Pilot Mindfulness Intervention for Children Born with

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strategies were also affected by the mindfulness-based program. Rumination .... dence on the school and family environments must be noted, particularly the time ... Reviews of the literature favorably and positively regard the implementation of ...
J Child Fam Stud DOI 10.1007/s10826-017-0657-0

ORIGINAL PAPER

Pilot Mindfulness Intervention for Children Born with Esophageal Atresia and Their Parents Eva Andreotti1 Pascal Antoine1 Manel Hanafi2 Laurent Michaud2 Fréderic Gottrand 2 ●







© Springer Science+Business Media New York 2017

Abstract A pilot mindfulness home intervention was conducted for 6 weeks among 8–12-year-old children born with esophageal atresia and their parents. Participants were randomly assigned to a waiting list control (WLC) group (n = 8) and an experimental group (n = 12). When all participants had completed the mindfulness-based program, data were pooled and treated for the entire sample (n = 19). Selfassessment measures included the Mindful Attention Awareness Scale for Adolescents (MAAS-A), the Child and Adolescent Mindfulness Measure (CAMM), the modified Spielberger State-Trait Anxiety Inventory—Child (STAIC), the Children’s Depression Inventory (CDI), and the Cognitive Emotion Regulation Questionnaire Kids version (CERQ-k). Parental assessment measures included the modified STAI-C. The results underlined the program’s feasibility and acceptability. Clinical effects of the mindfulness-based program were observed. Self-assessed data for children who had practiced mindfulness compared to the WLC group showed increased mindfulness and decreased depression. Reduced anxiety was found in all groups. Positive affect tended to improve from pre-test to post-test in children who had practiced mindfulness compared to the WLC group. Parental assessments showed significant improvement in positive affect and decreases in anxiety and negative affect in the intervention group compared to the WLC group. Cognitive emotion regulation

* Eva Andreotti [email protected] 1

Universite de Lille, CNRS, UMR 9193—SCALab—Sciences Cognitives et Sciences Affectives, Lille F-59000, France

2

CHU Lille, Centre de Reference des Affections Congénitales et Malformatives de l’Oesophage, Pôle Enfant, Lille F-59000, France

strategies were also affected by the mindfulness-based program. Rumination scores significantly decreased from pre-test to post-test in the intervention group. This preliminary study suggests that regular mindfulness practice presents a promising approach to reducing the burden of this neonatal malformation. Keywords Mindfulness Children Depression Emotion regulation Anxiety ●







Introduction Esophageal atresia is a rare congenital malformation of the esophagus and trachea involving an interruption in the continuity of the esophagus, with or without persistent communication with the trachea. Associated anomalies occur in 50% of cases and affect the urinary, respiratory, cardiac and digestive systems as well as the skeleton—also called VACTERL syndrome (Gottrand et al. 2008). Following surgical repair of the malformation, long-term complications can be observed in association with respiratory and nutritional problems, dysphagia, esophageal stenosis and gastro-esophageal reflux (Legrand et al. 2012; Schneider et al. 2014). Children born with esophageal atresia require intensive medical and surgical care from birth (Sfeir et al. 2013). These children are an at-risk population in terms of mental health and psychological adjustment (Faugli et al. 2009a, 2009b). When assessed by their parents, children who have experienced complex and aversive medical events are identified as being at greater risk of emotional and

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behavioral issues between the ages of eight and twelve years compared to the general population (Bouman et al. 1999). Parents of nine to eighteen-year-old children who have undergone surgery for esophageal atresia also report more anxiety and depressive symptoms in their children than parents from a control group. These results are not related to gastro-intestinal and respiratory symptoms (Peetsold et al. 2010). Children who experience physical symptoms tend to describe themselves as more depressed and worthless and assess their physical appearance more negatively than those who do not (Bouman et al. 1999). According to Kubota et al. (2011), 35% of children who had major neonatal surgery for esophageal atresia show more behavioral issues than the general population, as assessed by their parents. Furthermore, the quality of life of 81 patients aged nine to eighteen years who had surgery for esophageal atresia was significantly inferior to that of healthy children (Legrand et al. 2012). Caplan (2013) emphasized the presence of “psychological comorbidities” linked to esophageal atresia in both children and their families. The results of studies on psychological issues among children with esophageal atresia in the medium to long-term remain controversial depending on population age and suggest an impact on mental health associated with developmental delays or an absence of differences compared to control groups (Faugli et al. 2009a). Children with esophageal atresia who exhibit anxiety and depressive symptoms are likely to respond to mindfulness-based interventions because such interventions assist patients with emotion regulation. Mindfulness is a way of paying attention on a moment-to-moment, non-judgmental basis, with curiosity and without trying to control the here and now experience (Kabat-Zinn 1996). With mindfulness practice, individuals can become observers of their inner physical sensations, feelings and cognitions or the environmental events that constitute their experience (Greco et al. 2011). Mindfulness can be taught through formal methods such as mindfulness meditation guidance or through informal methods that are part of daily life (Hayes and Shenk 2004; Kabat-Zinn 1996). Mindfulness cultivates non-reactivity and acceptance and can be maintained through “mental training” to reduce individual vulnerability to stress, automatism and emotional distress by promoting a shift in focus and distancing (Bishop et al. 2004). With mindfulness, children can learn how to know, understand and accept themselves and their thoughts and feelings to react more calmly to life events (Snel 2013). Although mindfulness interventions were initially conducted for adults, this approach has recently been adapted for children and adolescents (Greco and Hayes 2008). Mindfulness for children has also been generalized for the public (Greenland 2014; Snel 2013). Young children are

spontaneously able to be mindful and to “be” in the present moment without anticipating the future or ruminating on the past (Hooker and Fodor 2008). Nevertheless, adapting mindfulness-based programs to children requires specific attention to the developmental level and needs of this particular population. It is important to account for children’s attentional, cognitive and relational abilities. The dependence on the school and family environments must be noted, particularly the time allocated for mindfulness practice in addition to the usual time needed for homework and family life (Saltzman and Goldin 2008; Semple and Lee 2008). In school settings, a large number of studies have been conducted on mindfulness for children (Burke 2010). These studies have led to decreases in anxiety and depressive symptoms, such as rumination, intrusive thoughts and suicidal ideation. Moreover, increases in attentional, social and emotional abilities and increased positive affectivity have been found (Black and Fernando 2014; Britton et al. 2014; Liehr and Diaz 2010; Mendelson et al. 2010; Napoli et al. 2005; Schonert-Reichl et al. 2015). Familial specificities have been considered by some authors. Studies have shown that programs targeted to nine to twelve-year-old children and their parents have led to improvements in attentional abilities and compassion as well as decreases in emotional negative reactivity with regard to threatening events and judgmental attitudes toward oneself (Felver et al. 2014; Saltzman and Goldin 2008). Among the clinical population, children who suffer from psychopathologies or emotional disorders, such as anxiety disorder, present a lower level of behavioral and mood disorders after practicing mindfulness, with decreases in anxiety, depression levels and behavioral issues as well as increases in attentional abilities (Joyce et al. 2010; Semple et al. 2005, 2006). Children who have undergone difficult life journeys (e.g., abandonment, abuse) and who have low self-esteem also benefit from mindfulness, exhibiting a decrease in emotional reactivity (Coholic et al. 2012). For children with attention and hyperactivity disorders, mindfulness practice may lead to attentional and self-esteem increases as well as improvements in sleep quality and decreased anxiety (Harrison et al. 2004). On the somatic level, only one study has reported a case of mindfulness practice and examined a nine-year-old girl who suffered from gastroesophageal reflux (Ott 2002). Practicing mindfulness helped reduce her subjective experience of pain and distress as well as somatic symptoms and sleep disorders. Reviews of the literature favorably and positively regard the implementation of mindfulness interventions for children, with no negative effects observed (Burke 2010). Mindfulness practice can therefore lead to improvements in physiological and psychological health (Black et al. 2009).

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Mindfulness practice can be especially effective in decreasing anxiety and depression for children and teenagers and improving emotion regulation skills (Meiklejohn et al. 2012; Waters et al. 2015). In the present study, we developed and evaluated the effects of a mindfulness-based intervention for emotion regulation among children with esophageal atresia. The first aim of the study was to test its feasibility with these children’s families, and we hypothesized that the mindfulnessbased program would be favorably accepted. The second aim of the study was to assess whether the mindfulnessbased intervention would effectively promote clinically positive outcomes. We hypothesized that children who practiced the mindfulness-based program would demonstrate significant improvement over the course of the intervention in specific clinical variables such as mindfulness, positive and negative affectivity, state and trait anxiety and depressive symptoms compared to a control group. Finally, we evaluated the children’s cognitive emotion regulation strategies as potential mechanisms linked to mindfulness and their evolution during the course of the mindfulness-based program.

At the beginning of the study, all participants were invited to complete pre-test questionnaires. The participants in the experimental group then started the mindfulnessbased program for 6 weeks. A follow-up phone call was organized once a week or once every 2 weeks depending on the families’ schedules. This call provided an opportunity for the participants to share their experience, express their feelings and receive answers to any questions. In this way, we were able to obtain a better understanding of the participants’ feelings. After the 6-week mindfulness-based program, the participants in the experimental group completed the same questionnaires in an immediate post-test assessment. Meanwhile, the participants in the WLC group completed the same set of questionnaires for the second time before they began the 6-week mindfulness-based program. 6 weeks later, the participants in the experimental group were invited to complete, for the third and last time, the same questionnaires, representing the delayed post-test. Participants in the WLC group also completed the set of questionnaires, thereby concluding their participation in the study. Participants who wished to be informed about the outcomes of the study were sent the results.

Mindfulness-Based Program Overview

Method Participants Participant characteristics are presented in Table 1. The participants in this pilot study were eight to twelve-year-old children who were recruited from the Reference Center for Congenital Malformations of the Oesophagus (CRACMO) database and were accompanied by their parents. The children were diagnosed with esophageal atresia at birth and had underwent surgery. The exclusion criteria were Axis 1 disorders and deafness.

Procedure This study was approved by the Ethical Committee of the University of Lille Human and Social Sciences. Information about the project was shared through phone calls and letters. The parents and children had 10 days to decide whether to participate in the project. As shown in Fig. 1, 81 families were contacted, and 38 agreed to take part in the study. Written informed consent was obtained from the children and parents included in the study. Participants were randomly assigned to the experimental and the waiting list control (WLC) groups. We ensured that the two groups were well balanced in sex and age for comparisons. The participants were volunteers and were not remunerated.

The participants were mailed our 42-day mindfulness-based program, including detailed instructions and the investigators’ contact information. This daily program consisted of formal mindfulness practice with audio guidances that varied in length from 4 to 12 min and were displayed on and easily downloadable from a dedicated website. The program included a body scan (guided with or without gestures), a meditation centered on thoughts, three mindful breathing meditations, a mindful walking meditation, a sound meditation and a visualization meditation to help children focus their attention. Informal mindfulness training was also part of the program and consisted of observations of the environment, mindful tooth brushing and eating, drawing and observations of a familiar route. Every 6-day period focused on a formal practice experience, and each week represented a specific mindfulness theme (breath, body, sounds, thoughts, walking, and visualization) with reminders of the previous mindfulness guidance. The design of the mindfulness-based program was inspired by Britton et al. (2014), Fisher (2006), Hooker and Fodor (2008), KabatZinn (1990), Napoli et al. (2005), Saltzman and Goldin (2008) and the mindfulness-based cognitive therapy (MBCT) program for children (Semple and Lee 2008; Semple et al. 2006; Semple et al. 2010). Following Semple and Lee (2008) and Semple et al. (2006), we created small picture memos to remind the participants about the daily mindfulness practice.

J Child Fam Stud Table 1 Participants’ characteristics

WLC (n = 8)

Experimental (n = 12)

Variables

Value

Sex

Female (%)

Age

Mean (Range)

Mindfulness practice before

Yes (%)

1 (12.5)

3 (25)

Pathologies

Esophageal atresia III (%)

8 (100)

11 (92)

50 10.7 (9–12)

Esophageal atresia IV (%) Preterm birth

50 10.0 (8–12)

1 (8)

VACTERL (%)

1 (12.5)

2 (16)

Prematurity (%)

4 (50)

4 (33)

Term infant (%)

3 (37.5)

8 (67)

No information (%)

1 (12.5)

Birth weight

Mean in g (Ranges)

Gastroesophageal reflux

Yes (%)

5 (62.5)

3 (25)

Anti-reflux surgery

Yes (%)

2 (25)

1 (8)

Gastrostomy

Yes (%)

2 (25)

1 (8)

Esophageal dilation

Yes (%)

4 (50)

Parental status

Divorced (%) Married (%)

Homework time spent

Mean in min (Range)

Measures Acceptability and observance The extent to which children and parents followed the mindfulness practice was assessed with a daily follow-up sheet that was returned at the end of the 42 days. The follow-up sheet allowed us to assess whether children had completed the daily mindfulness practice independently or with assistance.

2315 (1235 3260)

0 8 (100) 37 (12 80)

2788 (1760 3803)

6 (50) 1 (9) 10 (91) 36 (5 90)

To complete the assessment, we used the Mindful Attention Awareness Scale for Adolescents (MAAS-A; Brown et al. 2011). This scale is based on a unidimensional concept of mindfulness. The fourteen items inquire about attention and consciousness in the present moment. Respondents were invited to answer on a 6-point Likert scale. This scale was validated in fourteen—to eighteenyear-old individuals, with a consistency of .82 and .84 (Brown et al. 2011).

Socio-demographic variables and medical information

Affect

Parents and children’s age, sex and education level as well as previous mindfulness practice were collected with a questionnaire. Parents were questioned about their family situation. Medical information (e.g., pathologies, premature birth, birth weight, nutrition type, esophageal dilation, gastroesophageal reflux, anti-reflux surgery) was collected from the Lille hospital center.

To obtain positive and negative affect scores as well as trait and state anxiety scores, we used a nonclinical measure, the modified Spielberger State-Trait Anxiety Inventory—Child version (STAI-C modified) (O’Neil et al. 1969; Turgeon and Chartrand 2003). This self-assessment scale measures anxiety for children aged eight to thirteen years old. It includes two series of twenty items assessing state and trait anxiety. The state-anxiety scale measures the intensity of emotional reactions. It encompasses ten positive affect items and ten negative affect items. Trait anxiety takes into account the frequency and tendency to consider the environment threatening and dangerous (Spielberger 1979). To improve the sensitivity of the self-assessment, we used a visual analog scale for the state-anxiety scale that inquired about the frequency of affect experienced, published by Britton et al. (2014). Positive and negative affect scores were computed in addition to state and trait anxiety measures.

Mindfulness measure The Child and Adolescent Mindfulness Measure (CAMM; Greco et al. 2011) was used to estimate mindful tendencies based on two constructs: acting with awareness and showing non-judgmental acceptance toward internal experiences. Answers to this 10-item scale were given on a 5-point Likert scale. This questionnaire was validated in nine to seventeen-year-old children, with a consistency of .81 (Greco et al. 2011).

J Child Fam Stud Fig. 1 Procedure flowchart Enrolment

Assessed for eligibility (n = 81)

Did not meet inclusion criteria (n =1) Declined to participate (n = 42)

Pre-test

Assignment

Randomized (n = 38)

Assigned to the Waiting List Control group (n = 19)

Assigned to experimental group (n = 19)

Pre-test assessment returned (n = 8) Lost to follow-up (n = 11)

Pre-test assessment returned (n = 15) Left the program the first week (n = 1) Lost to follow-up (n = 3) Received the intervention (n = 15)

Follow-up

Second set of questionnaires returned (n = 8) Lost to follow-up (n = 0) Received the intervention (n = 8)

Analyses

Immediate post-test returned (n = 8) Excluded from analyses (n = 1) Total lost to follow-up (n = 11)

Depressive symptomatology We used the Children’s Depression Inventory (Kovacs and Beck 1977; Mack and Moor 1982). This 27-item selfassessment measure aims to determine various aspects of depressive symptomatology. Respondents choose the statement that most closely matches their state of mind in the past 2 weeks out of three possible statements. Answers are rated from 0 (none) to 2 (severe depressive symptom). The global score is computed by adding the scores of all items.

Immediate post-test returned (n = 13) Excluded from analyses (n = 1 or 2 according to the variables) Lost to follow-up (n = 1)

Delayed post-test returned (n = 8) Excluded from analyses (n = 3) Total lost to follow-up (n = 9)

Whole sample (n = 21) Excluded from analyses (n = 2 or 3 according to the variables)

2007). Nine types of coping strategies are measured in this instrument with 36 items. Answers are given on a 5-point Likert scale ranging from 1 (almost never) to 5 (almost always). The aim is to evaluate the types of thoughts that appear after difficult life events. The subscales are SelfBlame, Other-Blame, Acceptance, Rumination, Positive Refocusing, Planning, Positive Reappraisal, Putting into Perspective and Catastrophizing. The internal consistency varies from .68 to .85, and the general validity was evaluated at .97 (Garnefski et al. 2007). Data Analyses

Cognitive emotion regulation measure To identify some of the processes linked to mindfulness practice, we used the Cognitive Emotion Regulation Questionnaire—Kid’s version (CERQ-k, Garnefski et al.

Data for any changes across the study periods were analyzed with Statistica 10 software. Self-assessment data covered levels of mindfulness, anxiety, depressive symptoms and cognitive emotion regulation strategies as well as

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positive and negative affect. Parental assessment data focused on anxiety and affectivity. Given the sample size, paired-sample Wilcoxon-Signed Rank tests were used to identify differences across the intervention periods at a group level to indicate any significant variation. Data analyses were conducted for eight children and parents in the WLC group and for twelve children and parents in the experimental group (Fig. 1). Due to missing responses, the quality of the data was insufficient to conduct analyses of two individuals from the WLC group for the selfassessment and parental assessment. For the experimental group, one set of data had to be withdrawn from the analyses for the same reason.

Results Feasibility and Qualitative Aspects Participants in the experimental group (n = 12) completed an average of 82.9% of the mindfulness exercises. On average, 57% of the exercises were completed with one or two parents. Within the entire sample, information was obtained for 16 of the participants who regularly practiced mindfulness during the program (84% of the total population). Mindfulness exercises were performed by 84.5% of the children (ranging from 64.3 to 100%). Two participants completed less than 70% of the exercises, four completed between 70% and 80%, two completed between 80 and 90%, and eight completed between 90 and 100%. The mean score of exercises completed with the help of one or two parents was 67.3%. Four children were routinely accompanied by their parents, and three of them always completed the exercises independently. During the phone calls, the participants raised a few points about their impressions of the program. This information enlightened us about the difficulties and fulfilment that they experienced during the study. Almost 1 week of implementing the daily mindfulness practice was needed for the family’s schedule to adjust and to find a rhythm, as some parents noted. A few parents were surprised by the effort needed to assist their child during the program. However, others were pleased by the low effort required. Among the parents who participated in the program, some noted that the intervention could promote family bonding. Increases in calm feelings were also noted within the family and professional settings. One child stopped the program during the 1st week because he said he could not perform the mindfulness practice; the experience seemed “not natural”. The artificial aspects of the program appeared to cause him discomfort. Some children were surprised by the repeated guidance. Some of

them became bored and impatient, whereas others enjoyed discovering new aspects during each practice and deepened feelings of concentration and perception. Less appreciated practice guidance, which differing depending on the child, was often qualified as being “too long”. A majority of children said that they used mindfulness practice in their daily life, especially during key times, such as sleeping, in interpersonal conflicts, and in medical and school exams. Nine children said that they were “calmer” after the mindfulness practice, and seven said that they were “more relaxed” and “less stressed”. Some children noticed feeling less upset and irritated, whereas others noticed increased concentration abilities, particularly during school situations. Some children discussed ways in which the mindfulness practice allowed them to grasp emotions, particularly anger, and to identify and live with these emotions. The children were surprised to notice shifts in their perceptions and behavior following the mindfulness-based program, especially in negative affect. One of the children claimed that practicing mindfulness helped her step back and keep things in perspective. One child noticed that the intervention helped her “better understand what she did and how she reacted”. Another said that she “had grown up” during the program. She explained, “It was fine for children who had lived through shocks that impacted them. I went through that, and I realized I had lots of thoughts in my head”. Another child noticed that the program “helped [her] to identify [her] emotions and get calmer”. Some participants were intrigued by the novelty of the program and how it allowed them to have “new experiences” with only what they already had. A majority of children said that they became more aware of their thoughts and affect during the mindfulness practice. This information supported the feasibility and acceptability of the mindfulness-based program.

Program Efficacy and Mindfulness, Affect and Depression Variables As shown in Table 1, the experimental and WLC groups did not differ by sex ratio or age. There were also no differences between the experimental and the WLC groups in self—or parental assessment data at pre-test, except for self-assessed negative affect. The outcomes of the analyses between the pre—and post-test self-assessments for the experimental and WLC groups are shown in Table 2. The WLC group presented no significant improvement in mindfulness variables (MAAS-A, CAMM), positive affect or depression symptomatology variables, although the means of the negative affect and anxiety measures decreased. In the experimental group, a significant improvement in

J Child Fam Stud Table 2 WLC group compared with the experimental group—Self-assessment Variables

Mindfulness

MAAS-A CAMM

Affectivity

Positive affect Negative affect

Anxiety

STAI-State STAI-Trait

Depression Cognitive

CDI Self-blame

emotion regulation

Acceptance

strategies Rumination Positive refocusing Planning Positive reappraisal Putting into perspective Catastrophizing Other-blame

Group

N

Pre-test M (SD)

Post-test M (SD)

Z

p-value

58.4 (11)

65.9 (10)

2.123

0.032*



57.6 (16.8)

61.9 (15.1)

1.403

0.161

=

2.073

0.038*



1.890

0.058

=

1.765

0.077†



1.400

0.161

=

EXP

12

WLC

8

EXP

12

25.6 (5.5)

29.4 (5.8)

WLC

8

22.6 (4.1)

26.4 (4.3)

EXP

12

68.8 (18)

78.2 (8.9)

WLC

8

73.8 (23)

78.2 (19)

EXP

12

16.1 (11.1)

10.9 (7)

1.333

0.182

=

WLC

8

43.6 (29.2)

22.1 (19.8)

1.960

0.049*



EXP

12

46.4 (25)

32.6 (14)

1.961

0.049*



WLC

8

69.9 (47.7)

43.8 (37.5)

2.520

0.011*



EXP

12

35.8 (6.7)

27.7 (2.9)

2.784

0.005**



WLC

8

35.6 (7.6)

33.4 (7.5)

1.224

0.046*



EXP

12

7.2 (3.8)

4.3 (3.1)

2.311

0.020*



WLC

8

9.4 (4.7)

9.5 (5.4)

0.209

0.833

=

EXP

11

7.5 (2.1)

6.4 (1.8)

1.362

0.173

=

WLC

8

10.7 (5.4)

9.1 (3.6)

1.467

0.142

=

EXP

11

12 (3.5)

9.1 (3.6)

2.201

0.027*



WLC

8

11.1 (4.2)

8.1 (1.9)

1.960

0.049*



EXP

11

10.1 (3)

7.8 (2)

1.987

0.046*



WLC

8

11 (4.3)

9.4 (3.3)

1.362

0.172

=

EXP

11

14.2 (2.9)

11.6 (3.3)

2.369

0.017*



WLC

8

13.1 (5.6)

12.4 (5.2)

0.000

1.000

=

EXP

11

12.9 (3.4)

10.8 (3.6)

1.572

0.115

=

WLC

8

13.7 (2.8)

13.1 (4.4)

0.420

0.674

=

EXP

11

10.9 (2.5)

9.3 (3.2)

1.421

0.155

=

WLC

8

9.6 (3)

9.4 (2.4)

0.419

0.674

=

EXP

11

12.4 (3.6)

9.8 (4.1)

1.717

0.085†



WLC

8

10.2 (4.2)

1.014

0.310

=

EXP

11

8.1 (2.9)

6 (1.9)

1.895

0.058†



WLC

8

11 (5)

10 (4.8)

0.676

0.498

=

EXP

11

8 (3.3)

6.4 (2)

1.680

0.092†



WLC

8

8.2 (4.3)

5.6 (1.6)

1.603

0.108

=

11.2 (4)

EXP Experimental group, WLC Waiting list control group, MAAS-A Mindful Attention Awareness Scale for Adolescents, CAMM Child and Adolescent Mindfulness Measure, STAI-C State-Trait Anxiety Inventory-Child modified, CDI Children’s Depression Inventory, M mean, SD standard deviation †

p < 0.10, tendency, *p < 0.05, **p < 0.01, two-sided

↗: increase in the mean from pre-test to post-test, ↘: decrease in the mean from pre-test to post-test, =: no significant change

mindfulness variables and a decrease in anxiety and depression were observed. Positive affect tended to improve over time. For the parental assessments, as reported in Table 3, we observed no significant change in the WLC group, whereas external observers reported a significant increase in positive affect and a decrease in anxiety and negative affect in children in the experimental group.

As shown in Table 4, we found that for the entire group of children (n = 19) who completed the mindfulness-based program, the average MAAS-A scores increased significantly, whereas the CAMM scores remained stable. We observed a tendency toward an increase in the average scores for positive affect, whereas the negative affect scores decreased significantly, as did the depression symptom scores and the trait—and state-anxiety variables.

J Child Fam Stud Table 3 WLC group compared with the experimental group— parental assessment

Variables

Positive affect Negative affect STAI-C State STAI-C Trait

Group

N

Pre-test M (SD)

Post-test M (SD)

Z

p-value

EXP

11

18.7(3.4)

22.6 (3.9)

2.934

0.003**



WLC

8

18.9 (2.5)

18.6 (3.1)

0.209

0.833

=

EXP

11

14.2 (4.6)

10.6 (1)

2.520

0.011*



WLC

8

15.2 (3.7)

13.5 (4.7)

1.153

0.248

=

EXP

11

25.4 (6.3)

18 (4.3)

2.934

0.003*



WLC

8

24.9 (7.3)

0.674

0.500

=

EXP

11

32.5 (6.5)

28.4 (3.9)

2.578

0.009**



WLC

8

37.7 (6.4)

37.2 (6.1)

0.676

0.498

=

26.4 (5)

EXP Experimental group, WLC Waiting list control group, STAI-C State-Trait Anxiety Inventory—Child modified, CDI Children’s Depression Inventory, M mean, SD standard deviation *p < 0.05, **p < 0.01, two-ways ↗: increase in the mean from pre—to post-test, ↘: decrease in the mean from pre-test to post-test, =: no significant change

Table 4 Pre-test and post-test self-evaluation during the mindfulness intervention

Variables

N

Pre-test M(SD)

Post-test M (SD)

Z

p-value

Effect size

MAAS-A

19

60.5 (12.5)

66.6 (9.5)

2.385

0.017*



CAMM

19

25.8 (5)

27.7 (6.4)

1.491

0.135

=

Positive affect

19

71.6 (18.6)

79.2 (12.1)

1.951

0.050†



0.45

Negative affect

19

18.1 (15.3)

10.9 (10.1)

2.535

0.011*



0.58

0.54

STAI-state

19

45.9 (30.4)

31.7 (20.2)

2.615

0.008**



0.60

STAI-trait

19

34.5 (7)

28.6 (4.7)

3.219

0.001**



0.73 0.76

CDI

18

7.9 (4.5)

4.2 (3.2)

3.218

0.001**



Self-blame

18

7.9 (2.6)

7 (2.9)

1.448

0.147

=

Acceptance

18

10.4 (3.5)

8.8 (3.3)

1.733

0.082†



0.40

Rumination

18

9.7 (3.1)

7.6 (2.3)

2.726

0.006**



0.64

Positive refocusing

18

13.4 (4.1)

11.8 (3.5)

1.703

0.088†



0.40

Planning

18

12.8 (3.8)

11.3 (3.4)

1.607

0.107

=

Positive reappraisal

18

10.3 (2.6)

9.1 (3.2)

1.680

0.092†



Putting into perspective

18

11.2 (3.9)

10.2 (3.3)

0.904

0.365

=

Catastrophizing

18

8.9 (4)

7.7 (3.8)

1.396

0.162

=

Other-blame

18

7.1 (3)

6.8 (2.8)

0.803

0.421

=

0.39

MAAS-A Mindful Attention Awareness Scale for Adolescents, CAMM Child and Adolescent Mindfulness Measure, STAI-C State-Trait Anxiety Inventory—Child modified, CDI Children’s Depression Inventory, M mean, SD standard deviation †

p < 0.10, tendency, *p < 0.05, **p < 0.01, two-sided

↗: increase in the mean from pre-test to post-test, ↘: decrease in the mean from pre-test to post-test, =: no significant change

For the parental assessments (reported in Table 5), a significant increase in positive affect as well as significant decreases in negative affect and state—and trait-anxiety variables were observed. Potential Underlying Mechanisms The analysis of the CERQ-k (Table 2) showed changes in several sub-scales over time. The measurements remained

unchanged in the WLC group, with the exception of a significant decrease in the Acceptance sub-scale. The experimental group showed significant decreases in the Acceptance, Positive Refocusing and Rumination scores. A decreasing tendency was found for the Putting into Perspective, Catastrophizing and Other-Blame sub-scales. Analyses of the entire sample (reported in Table 4) were slightly different. There was a non-significant tendency toward a decrease in Acceptance over time. We also

J Child Fam Stud Table 5 Mindfulness intervention evolution from pretest to post-test in the parental evaluation (n = 18)

Variables

Pre-test M (SD)

Positive affect

18.4 (3)

22.1 (3.7)

3.516

0.000**



0.82

14 (4)

10.7 (1.1)

3.059

0.002**



0.72

STAI-State

25.6 (6.5)

18.6 (4.3)

3.723

0.000**



0.87

STAI-Trait

33.9 (6.3)

29.3 (3.6)

3.396

0.000**



0.80

Negative affect

Post-test M (SD)

Z

p-value

Effect size

M mean, SD standard deviation *p < 0.05, **p < 0.01, two-sided ↗: increase in the mean from pre-test to post-test, ↘: decrease in the mean from pre-test to post-test

observed a decreasing tendency for the Positive Refocusing and the Positive Reappraisal sub-scales. The decrease in Rumination remained significant.

Discussion The purpose of this study was to examine the acceptability and the clinical effectiveness of a 6-week daily mindfulness home intervention for eight to twelve-year-olds born with esophageal atresia and their parents. The feasibility and acceptability of the intervention were supported by the data obtained from the participants who regularly practiced mindfulness, thus confirming our hypothesis. Clinical variables in children such as anxiety, depression and positive and negative affectivity, which were assessed through self-assessment and parental assessment, changed during the course of the program. Few significant clinical changes were found among the WLC group; these changes involved negative affectivity and anxiety. Consequently, our hypothesis was partially verified for the self-assessment measures and confirmed for the parental assessment outcomes. Among all participants who practiced mindfulness, clinical effectiveness in children was apparent for several variables, such as negative affectivity, positive affectivity, anxiety and depression, based on the perceptions of the children’s parents as external observers of the phenomenon. In contrast to Britton et al. (2014), our findings supported the idea that mindfulness practice might be a mediator of increased positive affectivity (Fredrickson 2001; Nyklícek and Kuijpers 2008). Consistent with the literature, it was difficult to identify a link between decreased self-assessed anxiety measures and the intervention (Liehr and Diaz 2010; Semple et al. 2010), although the parental assessment data showed significant changes in this variable. This discrepancy between the measures according to the children and their legal guardians was previously noted by Bouman et al. (1999). This finding led us to wonder whether our observations could be based on parental expectations of

children’s participation in mindfulness-based programs or parents’ openness toward their children’s emotional experience. The latter could be consistent with Harrison et al. (2004), who found that some parents mentioned a new awareness of positive changes in their relationship with their children. An effect of mindfulness on levels of depression was also found in the literature (Joyce et al. 2010; Liehr and Diaz 2010; Schonert-Reichl et al. 2015). Examining the potential underlying mechanisms, we observed an increase in mindfulness measured with the MAAS-S, and this finding was consistent with Saltzman and Goldin (2008) and with Felver et al. (2014). These improvements in mindfulness may relate to increases in attentional abilities toward here-and-now aspects (Brown et al. 2011). We also found that rumination scores decreased over time with the mindfulness practice. A decreased tendency appeared in the acceptance, positive refocusing and positive reappraisal cognitive emotion regulation strategies. These results are not consistent with the findings of Schonert-Reichl et al. (2015). Interpreting the acceptance scores is complicated because of the CERQ-k subscale items (Garnefski et al. 2007), which more address “resignation” aspects, such as duty to accept negative events, than the capacity of the individual to stay in touch with experiences involving emotions, sensations or cognition without constraints or avoidance. Decreased rumination has been attributed by Mendelson et al. (2010) to a modification of the usual response to negative stimuli. Legerstee et al. (2010) suggested that the low anxiety and limited experience of rumination and catastrophizing among nine to eleven-year-olds were linked. The need for emotion regulation appears when one’s emotions seem unsuitable for the context, when they limit access to specific aims or when they do not respond to social norms (Gross and Thompson 2007). Considering our observations, we asked whether the participants’ experience with mindfulness involved emotion avoidance, instead of non-judgmental acceptance, if individuals attempted to avoid unpleasant stimuli by directing their attention toward the present moment. Mindfulness practice could be diverted

J Child Fam Stud

and be associated with attention withdrawal from negative stimuli toward more gratifying events (Felver et al. 2014). Furthermore, we asked whether the mindfulness learning had led children to develop specific meta-cognitive attention toward the type of cognitive emotion regulation strategies that they initially used and whether they might specify their perceptions of this use. Finally, we asked how relevant the need for cognitive emotion regulation strategies was while practicing mindfulness. Shapiro et al. (2006) considered “reperception” as the meta-mechanism through which mindfulness provided a non-judgmental authentic observer position for the meditator. Thus, no cognitive reconstruction was necessary when practicing mindfulness. The nature of the experience does not need to be changed or controlled. This might contribute to reinforcing subjective distress (Moses and Barlow 2006). High-frequency mindfulness practice may influence well-being and emotion regulation capacity (Waters et al. 2015). Mindfulness practice appears to be related to the development of mental abilities and improvements in health and behavior (Fisher 2006). Emotional, attention and executive regulation processes are likely to benefit from mindfulness practice and to contribute to the individual’s development (Davidson et al. 2012). Through mindfulness, self-regulation abilities may be developed, top-down processes such as attention and cognitive flexibility may be facilitated, and bottom-up processes such as judgments, emotional reactivity, and rumination may be diminished (Zelazo and Lyons 2012). Mindfulness contributes to attentional processes and their regulation at both the theoretical and empirical level (Shapiro et al. 2006; Semple et al. 2010). As children grow up, their cognitive capacities mature and they are progressively able to obtain an increasingly fine level of detail in their emotional states (Legerstee et al. 2010). Although they initially center on behavioral emotion regulation strategies, from six to nine years old, children develop more internalized strategies that are linked to cognitive resources (Aldwin 2007, p. 284). At approximately eight or nine years old, children are able to regulate their emotions with cognitions and thoughts about their feelings, themselves and others (Garnefski et al. 2007; Muris et al. 2007). We chose to develop a mindfulness intervention that addressed eight to twelve-year-old children with esophageal atresia. This developmental period seemed appropriate relative to children’s increased self-regulation abilities (Mendelson et al. 2010). Children with esophageal atresia and their families encounter a life-threatening situation that requires surgical intervention within a short period of time. Moreover, in 50% of cases, these children are affected by several malformations (Gottrand et al. 2008). Parental expectations of the prospect of a healthy child vanish brutally, and parents have very little time to adjust. Family structures and

dynamics are affected by the threat of malformation to the child’s life (Leblanc et al. 2003). Parents must face the prospect of their children requiring feeding intravenously or with a gastrostomy tube that will remain until the children are able to feed themselves per os. During the healing process, the children must frequently return to the hospital for medical examinations with radiography and endoscopy, which can present an ongoing stressor. During the first year following surgery, respiratory and gastric complications can occur that may also affect the children’s quality of life (Legrand et al. 2012; Schneider et al. 2014). The aftereffects can lead to a recurrence in clinical symptoms, and children may show a lack of self-esteem and suffer from decreased body image (Bouman et al. 1999). In pediatrics, great care must be taken with families and children who are potentially at risk of developing psychopathology (Faugli et al. 2009b). Although some children and families can adjust and develop a fulfilling life after encountering a medically challenging life onset, others are likely to develop anxiety and depressive symptoms, acute stress or post-traumatic stress disorder when facing key moments of the child’s care, such as first symptoms, diagnosis, medical interventions, hospitalizations, and followups (Caplan 2013). An intense emotional range may be experienced by parents and children facing pain and distress, including hopelessness and fear. It is quite common for subclinical post-traumatic stress disorder with reexperiencing, rumination or avoidance symptoms to appear among families. Regarding the risks of psychopathology in these families, it seems of the utmost importance for medical and health teams to play a preventive role. Whether within the context of such an experience or in the distance conferred by time from these potentially traumatic events, it is relevant to suggest mindfulness practice as a resource that is accessible at any time for children and parents. Thus, we offered parents the opportunity to join their children in mindfulness practice. Moreover, parents can function as effective partners in mindfulness practice by encouraging children, reminding them of the practice, and even practicing with them (Semple et al. 2006). Our qualitative understanding of the work with families allowed us to observe that parents as well as children appeared to benefit from the mindfulness practice. Mindfulness practice within the family seems particularly interesting due to the development of inter-personal and attachment aspects, such as reinforcement of family bonding (Duncan et al. 2009). This practice encourages careful listening, non-judgmental acceptance of both the parent’s needs and the child’s needs, self-regulation in the parental relationship and compassion for oneself and for one’s child. Parents can also provide their children with a way of recognizing and express emotions, thereby promoting self-regulation abilities (Duncan et al. 2009).

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There are several limitations regarding the generalization of the current results. The WLC group was “waiting” and did not perform an activity during the experimental procedure. Furthermore, we did not investigate the children’s social and economic background. Underprivileged backgrounds appear to be correlated with emotion regulation difficulties (Mendelson et al. 2010). The recruitment conditions were subject to self-selection bias. The inclusion phase also involved an intentional stage that was necessary to implement mindfulness and that may change as the practice proceeds (Shapiro et al. 2006). The measures used in this study were subjective, based on questionnaires, and we therefore must consider the potential risk of bias, such as social desirability and expectation effects. However, we believe that the individuals were capable of determining and sharing their own experiences. Due to the use of the modified STAI-C, the results cannot be easily generalized. The use of a scale that differentiates emotions based on valence and activation level may have produced greater precision in the nature of the changes we observed. The specificity of the sample and its small size also represent limitations in generalizability. Feasibility information was gathered through phone calls. In the future, more systematic measures could be used, perhaps with quantitative information. With regard to the intervention limitations, a few participants had difficulty accessing the online website to download the guidances. Three children had difficulty understanding some terms. The aspects of the guidances that were commented on will be modified in the future. Despite the fact that the literature recommends implementing interventions with experienced meditators for mindfulness-based programs (Hooker and Fodor 2008; Saltzman and Goldin 2008), the telephone support was provided by a young meditator. Questions remain after this study, such as the “quantity” of mindfulness practice necessary to measure an impact in children. Moreover, it seems essential to identify the pretest characteristics and predictive factors of the success or failure of mindfulness-based programs among children (Britton et al. 2014). The level of executive functioning (Flook et al. 2010), developmental age (Schonert-Reichl and Lawlor 2010), socio-economic level (Mendelson et al. 2010) and familial environment (Barnes et al. 2010; Roeser and Eccles 2015) may play a key role in the way children benefit from interventions. Investigating the mechanisms underlying the mindfulness process in connection with emotion and attention regulation seems to be a promising approach (Britton et al. 2014; Felver et al. 2014; SchonertReichl and Lawlor 2010). Understanding the way mindfulness typically develops within the framework of socialization, families, school and medical conditions could be an interesting area of research to better define appropriate methods of encouraging the use of mindfulness.

The specific design of our study confirmed that it is possible to deliver a mindfulness guidance and training program as a self-help tool outside of the hospital setting. This intervention, which is free of cost for families, has several strengths. The addition of a WLC group, as recommended by Burke (2010), allowed for a differentiation of the effects of the mindfulness-based program from those linked to placebo or maturation. This program consisted of short practices that were repeated over time with a few trainings. It encouraged learning and the development of mindful awareness toward internal or external stimuli from multi-sensorial modalities to ensure that participants could implement mindfulness in their daily lives. The daily follow-up helped us evaluate the amount of exercises completed by each participant. To help participants remember to perform their daily practice, we provided them with small pictures to color and stick on significant places as a reminder. Unfortunately, only a small quantity of follow-up questionnaires for the experimental group was returned, and we could therefore not conduct analyses about the persistence of effects. Future studies would benefit from a larger sample to understand the impact of mindfulness (Meiklejohn et al. 2012; Waters et al. 2015). Compliance with ethical standards Conflict of Interest peting interests.

The authors declare that they have no com-

Ethical Approval All procedures performed in studies that involved human participants were in accordance with the ethical standards of the institutional and/or national research committee and the 1964 Helsinki declaration and its later amendments or comparable ethical standards.

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