Introduction

Over the last decade, there has been growing appreciation of the relationship between oral health status and children’s quality of life or well-being. This has led to a wealth of studies exploring the impact of dental disease, dentoalveolar trauma, malocclusion, and dental or craniofacial anomalies on both oral health-related and general health-related quality of life [1–6]. Furthermore, greater emphasis is now being placed on young patients’ perspectives and experiences of dental care and their participation in decision-making [7]. It is speculated that changes in how children are viewed and engaged in dentistry may underpin and facilitate future enamel defect-related research and service evaluation.

Research into the impacts of DDE may be driven by different agendas. Firstly, at the population level, there has been a need to establish whether DDE constitute a public health concern in the ongoing debate about the risks/benefits of water fluoridation. Indeed, the majority of psychosocial DDE-related research to date has been conducted in areas of natural or artificial water fluoridation in both developed and developing countries. Secondly, at the patient level, clinicians need to fully understand the impacts of DDE for each individual in order to better meet their needs and treatment expectations.

The media and celebrity industry also play a key role in shaping how society views and values appearance. Many children are growing up in a world where unrealistic body images are portrayed as desirable. A “perfect” smile of straight and artificially white teeth is inherent to this globalized representation of beauty. There is little tolerance for tooth color that deviates from the norm, placing a greater psychosocial impact on the individual and presenting more complex clinical and ethical challenges for the dentist.

Child-Centered Approaches to Research and Service Evaluation

A variety of methods, both qualitative and quantitative, may be used to engage children in research about their oral health and experiences of treatment [8]. Oral health-related quality of life (OHRQoL) questionnaires have dominated quantitative research in this field. The most frequently used child measures include the age-specific Child Perceptions Questionnaires (CPQ11–14 and CPQ8–10) [9–11], the Child Oral Health Impact Profile (Child OHIP) [12], and the Child Oral Impacts on Daily Performances (Child OIDP) Questionnaire [13]. It should be borne in mind that these instruments are generic OHRQoL measures, and although they have been used with children with DDE [1, 14–16], they are not specific for enamel conditions. Studies have also employed proxy (parent) assessments of child OHRQoL in relation to DDE [2], but these will not be reviewed in this chapter as the focus is on children’s self-reported impacts. Multidisciplinary research has also examined the relationship between the impact of DDE and various psychological constructs such as coping styles or self-esteem [17, 18].

In view of the recognized limitations of questionnaires in capturing the full spectrum of an individual’s experiences, qualitative methods have been used to gain a more detailed insight into the impacts of DDE. Common approaches have included interviews, focus groups, and written diaries [16, 19–22], but there is considerable scope to utilize more diverse participatory activities such as video diaries, photo-elicitation, creative writing, drawings, and role play.

Psychosocial Impacts of Enamel Defects on the Individual

Background

As mentioned previously, both qualitative and quantitative approaches have been employed to determine the impacts of DDE on children, with greatest insights gained from studies which have incorporated a mixed method approach. A key finding to emerge from the literature is that there is no clear relationship between the severity of the condition, as measured by normative data (e.g., epidemiological indices of enamel defects), and the impact of DDE from the child’s own perspective [23, 24]. This may be attributed to the complex interplay between individual characteristics such as coping strategies and concepts of the self (e.g., self-esteem and self-confidence) which may all modify a person’s response to having a visible difference [20, 24–26]. One area which remains completely un-researched is whether the etiology of the DDE, inherited or environmental, has any bearing on the reported impacts. The prevalence of DDE in the general population has, however, been considered as a modifying factor on how children feel about their teeth. Interestingly, there are conflicting reports as to whether children living in areas where DDE are the “norm” (regions of endemic dental fluorosis) actually experience any less negative impacts relating to social interactions and satisfaction with their appearance [27–29]. The following sections will review some of the key studies which have described or measured impacts in young populations relating to a variety of DDE.

Impacts Relating to Dental Fluorosis

In the main, the psychosocial literature has focussed on children with DDE on permanent teeth attributed to dental fluorosis [28] and less is known about how conditions such as amelogenesis imperfecta (AI), molar incisor hypomineralization (MIH), or trauma-related sequelae affect individuals.

One of the earliest studies to consider the psychosocial impact of severe dental fluorosis was conducted with 13- to 15-year-olds living in Tanzania [27]. The main finding was that 70 % of respondents said that the way their teeth looked hindered them from smiling freely. Interestingly, the negative impact of DDE on smiling has been a common theme throughout the literature.

A more detailed investigation was later carried out in Tanzania, in an area where 75 % of the population had a Thylstrup-Fejerskov Index (TFI) score of ≥2 [14]. To appreciate the appearance of teeth which may be classified as having a TFI of 4 or 9, see Figs. 7.1 and 7.4, respectively. A sample of 478 young people, with a mean age of 15.7 years, completed a modified version of OIDP. They were asked how often they had experienced problems with their mouth or teeth in relation to seven daily performances: eating, speaking and pronouncing clearly, cleaning teeth, sleeping and relaxing, smiling without embarrassment, maintaining emotional state, and enjoying contact with people. Significantly more females than males reported themselves to be affected on at least one daily performance. Overall, young people who reported either oral impacts on their daily performance and/or discoloration of upper anterior teeth were more likely to be dissatisfied with both their oral condition and their dental appearance.

Fig. 7.1
figure 1

Photograph of teeth of a participant with development enamel defects who said: “I always wanted to change my teeth, they are like multi-coloured… I’m loads bothered” (girl, aged 14)

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A similar study in Brazil also investigated the impact of fluorosis using a modified version of OIDP, this time with 513 schoolchildren aged 6–15 years [15]. No differences in impact on daily activities (eating, speech, oral hygiene, sleep, smiling, emotional stability, studying, and socializing) were found between those normatively assessed as having fluorosis or not. Robinson and coworkers [30] issued the CPQ11–14 to a sample of 174 12-year-old Ugandan children, 24 % of whom had dental fluorosis. They reported that more participants with socially noticeable fluorosis (TFI>2) had impacts on their OHRQoL than those with mild or no fluorosis, although the difference was not significant.

Studies in developed countries, where any fluorosis is likely to be less severe, have suggested that there is no impact on OHRQoL [1, 31]. Indeed, a study of 8- to 13-year-old Australian children found that those with a TFI of 2 had better OHRQoL than their peers with normal enamel [1]. However, it was not clear whether this could be attributed to the “whiteness” of affected teeth or reduced caries incidence.

Impacts Relating to Amelogenesis Imperfecta

Although there have been comparatively few studies on the psychosocial aspects of AI [32], it is evident that this condition has far greater impact than dental fluorosis. This is not surprising in view of the often severe functional problems, relating to dental hypersensitivity and tooth tissue loss, which may accompany compromised aesthetics. The first, and landmark, study of the psychosocial impacts of AI was conducted with a mainly adult population, although a few adolescent participants were included [19]. Interestingly, social avoidance and distress were found to be greatest among younger patients. More recently, a mixed method study involving children from a UK dental hospital has made a significant contribution to our understanding of how AI affects younger people [16]. In-depth interviews were conducted with seven children, aged 13–16 years. A further 40 AI patients, aged 10–16 years, completed the CPQ11–14 as well as some additional questions generated from the previous interviews. Participants reported impacts relating to aesthetics as well as function. Children did not like to show their teeth, especially for photographs and in social settings.

I don’t like smiling with my teeth because I don’t like them (girl, aged 13)

When all my friends are talking I’d want to join in but don’t want to show my teeth (girl, aged 16)

For some children, tooth sensitivity was actually more of an issue than aesthetics.

It is the sensitivity more than the colour, the colour doesn’t bother me, it’s more the sensitivity (girl, aged 15)

If there was no problem with sensitivity, I’d drink faster and bite down on ice lollies and not cringe when I think of it (girl, aged 13)

Impacts Relating to DDE of Unknown Etiology

The only study to utilize in-depth interviews to explore the impact of DDE was conducted in the UK with populations receiving either a non-fluoridated or fluoridated (1 ppm) water supply [23]. A theoretical framework, based on symbolic interactionism was used to guide and interpret the qualitative data. Twenty-one 10- to 15-year-old young people were interviewed, with TFI scores ranging from 0 to 5. The impact of DDE was frequently described in terms of how much dental appearance “bothered” the individual. Photographs of the teeth of two participants have been included to illustrate the clinical severity of the DDE.

I don’t like the colour, I’m conscious about it, when I am talking I don’t like showing them…. I’m actually quite bothered (boy, aged 15)

I always wanted to change my teeth, they are like multi-coloured… I’m loads bothered (girl, aged 14) (Fig. 7.1)

I’ve got some marks on the side of these two teeth, but they don’t bother me (girl, aged 13)

Interestingly, DDE impacted most on individuals whose sense of self was defined by appearance and who depended on perceived approval from others about appearance. No association was found between gender, age, severity of DDE, and the reported impacts. However, this research was the first to identify that a sense of self may explain variation in the impact of DDE on young people. The following selected quotes illustrate how DDE were found to impact variably on the participants, in terms of how they felt about their own dental appearance.

I don’t want to look at myself in the mirror ‘cos I know I’ll look horrible…. (girl, aged 14)

I’m happy with the way I am, it doesn’t really matter what you look like, its personality. (girl, aged 12) (Fig. 7.2)

Fig. 7.2
figure 2

Photograph of teeth of a participant with developmental enamel defects who said: “I’m happy with the way I am, it doesn’t really matter what you look like, its personality” (girl, aged 12)

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Another important finding was the degree to which children were teased or questioned about the marks on their teeth, particularly when new people were encountered. Again, there was considerable variation in how teasing and name-calling impacted on young people, which may have related to how much importance the individual placed on their appearance as a whole.

I don’t’ like the thing on my tooth and I don’t like my big ears. Whenever I get into an argument with someone that’s the first thing they go on about (boy, aged 11)

When people are being nasty they always say things about my teeth, I don’t really care (girl, aged 12)

Whenever I go on holiday or when I meet some new friends I don’t know they say “I don’t mean to be bad, but I think you’ve got something on your teeth there”… They thought I wasn’t brushing them and stuff… (boy, aged 11)

Teasing and Bullying

Teasing, name-calling, and bullying are a common occurrence in childhood and adolescence. Lovegrove and Rumsey [26] found that over half of their sample of 654 British 11- to 14-year-olds had encountered appearance-related teasing or bullying. In a study of children with AI, 50 % reported being teased about their teeth [16]. This figure concurs with findings from an earlier study of children with a range of DDE, where 56 % stated that they had been subject to unkind remarks about their teeth [22]. Although Marshman and colleagues [23] found variation in how teasing and name-calling impacted on young people, for some young people, being subject to teasing may be the motivation for seeking cosmetic treatment, and the clinician needs to be sensitive to such issues. However, young patients may be self-conscious and not always forthcoming about this matter, especially when meeting a dentist they do not know well. An empathetic approach is clearly needed, and the dentist should be able to direct patients to available support services, such as online anti-bullying resources (www.kidscape.org.uk).

The Impacts of Treatment for Enamel Defects

Dental Anxiety

The management of DDE encompasses a wide range of treatment regimens, which broadly aim to preserve tooth structure, prevent dental caries, reduce symptoms such as thermal hypersensitivity, and improve dental appearance. Some children face a lifetime of dental interventions in order to maintain their compromised dentition, and this may present considerable demands in terms of treatment compliance and costs for affected individuals and their families. Jälevik and Klingberg [33] highlighted the impacts of repeated treatment episodes for 9-year-old Swedish children with molar incisor hypomineralization (MIH). They found that children with MIH were more dentally anxious and were more likely to exhibit clinical behavior management problems than children in a control group. Interestingly, their longitudinal findings for the same group of children, 9 years later, revealed that although patients with MIH had poorer oral health (a significantly higher DMFT), they were no longer more dentally anxious then their non-MIH controls [34]. Clinical impressions suggest that children with AI may also be more dentally anxious than their peers [35], but this has yet to be evidenced by research findings. It is hypothesized that because these children frequently experience and anticipate oral pain from normally innocuous thermal, mechanical, and osmochemical stimuli, they understandably become more dentally anxious.

Psychosocial Outcomes

Patient-reported outcome measures are becoming more recognized within pediatric healthcare [36] but have yet to be well developed within dentistry. A recent Cochrane systematic review sought to compare clinical-reported and patient-reported outcomes (satisfaction with esthetics and sensitivity) following restorative care of children with AI [37]. Unfortunately, no studies met the inclusion criteria, but it was encouraging to see that the authors had recognized the need to seek children’s views on what treatment outcomes were important to them.

To date, only one study appears to have considered the impact of treatment on young people with DDE [22]. This service evaluation sought the views of 67 young people, aged 7–16 years, who had received microabrasion and/or composite restorations on their upper incisors for a variety of visible enamel defects. Participants completed a simple 10-item questionnaire which was developed with service users. There was also a free text box for patients to write further comments. Prior to any intervention, children reported high levels of worry and embarrassment. However, following treatment, children said they felt happier and were more confident.

I am a lot happier now, people don’t pick on me (girl, aged 10)

I cannot fault my treatment which has made me gain some confidence, which has helped me in this difficult year of exams (girl, aged 16)

Interestingly, the study also revealed unmet treatment expectations as some young people reported disappointment that their teeth “weren’t perfect” after interventions. This finding clearly highlights the need for clinicians to communicate effectively and manage patient expectations more realistically at the outset of treatment. The media and celebrity culture may be influencing children to desire an unnaturally white and uniform dentition, which presents clinicians with ethical, financial, and clinical concerns.

I was looking forward for seeing my teeth completely white but they were not completely white. It looked better than it was but they should have said that it wasn’t going to do all my teeth white (boy, aged 14)

It is encouraging to see that a more recent study, conducted with children with AI, did seek young patients’ expectations at the outset of treatment [16]. The authors asked 40 children what was the single most important thing they wanted from their course of treatment? The majority (63 %) stated that they hoped for an improvement in the color of their teeth, and 18 % said they wanted a better smile. For 10 %, a reduction in tooth sensitivity was the most important outcome.

Timing of Interventions

Another consideration is how the timing of any dental treatment, particularly for largely esthetic interventions, impacts on the individual. Clinical anecdote suggests that some children, who have been previously unconcerned about their dental appearance, are prompted to request treatment because of a significant social event (e.g., being a bridesmaid at a wedding) or change in their circumstances.

One study explored the experiences of 11- to 12-year-old children with a variety of visible facial and dental differences, including DDE, during their transition to secondary (high) school [17, 20, 22]. A 2-week diary was developed with children and incorporated both open and closed questions with space to include drawings. Children also completed the CPQ11–14. Participants discussed a variety of aspects about the transition to secondary school including concerns about their appearance in their new social environment.

I had like brown marks on the front two teeth, I didn’t know what they were going to say about them…. it was a worry before I went to big school. (girl, aged 11)

Some children discussed their oral conditions such as cleft lip and how they dealt with questions from peers about it, while others reported having sought treatment to improve the appearance of enamel defects prior to starting secondary school (Fig. 7.3).

Fig. 7.3
figure 3

Extract from the diary of 11-year-old girl who had undergone microabrasion of her incisal opacities prior to moving to her new school

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It should be noted, however, that heightened appearance-related concerns were not limited to oral conditions as some children stated that they had tried to lose weight, have their hair cut, or not to wear glasses prior to the transition. This enquiry demonstrated how impacts from DDE may be more or less important to a young person at different times during their life course, and dental care professionals should be sensitive to these issues when planning courses of treatment.

Another important consideration, relating to the provision of dental treatment, is how this may impact on a child’s school attendance and social activities. The need for frequent dental visits, often at a specialist practice some distance from home, is likely to have a social impact on the child and their family. However, our review of the literature failed to reveal any studies in this area.

In general, the evidence base is lacking to support the psychosocial benefits of DDE-related treatment for young people. This should be a priority for future research as it becomes increasingly important that treatments are justified and evaluated in terms of patient benefit. Longitudinal studies are paramount to consider both the short- and long-term psychosocial effects of different dental interventions and thereby develop and safeguard services for young people with DDE.

Social Judgments in Relation to Enamel Defects

Appearance-Related Judgments

There is a wealth of literature to support the association between an individual’s appearance, especially facial appearance, and how they are perceived by others [38]. A fascinating study by Dion showed that children who were considered less attractive than other children would be viewed as more likely by an adult to have committed a misdemeanor and were viewed as less honest [39]. The seminal work by Langlois and colleagues [40] also demonstrated that attractive children were rated more positively for a number of personal attributes than those considered unattractive. Regrettably, we live in a society where appearance does matter, and those who look different may elicit negative judgments which have important social consequences, including even future job prospects.

Children’s Views of Other Children with Enamel Defects

The first study to consider how a child’s dental appearance influenced how their peers and adults viewed them was conducted by Shaw [41]. Children, depicted in photographs, as having well-aligned teeth and normal facial appearance were judged to be better looking, more desirable as friends, more intelligent, and less likely to behave aggressively than children with a malocclusion or cleft lip.

More recently, a similar methodology was employed to determine whether, or not, young people made value judgments, or ascribed certain social attributes, to other young people with visible enamel defects [21]. Focus groups were first conducted with children aged 11 to 16 years to identify what terminology they used, or judgments they made, in relation to photographs of teeth with a range of DDE. Two common themes emerged from these discussions with participants perceiving that people with DDE were “lazy” or “did not care about their appearance” (Fig. 7.4).

Fig. 7.4
figure 4

Children’s judgments relating to this photograph of a child with severe dental fluorosis included: [this person] “has no personal hygiene,” “has eaten too much syrup,” and “is lazy”

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The investigators then recruited 547 school children, aged 11–12 and 14–15 years, to rate full-face photographs of a boy and girl using a social attribute questionnaire that had been previously developed with young people. The 11 social attributes included: kind, rude, clever, honest, cares about appearance, careful, lazy, confident, helpful, stupid, and naughty. Half the participants were shown photographs of the boy and girl with normal teeth, and half were shown digitally modified photographs of the same children with a visible enamel defect affecting one upper incisor. The key finding was that young people did make negative social judgments on the basis of DDE. Age and socioeconomic status did not have an effect on raters’ scores, but girls were more positive in their assessment than boys. Affected individuals may be unfairly judged by others which may, in turn, impact on their self-esteem, social interactions, and life prospects.

Summary and Clinical Implications

This brief review of the literature, albeit an emerging literature, suggests that having a DDE can impact on children and young people in a variety of ways. Generic oral health-related quality of life questionnaires have provided conflicting findings, but qualitative enquires have given a richer and more meaningful insight into the range and severity of psychosocial impacts. Appearance-related impacts are foremost with some children reporting embarrassment, upset, and reluctance to smile or show their teeth in normal social interactions. In addition, some children may experience dental hypersensitivity as well as compromised esthetics, which are likely to have negative impacts on daily activities. For young patients requiring extensive dental treatment, there is the potential to develop dental anxiety. Studies have also shown that some children with DDE are subject to dental appearance-related teasing and may incur negative social judgments from their peers. Most of the research to date has been conducted on the impact of DDE in the permanent dentition with little knowledge about the nature or severity of the impact of DDE on younger children.

Some individuals with a visible difference cope better than others but the reasons for their resilience have not been fully elucidated. This observation is supported by the finding that there is no simple correlation between the severity of the enamel defect and the resultant impact. Further research, in collaboration with social science experts, is indicated to explore these complex and dynamic relationships, throughout childhood and adolescence.

The “take-home” message for clinicians is that children and young people with DDE require an empathetic and insightful approach. Every effort should be made to provide timely and aesthetic care, which addresses the young patient’s individual concerns and circumstances. However, unrealistic treatment expectations may be encountered, and these need to be met with good communication and appropriate support. It is important that robust pediatric patient-reported outcome measures are developed for future use, so that we can be sure that our treatment is actually helping to reduce the psychosocial impacts of having an enamel defect.