The Cleft Palate–Craniofacial Journal 52(4) pp. e73–e80 July 2015 Ó Copyright 2015 American Cleft Palate–Craniofacial Association

ORIGINAL ARTICLE Longitudinal Changes in Dental Fear and Coping Behavior in Children, Adolescents, and Young Adults With Cleft Lip and/or Cleft Palate Janneke B. Krikken, M.Sc., Ph.D., D.D.S., Ad de Jongh, M.Sc., Ph.D., D.D.S., Jaap S.J. Veerkamp, D.D.S., Ph.D., Wilma Vogels, M.Sc., D.D.S., Jacob M. ten Cate, M.Sc., Ph.D., Arjen J. van Wijk, M.Sc., Ph.D. Objective: To determine changes in dental anxiety levels of cleft lip and/or palate (CL/P) children and to explore the role of coping strategies in the development of their dental anxiety. Design: Prospective study. Setting: Free University Medical Center Amsterdam. Patients: A sample of CL/P children (at T1: n ¼ 153, 4 to 18 years, 67 girls; at T2: n ¼ 113, 7 to 21 years, 51 girls). Data were available at both time points for 102 children. Measures: Dental anxiety and coping strategies were assessed at the start of the study (T1; mean age: 9.8 years, standard deviation 4.1) and 3 years later (T2; mean age: 13.4 years, standard deviation 3.8). These scores were compared to a normative group of Dutch children. Main Outcome Measure(s): The severity of dental anxiety was indexed using the Parental Version of the Dental Subscale of the Children’s Fear Survey Schedule. Dental coping strategies were assessed with the Dental Cope Questionnaire. Results: Overall, dental anxiety decreased to a level equal to normative scores of Dutch children. However, 5% of the children became more anxious. At T2, children used significantly fewer coping strategies. Children whose level of dental anxiety increased significantly used more destructive coping strategies than children whose level of dental anxiety decreased significantly or remained stable. Conclusions: Results suggest that dental anxiety levels of most CL/P children gradually decline over time. Whereas some coping strategies have the potential to be protective, more destructive coping strategies may put children at greater risk for developing and maintaining their dental anxiety. KEY WORDS:

cleft lip and/or cleft palate, coping behavior, dental fear, Dental Subscale of the Children’s Fear Survey Schedule

Medical procedures and dental treatments can be potentially distressing for children since they can include experiences with a number of adverse stimuli. In addition, such experiences can activate memories of earlier unpleasant or painful dental or medical treatments (de Jongh et al., 2006). Children who are not able to cope with distressing events are at risk of developing dental anxiety, which is a

common phenomenon in children and adolescents (Chhabra et al., 2012; Salem et al., 2012). It is estimated that in a Western country such as the Netherlands, about 14% of the children are either vulnerable to developing, or already suffer from, high levels of dental anxiety (ten Berge et al., 2002b; Krikken et al., 2013). Because dental anxiety may cause people to avoid going to the dentist, with negative consequences for their dental health, this represents a problem for both dentists and patients (Taani et al., 2005; de Jongh et al., 2011). With regard to the development of dental anxiety, a large array of studies suggest that overwhelming or adverse experiences make people vulnerable and increase the risk of developing dental anxiety (de Jongh et al., 1995; Oosterink et al., 2009). In particular, the experience of helplessness during a previous medical or dental treatment has been found to potentially predispose people to pathological levels of dental anxiety (Oosterink et al., 2009). Studies in children show inconsistent results (King and Ollendick, 1989; Klingberg et al., 1995; ten Berge et al., 2002a; Versloot et al., 2004; Krikken and Veerkamp, 2008; Gustafsson et al., 2010b; Krikken et al., 2010) but appear

Dr. Krikken, Dr. Veerkamp, and Dr. Vogels are Paediatric Dentists at the Department of Cariology, Endodontics, and Pedodontology, Academic Centre for Dentistry Amsterdam (ACTA), University of Amsterdam and VU University Amsterdam, the Netherlands. Dr. de Jongh is Psychologist, Dentist, and Professor, and Dr. van Wijk is Psychologist and Statistician, Department of Social Dentistry, ACTA, University of Amsterdam and VU University Amsterdam, the Netherlands. Dr. ten Cate is Professor, Department of Preventive Dentistry, ACTA, University of Amsterdam and VU University Amsterdam, the Netherlands. Submitted October 2012; Revised February 2013, September 2013, May 2014, July 2014, October 2014, December 2014; Accepted December 2014. Address correspondence to: Dr. J.B. Krikken, ACTA, Department of Pedodontology, Gustav Mahlerstraat 3004, 1081 LA Amsterdam, the Netherlands. E-mail [email protected]. DOI: 10.1597/12-262 73

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to indicate that exposure to distressing medical or dental procedures cannot fully explain the development of their dental anxiety and suggest that other determinants may also play a role. These include the child-rearing style of the parents (Krikken and Veerkamp, 2008) and a child’s personality characteristics (King and Ollendick, 1989; Klingberg et al., 1995; ten Berge et al., 2002a), such as temperament (Gustafsson et al., 2010b) and negative emotionality (Krikken et al., 2010). There is evidence to suggest that such characteristics have a negative effect on a child’s coping ability, i.e., the capacity to cope with adverse events (Versloot et al., 2004). Cleft lip and/or cleft palate (CL/P) is a common congenital anomaly, with an overall prevalence of 1.7 per 1000 live births in the Netherlands (Rozendaal et al., 2012). CL/P is considered to have a multifactorial origin involving a combination of genetic and environmental influences. Repairing the anatomical defects and treating the related functional problems involves a series of procedures such as reconstructive surgery, speech therapy, orthodontics, and dental treatment. Therefore, children with CL/P are exposed to a number of surgical and dental procedures. For example, in the Netherlands, CL/P children undergo repair of the lip defect at the age of 3 months, repair of the soft palate at 9 months, and closure of the jaw defects at approximately 9 years of age. Before this last procedure, most CL/P children have already been treated by the orthodontist to align teeth. Therefore, CL/P children should be considered vulnerable to the development of dental anxiety, as has been demonstrated in the literature (Davey, 1989; de Jongh et al., 1995; ten Berge et al., 2001). Accordingly, it is conceivable that children and adolescents with CL/P would show more severe levels of dental anxiety than children without this condition. Although this hypothesis was supported by the results of a previous study (Vogels et al., 2011), another study yielded contrasting results (Dogan et al., 2013). In the latter study, CL/P children were found to be less dentally anxious than their healthy counterparts, a result that was attributed by the authors to previous exposures to the dental setting (Dogan et al., 2013). As noted, the limited number of studies regarding CL/P and dental anxiety show inconsistent results; the same holds true for literature regarding the association between CL/P and other types of anxiety. For example, while some studies found that CL/P children report higher levels of social anxiety (Berk et al., 2001; Tyler et al., 2013) or separation anxiety (Tyler et al., 2013), a review done by Hunt et al. (2005) showed that, although CL/P children and young adults reported more symptoms of depression and had been teased more often, they did not suffer from more symptoms of trait and state anxiety (Hunt et al., 2006). One explanation for these conflicting results may be that some children are more vulnerable to the development of dental anxiety and are less resilient following CL/P-related surgery than others.

The use of coping strategies is another factor with the potential to affect the development of dental anxiety in children. It has been hypothesized that internal or external stressors can be managed by using different coping strategies to reduce levels of psychological distress after exposure to a distressing event (Folkman & Lazarus, 1991). However, not every form of coping is equally helpful in alleviating symptoms of dental anxiety or distress. For example, there is evidence to suggest that certain coping strategies (e.g., the suppression and avoidance of thoughts and/or feelings related to a distressing experience) put children at greater risk of psychological dysfunction (Kaplow et al., 2005; Simon et al., 2010), whereas others (i.e., expressiveness and emotional openness) have the potential to be protective (Gutner et al., 2006). Research examining the effect of coping strategies of children exposed to the dental setting indicates that the choice of coping strategies seems to be determined in part by the level of dental fear (Versloot et al., 2004). It was found that fearful children use more coping strategies, particularly external coping strategies, than less fearful children. This suggests that fearful children lack the capacity to deal with the situation and seek external support from their parents or dental staff (Versloot et al., 2004). The present study has a twofold aim. First, it sought to measure changes in CL/P children’s dental anxiety during a 3-year follow-up period (both in terms of differences in means and the percentage of patients who showed reliable change); these data were then compared with normative data of the Dutch population. Second, this study explored the relationship between dental anxiety and coping style. More specifically, it was hypothesized that the child’s coping styles (i.e., the frequency of internal, external, and destructive coping strategies, as indexed by the Dental Cope Questionnaire [DCQ]) would be significantly associated with the severity of their dental anxiety. MATERIALS

AND

METHODS

Participants and Procedure In the Netherlands, all children born with CL/P are treated by 1 of 16 CL/P teams. The eligible participants in the present study were the children (n ¼ 200) and their parents receiving treatment from one of these teams (VU Medical Center, Amsterdam) at the time of the investigation. Exclusion criteria were an inability to read and write and insufficient comprehension of the Dutch language. However, as none of the participants fulfilled the exclusion criteria, all individuals who initially were approached participated in the study. As part of the treatment by the VU CL/P team, the children were surgically treated at the age of 3 months for closure of the lip and at the age of 9 months for closure of the soft palate. From the age of approximately 2 years, the children visited their family dentist twice a year for an

Krikken et al., LONGITUDINAL CHANGES IN DENTAL FEAR AND COPING BEHAVIOR

examination and prophylaxis. At the age of 7 to 9 years, the children were surgically treated for closure of the hard palate and jaw. Between these visits, the children were seen for assessments of their hearing and speech. At the start of our study (T1) the children and their parents were sent questionnaires by mail and were asked to complete and return them by mail. At that time, the mean age of the participants was 9.8 years (standard deviation [SD] ¼ 4.1). Three years later (T2), the same participants received the same questionnaires. At that time, the mean age of participants who responded was 13.4 years (SD ¼ 3.8). All participants at both time points were asked to fill out the Dental Subscale of the Child Fear Survey Schedule (CFSS-DS) to assess the severity of their dental anxiety (Cuthbert and Melamed, 1982). Children 6 to 18 years old were asked to complete the DCQ to determine which coping strategies they would use in a dental situation. This was done at both T1 and T2. No differences in age were found between boys and girls at T1 and T2. Because all children visited their own family dentist for dental checkups and treatment, no exact data on current caries experience was available at the time of the study. For about half of the children, this could be determined retrospectively. Of these children, almost 31% had caries and were treated for this condition. This study was approved by the medical ethical committee of VU Medical Center Amsterdam, the Netherlands (ref 05/121). Measures Severity of dental anxiety was measured using the Dutch version of the CFSS-DS (Cuthbert and Melamed, 1982). The CFSS-DS is a questionnaire with sufficient validity and reliability (Aartman et al., 1998; ten Berge et al., 1998). It consists of 15 items to be answered on a 5-point scale ranging from 1 (not afraid at all) to 5 (very afraid), so that a total score ranges from 15 to 75. One item was added so that parents could rate their own level of dental fear on the same 5-point scale. It has been shown that the majority of parents are able to estimate their child’s level of dental anxiety with the CFSS-DS (Gustafsson et al., 2010a; Krikken et al., 2013). Research suggests the following classification of scores for Dutch children: a nonclinical range (not anxious, scores below 32), a borderline range (scores between 32 and 39), and a clinical range (very anxious, scores of 39 and higher) (ten Berge et al., 2002b). Cronbach’s alpha for the CFSS-DS completed at T1 was 0.93 and it was 0.94 at T2. The coping strategies of the children were assessed using the DCQ. The DCQ is a self-report checklist that requires the child to imagine a painful situation at the dentist and assess which type and frequency of coping strategies he or she would use. Therefore, this question-

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naire can only be used for children who are at least 6 years old. It is a revised version of the Kidcope, which was developed as a specific pain coping questionnaire for children (Spirito et al., 1988). The survey starts with the question: ‘‘Have you ever had pain at the dentist?’’ (scoring: often, sometimes, never). This question was followed by 15 coping strategies related to the dental setting; for example: ‘‘When I am in pain at the dentist’’ possible strategies include ‘‘I tell myself it will soon be over,’’ ‘‘I think about something else,’’ and ‘‘I get angry at the dentist.’’ The child is asked to rate both the use of each strategy (part A: yes or no) and the perceived effectiveness of each strategy (part B: not at all, a little, or a lot) (Spirito et al., 1988). Three groups of strategies are used in the analyses: internal coping strategies (six items; e.g., ‘‘When I am in pain at the dentist, I tell myself it will be over soon’’), external strategies (five items; e.g., ‘‘When I am in pain at the dentist I ask the dentist what he’s doing’’), and destructive strategies (four items; e.g., ‘‘When I am in pain at the dentist, I close my mouth’’) (Versloot et al., 2004). Cronbach’s alpha for the DCQ filled out at T1 was 0.39 (0.40, 0.30, and 0.71 for internal, external, and destructive strategies, respectively) and it was 0.41 at T2 (0.47, 0.43, and 0.78, respectively). Data Analysis All statistical analyses were performed using SPSS version 18.0 (SPSS Inc., Chicago, IL). Ten percent of the CFSS-DS questionnaires were excluded because too many items had been left unanswered. The other questionnaires were filled out completely or had one or two missing items (n ¼ 10 at both T1 and T2). Given this relatively low number, missing values were replaced by an item mean. Analyses of variance, independentsamples t tests, and paired-samples t tests were performed to test for equality of means. For nonparametric data, Kruskal-Wallis, Mann-Whitney U, Wilcoxon signed rank, and chi-square tests were used. The onesample t test was used to test equality with normative data. Linear associations were determined using Pearson and Spearman correlations. A Reliable Change (RC) index was calculated to determine whose CFSSDS score changed beyond a level that could be attributed to measurement error alone (Evans et al., 1998). For this purpose, the standard error (SE) of measurement of the difference was used. The formula is: SEdiff ¼ SD1=2 =1a, where SD1 is the SD of the baseline observations and a is the reliability of the measure (Cronbach’s coefficient alpha). It is assumed that change that exceeds 1.96 times this SE (i.e., the RC index) is unlikely to occur more than 5% of the time by unreliability of the measure alone (Evans et al., 1998). The level of significance was set at alpha ¼ .05.

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TABLE 1

Comparison of Mean CFSS-DS Scores at T1 and T2, for Different Age Groups, and to Dutch Normative Data

Age, y

Norm

N

Mean

SD*

Age, y

N

Mean

SD*

N

Mean

SD*

Mean

SD*

4–6 7–9 10–12 13–15 16–18 Total

24.6 23.6 23.4 na na

38 29 40 30 16 153

31.0 23.4 23.1 20.8 23.4 24.8

13.5ठ6.4 9.5 6.3 8.1 10.1

7–9 10–12 13–15 16–18 19–21

31 23 30 19 10 113

26.7 21.0 22.9 18.3 23.9 22.9

13.1‡ 6.0 9.3 3.5 12.8 10.0

24 21 29 19 9 102

29.5 23.6 23.5 21.6 25.2 24.7

12.9 7.0 10.7 6.4 10.6 10.2

28.6 20.9 23.1 18.3 24.8 23.2

14.2 6.1 9.4 3.5 13.3 10.3

T1*

T2*

T1 Versus T2*†

* T1 ¼ CFSS-DS scores of CL/P children at baseline; T2 ¼ CFSS-DS scores of the same CL/P children 3 years later; SD ¼ standard deviation; na ¼ not available. † T1 versus T2, mean scores based on subjects with data at both time points. Significant differences (,.05) shown in boldface. ‡ Compared to the older age groups. § Compared to normative data.

RESULTS Response The majority of the questionnaires were completed by the parents of the children (T1: 74.8%; T2: 69.0%), and the remaining questionnaires were filled out by the children themselves. At T1, 178 of the 200 participants responded (89%). The data of seven participants could not be used for analysis, due to missing demographic data. The remaining group of 171 participants consisted of 99 boys and 72 girls (mean age 9.8, SD 4.1; age did not differ between boys and girls). Of the original 200 participants, 29 addresses had become unavailable at T2. The remaining 171 participants were asked to complete the questionnaires; 118 responded (69%; 65 boys and 53 girls; mean age 13.3, SD 3.9; age did not differ between boys and girls). Dental Anxiety The comparison of anxiety levels between T1 and T2 is based on those participants who provided data at both time points (N ¼ 102; Table 1). At T1, 18 questionnaires contained too many missing (.2) data, and at T2 another 5 questionnaires were excluded because of too many missing data. At T2, children reported a significantly lower level of dental anxiety (mean 24.7, SD 10.2 versus mean 23.2, SD 10.3) compared to T1 (t(101) ¼ 2.54, P ¼ .01). These differences could mainly be explained by the declines in anxiety scores of the children in the age groups of 7 to 9 years and those of the group of children 13 to 15 years of age. Dental anxiety was significantly higher in the youngest group (4 to 6 years old) than in the other age groups. This was true for the severity of anxiety at both T1 and T2 (F(4148) ¼ 5.87, P , .01; and F(4108) ¼ 2.55, P ¼ .04) (Table 1). Parental dental anxiety remained stable between T1 and T2 (Z ¼ 0.35, P ¼ .72). Significant differences between boys and girls were found, with girls reporting a higher level of dental

anxiety than boys at both T1 (t(151) ¼ 2.09, P ¼ .04) and at T2 (t(111) ¼ 3.09, P , .01). The difference between boys and girls was most prominent in the age group of 10 to 12 years. A comparison of the CFSS-DS scores with normative data of Dutch children showed no differences in CFSSDS scores between CL/P children and children in the normative group for the 7- to 9-year-old and 10- to 12year-old children at both T1 and T2. However, the children ages 4 to 6 years reported a significantly higher level of dental anxiety at T1 than children in the normative comparison group (ten Berge et al., 2002b) (t(37) ¼ 2.92, P , .01). By T2, their dental anxiety had declined to a level equal to a normative Dutch group (Table 1). The CFSS-DS scores at T2 of all patients were subtracted from their scores at T1 to derive a difference score, which shows increased, stable, or decreased dental anxiety as determined by the RC index. Table 2 shows the percentage of patients who experienced significantly decreased, increased, or stable dental anxiety scores. Based upon RC as criterion for significant change, 13 patients (12.7%) reported a significantly lower dental anxiety score, whereas 5 patients (4.9%) had a higher level of dental trait anxiety at T2. Significant moderate negative correlations were found between the age and the CFSS-DS score of the children (T1: r ¼.31, P , .05; T2: r ¼.19, P , .05), indicating a reduction of dental anxiety with increasing age. Further, moderate, albeit significant, correlations were found between dental anxiety of parents and the CFSSDS score of their child (Spearman correlation; T1: r ¼ .24, P , .05; T2: r ¼ .32, P , .01). TABLE 2 Percentage of Children Experiencing Decreases, Stable, or Increased Dental Anxiety (CFSS-DS Scores) According to Either Their Absolute Scores or the RC Index

Absolute scores RC Index

Decreased

Stable

Increased

56.9% (1 to 17) 12.7% (8 to 17)

8.8% 82.4%

34.3% (1 to 20) 4.9% (7 to 20)

Krikken et al., LONGITUDINAL CHANGES IN DENTAL FEAR AND COPING BEHAVIOR

TABLE 3

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Percentage of Children Reporting Different Coping Strategies and Mean Number of Coping Strategies Based on the DCQ T1 When I Am in Pain at the Dentist. . .

T2

T1 Versus T2

All Boys Girls All Boys Girls (n ¼ 109) (n ¼ 62) (n ¼ 47) P* (n ¼ 92) (n ¼ 53) (n ¼ 39) P*

P*

Destructive strategies I get angry at the dentist I think of a reason to sneak out I close my mouth I get angry at mum and dad Mean number of destructive strategies used

9.2 5.5 12.8 8.3 0.36

6.5 4.8 8.1 8.1 0.27

12.8 6.4 19.1 8.5 0.47

.26 .73 .09 .93 .11

5.4 1.1 4.3 5.4 0.16

1.9 1.9 3.8 3.8 0.11

10.3 0 5.1 7.7 0.23

.08 .39 .75 .41 .14

1.00 1.00 .10 .66 .57

External strategies I ask what the dentist is doing I look at the mirror I tell the dentist It is good to have friends with me I like it when the nurse holds my hand Mean number of external strategies used

53.2 52.3 65.1 21.1 26.6 2.18

51.6 46.8 66.1 14.5 22.6 2.02

55.3 59.6 63.8 29.8 31.9 2.40

.70 .19 .80 .05 .28 .11

51.1 55.4 62.0 8.7 21.7 2.00

49.1 52.8 58.5 7.5 20.8 1.89

53.8 59.0 66.7 10.3 23.1 2.13

.65 .56 .43 .65 .79 .41

.04 .70 .71 .01 1.00 .50

Internal strategies I think of other things I tell myself it will be over soon I do what the dentist tells me I tell myself I have to do this because it is good for my teeth I think it is my own fault I have cavities I think it is part of dentistry Mean number of internal strategies used

57.8 50.5 97.2 91.7 56.0 71.6 4.25

59.7 50.0 98.4 93.5 51.6 79.0 4.32

55.3 51.1 95.7 89.4 61.7 61.7 4.15

.65 .91 .40 .43 .29 .05 .68

56.5 43.5 96.7 63.0 39.1 63.0 3.62

54.7 37.7 98.1 67.9 39.6 69.8 3.68

59.0 51.3 94.9 56.4 38.5 53.8 3.54

.68 .20 .39 .26 .91 .12 .59

.49 .58 1.00 ,.01 .02 .20 .01

6.79

6.61

7.02

.25

5.77

5.68

5.90

.87

.01

Mean number of coping strategies used * Significant differences (,.05) are shown in boldface.

Coping Only children older than 6 years filled out the DCQ. Some questionnaires were excluded because of missing values, so that not all participants had data available for this analysis (T1: n ¼ 109; T2: n ¼ 92; both time points: n ¼ 59). Participants reported that, on average, they would use 6.8 (SD 2.0) coping strategies, as indicated by the DCQ, at T1 (when they filled out the DCQ at T1) and 5.8 (SD 2.0) at T2 (when they filled out the DCQ at T2). For children who filled out the DCQ at both T1 and T2, at T2 significantly fewer coping strategies were indicated as compared to T1 (Z ¼ 2.59, P ¼ .01 [6.5 versus 5.7]). There was an increase in the number of internal coping strategies used (Table 3). No differences were found in the number of coping strategies used among the four age groups (Kruskal-Wallis test; chi-square(4) ¼ 1.78, P ¼ .78; and chi-square(4) ¼ 2.69, P ¼ .61). The three most frequently used coping strategies at T1 and T2 were ‘‘I do what the dentist tells me to do,’’ ‘‘I think it is good for my teeth,’’ and ‘‘I think it is part of dentistry.’’ These data are presented in Table 3. No significant differences were found in coping strategies between boys and girls. Dental Anxiety and Coping At T1, data were available for both the CFSS-DS and the DCQ on 103 children, and at T2 data were available for 90 children. Children were categorized as not

anxious or highly anxious using a CFSS-DS score of 32 as a cutoff (ten Berge et al., 2002b). This implies that children within the so-called borderline range were included in the highly anxious group. Highly anxious children used more coping strategies at T1 than their nonanxious counterparts. They especially used more destructive and external coping strategies and fewer internal coping strategies (Table 4). The nonanxious children used fewer internal coping strategies at T2 than at T1, and the highly anxious children used fewer external coping strategies at T2 compared to T1. To relate the RC in dental anxiety to the use of different coping strategies, each strategy was recoded (1 ¼ no such coping strategy used, 2 ¼ one or more of such coping strategies used). The latter variable was related to the changes in dental anxiety using the chi-square test. A significant association was found between the use of internal coping strategies (at T2) and the change in dental anxiety (chi-square(2) ¼ 16.99, P , .001). Inspection of the adjusted standardized residuals showed that the group that did not change in dental anxiety used internal coping strategies relatively more often, while the group that became more anxious did not use internal coping strategies at all. A significant association was also found between the use of external coping strategies (at T2) and the change in dental anxiety (chi-square(2) ¼ 6.41, P , .001). The children who became more anxious used external coping strategies relatively more often. Comparing the three groups (increased, stable, or decreased dental anxiety)

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TABLE 4

Mean Number of Coping Strategies Used Based on the DCQ

Time/Coping Strategies

LAC (n ¼ 91)*

HAC (n ¼ 12)*

P*

D*

Dþ*

P*

T1 All coping strategies External coping strategies Internal coping strategies Destructive coping strategies

6.67 2.11 4.37 0.19

7.75 2.67 3.33 1.75

,.05 ns ns ,.05

6.36 1.89 4.18 0.29

7.03 2.28 4.28 0.47

ns ns ns ns

T2 All coping strategies External coping strategies Internal coping strategies Destructive coping strategies

5.63 1.85 3.67 0.11

6.40 2.70 3.10 0.60

ns ns ns ,.05

5.38 1.83 3.49 0.06

6.14 2.11 3.70 0.32

ns ns ns ,.05

* LAC ¼ low (non)anxious children (CFSS-DS , 32); HAC ¼ highly anxious children (CFSS-DS  32); D ¼ children whose dental anxiety decreased between T1 and T2; Dþ ¼ children whose dental anxiety increased or did not change between T1 and T2; ns ¼ not significant.

with respect to the mean number of coping strategies used, with a one-way analysis of variance or its nonparametric alternative (Kruskal-Wallis), we reached identical conclusions. DISCUSSION In this prospective study, dental anxiety in children who had been surgically treated for CL/P during childhood declined over a 3-year follow up period. On average, a significant reduction in dental trait anxiety levels occurred, particularly in the youngest age group, with scores dropping to a level equal to normative scores from Dutch children. Although there was a significant reduction in the average level of dental anxiety, when RC was used as a criterion for significant change, only 13% of the children reported an actual decline in dental anxiety scores, whereas about 82% of the anxiety scores remained stable over time. About 5% of the children reported higher anxiety levels. Interestingly, these children appeared to have applied more destructive coping strategies than children whose dental anxiety declined or remained stable. Perhaps the overall decline in dental anxiety scores can be explained not only as a result of natural recovery (ten Berge et al., 2002b) but also by extinction caused by the repeated exposures to cues comprising the dental setting during the regular dental checkups the children received, as was suggested in an earlier study (Dogan et al., 2013). The latter explanation is supported by the finding that the reduction in anxiety scores could be attributed largely to lower mean scores on CFSS-DS items relating to procedures that are most frequently used during regular dental checkups of children. The present results appeared to be supportive of our hypothesis that each child’s coping style would be associated with the severity of dental anxiety. To this end, the results are in line with those of Versloot and colleagues (2004), who also found that anxious children applied coping strategies more frequently than their nonanxious counterparts. Another important finding was that, at 3 years after baseline, a significant reduction in the amount of coping

strategies used for dealing with dental pain had occurred. While the results showed a significant reduction in the mean number of external and destructive coping strategies, the number of internal coping strategies appeared to increase significantly. Apparently, children gradually learned that getting angry (destructive coping) and needing external support (external coping) are less effective in coping with pain than internal coping strategies. The decline in the need for external support may be related to the maturity level of children, in that when children grow older, they learn to manage their problems themselves without needing help (e.g., from their parents). The finding that highly anxious children and those children who became more anxious during the course of this study used more destructive coping strategies may indicate that these children are less capable of selecting the most effective coping strategy to manage their dental anxiety. In this respect, it could be argued that the anxious children became more anxious because they used ineffective coping strategies. This would be in line with earlier studies that showed that avoidance-based coping was positively related to the severity of dental anxiety (van Meurs et al., 2005; Marsac and Funk, 2008). Conversely, the opposite may also be true, in that these children used ineffective coping strategies because they were anxious. However, the first of these explanations seems more likely, as our results showed that the children whose dental anxiety increased were neither more anxious than the children whose anxiety decreased at baseline, nor did they experience more pain at the dentist. Yet it should be noted that factors other than dental anxiety might explain why some children use ineffective coping strategies and other children use more effective coping strategies. These factors might include temperament of the child and psychological conditions (e.g., autism spectrum disorders, attention deficit/hyperactivity disorder) (King and Ollendick, 1989; Versloot et al., 2004). Some limitations of the study need to be discussed. First, since children from only one center for CLP treatment participated in the study, the generalizability of the results may have been influenced. Second, specific data about the type of dental treatments to which the children had been exposed in the period between T1 and T2 were not

Krikken et al., LONGITUDINAL CHANGES IN DENTAL FEAR AND COPING BEHAVIOR

available. Thus, possible positive or negative dental experiences could not be related to the development of dental anxiety. Third, although differences were found between responders and nonresponders at T2 on the CFSS and on the DCQ measured at T1, responding parents reported significantly lower levels of dental anxiety than their nonresponding counterparts. Thus, it could have been that the dental anxiety of parents played a negative role in the compliance of children in the clinical setting of the CL/P team, including a negative response that would avoid this setting entirely. It remains uncertain how the parents’ anxiety level and their avoidance tendencies might have influenced the development of dental anxiety in these children. Fourth, the differences in mean numbers of coping strategies used were rather small. Therefore, the clinical importance of these differences remains unclear. However, a shift from one ineffective coping strategy toward one effective coping strategy might distinguish an uncooperative child from a cooperative child on the individual level and may therefore be clinically relevant. Finally, the values for internal consistency (Cronbach’s alpha) of two of the three scales (internal and external coping strategies) of the DCQ were (unacceptably) low. However, this primarily implies that the set of items within each subscale does not reflect a strict one-dimensional concept, making subscale summation scores less interpretable. Nevertheless, a count of the number of strategies used within a certain category may still yield valid information. One additional limitation of the study that needs to be mentioned is that the questionnaires, in particular the CFSSDS of the younger children, were filled out by the parents of the children. Although former studies have indicated that parents are reasonable proxy measures of their children’s dental anxiety, this might have influenced the results of this study in an uncertain way (Krikken et al., 2013). The findings from our study suggest that, for almost all CL/P children, dental anxiety, if present, gradually declines over time. The results further suggest that, whereas some coping strategies have the potential to be protective, other more destructive coping strategies may put children at greater risk for developing and maintaining dental anxiety. Future research might investigate the possibility of teaching children how to use coping strategies more effectively to guide them through dental treatment and reduce dental anxiety (or prevent an increase in anxiety). This might especially be true for vulnerable children, including some CL/P children. Acknowledgments. We thank all children and parents for participation in this study.

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