Abstract
This pilot study aimed to explore the suitability of a preventative intervention for internalising problems in young children with chronic physical health conditions. The pilot study focused on a subsample of 27 children with chronic physical health conditions within a population-level randomised controlled trial of the Cool Little Kids parenting programme. The Cool Little Kids parenting programme aims to prevent the development of internalising problems in inhibited (shy/anxious) preschool children by educating parents to reduce young children’s avoidant coping styles and manage their anxiety/distress. The wider trial recruited 545 temperamentally inhibited preschool children. Measures included child health/development concerns and internalising symptoms at baseline, feedback on Cool Little Kids post-intervention and child internalising problems at one- and two-year longitudinal follow-up. At baseline, inhibited children with and without chronic physical health conditions had levels of internalising symptoms above the normative mean. At post-intervention, parents of children with chronic physical health conditions gave feedback that Cool Little Kids was helpful for managing their child’s emotional distress. At follow-up, significantly fewer intervention than control children with chronic physical health conditions had specific phobia after 1 year (25% vs 70%) and specific fear symptoms after 2 years (mean 9.57 vs 16.89). As the pilot findings are promising, a further trial of Cool Little Kids in a physical health treatment service with a larger sample of children with chronic physical illness diagnoses would be worthwhile.
Keywords
Childhood chronic physical health conditions have increased over recent decades as previously life-threatening conditions are successfully managed through medical advances (Compas et al., 2012). Children’s chronic physical health conditions are defined as having a prolonged duration (>3 months), impacting normal activity, requiring reliance on either medication, special diet, medical technology, assistive device or personal assistance to compensate and necessitating greater need for health-related services than is usually expected for children of a similar age (Van der Lee et al., 2007). The more common chronic physical health conditions in childhood are asthma, allergies, diabetes, epilepsy, inflammatory bowel disease, cancer, cystic fibrosis and cerebral palsy. Chronic health conditions in childhood also include visual and hearing impairments (Australian Institute of Health and Welfare, 2014).
While medicine has reduced child fatality rates, children with chronic physical health conditions are at increased risk for mental health problems and three times more likely to have internalising (anxious/depressive) problems than their physically healthy peers (Blackman et al., 2011). These children can be exposed to threatening symptoms, situations and medical procedures, face feelings of uncertainty and lack of control over their condition and treatment and experience restrictions on their educational and social functioning and social stigma associated with their condition (Pinquart and Shen, 2011).
Research with children in the general population shows that internalising problems can continue over time, from preschool age into mid-childhood and then into adolescence and adulthood (Bufferd et al., 2012; Carballo et al., 2010; Luby et al., 2014). Internalising problems are linked to impaired functioning in interpersonal, financial and health domains and increased mortality via suicide (Copeland et al., 2014; Fombonne et al., 2001; Jokela et al., 2009). Early detection and intervention are recognised to be important (Gleason et al., 2016).
Internalising problems of children with chronic physical health conditions may also have ongoing consequences (de Araujo Filho and Yacubian, 2013; Meuret et al., 2017). As described by Cottrell et al. (2015), conditions can lead to a cycle of worsening morbidity, whereby poor physical health adversely affects the child’s mental health, and in turn internalising problems exacerbate the physical condition symptoms. Ultimately, these symptoms impact children’s longer term behavioural, emotional, social and educational functioning and quality of life (Bennett et al., 2015; Blackman et al., 2011). In physical health treatment settings, children with comorbid physical and mental health problems tend to be identified as ‘complex cases’, and treatment of children’s physical conditions can take precedence over any psychological concerns, leading to mental health treatment being neglected (Katon et al., 2006).
A small body of research has developed to explore the efficacy of psychological interventions for school-aged children with chronic physical health conditions and comorbid internalising problems. Cognitive behavioural therapy (CBT) is adapted to address children’s illness-specific concerns including pain/symptom management, medication adherence, health misappraisals, coping with uncertainty related to a physical health condition and implications of living with a chronic physical illness (Blocher et al., 2013; Hains et al., 2001; McGrady and Hood, 2013; Reigada et al., 2013; Rosselló and Jiménez-Chafey, 2006; Scholten et al., 2013; Szigethy et al., 2014). These studies highlight some promising reductions in comorbid emotional distress. Recent systematic reviews have highlighted that CBT programmes are well accepted, with the added benefit of providing a space for sharing insights into the experience of dealing with a chronic physical health condition (Moore et al., 2019; Shaw et al., 2019). Further to this, another systematic review found that parents may also benefit from intervention by increased self-efficacy and positive parenting behaviours (Mitchell et al., 2019). However, the authors highlighted that most samples are small, most studies are uncontrolled, and they tend to focus on only one type of chronic physical health condition (e.g. epilepsy, asthma or diabetes). Efficacy of CBT for children with various chronic physical health conditions and comorbid internalising problems therefore remains uncertain. Additionally, as many studies used a CBT programme modified for chronic physical illness, it is unknown whether current existing CBT-based programmes may be effective for children with chronic physical health conditions without any modifications.
Effective intervention for internalising problems of children in the general population is more extensively researched (Cox et al., 2012; James et al., 2013). Intervening early with preschool-aged children who have chronic physical health conditions may hold potential to circumvent risk trajectories to anxiety and depression. The Cool Little Kids parenting programme is a CBT-based programme that aims to prevent the development of internalising problems in inhibited (shy/anxious) preschool children by educating parents to reduce young children’s avoidant coping styles and manage their anxiety/distress (Rapee et al., 2005). The wider randomised trial found intervention children had fewer anxiety disorders than controls at 1-year follow-up (50% vs 64%) (Rapee et al., 2005), effects were sustained at 3-year follow-up (Rapee et al., 2010) and adolescent depression was reduced for girls at 11-year follow-up (0% vs 16%) (Rapee, 2013). However, it is unknown whether Cool Little Kids (without modifications) could be effective for young children with chronic physical health conditions. This initial pilot study focused on preschool children with various chronic physical health conditions who were participating in a wider population-level randomised trial of the Cool Little Kids programme (Bayer et al., 2018).
Aim
The aim of this study was to evaluate (i) acceptability of the intervention for parents of children with chronic physical health conditions and (ii) randomised trial outcomes for a subsample of children with chronic physical health conditions in the intervention versus control arm at one- and two-year follow-up.
Method
Study design and participants
In the wider population trial of Cool Little Kids (Bayer et al., 2018), parents of children enrolled in their final preschool year across eight socioeconomically diverse districts of Melbourne, Australia, were sent a brief inhibition screening questionnaire. Inclusion criteria for participation in the trial were parents who scored their children above the ≥85th percentile on the child inhibition screening questionnaire. Parents were excluded from participation if their English was insufficient to complete questionnaires or if the child had a severe developmental/health disability rendering them unlikely to benefit from the trial (Bayer et al., 2018). For the current pilot study, this comprised a subsample analysed from the wider trial, the chronic physical illness group (n = 27), who had children with a chronic health condition (defined as a diagnosis of an ongoing health condition requiring long-term treatment, reported by parents at baseline). Parents randomly allocated to the intervention arm were invited to attend six, 90-min manualised Cool Little Kids parenting group sessions delivered after-hours (7–9 pm) at preschools in the local community. As described previously (Bayer et al., 2011), the parenting sessions involved CBT-based training on principles of anxiety management, instruction on setting up exposure-based tasks for their child to face their distress and methods for parents to reduce their own overprotective behaviours and distress (see online Supplemental Material Appendix A for an outline of programme sessions). In line with the aims of this study, no modifications were made to the programme for parents of the children with chronic physical health conditions. The control arm was usual care access to community services. Parents completed measures at baseline, post-intervention and at one- and two-year follow-up. Ethics committee approval was obtained from the Royal Children's Hospital Melbourne (30105A) and La Trobe University (HEDC13-022).
Measures
All measures were completed by the parent or primary caregiver of the child. 1
Inhibition
The Approach/Inhibition subscale of the Short Temperament Scale for Children (7 items) has good test–retest reliability (r =.90) and corresponds with observed child behaviour (Pedlow et al., 1993). Scores >30 (85 percentile) indicate an inhibited temperament (Rapee et al., 2005).
Chronic physical health condition
The Parents’ Evaluation of Developmental Status enquired if preschool children had been diagnosed with any major medical or developmental problems (10 items with comment) (Glascoe, 1997). This was used to determine the chronic physical health condition sample, based on the standard criteria in the literature reported above (Australian Institute of Health and Welfare, 2014). Since the present pilot study was a secondary analysis of the population trial data, separate independent corroboration by a medical professional was not possible.
Acceptability measures
Parent feedback was sought regarding the intervention via questionnaire items targeting perceived ‘usefulness’ (5-point Likert scale from ‘not at all useful’ to ‘extremely useful’) across five domains of understanding preschoolers’ shyness/inhibition/anxiety, precursors of preschoolers’ anxiety, encouraging brave behaviour, reducing anxious behaviour and changing parents’ own anxious/fearful thoughts. Open-ended probes also solicited ‘best’ and ‘worst’ aspects of the intervention and potential improvements, and parents were asked whether they would recommend the programme to others (yes/no).
Outcome measures
Parents completed the Strength and Difficulties Questionnaire (SDQ-Australian adaptation) (Goodman, 2001) at 1-year follow-up (and baseline), which has good internal consistency, 12-month test–retest reliability and convergent validity with teacher ratings (Hawes and Dadds, 2004). Emotional symptoms (internalising) was the subscale of interest (5-items). The Children’s Moods, Fears and Worries Questionnaire (CMFWQ, 34 items) was completed at both one- and two-year follow-up, as a detailed internalising measure with excellent internal consistency and sensitivity/specificity to detect clinical-level problems (Andrijic et al., 2013; Bayer et al., 2006). At 2-year follow-up, parents also completed the Preschool Anxiety Scale-Revised (PAS-R, 28 items) that has subscales for social, separation and generalised anxiety, specific fears and obsessive–compulsive symptoms. PAS-R has good internal consistency and construct validity (Edwards et al., 2010). The Child Anxiety Life Interference Scale-Preschool Version (CALIS-P, 20-items) was also completed at one- and two-year follow-up. CALIS-P measures the effects of anxiety symptoms on child and family life and has strong internal consistency (Kennedy et al., 2009).
Parents were also administered the Anxiety Disorders Interview Schedule for DSM-IV, Parent Interview Schedule (ADIS) (Silverman et al., 2001) at one- and two-year follow-up, via telephone by blinded interviewers. This included DSM-IV anxiety disorders most relevant to young children (separation anxiety disorder, social phobia, specific phobia and generalised anxiety disorder) (Cartwright-Hatton et al., 2006). The ADIS has been subjected to numerous reliability and validity studies, and findings indicate it has good-to-excellent test–retest reliability and fair-to-excellent interrater reliability (K = .45–.82), and telephone administration demonstrates validity with face-to-face interviews (Lyneham and Rapee, 2005).
Statistical analyses
Demographic characteristics for families of inhibited preschool children with and without chronic physical health conditions at recruitment.
All percent are valid percent.
Parent-reported child socioemotional functioning at recruitment and parent attendance at Cool Little Kids intervention.
Note: M = mean, SD = standard deviation, ES = effect size: Cohen’s r for U (small 0.1, medium 0.3, large 0.5).
Parents’ quantitative feedback on the Cool Little Kids intervention.
Note: M = mean, SD = standard deviation, ES = effect size: Cohen’s r for U (small 0.1, medium 0.3, large 0.5).
11=‘not at all useful’, 2=‘a little useful’, 3=‘quite useful’, 4=‘very useful’, 5=‘extremely useful’.
Intervention outcomes for children with chronic physical health conditions.
Note: All percent are valid percent. ADIS: Anxiety Disorder Interview Schedule; CALIS: Child Anxiety Life Interference Scale; CMFWQ: Children’s Moods Fears and Worries Questionnaire; PAS-R: Preschool Anxiety Scale-Revised; SDQ: Strengths and Difficulties Questionnaire; ES: effect size.
1phi (small 0.10, medium 0.30, large 0.50).
2Cohen’s d (small 0.2, medium 0.5, large 0.8).
Results
Of children with an inhibited temperament (11%), parents of 545 consented to participate (78% uptake) in the Cool Little Kids trial. Among 545 parents enrolled in the trial, 27 (5%) of their children had a chronic physical health condition (intervention n = 17; control n = 10) at baseline. Figure 1 presents the trial flow chart highlighting children with chronic physical health conditions. Trial retention was high, including the subgroup with chronic physical health conditions (n = 25 or 96% at 1-year follow-up and n = 24 or 89% at 2-year follow-up). The rate of chronic physical health conditions amongst the population sample of inhibited preschool children was 1:20 (n = 27, 5.0%). The most frequent condition was vision impairment (n = 12, 4.4%, e.g. microphthalmia, strabismus and stigmatism) with severity ranging from impairments that required corrective lens to those that needed multiple surgeries. Common conditions also included allergies (n = 5), asthma (n = 4) and hearing impairment (n = 4). The less-frequent conditions (n = 1 each) were kidney failure, cerebral palsy, epilepsy, glucose/galactose malabsorption (peg feed) and coeliac disease. Demographic characteristics of the sample are presented in Table 1 for families of children with and without chronic physical health conditions. While the trial sample was broadly population representative (Bayer et al., 2018), parents of children with chronic physical health conditions tended to have less-partnered relationships and household income. Participant flow chart for the trial highlighting the sample with chronic physical health conditions within the population trial (RC: remaining cohort).
At recruitment, there was a trend towards inhibited preschool children with chronic physical health conditions having higher internalising symptoms by parent report (p = 0.08; Table 2), although both groups were elevated relative to SDQ norms (borderline/abnormal range 74% with chronic physical health condition and 57% of physically healthy; no effect Cohen’s r = −0.08). The parents of children with chronic physical health conditions and those with physically healthy children attended the Cool Little Kids parenting group sessions at reasonably similar rates (means 3.29 vs 3.08, Cohen’s r = −0.06; Table 2). Most parents of children with chronic physical health conditions rated the Cool Little Kids programme as ‘quite’ to ‘very’ useful for each of the five domains (all means ≥3; Table 3). Most parents would recommend this programme to other families (93% chronic physical health condition; 95% physically healthy; phi = −0.02). Qualitative feedback from parents of children with chronic physical health conditions is presented in Appendix B (in Supplemental Materials). Parents most frequently reported that the ‘best thing’ about Cool Little Kids was the opportunity to interact with other families with inhibited children to share ideas and support. Parents also described the benefit of learning strategies to manage their young child’s anxious distress. More than a third of parents whose children had chronic physical health conditions indicated that no improvements to Cool Little Kids were necessary. However, a third of parents with these children would appreciate availability of day-time in addition to evening group sessions to increase accessibility.
For children with chronic physical health conditions in the intervention versus control arm of the wider randomised trial, internalising outcomes were then compared at long-term follow-up (Table 4). After 1 year, children with chronic physical health conditions in the intervention arm had significantly fewer specific phobia diagnoses than those in the control arm (n = 4, 25% vs n = 7, 70%, medium-to-large effect size phi =0.44). The intervention children (compared to their controls) also had trends with medium-to-large effect sizes towards less internalising problems, anxiety life interference and overall anxiety disorder. After 2 years, the children with chronic physical health conditions in the intervention arm (compared to their controls) had significantly fewer specific phobia diagnoses (n = 3, 20% vs n = 6, 75%, large effect size phi = 0.54) and specific fear symptoms (means = 9.57 vs 16.89, large effect size d = 0.98). Intervention children (compared to their controls) also had medium-to-large effect trends towards less separation anxiety symptoms and overall anxiety symptoms and disorders.
Discussion
The rate of chronic physical health conditions within the population sample of inhibited preschool children was one in twenty. Inhibited young children with and without chronic physical health conditions were well above population norms for internalising distress (inhibition can be an indicator of early internalising symptoms) (Andrijic et al., 2013). The pilot findings suggest that preschool children with chronic physical health conditions may be responsive to the Cool Little Kids parenting group programme, even with their additional medical conditions. Notably, at follow-up, children with chronic physical health conditions allocated to the intervention arm had significantly fewer specific phobia diagnoses and symptoms than children with chronic physical health conditions allocated to the control arm. There were also trends of substantive effect size for improved emotional functioning across other outcome measures (any anxiety disorder, overall anxiety/internalising symptoms and child/family life interference).
In this pilot, preventative intervention shows stronger results for the subsample with chronic physical health conditions than the wider trial for inhibited children in general. The wider trial’s 1-year follow-up rates of anxiety disorder were 44% intervention versus 50% control (Bayer et al., 2018), compared to 50% intervention versus 80% control for children with chronic physical health conditions. The wider trial’s 2-year follow-up anxiety disorder rates were 38% intervention versus 43% control (Bayer et al., 2020), compared to 40% intervention versus 75% control for children with health conditions. Specific phobia in particular showed positive intervention versus control effects at follow-up for children with chronic physical health conditions (1-year 25% vs 70%; 2-year 20% vs 75%), compared to the wider trial (1-year 20% vs 24%; 2-year 16% vs 20%).
Parents’ feedback showed that Cool Little Kids was acceptable for parents of young children with chronic physical health conditions. Their parents endorsed this parenting group programme as highly useful for understanding and managing their young child’s internalising distress. Parents of children with chronic physical health conditions appreciated the opportunity to interact with other parents to access support and share ideas. These findings are consistent with recent systematic reviews on interventions to improve the mental health of children with long-term physical conditions (Moore et al., 2019; Shaw et al., 2019). The parent’s main suggestion for programme improvement was to offer day as well as evening time options. It may be harder to find childcare and leave home in the evening with the additional demands of caring for young children with physical health conditions.
Strengths of this pilot study were the population sample of preschool children across diverse localities, randomised trial protocol, psychometrically sound child mental health measures, assessor-blind outcome (child anxiety disorders) and longitudinal follow-up over 2 years. The sample size was also comparable to (or larger than) most prior studies exploring psychological interventions for older children with chronic physical health conditions (Blocher et al., 2013; McGrady and Hood, 2013; Reigada et al., 2013). Nevertheless, as a pilot, this study was underpowered to establish statistical reliability for the substantive effect sizes detected between the intervention and control children at follow-up. The pilot findings need confirmation in research with larger samples of preschool children with chronic physical health conditions, along with independent corroboration of illness diagnosis by a medical professional.
As the pilot sample included several young children with low-severity physical health conditions, further research that is more focused on severe illnesses is required. To capture a variety of chronic physical illnesses including severe conditions (i.e. cancer and diabetes), recruitment could be conducted through physical health treatment settings such as tertiary hospital departments and community health centres. The parenting groups could be delivered at the health services where families are already attending (as suggested by Morawska et al., 2015), with day/evening times and on-site childcare to facilitate accessibility. If a programme was offered specifically for children with chronic physical health conditions, parents would also have this challenging experience in common to share. In research for older children with chronic physical illnesses, CBT modifications have been developed to address their illness-specific concerns (i.e. techniques to differentiate anxiety and physical health symptoms to reduce misappraisals when implementing exposure hierarchies) (Blocher et al., 2013; Hains et al., 2001; McGrady and Hood, 2013; Reigada et al., 2013; Rosselló and Jiménez-Chafey, 2006; Scholten et al., 2013; Szigethy et al., 2014).
While it appears that standard CBT exposure techniques are useful for young children with chronic physical health conditions, some modification for chronic physical illnesses could similarly be developed for Cool Little Kids. Interestingly, our pilot data suggest the programme may be effective without modifications. If the programme is effective without modification, then this has the advantage of being more cost effective as there is no need to develop and train clinicians in an alternative version. Still, some small improvements could be made without changing the intervention itself, such as offering the programme exclusively to a group of parents with children who have chronic physical health conditions (to draw on shared experiences) and providing more flexibility around times of programme sessions as was suggested from parent feedback in our study and others (Bennett et al., 2015).
Implications for practice
The current study highlights the need for CBT-based parenting intervention programmes to prevent internalising problems in young children with chronic physical illness, particularly those who are temperamentally inhibited. Clinicians working with children who have a chronic physical illness should be cognisant that internalising symptoms can be identified in preschool-aged children. Referring these at-risk children to targeted CBT-based preventative intervention programmes may reduce the likelihood of internalising problems in later childhood.
In conclusion, this pilot study offers initial support for the feasibility of Cool Little Kids for young children with chronic physical health conditions. This early intervention was acceptable to parents of such preschool children and may be efficacious to reduce their comorbid distress. The next step is for research to include more severe chronic physical illnesses and consider potential modifications to incorporate physical illness–specific concerns. Such research with young children is vital, given that comorbid physical and mental health problems have cycles of worsening morbidity that are known to impact on long-term quality of life.
Supplemental Material
sj-pdf-1-chc-10.1177_13674935211013192 – Supplemental Material for Preventing internalising problems in preschoolers with chronic physical health conditions
Supplemental Material, sj-pdf-1-chc-10.1177_13674935211013192 for Preventing internalising problems in preschoolers with chronic physical health conditions by Kate Noone, Ronald M Rapee, Maria Kangas, Vicki Anderson and Jordana K Bayer in Journal of Child Health Care
Footnotes
Declaration of Conflicting Interests
The author(s) declared no potential conflicts of interest with respect to the research, authorship and/or publication of this article.
Funding
The author(s) disclosed receipt of the following financial support for the research, authorship and/or publication of this article: The work was supported by Victorian Government’s Operational Infrastructure Support Program and National Health and Medical Research Council, the grant number is 607302.
Supplemental Material
Supplemental material for this article is available online.
Note
References
Supplementary Material
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