questionnaire regarding their psychosocial conditions, perceived parent–child relationships and the dyads’ demographic and clinical information. Research assistants will assist children in responding to the mood scale. An EXAT therapist, a trained research assistant or an EXAT trainee under supervision will administer the Face Stimulus Assessment. Teachers or social workers of each child will also be invited to fill the behavioural checklist. To enhance retention of the participants, data will be collected from places convenient for the participants, such as the dyads’ homes or special schools. Research assistants will send reminders to the participants to attend appointments for data collection.
Mothers’ psychosocial outcomes and perceived parent–child relationships
Parenting stress
The 36-item Parenting Stress Index (fourth edition, short form) will be adopted to assess parenting stress.46 The scale consists of three subscales, parental distress, parent–child dysfunctional interaction and difficult child, and each item is measured on a 5-point Likert scale. The official Chinese translated version from Psychological Assessment Resources will be administered.
Burn-out
The level of burn-out of mothers will be captured by the Chinese version of the six-item client burn-out subscale of the Copenhagen Burnout Inventory,47 with each item rated on a 5-point Likert scale.
Parent–child relationship
Parent–child relationship will be captured by two subscales with a total of 19 items from the Parent–Child Relationship Inventory: parent–child communication and satisfaction with parenting.48 The subscales assess attitudes towards parent–child relationship on a 4-point Likert scale. The research team has obtained permission from the publisher to translate the items into traditional Chinese and validate the translation.
Positive and negative affect
The mothers’ positive and negative affectivity will be assessed by the Chinese version of the 10-item International Positive and Negative Affectivity Schedule-Short Form.49 The scale has two subscales, one each for positive and negative affect, and each item is rated on a 5-point Likert scale.
Quality of life
The mothers’ quality of life will be captured by the 28-item Hong Kong Chinese brief version of the WHO Quality of Life Measure.50 It assesses the subjective quality of life in four domains: physical, psychological, social relationship and environmental.
Psychological well-being
The mothers’ psychological well-being will be documented by the Hong Kong Cantonese version of the five-item WHO Five Well-Being Index.51 It measures subjective psychological well-being on a 6-point Likert scale.
Children’s psychosocial outcomes
Mood states
The Ottawa Mood Scales will be adopted to measure the children’s mood states.52 It is a Visual Analogue Scale composed of five items that assess mood, anger, worry, stress and self-regulation. Relevant faces, icons or images will be shown to help children choose the most suitable answer that reflects their mood states. Despite this scale only being validated in a young adult sample,53 the pictorial features in a scale can draw interest from children and may help obtain a more relevant response from them.54 The research team has obtained permission to translate the scale into traditional Chinese and validate the translated version.
Emotional expression
The emotional expression of children with ID will be captured by the Face Stimulus Assessment.55 It is a projective drawing test that requires children to use the provided colour markers on three A4-size drawing templates: first on a predrawn face, second on an outline of a face and third on a blank sheet of paper. The sketches will be analysed based on the guidelines mentioned in the Face Stimulus Assessment E-Packet and Rating Manual (second edition), provided by the author of the assessment. The sketches will be digitally scanned to extract the patterns of colour usage.43 The research team has obtained permission to translate the guidelines into traditional Chinese and validate the translated version.
Child behaviour
The behaviours of children with ID will be assessed by the teachers or social workers via the Chinese version of the Child Behaviour Checklist Teachers’ Report Form (6–18) for school age children.56 The checklist yields aggregate scores on externalising, internalising and total behavioural problems, and on eight syndrome scales: anxious/depressed, withdrawn/depressed, somatic complaints, social problems, thought problems, attention problems, rule-breaking behaviour and aggressive behaviour.57
Socio-demographic and clinical information
Demographics
Sociodemographic information of the mothers, namely age, gender, education level, family composition and financial status, and the children, namely age, gender, grade and level of ID, will be collected from the mothers through self-report questionnaires.
Clinical information
Clinical profiles of the mother–child dyads, such as time since diagnosis of the children with ID, time of onset and history of psychiatric disorders, presence of any comorbidities (eg, physical disabilities, hypertension, diabetes mellitus, other cognitive disturbances), and history of community and rehabilitation service utilisation will be documented.
Individual semistructured in-depth interviews
A subset of mothers (ie, 30 mothers) from the intervention group will be randomly invited to attend individual, semistructured interviews at T1 and T3. The first interview will explore their experiences of participating in the intervention and whether there are any immediate changes. The second interview will investigate possible changes related to the intervention and whether these changes further induce changes in their daily lives and relationships with their children. The interviews will be conducted by the research team based on the interview guide (online supplemental file 1). The sample size will be set at 30 mothers as data saturation will be reached in under 25 interviews58 and to ensure sufficient data after potential drop-outs at T3.
Supplemental material
Data analysis
Quantitative analysis
The intention-to-treat principle will be used to address the effects of crossovers and dropouts. Missing data will be handled by full information maximum likelihood. Descriptive statistics will be used to summarise the sociodemographic characteristics of the participants at all time points. Treatment outcomes will be examined by computing mixed-effects regression models. This analytical method will analyse the repeated measures between the groups to obtain the time effect, group effect and time×group interaction effect. Demographic and clinical variables will be controlled for in the analysis. Path analysis will be adopted to examine the mediating role of the parent–child relationship and parenting stress on the effect of dyadic EXAT on psychological distress and caregiver burn-out. Mediation effects will be determined based on the significance of the indirect effects of the intervention on psychological distress and caregiver burn-out via the mediators. As the data distribution is expected to be skewed, bootstrapping will be used to estimate the confidence intervals of the indirect effects. Statistical tests will be performed by using Mplus V.8.3 (Muthén & Muthén, 1998–2017) and will be two tailed with statistical significance set at 5%.
Qualitative analysis
Audio recordings of the in-depth interviews will be transcribed verbatim by the research staff. All of the transcripts will be imported to NVivo V.12.0 (QSR International, 2018) or above for data analysis. The thematic analysis approach59 will be used to identify codes and themes related to the experiences of the dyadic EXAT and the possible mechanism of the intervention. Two research staff members will be involved in data analysis to ensure the credibility of the findings.
Study monitoring and data management
The progress and safety of the study will be monitored by the General Research Fund of the Research Grants Council (RGC). A mid-term progress report and interim analyses will be submitted after having implemented the study for 18 months. In case of RGC identifies any serious problem or unsatisfactory progress, the trial may be terminated. A final report will be submitted on completion of the study.
Regarding data entry, quantitative data collected from the participants will be entered into the dataset by research assistants. After inputting all of the data, the data analyst will perform data screening to check its validity. Invalid inputs will be cross-checked and resubmitted by another research assistant. Regarding qualitative data, the audio recordings will be transcribed into text files, and the sketches created by the children will be digitally scanned by research assistants. The data analyst will check the transcripts by concurrently listening to the audio and reading the transcripts. Amendments will be made if necessary.
Regarding data storage, all of the hard copies, after removing personal identifiers, will be stored in a locked cabinet at the PI’s research centre. Soft copies and other related electronic files will be encrypted and stored in a password-protected desktop computer at the PI’s research centre. All the hard and soft copies will keep for a maximum of 3 years after the publication of the first paper. Only the relevant research staff members, that is, the PI, the Co-Is, research assistants and data analysts, will be granted access to the raw data files and trial data set.
Patient and public involvement
Patients and the public were not involved in designing the protocol. The findings and protocols of the study will be disseminated to the participants on request.
Ethics and dissemination
Ethical approval has been obtained from the Human Research Ethics Committee of the University of Hong Kong (EA200329). The protocol of this study has been registered with the Clinical Trial Registry (NCT05214859). Any amendments to the protocol will be reported to the ethics committee and clinical trial registry. Trained research assistants will explain the rights of the participants and objectives of the study to participants and obtain their written consent before data collection. The data collection and dyadic EXAT are not expected to cause any physical or psychological harm. If the participants are disturbed by the data collection or intervention process, they can choose to skip or terminate any procedures. Professional referrals can be made on request. All such incidents will be reported to the PI. Regarding disseminating the findings, the team will present at internal or local conferences and will publish in peer-reviewed academic journals.
Discussion
The mothers of children with ID are under high caregiving strain, and the children themselves experience difficulties in channelling their emotions. This study hopes to raise awareness of the psychosocial well-being of both children with ID and their mothers. Interventions are needed to mitigate their psychological distress and boost their psychosocial well-being. As dyadic interventions, particularly art(s)-based dyadic interventions, have been limited, the proposed study will aim to fill this gap by providing evidence of the effectiveness of dyadic EXAT in improving the psychosocial well-being of children with ID and their mothers. The dyads are expected to benefit from the intervention in terms of nurturing mutual understanding, providing opportunities for expression and fostering psychosocial well-being. The findings will inform on the mechanism underlying dyadic EXAT and shed light on how arts can benefit mother–child dyads. In the future, art(s)-based interventions can be considered as one of the intervention strategies to provide psychosocial support for this population. Furthermore, the findings of this study can also contribute to the development and further application of art(s)-based interventions for children with ID and their families or for families with children with other disabilities or special needs.
Despite the strengths of the study, there are several limitations. Given the high comorbidity of diagnosis with ID and ASD, it is difficult to recruit sufficient participants with children with ID only. To ensure there are sufficient participants for the project, children diagnosed with ID and ASD will also be included. However, it will be complicated to differentiate whether the child’s behaviours are related to ID or ASD and if the maternal stress is caused by ID or ASD-related issues. This study will also include all primary school students aged from 6 to 12. While lower and higher primary school children may have different needs, the themes of the intervention shall be applicable to all primary school students. The therapist will fine-tune the process to meet the immediate needs and preferences of participants without changing the session’s overall objectives, themes and structure. The proposed randomisation procedures may cause contamination across groups as participants may know each other in the same school. However, participants in the control group will also be invited to join the intervention later, and the dyads will be reminded to keep their experiences of the intervention confidential to minimise contamination. Although the process of designing the intervention did not involve the public, the content of the intervention took references from previous local studies on applying arts as a medium in promoting the well-being of individuals with ID and facilitating the communication between mother–child dyads.36 43 44
Ethics statements
Patient consent for publication
Acknowledgments
Publication made possible in part by support from the HKU Libraries Open Access Author Fund sponsored by the HKU Libraries.
References
Supplementary materials
Supplementary Data
This web only file has been produced by the BMJ Publishing Group from an electronic file supplied by the author(s) and has not been edited for content.
Footnotes
TLTL and AHYW contributed equally.
Contributors RTHH and AHYW conceived the study. RTHH, AHYW, PKSW and HHML contributed to the study design, and they are the research grant holders (named investigators). TCTF, CKPC, PKSW and HHML provided methodological expertise and resources. TLTL, AHYW and RTHH prepared the draft of this study protocol. All the authors reviewed and approved the final manuscript.
Funding This work was supported by the General Research Fund, Research Grants Council of Hong Kong (GRF/HKU/1760121). The study funder has no role in the study design; collection, collection, management, analysis, and interpretation of data; writing of the report; and the decision to submit the report for publication.
Competing interests None declared.
Patient and public involvement Patients and/or the public were not involved in the design, or conduct, or reporting, or dissemination plans of this research.
Provenance and peer review Not commissioned; externally peer reviewed.
Supplemental material This content has been supplied by the author(s). It has not been vetted by BMJ Publishing Group Limited (BMJ) and may not have been peer-reviewed. Any opinions or recommendations discussed are solely those of the author(s) and are not endorsed by BMJ. BMJ disclaims all liability and responsibility arising from any reliance placed on the content. Where the content includes any translated material, BMJ does not warrant the accuracy and reliability of the translations (including but not limited to local regulations, clinical guidelines, terminology, drug names and drug dosages), and is not responsible for any error and/or omissions arising from translation and adaptation or otherwise.