While parents who have children with intellectual disability (ID) experience positive outcomes and have positive life and child-related experiences (Hastings
2016; Hastings and Taunt
2002), they are often also at increased risk for stress and mental health problems. This risk has been understood for several decades, and increasingly sophisticated research designs and representative population-based data confirm, for example, that UK mothers of children with ID are approximately twice as likely to have clinically concerning levels of psychological distress on a mental health screening questionnaire than other mothers (Totsika et al.
2011). In addition, compared with other carers from a representative survey of English households, carers (mostly parents) of children and adults with ID reported a higher caring load, more financial struggles, a more negative impact of caring on their personal life (e.g., social or leisure activities, reduced time spent with other family members/friends), and overall poorer health (e.g., disturbed sleep, feeling depressed) (Totsika et al.
2017).
Stress and mental health problems in parents of children and adults with ID are not only concerning directly for the quality of life and well-being of the parents themselves but also for the children. Longitudinal data, allowing causal inferences, confirm that children with ID whose parents (especially mothers) experience significant stress or mental health problems are more likely (than those with less severe difficulties) to experience increased behavioral and emotional problems over time (e.g., Bailey et al.
2019; Hastings et al.
2006; Lecavalier et al.
2006; Neece et al.
2012; Totsika et al.
2013). Such findings can be understood from a family systems perspective, such that the well-being of one family member will affect other individual family members and family sub-systems (such as couple relationships, parent-child relationship quality, and sibling relationship quality) (Cridland et al.
2014; Seligman and Darling
2009; Trivette et al.
2010).
Although carers’ needs for support are recognized by professionals and services, and in policy internationally, there is surprisingly little high-quality research evidence relating to effective interventions for reducing parental stress. A number of parenting programs adapted from mainstream versions have been shown to be potentially effective in terms of their impact on parents’ well-being, with some randomized controlled trials with positive results for the Stepping Stones Triple P program (Tellegen and Sanders
2013) and the Incredible Years parenting program (McIntyre
2013). However, parenting programs are focused mainly on child outcomes and parenting rather than directly targeting parent well-being. Psychological supports directly targeting the well-being of parents of individuals with ID have rarely been tested robustly in randomized trials (Glidden et al.
in press).
Mindfulness-based interventions are a worthwhile focus for improving parents’ well-being for a number of reasons. First, there is gathering evidence that mindfulness-based interventions are effective for improving well-being and reducing stress in a variety of settings and with a variety of populations (e.g., Chiesa and Serretti
2011; Kallapiran et al.
2015; Khoury et al.
2013; Piet et al.
2012). Second, the challenges faced by parent carers of children and adults with ID are often chronic and potentially less amenable to adjustment through approaches such as cognitive behavior therapy (Lunsky et al.
2017). Third, increased avoidant coping (Hastings et al.
2005) and decreased state or trait mindfulness and psychological acceptance (Jones et al.
2014; MacDonald et al.
2010; Weiss et al.
2012) have been shown to be associated with increased stress and mental health problems in parents of children with ID. Therefore, targeting these processes (reducing avoidance, and increasing mindfulness and psychological acceptance) could be appropriate targets for change. Fourth, there have been a number of recent positive evaluations of mindfulness-based interventions for parents of children with ID in which these processes were targeted, including some randomized trials (e.g., Dykens et al.
2014; Jones et al.
2018; Lunsky et al.
2017; Neece
2014).
Despite some encouraging outcome data, a significant issue with existing evaluations of mindfulness-based interventions for family carers of children and adults with ID is that the interventions are delivered either by experienced mindfulness teachers or those delivering the interventions need to be trained and supervised by experienced mindfulness teachers. In addition, family carers find it difficult to commit to attending a regular group session in person due to difficulties finding childcare and transport to the sessions, and child and parent health problems (Lunsky et al.
2017). Thus, there are considerable barriers to the wide-scale implementation of these interventions likely leading to only a small reach when it comes to addressing family carers’ needs. Online delivery of mindfulness-based interventions might offer a solution since the expertise is invested in the development of the intervention, but access is not limited by the lack of availability of mindfulness-trained individuals or the unpredictability of parental care commitments. We could find no data on the feasibility of online mindfulness interventions with parents of children or adults with ID and no studies of parents of children or adults with ID were identified in a recent systematic review of online mindfulness evaluations (Spijkerman et al.
2016).
Published evaluations of an online version of Mindfulness-Based Cognitive Therapy (MBCT), Be Mindful, support its acceptability for participants and also effectiveness in terms of reductions in stress, anxiety, and depression pre-post intervention (Krusche et al.
2013), and improvements in work-related well-being in a randomized controlled trial (Querstret et al.
2016). A recent meta-analysis of 15 randomized trials of online mindfulness interventions also supported their effectiveness especially for reductions in stress (summary effect size .51) (Spijkerman et al.
2016). A sub-group analysis suggested that online mindfulness interventions were more effective when the interventions were guided (i.e., where some support and contact were offered alongside the online course) (Spijkerman et al.
2016). Previous research using modified versions of MBCT (Jones et al.
2018; Lunsky et al.
2017) also suggested that parents value the chance to relate mindfulness intervention with their experiences caring for their offspring with ID. Existing research (Dykens et al.
2014; Lunsky et al.
2018) suggests that involving parents in intervention delivery with other parents adds value to the experience and outcome for participants.
The present study contributes to practice knowledge by using an established program with a new population, caregivers of people with ID. The overall aim of the current feasibility study was to examine whether Be Mindful can be delivered successfully to family carers of children or adults with ID, and whether it would be feasible to conduct a later definitive randomized controlled trial of the effectiveness and cost-effectiveness of Be Mindful. Based on the literature supporting the addition of guided peer support, we developed a peer mentoring “add-on”–guided support element for Be Mindful through a co-production process with family carers. Peer mentors were not intended to deliver a mindfulness-based intervention, but instead to provide peer support to participants who were engaging with the online intervention. The feasibility questions were addressed in the context of a two-arm trial comparing Be Mindful to Be Mindful with mentoring support. The feasibility questions were as follows: (1) What are the most effective pathways to recruit family carers of children or adults with ID, and what recruitment rate for family carers (including the proportion of mothers and fathers) can be achieved? (2) Can peer mentors be recruited and trained to deliver telephone guided support sessions for the study? (3) Are family carers willing to be randomized within the context of a randomized trial? What design would be acceptable to family carers for a future randomized trial? (4) Can peer mentors deliver telephone guided support sessions with a high degree of fidelity? (5) What proportion of family carers complete the Be Mindful online intervention in each arm of the trial, and what is the typical time taken to complete the intervention? What proportion of the telephone guided support sessions do family carers receive? (6) What proportion of family carers are retained in the research study to the 6-month post-randomization follow-up? (7) What stress/well-being interventions do family carers receive typically, and how is this “usual practice” different from the program content of Be Mindful? (8) Do family carers complete the outcome measures for the study? And, (9) What is the feasibility of collecting resource use and health-related quality of life data for family carers?
Methods
Participants
Participants were a self-selected sample who responded to recruitment advertisements, all of whom underwent a short screening telephone call to determine their eligibility for the project. Family carers were eligible for the study if they were 18 years old or over, a family carer of a person (child or adult) with ID who lived with them, not currently receiving individual or group therapy (including mindfulness-based interventions) for their mental health, able to access the online mindfulness intervention, and had not already completed Be Mindful. Previous engagement in mindfulness-based interventions was not a reason for exclusion. ID was defined administratively by family carers reporting that the child or adult had received a diagnosis and/or was in receipt of ID services. Foster carers were eligible for the study, provided the placement was not due to end during the study period. All participants completed the Vineland Adaptive Behavior Scales (VABS) (3rd edition) Domain Level Interview Form (Sparrow et al.
2016) during this telephone call to assess the level of adaptive functioning of the person with ID to enable description of the participating sample.
Participants were recruited during a 2 and a half month period (October–December 2018) and were randomized throughout the study period by an independent statistician at the University of Warwick on a 1:1 basis using minimization, balancing the age of the person with ID (under 18 years old; 18 years old and over) between the two arms of the trial (
Be Mindful vs Be Mindful plus guided telephone support [
Be Mindful+]). A power calculation was not undertaken for this study as it was a feasibility study to inform a later trial (Arain et al.
2010). The researcher responsible for data collection remained blind to the allocations until after the final data collection point. Participants could not be blinded to their allocation, but were recruited prior to randomization.
Participants (n = 60) were predominantly female (n = 55), White British (n = 48) family carers of people with ID with a mean age of 46.09 (SD = 7.71; range = 33–62). Participants were mostly educated to university degree level (n = 32), and most were either employed (n = 21) or looked after their home and family (n = 22). The majority of participants had never previously engaged in any mindfulness-based interventions (n = 46).
The people for whom the participants cared were mostly under 18 years of age (
n = 47), with a mean age of 13.73 (SD = 8.97; range = 1–55 years), and there was a fairly even split of males (
n = 33) and females (
n = 27). The mean Vineland Adaptive Behavior Scale (VABS) Domain Level ABC score was 42 (SD = 13.47; range = 20–69), and they had a range of diagnoses, including dual diagnoses, with the majority having diagnoses of ID (
n = 54) and/or autism (
n = 41). Separate demographic data for both arms of the trial are presented in Table
1.
Table 1
Participant demographics split by trial arm
Gender | | | | |
Male | 3 | 10 | 2 | 6.7 |
Female | 27 | 90 | 28 | 93.3 |
Mean age (SD) | 46.65 (7.3) | | 45.57 (8.17) | |
Relationship to child | | | | |
Biological mother | 25 | 83.3 | 27 | 90 |
Biological father | 3 | 10 | 2 | 6.7 |
Adoptive mother | 2 | 6.7 | 0 | 0 |
Other | 0 | 0 | 1 | 3.3 |
Ethnicity | | | | |
White British | 13 | 76.7 | 25 | 83.3 |
Asian/Asian British | 3 | 10 | 3 | 10 |
White Other | 2 | 6.7 | 2 | 6.7 |
Black British | 1 | 3.3 | 0 | 0 |
Mixed race | 1 | 3.3 | 0 | 0 |
Educational level | | | | |
Some GCSE passes | 2 | 6.7 | 4 | 13.3 |
5+ GCSEs at A* - C | 4 | 13.3 | 2 | 6.7 |
5 A/AS levels | 1 | 3.3 | 1 | 3.3 |
Other higher education below degree level | 6 | 20 | 8 | 26.7 |
Degree (bachelors) or higher | 17 | 56.7 | 15 | 50 |
Employment | | | | |
Currently working | 10 | 33.3 | 11 | 36.7 |
On maternity/paternity/parental leave | 0 | 0 | 1 | 3.3 |
Self-employed | 3 | 10 | 4 | 13.3 |
Full-time student | 0 | | 1 | 3.3 |
Voluntary work | 1 | 3.3 | 1 | 3.3 |
Look after home and family | 11 | 36.7 | 11 | 36.7 |
Other | 5 | 16.7 | 1 | 3.3 |
Child gender | | | | |
Male | 26 | 43.3 | 40 | 66.7 |
Female | 34 | 56.7 | 20 | 33.3 |
Child mean age (SD) | 13.63 (8.39) | | 13.83 (9.67) | |
Procedures
This feasibility randomized controlled trial (RCT) was retrospectively registered on 21st September 2018 (ISRCTN20615805) before recruitment and randomization commenced. A two-arm RCT design was used to examine the feasibility of delivering an online mindfulness intervention (Be Mindful), with or without additional guided telephone support, to family carers of children or adults with ID, and the feasibility of a definitive RCT examining effectiveness and cost-effectiveness. Ethical approval was granted by the University of Warwick’s Humanities & Social Sciences Research Ethics Committee (58/17-18).
Family carers who expressed interest in participating in the study after reading an information sheet underwent a short screening telephone call to determine their eligibility. Eligible participants were offered a choice of questionnaire completion method (postal, online, or telephone). Informed consent was taken by a researcher (for postal or telephone completions) or online, before participants completed the baseline questionnaire. Participants were then allocated randomly to either Be Mindful or Be Mindful+ and were informed of their allocation by an email invitation to start Be Mindful (Be Mindful arm) or a text message/telephone call from a Peer Mentor (Be Mindful+ arm). Participants in the Be Mindful+ arm received an email invitation to start Be Mindful after their first telephone mentoring session. Once enrolled on Be Mindful, participants used it at their own pace, with Be Mindful+ participants being offered a further two telephone mentoring sessions (as described below).
All participants received follow-up questionnaires at 12 weeks and 6 months post-randomization. Participants received a £10 high-street voucher for each time point they returned the questionnaires.
Results
Feasibility Outcomes
Feasibility of Outcome Measures
Participants were offered a choice of three methods of questionnaire completion: online, post, or telephone. Forty-seven participants elected to complete questionnaires online, 10 in the post, and three via telephone (one changed from telephone to online after the baseline questionnaire). Interviewed participants were happy with their completion method, with online completers stating that it was convenient, did not take very long, and it seemed easier than the other methods. Postal completers were generally reluctant to use technology to complete questionnaires, and were grateful for a postal option. No telephone completers were interviewed. Personal questions were difficult to think about for four interviewed participants, but this was not prohibitive of them completing questionnaires, nor was it detrimental to their overall sense of well-being.
At baseline, participants (n = 60) generally completed all outcome measures. However, there was a small amount of missing data for questions about finances (weekly income, n = 6; raising £2000 in an emergency, n = 2), participant-partner agreement (n = 1), partner relationship satisfaction (n = 1), and the number of people living in the household (n = 1). Some participants did not complete every question on the Parenting Efficacy Scale, so this could not be summed and used in the analyses for them (n = 3). One interviewed participant found the questions about finances to be intrusive, and so elected not to answer them; this may be reflective of other participants’ feelings about these questions.
Forty-seven participants completed outcome measures 12 weeks post-randomization, with a small amount of missing data for questions about participant-partner agreement (n = 1) and partner relationship satisfaction (n = 2). There were some missing data on the HADS (n = 2), Family APGAR (n = 4), Child relationship scale (n = 3), and the Parenting Efficacy Scale (n = 5), meaning that these could not be summed and analyzed for these participants.
Fifty participants completed outcome measures 6 months post-randomization, and three of these were minimum datasets (including only Positive Gains Scale, WEMWBS, and Family APGAR). Missing data on the HADS (n = 4), Family APGAR (n = 1), Child relationship scale (n = 4), and the Parenting Efficacy Scale (n = 5) meant that these could not be summed and analyzed for those participants.
Exploratory Analysis of Participant Outcomes
Discussion
The feasibility outcomes indicate that it would be possible to recruit and retain family carers to a definitive RCT of this intervention, and that the study design and methods (e.g., randomization, outcome measures) are acceptable to participants. Be Mindful and the additional Peer Mentor telephone calls were well received (in terms of intervention and telephone call adherence, and qualitative process evaluation data) by family carers of people with ID. This finding highlights that the additional element was not burdensome for participants; indeed, it was an additional motivation for participants to complete Be Mindful. The Peer Mentor telephone support sessions can be delivered with high fidelity to the manual, but this is inconsistent and requires further development and piloting work (e.g., increased training for Peer Mentors). Two-thirds of family carers in this study were not receiving any therapeutic intervention for their well-being at baseline and, as such, there would be little overlap with existing services and this intervention. From the preliminary comparison data, there are small, but non-significant, improvements for Be Mindful+ relative to Be Mindful over time.
The study design appears to be acceptable to participants, who were happy to be randomized and to complete questionnaires at all three time points with minimal missing data. It was feasible to collect data about resource use and health-related quality of life, although it was never the intention to analyze these data within this feasibility study. As it has been established that it is possible to collect these data, full health economic analyses can be undertaken in a future definitive trial.
Data pertaining to usual practice for well-being interventions indicates that two-thirds of participants are not receiving any support. Interviewed participants recognized that it is important for family carers of people with ID to look after their own mental health and well-being, but this can be difficult for many family carers to achieve due to competing commitments and being unable to attend regular in-person interventions (Lunsky et al.
2017). The provision of a straightforward, online intervention offers a potential solution to this, as it can be completed alongside other commitments in a flexible way. Interviewed participants indicated that Be Mindful was successful in improving their perceptions of their experiences, and in reducing stressful encounters with the person with ID for whom they cared. These findings provide merit to continuing research to establish an evidence-base for the use of online mindfulness interventions for family carers of people with ID.
When both groups were combined, there were significant improvements in participant-reported well-being, psychological distress, participant-child conflict, participant-partner relationship, and participant-partner agreement; however, these changed less over time. Preliminary analysis of the intervention effectiveness between groups should be interpreted with caution due to the small sample. The current study was not powered to detect differences in outcomes between the two arms of the trial and this was not the purpose of this feasibility study. Preliminary results indicate that there is a small, but non-significant, benefit to being in the Be Mindful+ arm across the outcomes. Mindfulness measures were not included in this feasibility study, and in a larger definitive trial, it will be important to include appropriate measures as it will then be possible to undertake exploratory analyses of intervention effects mediated by changes in key process measures.
Furthermore, there was no difference in the adherence to Be Mindful between the two groups, indicating that the additional element of telephone support was not burdensome. Telephone support, despite the inconsistencies in fidelity, was reported to be beneficial to participants’ motivation to continue with Be Mindful, and provided a space for reflection about their completion of the course. This finding is reflected in the wider literature about guided support for online interventions (Spijkerman et al.
2016). This additional element, therefore, provides the opportunity to tailor support to family carers in a way that appears to offer a small benefit over not receiving additional support and remains easy to access for family carers of people with ID. The addition of telephone support is a time- and cost-effective strategy of tailoring an existing intervention for family carers of people with ID, and should be extended and examined further in future research.
Limitations and Future Research
While recruitment was completed with minimal effort (online through a small number of sources) in a short space of time, most participants were mothers which does not provide data on pathways to recruit other family carers of people with ID. Recruitment efforts in a future trial should, therefore, be such to engage a wider cross-section of family carers (e.g., fathers, grandparents, adult siblings). Furthermore, study retention at 6 months post-randomization was high, suggesting that a larger trial is feasible in this respect. The fidelity to the Mentoring Manual was inconsistent, and perhaps an artifact of limited time available to train Peer Mentors in this study. Future research should seek to rectify this by providing ample training time for Peer Mentors and ensuring that all Peer Mentors understand the importance of fidelity.
It was possible to recruit Peer Mentors, but only 50% of the recruited mentors were trained and able to commit to the project before it began. We would likely need to over-recruit Peer Mentors to this role in a future study to ensure that there was an appropriate number of mentors to deliver telephone support. Sufficient time to complete training activities is also required for a future trial, and the time taken to do this should not be underestimated. The fidelity to the Mentoring Manual was inconsistent, and this would need reviewing before a future trial in conjunction with a revision of the Peer Mentor training activities. While there is increasing recognition of the importance of structured peer support from people share circumstances (e.g., they are a family carer of a person with ID) (Dykens et al.
2014; Lunsky et al.
2018), it is important to understand their training and support needs (Lunsky et al.
2018); this will be important to maintaining intervention fidelity. The consistently missing elements of the mentoring calls were directly related to the GROW model (Whitmore
1996) and, upon reflection, the importance of including these elements was perhaps not clear to the Peer Mentors; this would be rectified in future training activities. It is also clear, from these data in conjunction with the self-assessment data, that recordings are needed in a future definitive RCT to establish the fidelity to the manual, as there are inconsistencies between the scores using the two methods for fidelity (Mentor and Researcher ratings).
These results indicate that it is feasible to deliver Be Mindful with additional Peer Mentor telephone support to family carers of people with ID, and to research the effectiveness of this intervention using the design and methods in this feasibility trial. Further research should now be undertaken to answer some of the outstanding questions (e.g., recruitment of a wider cross-section of family caregivers) and to evaluate the effectiveness and cost-effectiveness of offering Be Mindful plus additional telephone support to family carers of people with ID in improving their well-being.
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