Dental fear/anxiety and dental behaviour management problems in children and adolescents: a review of prevalence and concomitant psychological factors

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1 DOI: /j X x Blackwell Publishing Ltd Dental fear/anxiety and dental behaviour management problems in children and adolescents: a review of prevalence and concomitant psychological factors GUNILLA KLINGBERG 1,2 & ANDERS G. BROBERG 3 1 Department of Paediatric Dentistry, Faculty of Odontology, Malmö University, Malmö, 2 Mun-H-Center, National Orofacial Resource Centre, Göteborg, and 3 Department of Psychology, Göteborg University, Göteborg, Sweden International Journal of Paediatric Dentistry 2007; 17: Objectives. The objectives of this article were to examine the literature published from 1982 to 2006 and to evaluate prevalence of dental fear and anxiety (DFA) and dental behaviour management problems (DBMP) in children and adolescents, and their relationships to age, sex, general anxiety, temperament, and general behavioural problems. Methods. A broad search of the PubMed database was performed using three combinations of search terms. Results. A large proportion of the identified articles could not be used for the review owing to inadequate endpoints, measures or poor study design. Thirty-two papers of acceptable quality were identified and reviewed. The prevalence of both DFA and DBMP were estimated to 9%, with a decrease in prevalence with age. DFA/DBMP were more frequent in girls. DFA/DBMP were related to general fear and both internalizing and externalizing behavioural problems, although these relationships were not clear-cut. Temperament was related to both DFA and DBMP but with different temperamental characteristics, while general behavioural problems mainly correlated with DBMP. Conclusions. DFA/DBMP are common, and several psychological factors are associated with the development of these problems. In order to better understand these relationships, a number of issues concerning design of research and measurement of DFA/DBMP have to be dealt with. Introduction In 1982, Winer published a review in Child Development, a leading developmental psychology journal, regarding children s fearful behaviour in dental settings 1. The review concerned measurement of dental anxiety and uncooperative behaviours in relation to dental treatment, incidence of dental fear, age changes, and correlations to possible aetiological factors. The review found considerable evidence that positive behaviour increased between ages 3 and 6. But there were also a few studies not showing changes in dental anxiety with age, and even some studies showing an increase in dental anxiety with age. Winer suggested that anxiety in the dental setting reflected a more general and basic type of anxiety, and that aspects of Correspondence to: Gunilla Klingberg, Mun-H-Center, National Orofacial Resource Centre, Odontologen Göteborg, Medicinaregatan 12 A, SE Göteborg, Sweden. gunilla.klingberg@vgregion.se the child s emerging personality (e.g. impulse control and cognitive functions) might lead to a decline in fear in older preschoolers. He presented findings of several correlations between dental and nondental anxieties that supported the hypothesis that dental anxiety is not highly specific, and he also pointed to difficulties interpreting the relationship between dental and general anxiety 1. According to the ISI Science Citation Index, Winer s review has been cited at least 40 times, which probably underestimates the impact of the review, since the ISI system does not include all scientific journals. Mild fear and anxiety are expected experiences, consistent with normal development, but they become a concern and potentially in need of treatment when the fear or anxiety is disproportionate to the actual threat, and daily functioning becomes impaired. One of the well-accepted statements about anxiety is that it is a multidimensional construct that consists of somatic, cognitive, and emotional elements 2. Dental fear (DF) is a normal emotional reaction to one or more specific threatening stimuli in the 391

2 392 G. Klingberg & A. G. Broberg dental situation. Dental anxiety (DA) denotes a state of apprehension that something dreadful is going to happen in relation to dental treatment, and it is coupled with a sense of loosing control. Dental phobia (DP) represents a severe type of dental anxiety and is characterized by marked and persistent anxiety in relation either to clearly discernible situations/objects (e.g. drilling, injections) or to the dental situation in general. According to the Diagnostic and Statistical Manual of Mental Disorders (DSM IV) 3 published by the American Psychiatric Association, the criteria for a diagnosis of a specific phobia are (i) marked and persistent fear that is excessive and unreasonable; (ii) exposure to the phobic stimuli almost invariably provokes an immediate anxiety response; and (iii) the person recognizes that the fear is excessive or unreasonable. (Note that the third criterion may be absent in children); (iv) the phobic situation is avoided or else endured with intense anxiety or distress. In addition to the criteria for a specific phobia, the general criteria for a mental disorder, which is common to all Axis I diagnoses, also has to be met. This definition includes the criterion disability (i.e. impairment in one or more important areas of functioning). For a diagnosis of dental phobia thus, the dental fear/dental anxiety must result either in avoidance of necessary dental treatment all together or enduring treatment only with dread and in an adjusted treatment situation (e.g. specialized paediatric dentistry). The concepts dental fear, dental anxiety, and dental phobia are often used interchangeably within the dental literature. The term dental fear and anxiety (DFA) will be used throughout this paper when we refer to strong negative feelings associated with dental treatment among children and adolescents whether or not the criteria for a diagnosis of dental phobia are met. Dental behaviour management problems (DBMP i ), on the other i The term behaviour management problems is used in the developmental psychopathology literature to denote externalizing behavioural problems in general. In accordance with the distinction between general fears and dental fears, we use DBMP to refer specifically to behaviour management problems related to the dental situation. Children with DBMP may or may not have behaviour management problems in other situations (home, school, etc.). hand, are defined by the dentist s experience when treating the patient. It is a collective term for uncooperative and disruptive behaviours, which result in delay of treatment or render treatment impossible, regardless of the type of behaviour or its underlying mechanism(s) 4. Many measurement techniques have been proposed to assess DFA and DBMP: behavioural ratings, psychometric scales, physiological measures, and projective techniques. Two broad types of measurement techniques are most frequently used in research: (i) observation of the child s reaction/behaviour by dentist or other person during dental treatment (behavioural ratings) or (ii) reports of anxiety made by the child him- or herself or by the accompanying parent (most often the mother) using psychometric scales. Self-reports are most often used when studying adolescents, while parental reports are normally used with children under 13 years of age. Based on research on psychological problems in general (de Los Reyes & Kazdin 5 ), the agreement between informants (parent and child) with regard to the child s level of dental anxiety can be expected to be far from perfect. There are ongoing studies within the field of DFA looking into the relationships between parental and child ratings on the Dental Subscale of Children s Fear Survey Schedule (CFSS-DS), and so far unpublished results point in the same direction (Annika Gustafsson, personal communication, May 2007). Reports using different informants should therefore not be mixed when, for example, trying to establish prevalence rates. Behavioural ratings play a special role in general dentistry as they may provide an aid to classifying behaviour and cooperation of child patients. Ratings should have clear definitions of behaviour and should be easy to use. In the 1980s, Holst et al. published a series of reports where ratings of behaviour in more than 5000 children were performed in a public dental service setting by a large number of individual dentists 6,7. The dentists were trained to assess behaviour according to the scale described by Rud and Kisling 8, which is rather similar to the more commonly used Frankl scale 9. This was conducted as a community trial and unfortunately no data is available on properties of inter- and intra-examiner reliability. But there are several

3 Dental anxiety a review 393 other reports on high reliability, often over 0.90 (e.g. Frankl et al. 9 ). The Frankl scale 9 is probably the most frequently used behaviour rating scale. Other scales have been presented by, for example, Venham et al. 10 and Melamed et al. 11. Many studies using dental records and a retrospective design have used notes in the dental records clearly expressing severe disruptive behaviours resulting in delay in treatment or rendering the treatment impossible as the definition of DBMP 4. Several psychometric tests have been used in child populations, and their properties vary. In some cases, measures developed for adults or older adolescents [Corah Dental Anxiety Scale (DAS) and Dental Fear Scale (DFS)] 12,13 have been employed, sometimes somewhat altered (MDAS) 14. Other measures include the Venham Picture Test (VPT) 10. There is, however, one psychometric scale that is widely used in paediatric dentistry research: the Children s Fear Survey Schedule Dental Subscale (CFSS-DS), initially presented by Cuthbert and Melamed 15. It has been used in Croatia 16, Denmark 17, Finland 18, Japan 19, the Netherlands 20, Norway 21, Singapore 22, Sweden 23, and in the USA 24, and mainly in larger patient samples. It is considered to work well on a group basis, but has not been evaluated as a diagnostic tool on an individual level. In a report by Aartman et al. 25 comparing properties of different self-report measures, it was concluded that CFSS-DS was preferred as it has better psychometric properties, measures dental fear more precisely, covers more aspects of the dental situation, and since normative values are available. Furthermore, Aartman et al. 25 referred to several studies showing high test retest reliability for the CFSS-DS. Regarding validity (mostly evaluated as comparisons with behavioural ratings or congruence with other psychometric measures for child dental fear, and a few comparisons with physiological measures), there was more variation but several reports of moderate to good correlations 2. The CFSS-DS consists of 15 items scored on Likert-type scales ranging from 1 (not afraid at all) to 5 (very afraid) with as possible total score range. It was originally described in a version where young children answered the test using a fear thermometer. Today the test is mainly used in two versions: a self-report by the child him- or herself and a parental version. Using parental ratings, scores equal to or exceeding 37, 38, or 39 have been correlated to clinical assessments of DFA when comparing CFSS-DS in children with DFA (usually patients referred to specialist paediatric dentists) and non-anxious children, and thus used as cutoffs for dental anxiety 20, For the child version, 37 was reported as the cut-off in one group of fearful children 29, and based on statistics (i.e. mean score plus 1 standard deviation), 42 has also been suggested as the cut-off in the child version 22. Ten Berge et al. 30 reported a high negative predictive value, but a relatively low positive predictive value on the parental version of CFSS-DS using 39 as the cut-off when comparing with behavioural ratings in 718 Dutch children aged 4 12 years. The same comparison was used by Lee et al. 28 in to 8-year-old Taiwanese children, who reported high sensitivity and specificity on the parental version using 39 or higher as the cut-off. based mean scores on the parental version of CFSS-DS vary from 23.1 to ,20,23,28, and on the child version from 22.1 to ,18,19,22, even up to 32.7 in a low-income population 31. Later studies have shown that DFA and DBMP are likely to have multifactorial origins, and several potential aetiological factors have been proposed, including general fear 32 34, temperament 31 38, general behaviour, and attention 39,40. Pain and negative experiences from dental treatment are considered major reasons for DFA and DBMP 21, Issues related to socio-economical factors 23,44 culture, family and child-rearing 45,46, and parental dental anxiety 13,23,44 should also be recognized as influencing child behaviour during dental treatment and as being potential risk factors for the development of DFA/DBMP. In the review by Winer 1, the possible relations between dental fear and general behavioural and temperamental factors were not investigated, but since then these aspects have also attracted some researchers. The logical basis for this is to investigate if DFA/DBMP is a narrow concept, specific to the dental situation, or if it reflects more general personality and/or behavioural traits in the affected child. This research will be summarized further on in the current paper. The objectives of this review were to examine the literature published from 1982 (at the

4 394 G. Klingberg & A. G. Broberg time of the review by Winer 1 ) up to the end of 2006 and to especially evaluate literature on (i) the prevalence of DFA/DBMP in children and adolescents, and (ii) concomitant factors, i.e. relationships between DFA/DBMP and age, sex, general anxiety, temperament, and general behavioural problems. Methods A broad search of the PubMed database was performed in February 2007 using the terms shown in Box 1. The search was limited to publications in English including children aged 0 18 years, and only studies published between 1982 and 2006 were included. Relevant publications were identified after having reviewed the abstracts. Editorials and case reports, as well as publications on intervention and treatment of DFA/DBMP, were excluded, as were studies including a wide range of age groups where no specific data were available for the target age group (0 18 years). To be included, defined endpoints for DFA and DBMP had to be used, and results on the specific aetiological factors or prevalence had to be clearly retrievable within the papers. Three different literature searches were performed to cover (i) prevalence by age and gender; (ii) general fears and anxieties and general behavioural problems; and (iii) temperament. In each of these, additional exclusion criteria were used ii. The following psychometric scales were accepted as endpoints for DFA: the CFSS-DS 15, Dental Fear Scale (DFS) 13, Dental Anxiety Scale (DAS) 12, Modified Dental Anxiety Scale (MDAS) 14, and the Venham Picture Test (VPT) 10. Endpoints for DBMP were defined as behaviour ratings based on: Frankl scale 9, Rud and Kisling scale (R&K) 8, Venham Clinical Anxiety Rating Scale (VCAS) 10, and the Behaviour Profile Rating Scale (BPRS) 11. Furthermore, registrations based on dental records as described by Klingberg et al. 4 were also included as endpoints, since several epidemiological studies have used this method for compiling data. ii A complete list of excluded articles can be obtained upon request from the first author. In order to avoid double reporting, information was compiled based on only one publication if there was more than one report from the same study population. Exclusion criteria for studies on prevalence were studies including fewer than 100 subjects, studies on referred groups of patients, and studies where selection of patient materials did not have a populationbased approach. Including fewer than 50 individuals or not using well-defined measures of general fear/anxiety, general behavioural problems, or temperament was considered exclusion criteria for studies on general fear/anxiety, general behavioural problems, and temperament. Box 1. Terms used for literature search. No. Terms 1 Dental anxiety OR dental fear OR dental phobia OR dental behaviour management problem(s) 2 Prevalence 3 General fear(s) OR general anxiety(anxieties) OR anxiety disorder(s) OR SDQ OR strengths and difficulties questionnaire OR CBCL OR child behaviour checklist OR PBQ OR preschool behaviour questionnaire OR externalis(z)ing OR internalis(z)ing OR emotional problems OR ODD OR oppositional defiant disorder OR CD OR conduct disorder OR ADHD OR attention deficit hyperactivity disorder OR depression 4 Temperament OR shyness OR emotionality Results Prevalence of DFA and DBMP Combining search terms nos 1 and 2 from Box 1 revealed 135 abstracts. Forty-six relevant papers with acceptable endpoints were identified and retrieved. Based on the lists of references in these publications, an additional six papers were retrieved. In some cases two publications for the same study population had to be used in order to access sufficient information regarding prevalence as well as differences based on age and sex. After critical assessment, 15 surveyed populations and 17 publications were used for this review (Table 1). The papers were published between 1987 and 2006, and the number of individuals surveyed for DFA or DBMP varied from 223 to 4061.

5 Table 1. Prevalence (%) of dental fear and anxiety (DFA) and dental behaviour management problems (DBMP). Author, year; country Study design N Informant Dentist Holst & Crossner 6 ; SE Klingberg et al. 4 ; SE* Parent CFSS-DS Klingberg et al. 23 ; SE* ten Berge et al. 20 ; NL Wogelius et al. 17 ; DK Child CFSS-DS Raadal et al. 31 and Milgrom et al. 24 ; US Nakai et al. 19 ; JP Chellappah et al. 22 ; SG Alvesalo et al. 18 ; SF DAS/MDFS/DFS Murray et al. 33 ; CA Taani et al. 47 ; JO Bedi et al. 44 ; GB (Sc) Milgrom et al. 42 ; SG Bergius et al. 48 ; RU Thomson et al. 49 ; NZ Skaret et al. 43 ; NO, low income based cohort based cohort Age (years) Prevalence DFA % DBMP % Boys vs. girls Comparisons Younger vs. older Observed by dentist/answered by child/parent Endpoints; cut offs y: g > b o: b > g y > o O: dentist R&K 8 negative or no accept of one or more treatment steps no difference y > o O: dentist BMP-DR 4 gender and age difference based on % tot no diff. 9 11: b > g y > o A: parent CFSS-DS 15 38; gender and age difference based on CFSS-DS scores g > b no difference A: parent CFSS-DS 15 39; gender and age difference based on CFSS-DS scores no difference y > o A: parent CFSS-DS 15 38; gender and age difference based on % g > b y > o A: child CFSS-DS gender and age difference based on CFSS-DS scores Not calculated g > b no difference A: child CFSS-DS 15 gender and age difference based on CFSS-DS scores g > b A: child CFSS-DS (based on mean + 1 SD) gender difference based on CFSS-DS scores Not g > b no difference A: child Gender and age difference based on calculated CFSS-DS 15 scores g > b A: child DAS 12 13; gender difference based on DAS scores g > b A: child Modified DFS 13 high dental fear gender difference based on DFS scores g > b A: child DAS 12 15; age difference based on % no difference A: child DFS 13 60; gender difference based on DFS scores g > b A: child DAS 12 15; gender difference based on % no difference g > b A: child DAS 12 13; gender difference based on %; gender difference based on DAS scores g > b A: child DFS 13 > 59; gender difference based on DFS scores * Indicates two publications from the same study population. BMP-DR, Behaviour Management Problems retrospectively from Dental Records; CFSS-DS, Children s Fear Survey Schedule Dental Subscale; DAS Dental Anxiety Scale; DFS Dental Fear Scale; Frankl scale; R&K, Rud and Kisling; c-s, cross-sectional; tot, total group; g, girls; b, boys; y, younger; o, older; O, observed by; A, answered by. Dental anxiety a review 395

6 396 G. Klingberg & A. G. Broberg There were two reports from cohort studies 33,49, while the remaining studies were cross-sectional. There were two reports from one study population focusing on DFA in children from low-income backgrounds in the USA, reporting prevalence in one publication 24 and relationships with age and sex in the other 31. The remaining 14 populations were selected to include samples more representative of the background population. The other double reporting was a cross-sectional study from Sweden where data on DFA and DBMP were published separately 4,23. There were 15 publications on DFA from 14 populations, and the most common psychometric measure was the CFSS-DS, followed by DAS, and DFS. CFSS-DS was used in younger populations, while the other scales were used with adolescents (with the exception of a cohort study from Canada where the children were 9 years old at the first assessments 33 ). As can bee seen in Table 1, different cut-offs were used. Two studies did not report prevalence, but were included since they investigated influence of age and sex on DFA 18,19. Prevalence figures for DFA varied from 5.7% to 19.5% (12 populations) with a grand mean over all applicable studies of 11.1% (Table 1). When excluding the study on a low-income population, the mean prevalence fell to 10.3% (range ; 11 populations). The pooled prevalence (totalled numbers of individuals DFA out of totalled numbers of individuals surveyed) was 9.4% when including all 12 populations, and 8.7% when excluding the lowincome population. Five studies using CFSS- DS reported mean prevalence figures varying from 5.7% to 19.5%, with a mean of 10.3% (pooled prevalence 8.2%). When excluding the study from a low-income population 24, the mean prevalence fell to 7.8% (pooled prevalence 6.8%). Prevalence based on children s selfreports on CFSS-DS was reported in only two studies, 13.5% in 10- to14-year-olds in Singapore 22 and 19.5% in 5- to 11-year-olds from a low-income population in the USA 24 (mean 16.5%, pooled prevalence 17.3%). The mean using parental CFSS-DS reports (three populations) was 6.2% (range ; pooled prevalence 6.3%). Using children s and adolescents self-reports on DAS or DFS (seven populations), the mean prevalence rate was 11.6% (range ; pooled prevalence 11.2%). Prevalence of DBMP was reported in only two population-based studies from Sweden 4,6, 8.5% and 10%, respectively (mean 9.3%; pooled prevalence 9.6%). DFA differences by age There were six reports relating DFA to age, all using the CFSS-DS (Table 1). They were based on comparisons of mean scores in five reports, and on prevalence in one. Dental anxiety was reported to be higher in younger children compared with older children in three populations 17,23,31, while no differences were found in three The two cohort studies also reported analyses with regard to age, both using DAS 33,49. This piece of data was omitted from Table 1. The study by Murray et al. found higher mean scores on DAS in the study group at age 12 compared with at age Thomson et al. did not find a different prevalence of DFA in their study population at age 15 compared with age 18, but when comparing DAS mean scores, the younger group scored higher 49. DFA differences by sex As can bee seen in Table 1, boys and girls were compared in all 14 populations, and 10 found more dental anxiety in girls than in boys (eight based on mean scores and two based on prevalence). Two studies did not find any differences 17,42 (one based on mean scores and one on prevalence). One study reported no differences in the total group, but higher scores on CFSS-DS in boys in an older age group (9 11 years olds) 23, and one study reported no differences in prevalence but higher mean scores on DAS in girls 49. DBMP differences by age and sex The two studies on DBMP both reported higher prevalence of DBMP in younger children 4,6. One study could not establish any differences in prevalence between boys and girls. The other study reported higher prevalence of DBMP in girls in younger children, but higher prevalence in boys aged 14 or older 6 (Table 1).

7 Dental anxiety a review 397 General fear and anxiety, and general behavioural problems Literature search for papers concerning correlations between DFA/DBMP and general fear/ anxiety and general behavioural problems were made combining search terms nos 1 and 3 from Box 1 and revealed 237 abstracts. Based on the abstracts, 45 relevant papers were identified as having acceptable endpoints and were retrieved. Furthermore, based on lists of references in these papers an additional six papers were retrieved. After critical assessments, 16 papers, based on studies in 11 populations ( individuals) published between 1990 and 2004, were used for this review (Table 2). General fears As shown in Table 2, seven papers on six patient samples provided information about general fear. There were six cross-sectional studies, and one case control study. One of the cross-sectional studies concerned a low-income population 24, one was based on a convenience sample of children with no previous experience of dental treatment 51, and one on a referred sample 52. The remaining studies were population based. One population was reported in two publications: one on DFA and one on DBMP 23,34. Five studies concerned DFA and general fear, and all but one 52 reported a positive relationship between DFA and general fear. One of the studies reported an association between high levels of DFA and high general fear using the χ 2 -test. Despite this, 64% of the children with DFA had low or moderate general fear 44. Two studies reported on DBMP and general fear, with conflicting results. In a population-based study including 2257 individuals, children with DBMP had higher scores for general fear 34, while a case control study did not report any differences in general fear between children referred because of DBMP and controls in a reference group 38. Internalizing problems Seven papers on six patient samples provided information about internalizing problems (Table 2). There were five cross-sectional studies, and two case control studies. In one of the cross-sectional studies, data from a referred sample were compared with national normative data 39. The remaining three cross-sectional studies were population-based. For one population (low-income population) the results were reported in two separate publications 24,31. Five studies concerned DFA and internalizing problems, and positive relationships were reported in two populations 39,61, while no association was reported in one case control study 56. One population was surveyed using CBCL and reported a weak but statistically significant correlation in one publication 31, but could not verify a relationship in terms of odds ratio in another publication 24. One population displayed an association between DFA and state anxiety but not with trait anxiety using odds ratios 22. There was only one study on DBMP 38, which reported children referred because of DBMP to have more internalizing problems than controls in a reference group 38. Externalizing problems Seven papers on five patient samples provided information about externalizing problems (Table 2). There were three samples studied in cross-sectional studies (four papers), and two samples studied in case control studies (three papers). In one cross-sectional study, data from a referred sample were compared with national normative data 39, and for one population (lowincome population) the results were reported in two separate publications 24,31. The remaining two cross-sectional studies were population based. In addition, one of the case control studies was reported in two separate publications 40,63. There were two case control studies focusing on neuropsychiatric problems 40,63. The first, which included children screened positive for attention problems, found higher frequencies of DBMP in the group screened positive compared with controls 40. The second study included subjects from the first study who had been diagnosed as having ADHD and also reported more DBMP (not shown in Table 2) 63. This study could, however, not find any differences concerning DFA in children with ADHD compared with controls 63. Of the remaining study populations, three concerned DFA (four publications) and one

8 Table 2. Relationships between general fears/anxieties/general behavioural problems and DFA or DBMP. Author, year; country Study design N Age (years) DFA DBMP Measures for General fears/internalizing/ Externalizing answered by child or parent Endpoint; cut-offs DFA/BMP answered by child/parent General fears Klingberg et al. 23 * c-s based correlation r = 0.48 CFSS-SF 23 CFSS-DS 15 ; 38; parent Klingberg et al. 34 *; SE c-s based Multiple regression Higher scores Parent BMP-DR 4 ; dentist R 2 = for CFSS-SF Milgrom et al. 24 c-s based OR 1.8 CFSS 50 ; parent CFSS-DS 15 : 40; child Low income population Arnrup et al. 38 ; SE case control Referrrals + reference group No difference CFSS-SF 23 ; parent BMP-DR 4 ; dentist Folayan et al. 51 ; NG c-s Convenience sample no correlation r = 0.58 CFSS-SF 23 ; child CFSS-DS 15 ; child previous dental treatment Townend et al. 52 ; GB (Sc) c-s Referred sample anxious 29 non-anxious 7 14 No difference FSS-II mod 53 ; child DAS 12 with pictures + item on inj; child 3 unsure class Bedi et al. 44 ; GB (Sc) c-s based DAS 15 associated GFS mod 18 items 54 ; child DAS 12 ; 15; child with high general fears CI 11 45%; 64% w DAS 15 had low or moderate fear Internalizing ten Berge et al. 39 ; NL c-s Referrals fearf consec comp Higher scores CBCL 55 ; parent Based on referral w normative values Raadal et al. 31 and c-s based correlation r = 0.34 CBCL 69 ; parent or guardian CFSS-DS 15 : 40; child Milgrom et al. 24 ; US c-s based OR, NS Low-income population Alwin et al. 56 ; GB (Eng) case control Referred samples 65 fearful 6 18 No difference CMAS 57 ; child VPT 10 ; child 42 controls + parent Arnrup et al. 38 ; SE case control Referrrals + reference group More internal Rutter 58 ; parent BMP-DR 4 ; dentist PBQ 59 ; parent Chellappah et al. 22 ; SG c-s based OR 2.8 state anxiety OR NS trait anxiety STAI-C 60 ; child CFSS-DS 15 ; 42; child Locker et al. 61 ; NZ c-s based OR 3.0 agoraphobia OR 2.9 social phobia OR 3.0 simple phobia Externalizing ten Berge et al. 39 ; NL c-s Referrals fearf consec comp w normative values DIS 62 patient DAS 12 15; patient Higher scores CBCL 55 ; parent Based on referral Raadal et al. 31 and c-s based correlation r = 0.14 CBCL 55 ; parent or guardian CFSS-DS 15 : 40; child Milgrom et al. 24 ; US c-s based OR, NS Low-income population Arnrup et al. 38 ; SE case control Referrrals + reference group More external Rutter 71 ; parent BMP-DR 4 ; dentist No difference in attention PBQ 72 ; parent Blomqvist et al. 40 and case control Suspected ADHD + controls More BMP Conner s 10-item 64 ; BMP-DR 4 ; dentist Parent Blomqvist et al. 63 ; SE case control ADHD + controls No difference DSM IV 3 for ADHD; medical doctor CFSS-DS 15 ; parent Locker et al. 50 ; NZ c-s based OR 5.0 conduct disorder DIS 62 ; patient OR 2.8 alcohol depend DAS 12 15; patient * Indicates two publications from the same study population. BMP-DR, Behavioural Management Problems retrospectively from Dental Records; CBCL, Child behaviour Checklist; CFSS, Children s Fear Survey Schedule; CFSS-DS, Children s Fear Survey Schedule Dental Subscale; CFSS-SF, Children s Fear Survey Schedule Short Form; CMAS, Children s Manifest Anxiety Scale; Conner s 10-item; DAS, Dental Anxiety Scale; DIS, Diagnostic Interview Schedule; DSM IV, Diagnostic and Statistical Manual of Mental Disorders, 4th edn; FSS-II mod, Fear Survey Schedule II modified; GFS, Geer Fear Scale; PBQ, Preschool Behaviour Questionnaire; Rutter s Child Scale; STAI-C, State-Trait Anxiety Inventory for Children; VPT, Venham Picture Test; comp w, compared with; CI, confidence interval; OR, odds ratio; NS, not statistically significant; ini, injection. 398 G. Klingberg & A. G. Broberg

9 Dental anxiety a review 399 DBMP. Relationships between DFA and externalizing problems were reported in two studies 39,61. Again, one population was surveyed using CBCL and reported a weak but statistically significant correlation between DFA and externalizing problems in one publication 31, but could not verify a relationship in terms of odds ratio in another publication 24. The study on DBMP reported children referred because of DBMP to be more externalizing than controls in a reference group, while no difference was found regarding attention 38. Temperament A literature search for papers about associations between DFA/DBMP and temperamental factors were made combining search terms nos 1 and 4 from Box 1 and it revealed 10 abstracts. Based on the abstracts, eight relevant papers were identified and retrieved. Furthermore, based on lists of references in the retrieved papers, an additional four papers were retrieved. After critical assessment, seven studies (seven different study samples) were identified for the review, including one study using qualitative research methods comprising 14 informants 72 (Table 3). The seven papers were published between 1990 and 2006, and all used parental ratings of temperament. There were two crosssectional studies based on convenient samples (consecutive patients). Furthermore, there was one cohort study, three case control studies, and one study using a qualitative research method (content analysis). The numbers of individuals included in the studies using quantitative research methods varied from 50 to 203. Four studies reported on relationships with DFA, and six on relationships with DBMP (Table 3). Three of the studies included analyses reported on both DFA and DBMP. Three out of four studies that analysed DFA pointed to the importance of shyness, and negative emotionality in relation to DFA 36,37,71, while one study could not establish any correlations 69. The studies on DBMP all reported relationships with the children s adaptability, intensity, activity, and shyness. In the qualitative study, the mothers of children with DBMP identified temperament as an aetiological factor and mentioned shyness, aggressiveness, dependence, and difficult behaviour as examples 72. Discussion What can be concluded from this review? First, it indicates that DFA and DBMP are relatively common encounters in the dental setting, affecting approximately 9% of children in normal populations in Australia, Canada, Europe, and the USA. Differences between studies are possibly due to a mix of differences related to culture, study design, sampling methods, and measures of DFA and DBMP. DBMP seems to decline with age, while the relationship between age and DFA is more complicated. Both DFA and DBMP seem to be more frequent in girls and to be related to general fear and internalizing and externalizing behavioural problems even though these relationships are not clear-cut. Temperamental factors are related to both DFA and DBMP but with different temperamental characteristics, while general behaviour problems mainly correlate with DBMP. In this review, focus has been on psychological aspects of DBMP and DFA. Thus, the relationships solely reflect nondental aetiological factors. Several studies have reported the importance of dental factors, such as pain or perceived lack of control during treatment, for the development of DFA and DBMP 21, These relationships were, however, outside the scope of this review. The review was based on a number of literature searches using PubMed and the selection of papers to review was based on abstracts. Thus, there could be papers that should have been included that were not retrieved because their abstracts not giving enough information. Still, all retrieved papers were also searched for relevant references and new studies were included this way in the process of evaluation. In order to enable evaluation and comparisons, the studies had to provide information in relation to well-defined endpoints, as well as valid measurements of factors studied. In total, 32 papers were analysed, which is not a high number indicating a need for more well-designed studies with sufficient numbers of subjects using measures shown to be valid and reliable for the age-groups studied. The distinction between DFA and DBMP is important. Dentists are likely to identify DBMP more easily than DFA. Most studies on referred

10 Table 3. Studies on temperament in relation to dental anxiety and behaviour management problems. Author, year; country Study design N Quinonez et al. 36 ; CA Arnrup et al. 38 ; SE case control Klingberg & Broberg 37 ; SE c-s Consecutive 55 GA pat DBMP, low age, extensive treatment needs Referrals randomly selected + consecutive controls 86 DBMP 117 controls case control based 65 DFA 81 non-dfa Radis et al. 67 ; US c-s Consecutive 50 initial dental examination Age (year) DFA DBMP 2 5 Corr w CMAS (trait anx); emotionality r = 0.53 shyness r = 0.30 activity r = 0.23; sociability r = 0.46 Shyness discriminated for DBMP 4 12 Negative emotionality, impulsivity discriminated; 4 subgroups with difference temp and behavioural profiles could be identified 5 12 DFA associated with shyness and negative emotionality DBMP associated with activity months Temp easy, low, intermediate, and difficult predicted behaviour. Approach/withdrawal predicted quite in 3 years; intensity, activity predicted crying behaviour in 5-years-olds. Liddell 69 ; CA cohort based 179 x = 12 No corr. DBMP: activity r = 0.20 mood r = 0.24 task orientation r = 0.22 ten Berge et al. 71 ; NL de Oliveira et al. 72 ; BRI case control structured interviews qualitative study using content analysis Referrals + selected controls 67 high DFA 56 low DFA % indicative temp factors as reason for DFA; shy temp id by parents. Temperament measure; Informant child/parent EAS 65 ; parent EAS 65 ; parent Endpoint DFA/BMP Observed by dentist/ Answered by child/parent CMAS 57 ; parent BPRS 11 ; staff EAS 65 ; parent BMP-DR 4 EAS 65 ; parent EAS 65 ; parent BSQ 68 ; parent DOTS-R 70 ; parent Interviews with parents; parent 14 DBMP 4 14 DBMP: child temp id as aetiological factor: fearful, shy, dependent aggressive, difficult Interviews with mothers CFSS-DS 15 ; parent CDFP 66 ; child BMP-DR 4 ; dentist OSUBRS 78 ; staff DAS 12 ; child Upset behaviour during treatment; parent CFSS-DS 15 ; parent Interviews; parent BMP-DR, Behavioural Management Problems retrospectively from Dental Records; BPRS, Medical Behaviour Profile Rating Scale; BSQ, Behaviour Style Questionnaire; CDFP, Children s Dental Fear Picture test; CFSS- DS, Children s Fear Survey Schedule Dental Subscale; CMAS, Children s Manifest Anxiety Scale; DAS, Dental Anxiety Scale; DOTS-R, Revised Dimensions of Temperament Survey; EAS, Emotionality, Activity, Sociability (Impulsivity) temperamental survey; OSUBRS, Ohio State University behaviour Rating Scale; GA pat, patient scheduled for treatment under general anaesthesia; id, identified. 400 G. Klingberg & A. G. Broberg

11 Dental anxiety a review 401 samples are also based on DBMP discriminating referred patients from those not being referred. Still, these studies frequently assume that they are investigating DFA. This is a methodological question of concern. If designing a case control study on DFA, the sample selection probably has to be preceded by screening a larger population for DFA instead of including patients referred because of uncooperative behaviours. More than 10 years ago, it was shown that DFA and DBMP overlap only partially with 27% of children with DBMP also showing DFA and 61% of children with DFA also showing DBMP 34. More recent studies using cluster analyses have shown that children referred to specialist paediatric dentist because of DBMP form a heterogeneous group with DFA being only part of the problem 38,73. The most used measure CFSS-DS was designed for use with children up to 14 years of age, and several large population-based studies have provided normative data from different populations 17,18,20,23. This measure is problematic especially with regard to unsatisfactory validation of cut-offs, and lack of investigated level of congruence between child and parental versions. DAS and DFS were developed for adult populations and they mirror the dental treatment encounter from an adult perspective. There are no studies exploring how these measures function with children or adolescents; still they have been used in children as young as 9 (DAS) and 12 (DFS) years in two studies included in this review, which is far from ideal 33,47. Several studies have employed a retrospective design using dental records where DBMP has been defined based on notes in the records clearly describing behavioural problems resulting in delay of treatment or rendering treatment impossible. This type of registration is almost certainly biased. One can argue that this methodology tends to under-report rather than overreport DBMP on the basis that it probably takes quite a high degree of DBMP for it to be registered. In addition, there is a risk for another systematic bias using this methodology as the dentist could be influenced also by factors outside of the dental situation when recording DBMP. An inexperienced dentist will encounter more DBMP than a more experienced colleague. Furthermore, the experienced dentist will often sense the risk of DBMP and may take extra precautions to avoid the problems, or refrain from giving treatment in order to avoid DBMP. This is of course not ideal from an oral health point of view. This, however, pinpoints the importance of dental health professionals knowledge and understanding of psychological aspects of dental care for children. If a child does not cooperate, we as professionals must ask ourselves why this child cannot cooperate, and what we can do to help her or him. Children want to do well, but some children do not have the capacity. Thus, strictly speaking DBMP is not a quality of the child, but of the relationship between dentist and child. Still, DBMP is usually the reason for referral to specialist paediatric dental care, and DBMP in patients with dental caries constitute up to 37% of all referrals 74. It also underscores the importance of using valid and reliable measures of the child s ability and willingness to comply (i.e. cope with the different demands of the dental situation). Prevalence of DFA appears to vary between different studies. There could be several reasons for this. This review identified three measures of DFA of which two were designed for adults 12,13. The younger study populations were mostly surveyed using the CFSS-DS, while DAS and DFS were used with older children and adolescents. Based on the reviewed studies, it would be fair to surmise that approximately 9% of children and adolescents exhibit DFA. Even though studies using different measures are not comparable, the included studies give some support to a suggested decrease in prevalence of DFA with age. Still, there were three studies not showing this decrease, and one cohort study where DFA actually increased between 9 and 12 years of age. Regarding DBMP there were only two studies, and both showed a clear decline in DBMP with age. These findings are partly different from the results presented by Winer in He reported a decline in DBMP in young preschool children but also a more complex picture in older children and adolescents and even an increase in dental anxiety at an older age. This pattern may reflect differences in definitions. The findings in the earlier age groups could represent dental fear, which is the normal reaction to novel

12 402 G. Klingberg & A. G. Broberg stimuli. Fear is a normal reaction for young children in novel situations 75, and uncooperative behaviours can be viewed as adequate reactions for young children in situations where they experience lack of control, pain, or meet a nonfamiliar person that does not pay attention to or respect the child. A decrease in DFA and DBMP with age may thus reflect normal psychological development. The young child s understanding of dental treatment differs from an older child s perception, and younger children have fewer experiences from dental care implying more novel and fearprovoking situations. Pain, discomfort, and anxiety are abstract phenomena and coping with them requires advanced cognitive skills as well as a capacity for effortful control and emotion regulation that the young child is not yet mature enough to fully manage. In the present review, there was a clear trend with girls being both more dentally anxious and also presenting more DBMP as compared with boys, a finding that is more in line with our current knowledge regarding sex differences with regard to fearfulness and anxiety disorders in children and adolescents 76. This is in contrast with the review from 1982 where there was no clear relationship between DFA and sex 1. The present review found some evidence of a relationship between DFA and general fear and anxiety, which is consistent with Winer 1. Specific fears change as the child grows older in a developmentally meaningful manner (i.e. from separation anxiety in 1-year-olds to fears of the dark and of animals in the preschool age to social anxieties in school-aged children and adolescents) 77. Young children are likely to have more fears, and probably experience higher levels of fear than older children 75. General fearfulness (or trait anxiety) must be distinguished from fears in general (i.e. different specific but common fears) and both can manifest themselves as state anxiety (i.e. an immediate fear reaction). This distinction is not always made when relating dental fear to general fear(s). Another problem is that parents are often used as proxy for the child and, furthermore, that responses to psychometric measures regarding dental fear and fears in general are collected: (i) from the same informant (usually the accompanying parent), (ii) at the same time, and (iii) in the same situation (the dentist s waiting room). This maximizes the risk for a halo effect, inflating the relationship between dental fear and fears in general. The present review gives some support to a relationship between general behavioural problems and both DBMP and DFA, areas that were not covered in Winer s review. Children at risk of developing internalizing disorders (anxiety, depression, psychosomatic problems, etc.) tend to score high on measures of DFA, while the picture is more complicated with regard to DBMP, due to the heterogeneity of children classified as having DBMP. For some children, the origin of their DBMP is due primarily to previous negative experiences with dental care. These children need not have either internalizing or externalizing problems in general. Other children are probably genetically prone to react with fear and anxiety to a variety of threatening situations, and among them we expect to see a relationship between DBMP and internalizing disorders. Still, other children react to a variety of frustrating external demands (e.g. sitting still in the dentist s chair) with anger and oppositional tendencies. Among them we expect to see a relationship between DBMP and externalizing disorders (Oppositional Defiant Disorder, Conduct Disorder). It is also likely that children with neuropsychiatric disorders may present with DBMP as part of their diagnosis. Children with attention difficulties of various sorts, and difficulties adjusting their activity level to situational demands have more difficulties complying with the dental situation. In this review two studies actually looked at children with ADHD, but it is likely that children with problems within the neuropsychiatric spectrum are included in many other studies especially those on referred patients. In many cases a neuropsychiatric diagnosis has not yet been established, or is not known to the dentist. Since neuropsychiatric disorders constitute a substantial group of diagnoses, affecting up to 5% of the child population 78, it is likely that dentists frequently meet children and adolescents with ADHD, autism, Asperger syndrome, or Tourette syndrome. Thus, the dental teams may meet and treat several children who have not yet received a child psychiatric diagnosis, but present with such problems

13 Dental anxiety a review 403 and need special care to be able to comply with the demands of the dental situation. There was clear evidence of relationships between temperament and DFA and DBMP. As with internalizing problems, DFA was more connected with temperamental traits like shyness, inhibition, and negative emotionality, while DBMP was more often associated with activity and impulsivity. Temperament, which was not part of Winer s review 1, has become an increasingly important concept in developmental psychology and psychopathology. With the growth of developmental psychopathology as a discipline, a renewed interest in the transaction between the child s inborn tendencies (e.g. temperament, neurological vulnerability) and the caregiving environment has followed. Over the last decade the interest in temperament in terms of reactivity (i.e. how quickly and intense different parts of the nervous system reacts to external stimuli) has been supplemented with an interest in temperamental aspects of the regulation of affect and behaviour (i.e. how well the child is able to regulate his or her activated nervous system). Concepts such as emotional regulation and effortful control are used to tap these temperamental constructs. Future studies of temperament in relation to DFA and DBMP should also include measures of the aspects of temperament in order to better understand how and why children s temperament affect behaviour in the dentist s office. To conclude, first of all, despite the fact that two scales are approaching gold standard when measuring dental fear in children (CFSS-DS) and adolescents (DAS), none of them are sufficiently validated for this purpose. Cut-off scores are not differentiated with regard to children s age or sex. Even more disturbingly they are not differentiated with regard to informant (child s self-report or report by the accompanying parent), despite extensive research in clinical child psychology and developmental psychopathology showing that the correlation between child and parental ratings of the child s internalizing problems is much too low (typically ) to make them interchangeable 79,80. We cannot come close to establishing the incidence or prevalence of clinically relevant dental anxiety (i.e. a level that hampers the child s capacity to endure treatment whether in ordinary dental treatment or when referred to a specialist) in girls and boys of different ages until the groundwork of validating at least one measure of dental fear in relation to children s age and gender has been done. Second, most studies in this review addressing the issue of prevalence were not based on large enough population-based samples. The field needs to move on from convenience to representative samples if we are to understand potential differences in prevalence between cultures and within cultures over time and across dental practises. It is important to distinguish between DFA and DBMP when designing studies. If the aim is to study DFA, a referred patient sample is not ideal as most referrals are based on DBMP. Third, future studies of the aetiology and concomitants of DFA and DBMP must be theory-driven to a much higher extent than has hitherto been the case. Most aspects of children s behaviour in the dentist s office (attachment, temperamental reactivity, emotional regulation, effortful control, internalizing and externalizing psychopathology to name only a few) are core aspects of clinical child psychology, and consequently theories and measures developed within that field have a strong potential to enrich studies of DFA and DBMP. Children and adolescents represent a huge variation in age, competence, maturity, personality, intellectual capacity, temperament and emotions, experience, oral health, family background, culture, etc. All these aspects influence the child s ability to cope with dental treatment. Some children are robust and tolerant in stressful situations and are not likely to present problems to the treating dentist, while others are vulnerable and may need more attention and time in order to feel at ease and to cooperate to dental treatment. Due to their genetic vulnerability a small minority of children will always remain a great challenge to every dentist. It is important that dentists acknowledge that uncooperative children are not difficult children. Instead they have a personality, a special need, or a disability that requires both knowledge and special attention from the dentist, something that a dentist working with children and adolescents must be prepared to offer. Incorporating knowledge from developmental and clinical child psychology as