The burden of parents of children with ASD

Autism spectrum disorder (ASD) is defined as a group of complex neurodevelopmental disorders, including autism, Asperger syndrome, pervasive developmental disorder not otherwise specified (PDD-NOS), and other relevant symptoms and conditions [1]. According to a most updated systematic review, the prevalence of ASD among children aged 4 and 8 years in the United States was 1.70 and 1.85 percent, respectively, while the prevalence in Europe ranged from 0.38 to 1.5 percent [2]. Another meta-analysis reported a pooled prevalence of ASD of 0.27% (95% CI: 0.19–0.35%) in Mainland China, Hong Kong, and Taiwan [3]. The General Household Survey in Hong Kong conducted in 2019 and 2020 by the Census and Statistic Department reported that the prevalence rate of autistic spectrum disorder (ASD) among the local population below 15 is estimated at around 1.4% [4].

To take care of children with ASD, parents may suffer from different hardships. A most recent meta-analysis reported that around 31% (95% CI: 24–38%) and 33% (95% CI: 20–48%) of parents of children with ASD suffer from depression and anxiety, respectively [5]. In the Chinese context, where special education service organizations are limited [6], parents have to make much more effort to educate, coach, and support their children and prepare the independent living of their children with ASD compared with other parents [7]. Families of children with ASD may experience more parenting stress than families of children with other disabilities [8]. Parental psychological distress may also reduce the effectiveness of early teaching interventions on their children and increase children’s behavior problems [9].

Sources of the psychological distress among parents of children with ASD

Commonly, parents of children with ASD may face objective burdens, such as disturbed family relationships, constraints in social, leisure, and work activities, financial difficulties, and subjective burden, such as loss of hope, dreams, and expectations, and embarrassment in social situations [10]. A recent systematic review of 24 studies conducted with Chinese parents with children of ASD summarized four main categories of potential life events of parental stress, including (i) cultural factors derived from Confucianism, which mainly lead to the public’s supernatural etiological beliefs and public stigma toward ASD [11]; (ii) parents’ own psychopathological symptoms and autistic traits before their child with ASD was born [12]; (iii) the severity of children’ symptoms of problem behaviors [13]; and (iv) caregiver burden, including physical, psychological, financial, social and time burden [14].

While these four major categories of life events were found to be related to the psychological distress of parents, there was also evidence that parents would cultivate and develop resilience from these factors. A qualitative interview study with Chinese parents of children with ASD suggested that parents would improve their moral character to achieve inner peace and mental health based on Confucianism teaching [15]. Chinese culture’s collectiveness also helped parents seek family and social support, and this support significantly alleviated the psychological distress of parents.

The transaction theory of stress and coping

The transaction theory of stress and coping [16] provided the theoretical framework for comprehending why stressful life events would behave differently in different parents of children with ASD under similar circumstances, as well as why parents would develop resilience from stressful life events.

Based on the transactional theory, two kinds of cognitive appraisals are identified: primary and secondary appraisals. Primary appraisal refers to individuals’ judgment of whether an encounter is irrelevant, benign-positive, or stressful. If the individual adopts the stressful appraisal, it can be perceived in four forms, including harm/loss (damage already exists), threat (anticipated harms or losses), challenge (events that hold the potential of mastery or gain), and irrelevant [17]. Secondary appraisal denotes individuals’ judgment concerning what might and can be done. It includes an evaluation of whether a given coping option will accomplish what it is supposed to, that one can apply a particular strategy or set of strategies effectively, and an evaluation of the consequences of using a specific strategy in the context of other internal and/or external demands and constraints [16, 17].

Researchers have found that positive reappraisal comes from positive beliefs, optimism, parents’ cognitive reframing of life events, emotional acceptance and understanding, and adaptability toward having a child with ASD served as coping strategies to deal with psychological distress in Chinese parents of children with ASD [18].

Mindfulness-based intervention (MBI) and cognitive appraisal of daily life events

According to the transaction theory, the shift of individuals’ cognitive appraisal from threat to positive involves the process of metacognition, which is a form of awareness associated with mindfulness [19]. Originated from Buddhism, mindfulness, defined as “the awareness that arises from paying attention, on purpose, in the present moment and non-judgmentally” [20], was adopted in psychological intervention by John Kabat-Zinn and its effectiveness in improving psychological well-being has been approved by different meta-analysis [21, 22].

According to Garland [23] and his Mindful Coping Model [19], mindfulness facilitates participants to transit from stress appraisals into the non-evaluative state of mindfulness, which would interrupt participants’ automatic emotional reactions and broaden participants’ scope of attention to encompass previously ignored contextual information [24]. Subsequently, when participants’ habitual responses get disrupted, they will be able to access a broader set of information from which novel situational appraisals can be generated [24]. In a nutshell, mindfulness in this process enhances cognitive flexibility and leaves participants the space for selecting adaptive reappraisals and encouraging productive re-engagement with stressful life events [24].

On the one hand, mindfulness practice may facilitate positive reappraisal, i.e., after practicing mindfulness, individuals would re-construct stressful events as benign, beneficial, and/or meaningful [25] and further lead to reduced distress and improved mental health outcomes [26]. On the other hand, the self-compassion cultivated from mindfulness practice would alter individuals’ secondary appraisal and function as a coping strategy for stress reduction [27]. After recurrent engagement of the mindful reappraisal process, the participants’ neuroplasticity and certain brain regions may thus be altered for top-down emotion regulation and positive affectivity. Gradually, mindfulness can function as a transformational practice for replacing maladaptive cognitive habits with healthy and adaptive ones [28]. (The framework can be found in S1 Fig).

MBI and psychological well-being

Besides the theoretical functions of mindfulness, scholars also examined the clinical effectiveness of mindfulness practice in reducing parents’ psychological distress. Existing research has provided evidence that mindfulness practice is effective in reducing stress and enhancing psychological well-being in a variety of clinical and non-clinical groups, particularly with individuals under chronic stress conditions, such as chronic disease, burnout, and caring for individuals with chronic conditions [29–31]. Mindfulness-based practice is also found to be associated with neural changes in specific brain regions, with subsequent effects on attention, affective regulation, mood, psychological well-being, and behavior [32, 33].

Adding mindfulness-based components to existing behavioral knowledge resulted in measurable positive changes and reduced parental stress for parents of children with developmental disabilities [34]. There was also specific clinical evidence showing the effectiveness of MBI on parents of children with ASD. The effect size of a mindfulness intervention on the subjective well-being of caregivers of children with ASD was 0.79 in single-group design (p < 0.01) and 0.43 in randomized control trials (p < 0.001) [35].

Ecological momentary intervention and assessment (EMI/A)

There are three main research gaps in understanding the mechanisms of psychological distress in parents of children with ASD and the effectiveness of using MBI for reducing psychological distress. First, due to significant recall bias, traditional observational studies cannot accurately quantify the relationship between life events, cognitive appraisal, and psychological distress. Second, the moderating effects of MBI on modifying parents’ cognitive appraisal and psychological distress have not yet been explored with a robust methodology. Third, the traditional delivery face-to-face mode of mindfulness, was hard to attract a large population of parents of children with ASD as these parents are already occupied by their duties of parenting children and maintaining the function of the whole family. Therefore, a newly developed ecological momentary intervention (EMI) and assessment (EMA) platform to deliver MBI is worth exploring.

Patrick et al. first raised the term “ecological momentary intervention” in 2005 [36], which was later defined as “treatments that are provided to people during their everyday lives (i.e., in real-time) and in natural settings (i.e., real-world)” [37]. Normally, using EMI means delivering treatment on mobile phones and being implemented standalone or as a supplement to existing treatments. Meanwhile, EMA is defined as a method using wearable electronic devices to repeatedly document and collect human thoughts, emotions, behavior, and physiological states in the natural environment context [38]. As a special observational study technique, EMA can minimize recall bias, maximize ecological validity, help understand the dynamic interplay between events of interest, and enable ecological momentary interventions [39].

There are several benefits of using EMI and EMA. First, the application of EMI enables researchers to reach large populations easily, rapidly, and cheaply [40]. Secondly, using EMI can modify the contents of intervention based on individual and contextual factors that vary across time and to fit individual needs and backgrounds. Thirdly, By using EMI, participants can practice the skills and exercises in real-life settings that fit their day-to-day routine, reducing the burden on parents of children with ASD to change their daily schedule [40]. Lastly, using EMA enables researchers to obtain information with maximized ecological validity and to track behaviors and experiences change over different periods and contexts [41].

Existing research has offered clinical evidence supporting the effects of EMI and EMA in promoting healthy behavior and psychological well-being [40, 42]. Yet, the literature search on PubMed found no prior study examined the effects of MBI using EMI for parents of children with ASD. Only one study protocol was proposed to explore mindfulness intervention and EMI to decrease adolescent stress and anxiety [43]. The knowledge in filling the above research gaps is essential for examining the mechanism between life events, cognitive appraisal, psychological distress, and methods for reducing the psychological distress of parents of children with ASD.

This study aims to develop and implement the MBI using EMI and EMA platform (EMI/A-MBI) and to assess the effectiveness of this newly developed EMI/A-MBI on reducing psychological distress in parents of children with ASD. Specifically, we aim:

1. to identify the daily sources of depression, anxiety, and stress in parents of children with ASD and what are the protective factors for resilience;
2. (comparison of the intervention and control group) to evaluate the effectiveness of EMI-MBI in improving the psychological well-being of parents of children with ASD;
3. (within the intervention group) to explore whether the intensity and frequency of mindfulness practice moderate the participants’ cognitive appraisal of daily life events and their psychological distress and mental well-being.

Research hypotheses

The research hypotheses of this proposed study include the following:

1. The intervention group would show a significant reduction in depression, anxiety, and stress than the control group right after intervention and during follow-up.
2. The intervention group would significantly increase life satisfaction and empowerment more than the control group right after intervention and during follow-up.
3. The intensity and frequency of mindfulness practice would moderate the relationship between participants’ cognitive appraisal of daily life events and psychological distress and mental well-being in the intervention group.

Trial design

This research adopts a parallel-armed, randomized controlled trial (RCT) design. The intervention group will receive a combination of (1) a time-based system-triggered EMA, which will collect participants’ sources and status of depression, anxiety, and depression in daily life; (2) EMI-delivered MBI, and (3) a follow-up survey of long-term effects of EMI/A-MBI. The participants will complete a baseline questionnaire and then participate in EMI via a smartphone application (App) for eight consecutive weeks and receive the daily exercise prompts. The EMA will include questions about self-reported depression, anxiety, and depression. After the 8-week EMI/A-MBI, the participants will be invited to complete a post-experimental survey with similar questions in the baseline questionnaire. Two months after completing the EMI/A-MBI, participants will be contacted to complete a telephone follow-up survey with similar questions in the baseline questionnaire. The control group will receive the longitudinal survey exactly the same as the intervention group and 8-week mindfulness-based short messages sent by the research team on a daily basis. The messages will contain audio-based instructions for mindfulness-based practice, which will be the same as the intervention group. The SPIRIT schedule of enrolment, interventions, and assessments can be found in Fig 1 and the flowchart of the design can be found in Fig 2. (The SPIRIT checklist can be found in S1 File).

Download:

• PPT
  PowerPoint slide
• PNG
  larger image
• TIFF
  original image

Fig 1. SPIRIT schedule of enrolment, intervention and assessment.

https://doi.org/10.1371/journal.pone.0291168.g001

Download:

• PPT
  PowerPoint slide
• PNG
  larger image
• TIFF
  original image

Fig 2. Flowchart of participants through the trial.

https://doi.org/10.1371/journal.pone.0291168.g002

Outcomes.

At baseline, post-test, and follow-up survey, the questionnaire contains the same questions concerning demographic information (will only ask at baseline). Psychological distress and mental well-being will be administered to participants in both intervention and control groups. Demographic information, including parent and children’s gender, parent and children’s age, years of children diagnosed with ASD, number of children living in the same household, family monthly income, parents’ education level, parents’ employment status, and average time spent with children daily. The primary outcome will be participants’ psychological distress measured by the depression anxiety stress scale. The secondary outcomes will include participants’ subjective well-being, measured by the satisfaction with life scale, and level of resilience, measured by the psychological empowerment scale. (Detailed measurement information can be found in Fig 3)

Download:

• PPT
  PowerPoint slide
• PNG
  larger image
• TIFF
  original image

Fig 3. Summarizations of measurements.

https://doi.org/10.1371/journal.pone.0291168.g003

The cognitive Appraisal of Health Scale will be used to measure participants’ cognitive appraisal of daily life events. This scale includes 28 items rated from 1 strongly disagree to 5 strongly agree. This scale was developed based on the transactional theory of stress and coping and thus consists of subscales of primary appraisal (threat, challenge, harm/loss, and benign/irrelevant) and secondary appraisal. The internal consistencies were all greater than 0.70 [46].

The Five Facet Mindfulness Questionnaire (FFMQ)-short version [47, 48] will be used to measure participants’ level of mindfulness before and after practicing. It includes 15 items measuring the five facets of mindfulness: observing, describing, acting with awareness, non-judging of inner experience, and non-reactivity to inner experience. This scale was further validated [49] and yielded good convergent validity and internal consistency [49].

Parents’ psychological distress will be assessed in the domains of depression, anxiety, stress, and mental well-being, indicating participants’ life satisfaction and empowerment.

The Depression Anxiety Stress Scale (DASS) [50] is a self-report questionnaire that measures the emotional states of depression, anxiety, and stress. This scale contains 21 questions rated from 0 (did not apply to me at all) to 3 (applied to me very much, or most of the time). There were seven questions for each sub-scale of stress, depression, and anxiety [51]. This scale was reliable (alpha = 0.96 and 0.80 for the depression and anxiety subscales, respectively). Cronbach’s alpha was high for the three subscales in parents of children with ASD: 0.91 for stress, 0.87 for anxiety, 0.94 for depression, and 0.92 for full scale [51]. It has also been validated in the Hong Kong population [52].

Satisfaction with Life Scale (SWLS) [53]. The SWLS has five items scored on a 7-point scale (1 = strongly disagree to 7 = strongly agree) measuring participants’ satisfaction with life and has a Cronbach’s alpha for internal consistency of 0.87 and a test-retest correlation of 0.82 [53]. This scale has also been validated in Chinese [54] and Hong Kong populations [55].

Psychological Empowerment Scale for parents of children with a disability (PES) [56]. It is a 32-item questionnaire on a 5-point scale (1 = strongly disagree to 5 = strongly agree) with four underlying sub-scales: (a) attitudes of control and competence, (b) cognitive appraisals of critical skills and knowledge, (c) formal participation in organizations, and (d) informal participation in social systems and relationships (57). This scale showed high-reliability coefficients (0.90–0.97) [56].

Parents’ life events will be prompted daily in the EMI/A app, and questions will be modified from the systematic review that summarizes the risk and protective factors of psychological distress [57]. The shortened version of the scale measuring mindfulness and psychological distress will be embedded in EMI/A and prompt out every day for future analysis.

Confidentiality

Data management

In consultation with SMN and XCZ, the principal investigator (QW) will be responsible for program coordination and data collection. All digital data, including questionnaires, consent forms, and data from the EMA, will be encrypted and stored on a hard drive following the data management guidelines of Lingnan University. Personal data that can be identified will be stored separately from anonymous data to prevent the identification of personal data throughout the project processing period. The principal investigator will also analyze the data following the guidelines of all phases of data collection, with no interim data analysis. Data access information can be found in data availability statement.

Data analysis

Data analysis will be conducted using STATA 17 [60]. All significance tests were two-sided with a 5% level of significance. Missing data will be calculated using intention-to-treat approach. Participants who failed to answer the prompted questions will be treat as no improvement on the outcomes. The characteristics of the observed data and the missing data will be compared to determine whether there are any systematic differences. If tested that the missing data will be missing not at random, imputation methods, such as multiple imputation (MI) or maximum likelihood estimation (MLE), will be adopted to estimate the missing values based on the observed data and the relationships between variables. Moreover, sensitivity analysis will be adopted to assess the impact of the missing data on the study results and to determine the robustness of the findings. Complete case analysis and multiple imputation will also be conducted as sensitivity analysis. The internal consistency of the constructs for mindfulness, depression, anxiety, stress, and life satisfaction will be assessed with Cronbach’s alpha. In each construct, the PI will sum all items to build the score for that attribute in each participant.

To answer the first and second questions, first of all, the effect size (Heges’ g) of the outcome for both the intervention and control groups will be calculated. Subgroup analysis will be conducted when necessary. Next, linear mixed models will be conducted to compare the effectiveness of the mindfulness intervention between the two treatment groups. Linear mixed models are particularly suitable for analyzing data collected via ecological momentary assessment, as they can account for the nested and repeated nature of the data. The main predictor variable will be the treatment group (intervention vs. control), and the model will also include baseline DASS scores and any relevant demographic variables (e.g., age, gender) as covariates. The linear mixed model will include random intercepts and slopes for each participant to account for individual differences in the baseline scores and the rate of change in scores over time. The treatment group effect will be assessed using a fixed effect, and the model will also include an interaction term between treatment group and time to investigate any differential effects of the intervention over time.

To answer the third question, structural equational modelling will be adopted to examine the pathways of the relationship between daily life events, cognitive appraisal, and psychological distress. Moderation analysis will also be used to explore the effects of mindfulness. The model will be adjusted for the participants’ role of mother or father. Bootstrapping with size 10,000 will be used to estimate the 95% confidence interval of the parameters. After checking data distribution normality, several fit indices will be served as the judgment of the fit of the model. A non-significant chi-square (X²) is desired, but the test is sensitive to erroneously reject a correct model when the sample size is not large enough [61]. The Comparative Fit Index (CFI) and the Non-Normal Fit Index (also known as the Tucker-Lewis Index, TLI), ranging from 0.00 to 1.00, with higher values representing better fit, will be adopted [62]. The standardized root-mean-square residual (SRMR) and root mean square error of approximation (RMSEA) will be used to detect model misspecifications, with values below 0.08 considered desirable [62]. The indirect effect of the model will be checked with the Sobel test.

Oversight and monitoring