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J Am Dent Assoc, Vol 136, No 5, 620-630.
© 2005 American Dental Association
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RESEARCH

The psychosocial impact of developmental dental defects in people with hereditary amelogenesis imperfecta



KRISTINA D. COFFIELD, D.D.S., M.S., CEIB PHILLIPS, M.P.H., Ph.D., MELISSA BRADY, MICHAEL W. ROBERTS, D.D.S., M.Sc.D., RONALD P. STRAUSS, D.M.D., Ph.D. and J. TIMOTHY WRIGHT, D.D.S., M.S.


   ABSTRACT
TOP
 ABSTRACT
AMELOGENESIS IMPERFECTA
 METHODS AND MATERIALS
 RESULTS
 DISCUSSION
 CONCLUSIONS
 REFERENCES
 
Background. Amelogenesis imperfecta (AI) is a hereditary dental condition with poor esthetics and dental sensitivity that frequently requires extensive dental treatment. The authors hypothesized that AI is associated with a negative psychosocial outcome.

Methods. Family members with and without AI completed a questionnaire including demographic and dental history questions, as well as a number of psychometric scales. The authors investigated the effects of AI status (with versus without), sex and age on each of the psychosocial outcomes using a generalized linear model.

Results. Subjects with AI (n = 30) had higher levels of social avoidance and distress, as well as higher levels of dysfunction, discomfort and disability attributable to their oral condition compared with subjects without AI (n = 29). The relationship of AI status to fear of negative evaluation, mastery and self-esteem was age-dependent. Younger subjects with AI tended to have higher fear of negative evaluation scores, while older subjects without AI tended to have higher fear of negative evaluation scores. Additionally, subjects without AI showed a definite decrease in mastery and self-esteem scores with age, while subjects with AI tended to show an increase in mastery and self-esteem scores with age.

Conclusions. These results indicate that having AI has a marked impact on the psychosocial health of affected people comparable with the impact of systemic health conditions, especially at younger ages.

Clinical Implications. Dental coverage for AI traditionally is excluded by third-party payers as being solely for esthetic reasons. The authors’ study shows that AI has marked psychosocial effects, which suggests that dental treatment could be medically necessary and has far-reaching implications for the affected person’s overall health.

Key Words: Amelogenesis imperfecta; psychosocial effects; fear; distress

Esthetic dentistry has become a prominent force in today’s popular culture, with treatments ranging from bleaching to porcelain veneers to "invisible" braces being commonplace. Unquestionably, a healthy, attractive smile is valued in American society. Given our current focus on esthetics, it is reasonable to speculate that deviations from "ideal" or "normal" dental or orofacial esthetics could be detrimental to a person’s psychosocial well-being. A number of investigations of craniofacial malformations (such as cleft lip with or without cleft palate) have sought to elucidate the psychosocial impact these conditions have on affected people.1–3 Generally, these people are well-adjusted psychosocially; however, they reportedly have decreased social interactions, fewer close relationships and feelings of self-consciousness regarding their facial features.1–3 For these reasons, patients with clefts should be treated early to prevent or interrupt negative psychosocial outcomes.4

Amelogenesis imperfecta has marked psychosocial effects, which suggests that dental treatment could be medically necessary.

Research has documented the effects of malocclusion and motivations for orthodontic treatment and orthognathic surgery. Malocclusion is identified as a potential threat to one’s body image and self-concept in both adolescence and adulthood.5 Many orthognathic surgery patients have primarily a self-image motivation for treatment6 and frequently report experiencing a level of psychological distress concerning their skeletal disharmony that warrants intervention.7

Given the tremendous emphasis society places on facial esthetics, we have come to question what effects an isolated hereditary developmental defect of teeth have on a person’s psychosocial health. It is essential that health care providers understand how dental health and appearance fit into the larger picture of overall health and well-being. Yet, to our knowledge, there are no data on the psychosocial influence of severe hereditary dental defects. Therefore, we undertook a potentially innovative study to provide information regarding the importance of teeth and oral esthetics in normal psychosocial development.


   AMELOGENESIS IMPERFECTA
TOP
 ABSTRACT
 AMELOGENESIS IMPERFECTA
METHODS AND MATERIALS
 RESULTS
 DISCUSSION
 CONCLUSIONS
 REFERENCES
 
To study the psychosocial effects of hereditary dental defects, we selected a population with a severe dental malady, amelogenesis imperfecta (AI), which includes no other systemic health problems. We studied a large, well-characterized population of people with AI. This provided us with a unique opportunity to examine a hereditary condition that affects primarily the dentition and thereby gives insight into the psychosocial impact of lifelong dental disabilities.

AI is a group of inherited disorders characterized by abnormal enamel formation. These non-syndromic disorders are not associated with defects of other parts of the body or any other health problem.8 Inheritance patterns for AI include autosomal dominant, recessive and X-linked modes of transmission.9 Two genes are known to be associated with AI, including the amelogenin gene and the enamelin gene. However, most forms of AI do not have a defined molecular basis at this time.10–13

The enamel defects associated with AI are highly variable and are described as hypoplastic, hypocalcified or hypomature.14,15 Hypoplastic defects represent deficiencies in the amount of enamel, characterized by thin enamel or enamel of normal thickness with pits or grooves. These teeth can have small crowns and have normal to opaque white or yellow-brown color. In contrast, hypocalcified and hypomature AI have a normal enamel thickness but poorly mineralized enamel. Hypocalcified AI is thought to result from a defect in initial crystallite formation followed by defective growth. AI involving hypomaturation is caused by a defect in final growth and maturation of enamel crystallites. In both situations, the hypomineralized enamel often abrades and chips easily, leaving exposed dentin. The enamel’s color ranges from opaque white to yellow-brown, and its surface is soft and rough. Dental sensitivity is a frequent complaint for people with these types of AI.14,15

Skeletal/dental open bite is a common finding in people with AI (25–35 percent).16–19 Open bite is more common in hypomature and hypocalcified types and is highly variable even within families. The etiology is unknown, but it may be caused by severe dental sensitivity and resultant tongue positioning, or by pleiotropic effects of the gene of major effect. Treatment can involve orthodontics or, in severe cases, orthognathic surgery.16–19

Treatment of AI depends on the individual diagnosis and phenotype. Ideally, infants with enamel defects should be referred for evaluation and early assessment before 1 year of age.20 Key clinical problems include diagnostic difficulties, esthetics, dental sensitivity and loss of vertical dimension of occlusion owing to rapid wear of the dentition. Hypoplastic AI tends to manifest itself with the least severe clinical problems. Hypoplastic enamel often is well-mineralized, making it feasible to bond the teeth with resin-based composite for protection and enhanced esthetics.21 In contrast, hypocalcified and hypomature AI tend to present complicated clinical challenges. Full-coverage restorations typically are required in lieu of intracoronal restorations to provide maximum protection, esthetics and function.21 People with severe hypocalcification or hypomaturation defects also frequently are prone to heavy formation of calculus that can cover the teeth and cause gingival inflammation. Therefore, more frequent recall appointments involving scaling may be indicated. It is critical that the gingiva be in optimal health before restorations are placed, to reduce gingival bleeding and inflammation as well as to enhance efforts to rehabilitate the patient’s dentition.10

The poor esthetics, dental sensitivity and requirement of extensive restorative treatment associated with AI led us to hypothesize that dental conditions such as these are associated with a negative psychosocial outcome. The purpose of this study was to assess self-image, quality of life and social interaction–related anxiety of people with AI when compared with people without AI.


   METHODS AND MATERIALS
TOP
 ABSTRACT
 AMELOGENESIS IMPERFECTA
 METHODS AND MATERIALS
RESULTS
 DISCUSSION
 CONCLUSIONS
 REFERENCES
 
Participant enrollment and data collection. We recruited as subjects people with AI and their unaffected family members from among people enrolled in a parent investigation studying the molecular basis of AI. All subjects with AI had either a confirmed genetic diagnosis based on DNA testing or a clinical diagnosis based on phenotype if the specific gene mutation was unknown. The research proposal was reviewed and approved by the University of North Carolina Institutional Review Board, and all participants gave additional written informed consent to participate in this psychosocial investigation before enrolling in the study.

We mailed a questionnaire to all potential participants. If the questionnaire was returned owing to an incorrect address and the correct address could not be determined, we dropped the person from the study. We sent three mailings to give the recruits multiple opportunities to respond, and one of the authors (K.D.C.) made at least one attempt to call potential subjects to inform them about the study and to answer questions they had regarding the study.

Instrument design. Multiple psychometric scales were used to measure psychosocial parameters such as social interaction anxiety, self-image and self-esteem, and self-perceived quality of life with regard to dental issues. In addition to general demographic and dental history questions, we incorporated into the questionnaire the Social Avoidance and Distress (SAD) Scale,22,23 the Fear of Negative Evaluation (FNE) Scale,22,23 the brief FNE,24 the Oral Health Impact Profile (the shortened 14-item version) (OHIP-14)25,26 and the Feelings About Yourself (FAY) scale.27–30 Validation for these instruments has been established, and the instruments have been used in other studies involving subjects aged 14 years and older.30–33

Social interaction anxiety measures. The SAD Scale was developed by Watson and Friend22 in 1969 and has been used extensively to examine the effectiveness of clinical interventions for social anxiety.22,23 It assesses both social avoidance and social distress/anxiety. The 28 items can be answered in the original true/false response format or on a Likertlike scale of 1 to 5 (1 being "not at all characteristic of me" and 5 being "extremely characteristic of me"), the format we used in this study.

Fourteen items compose the SAD-Avoidance Subscale and the other 14 items make up the SAD-Distress Subscale. Total scores for each sub-scale ranged from 14 to 70, with higher scores indicating higher levels of social avoidance and distress. Cronbach {alpha} coefficient (a measure of internal reliability) has been reported at close to 0.90.22,23

Watson and Friend22 also developed the FNE Scale to assess the degree to which people worry about how they are perceived and evaluated by others.23 It focuses primarily on people’s concerns regarding interpersonal evaluations rather than on the tendency to feel anxious per se. It has 30 items that are answered on a Likertlike scale of 1 to 5, with roughly an equal number of positively and negatively (reverse-) scored items. This produces a range of total scores from 30 to 150. Higher scores are indicative of higher levels of fear of negative evaluation. We also used scores derived from a brief-form FNE Scale24 consisting of 12 of the original FNE Scale questions. The brief FNE Scale correlates highly (r = 0.96, P < .0001) with the full-length scale and has been reported frequently in recent literature. Cronbach’s {alpha} coefficients for both FNE scales have been reported at 0.90.22–24

Self-image and self-esteem measures. The FAY Scale is a combination of the Mastery Scale27,28 and the Rosenberg Self-Esteem Scale.29,30 The Mastery Scale was designed to measure "the extent to which one regards one’s life chances as being under one’s own control in contrast to being fatalistically ruled."27,28 There are seven items (two of which are reverse-scored) that are answered on a scale from 1 to 4; therefore, total scores range from 7 to 28. Higher scores indicate higher feelings of mastery of one’s life.27,28

The Rosenberg Self-Esteem Scale measures adolescents’ global feelings of self-worth or self-acceptance. It consists of 10 items answered on a scale of 1 to 4; total scores range from 10 to 40, with higher scores indicating higher self-esteem. Cronbach’s {alpha} coefficient has been reported between 0.77 and 0.88, depending on the sample.29,30

Self-perceived quality-of-life measures. The OHIP provides a comprehensive measure of self-reported dysfunction, discomfort and disability attributable to oral conditions.25 It provides information about the "burden of illness" within populations and the effectiveness of health services in reducing that burden of illness. The shortened 14-item version (OHIP-14) was used in this study.26 It is scored in a 0-to-4 response format, with total scores ranging from 0 to 56. Higher scores indicate higher levels of dysfunction, discomfort and disability attributable to oral conditions. Cronbach’s {alpha} coefficient ranged from 0.80 to 0.90, depending on the sample.25,26

Data analysis. We investigated the effects of AI status (with versus without), sex and age on each of the psychosocial outcomes (that is, SAD subscales, FNE Scale, brief FNE Scale, Mastery Scale, Rosenberg Self-Esteem Scale and OHIP-14) using a generalized linear model (GLM).34 In the original models, we included the interactions between age and AI status and sex and AI status. We removed interactions from the model if associated P values were greater than .10 and evaluated a reduced model. For the outcomes with a significant interaction between age and AI status, we reran the explanatory models—including sex and age as main effects—separately for affected and unaffected subjects. For the outcomes with no significant interactions, we performed an analysis of covariance (ANCOVA). We compared adjusted mean scores for the affected and unaffected groups using the least squares approach for PROC GLM.34 We set significance levels at P ≤.05.


   RESULTS
TOP
 ABSTRACT
 AMELOGENESIS IMPERFECTA
 METHODS AND MATERIALS
 RESULTS
DISCUSSION
 CONCLUSIONS
 REFERENCES
 
General demographic characteristics. Overall, 68 subjects with AI and 72 subjects without AI met the study’s inclusion criteria (confirmed diagnosis, address information current, age of 14 years or older). The questionnaire response rate was 44.1 percent (n = 30) for the group with AI and 40.3 percent (n = 29) for the group without AI. Among all the participants, 55.9 percent (n = 33) belonged to the same genetically related kindred. Among the affected group, 56.7 percent (n = 17) had the same AI diagnosis (autosomal dominant hypocalcified AI).

Fewer subjects with AI (66.7 percent) were married or in a committed relationship than subjects without AI (79.3 percent). More subjects with AI (43.3 percent) than without (17.2 percent) reported having no children. Of the six subjects with AI who reported that they planned to remain childless, four (66.7 percent) said that their dental condition affected this decision. In addition, subjects with AI reported less satisfaction with their education and current employment than did subjects without AI. Fewer subjects with AI had completed high school; however, more had education beyond high school. The age ranged from 16 to 71 years in subjects with AI and 20 to 82 years in those without the condition. Despite these slight differences in age range and mean, there were significant and frequently inverse relationships between age and self-image scores, as discussed below. General demographic characteristics are displayed in Table 1Go (page 623).


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  		TABLE 1 GENERAL DEMOGRAPHIC CHARACTERISTICS OF PATIENTS WITH AND WITHOUT AMELOGENESIS IMPERFECTA (AI).

 
Self-reported dental experience. Subjects with AI reported with greater frequency the presence of various factors relating to the extent and character of their dental experience (Figure 1Go). More subjects with AI reported feeling that they get "cavities" easier than others (51.9 percent) and that they had had more than 10 dental restorations in their lifetimes (70.8 percent) compared with subjects who did not have AI (34.5 and 18.2 percent, respectively). In addition, more subjects with AI reported having dental sensitivity (82.8 percent) and being teased about their teeth (93.3 percent) than did their unaffected counterparts (44.8 and 6.9 percent, respectively). Unhappiness with color, shape and size of their teeth was more prevalent in subjects with AI (79.3 percent, 51.7 percent, and 37.9 percent) than in subjects without (32.1, 21.4 and 0 percent, respectively). Approximately the same number of subjects in each group had experienced at least one dental abscess (55.2 percent for subjects with AI versus 51.7 percent for subjects without).



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  		Figure 1. Self-reported dental experience of subjects with amelogenesis imperfecta compared with their family members who do not have the condition.

 
Social interaction anxiety measures. Subjects with AI had higher mean scores for social avoidance and distress than did subjects without the condition (Table 2Go). Neither the age by AI status interaction nor the sex by AI status interaction was statistically significant (P ≥.19) for the SAD sub-scales. An ANCOVA analysis indicated a significant difference in SAD-Avoidance (P = .02) and SAD-Distress (P = .01) scores between subjects with and without AI. Subjects with AI had higher levels of social avoidance and distress than did those without the condition when evaluated at the same mean age. Neither sex nor age had a significant effect on either subscale score in the reduced models (P ≥.17) (Table 3Go, page 626).


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  		TABLE 2 MEAN TEST SCORES BY AMELOGENESIS IMPERFECTA (AI) STATUS (WITH OR WITHOUT).

 

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  		TABLE 3 SIGNIFICANCE LEVELS OF EXPLANATORY VARIABLES.

 
The relationship between age and FNE (or brief FNE) score was significantly different for the affected and unaffected groups (P = .04 for FNE and brief FNE scores) (Table 3Go). Although we found no statistically significant relationship between age and FNE (or brief FNE) score in the separate analyses for the subjects with (P = .22 for FNE score; P = .12 for brief FNE score) and without (P = .08 for FNE score; P = .16 for brief FNE score) AI, there was a tendency for an interaction of conflict (Figure 2Go, page 627). In the group with AI, younger subjects tended to have higher FNE (or brief FNE) scores, while in the group without AI, older subjects tended to have higher FNE (or brief FNE) scores. Sex was significant in both scales, but only for the subjects without AI (P = .04 for FNE score; P = .05 for brief FNE score). Among the subjects without AI, females had higher levels of fear of negative evaluation than did males (µ = 84.7 female versus 63.6 male for FNE score; µ = 33.6 female versus 24.5 male for brief FNE score). Difference between sexes was not significant for subjects with AI on either scale (P = .50 for FNE; P = .55 for brief FNE).



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  		Figure 2. The relationship of age to scores for subjects with and without amelogenesis imperfecta (AI) on three scales. A. Fear of Negative Evaluation Scale. B. Feelings About Yourself—Mastery Subscale. C. Feelings About Yourself—Rosenberg Self-Esteem Subscale.

 
Self-image and self-esteem measures. There was a statistically significant difference in the relationship between age and Mastery score for the affected and unaffected groups (P = .0007) (Table 3Go). However, when we evaluated the AI status groups separately for main effects (age and sex), we found that the age difference was significant in the group without AI (P = .0004) but not the group with the condition (P = .55). Similar to the FNE scale and brief FNE scale, an interaction of conflict was found. Correlation analyses showed a definite negative relationship of mastery score and age for the group without AI, but a slight trend toward a positive relationship of mastery score and age for the group with AI (Figure 2Go). There was no effect of sex on mastery scores in the subjects with AI (P = .95) or the subjects without AI (P = .34).

Similarly, the interaction of age and AI status was significant in the reduced model for the Rosenberg Self-Esteem Scale (P = .02) (Table 3Go). However, when the AI status groups were evaluated separately for main effects (age and sex), age once again was significant in the group without AI (P = .02), but not the group with AI (P = .33). Another interaction of conflict existed as correlation analyses revealed a negative relationship of age to self-esteem scores in the group without AI, and a slight positive relationship of age to self-esteem scores in the group with AI (Figure 2Go). Sex had no effect on self-esteem scores in the group with AI (P = .75) or the group without the condition (P = .12).

Self-perceived quality-of-life measures. Mean scores for the OHIP-14 were markedly higher for subjects with AI (µ = 25.2) than for subjects without (µ = 4.4) (Table 2Go). Neither the age by AI status interaction nor the sex by AI status interaction was statistically significant (P ≥.26); therefore, we evaluated a reduced model (Table 3Go). An ANCOVA analysis indicated a significant difference in OHIP-14 scores between subjects with and without AI when evaluated at the same mean age (P < .0001). Subjects with AI reported higher levels of dysfunction, discomfort and disability attributable to oral conditions than did their unaffected counterparts. Figure 3Go (page 628) shows a comparison of OHIP-14 normative scores26 with OHIP-14 scores for subjects with and without AI in the percentage of subjects reporting a negative impact for each item (by answering "occasionally," "fairly often" or "very often"). There was no significant effect of sex or age on OHIP-14 scores in the reduced model (P ≥.52).



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  		Figure 3. Percentage of subjects with and without amelogenesis imperfecta (AI) who responded "Occasionally," "Fairly Often" or "Very Often" on the Oral Health Impact Profile (14-item version),25,26 compared with normative data (Slade26).

 

   DISCUSSION
TOP
 ABSTRACT
 AMELOGENESIS IMPERFECTA
 METHODS AND MATERIALS
 RESULTS
 DISCUSSION
CONCLUSIONS
 REFERENCES
 
This is the first study that has attempted to objectively characterize the psychosocial effects that a hereditary developmental defect of the dentition can have on the affected people in terms of social interaction, anxiety, self-image, self-esteem and self-perceived quality of life. Despite a relatively small sample size, we noted significant differences between subjects with AI and subjects without AI. Because 56 percent of the affected respondents had autosomal dominant hypocalcified AI with a severe clinical phenotype, the results of this study are most likely to be valid for people with severe types of AI. People who have mild forms of AI, such as the localized hypoplastic or "snowcapped" forms, very likely might not show as marked a psychosocial impact as seen in our study population.

Social interaction anxiety. Subjects with AI had significantly higher levels of social avoidance and distress than did subjects without the condition. Our mean total SAD score for subjects with AI (µ = 72.3) was slightly higher than other normative scores reported in the literature for two groups of moderately socially anxious people in one study (µ = 71.1 and µ = 62.0).35 In another study that used a true-false format instead of the Likertlike scale that we used, Chinese subjects with clefts were found to have significantly higher levels of social avoidance and distress than were their unaffected siblings and age-/sex-matched control subjects.36 People with high levels of social avoidance and distress may avoid being with or talking to others and can become upset, tense or anxious in social interactions.22 In this context, we found it interesting that fewer subjects with AI were married or in a committed relationship than were their relatives without the condition. Researchers have found that physical attractiveness relates positively to the affective quality of social experiences for both sexes.37 Taken together, these results indicate that a dental defect such as AI can mar one’s facial attractiveness and pose a threat to one’s social life.

We found that subjects with AI can have extremely high levels of fear of negative evaluation, especially at younger ages. Our mean FNE score for subjects with AI (µ = 93.2) is far greater than the mean score reported for Chinese adults with orofacial clefts (µ = 62.9).36 Females had higher levels of FNE in the Chinese cleft population, which is consistent with females in the original FNE study of healthy subjects.22 In our study, this sex difference in FNE scores was found among subjects without AI, but not those with the condition. This suggests that the presence of AI can be so overwhelming that it masks any sex difference that typically exists regarding fear of negative evaluation. Additionally, the mean brief FNE score for the subjects with AI (µ = 38.7) was greater than the published norms of 35.724 and 35.1.38 Our subjects with AI also had brief FNE scores higher than those of a population of people with psoriasis (µ = 32.6).39 Investigators found that FNE exerted a particularly strong influence for people with severe cases of psoriasis owing to their concerns about how they were perceived and their belief that they had a disease that could taint others’ impressions of them.

People with high levels of FNE try to gain social approval pre-emptively as a way of avoiding disapproval.22 These people can display apprehension about others’ evaluations or have expectations that others would evaluate them negatively.22 In our study, subjects with AI displayed higher levels of FNE, suggesting they could be overly concerned about how they are perceived on a date or a job interview or while talking to their superiors as a result of their self-consciousness regarding their oral condition. This was especially true at younger ages.

Self-image and self-esteem. Mastery has been reported to decrease as age increases, and older people usually report increasing loss of control over their surrounding world owing to poor health, decreased functional status and financial concerns.28,40,41 In our study, we found a strong negative correlation between age and mastery scale scores in subjects without AI, but subjects with AI actually showed a slight trend toward a positive relationship between age and mastery scale scores. This contradictory finding also could suggest that the presence of AI masks the typical changes in mastery that occur as people age. Contrary to our original hypothesis, older subjects with AI, on average, had higher mastery scale scores than did subjects without it. One can speculate that, though the disorder is devastating at younger ages, people with AI come to accept their condition and even rise above it as they age. Alternatively, they could have had comprehensive dental treatment that helped them cope with their condition at an older age. High Mastery Scale scores, especially as age increases, bode well for people with AI, because researchers have shown that high mastery scale scores are associated with better overall health in people with chronic disease and disability.42

Our population of subjects with AI had a very high average self-esteem score (µ = 33.9), which was only slightly lower than that of a healthy normative population (µ = 34.4).43 In addition, the subjects with AI had higher mean self-esteem scores than did subjects in another study who had facial malformations such as orofacial clefting (µ = 29.0)36 or unattractive skin conditions such as severe acne (µ = 29.0).44 Another study reported that 26 percent of young adults with chronic health conditions had low self-esteem (that is, a self-esteem score of 28 or less).45 In contrast, we found low self-esteem in only 10.3 percent of subjects with AI. While the self-esteem scores were similar for subjects with and without AI, it is of interest that younger subjects with AI had lower scores than did older subjects with the condition. The reverse was true in the unaffected population, in which self-esteem decreased with age.

Self-perceived quality of life. As we expected, we found the most significant difference in the reduced quality of life with regard to dental issues in people with AI, as measured by the OHIP-14. Unaffected subjects had similar scores to those reported in the general population.26 In contrast, more than 90 percent of subjects with AI reported feeling self-conscious, tense or embarrassed about their teeth, and 60 percent said that life had been less satisfying as a result of problems with their teeth (Figure 3Go).

Compared with people seeking implant treatment for edentulism, approximately the same percentage of subjects with AI reported feeling self-conscious (80 versus 81.6 percent) or tense (58.6 versus 55.1 percent) about their teeth. However, many more subjects with AI reported being embarrassed (70 versus 42.9 percent) or irritable (33.3 percent versus 18.4 percent) about their teeth than did those seeking implants.46 Both groups (those with AI and those seeking implants) reported feeling that life had been less satisfying as a result of their dental condition (33.3 versus 32.7 percent).46 Interestingly, the mean OHIP-14 score for our subjects with AI (µ = 25.2) was much higher than that of subjects in another study immediately after undergoing third-molar removal (µ = 18.0)47 and that of subjects in a third study who had oral lichen planus (µ = 17.1).48 These two situations are thought to produce discomfort and dysfunction that could be attributed to the person’s oral condition.

Interventions and future directions. Treatment of the underlying defect can make a substantial difference in the psychosocial functioning of people with any of various physical defects. In a study of the psychosocial effects of orthognathic surgery, researchers found that FNE and SAD scores decreased postoperatively (but only the SAD score was significant).49 Since treatment could decrease levels of social interaction–related anxiety, it is important for dental practitioners to provide timely and effective treatments that maximize function and esthetics for individuals with hereditary dental defects such as AI. However, treatment alone may not be sufficient in dealing with the psychosocial effects of various dental defects. A study evaluating the influence of a social-interaction skills workshop on a group of facially disfigured patients found significant decreases in SAD scores at six-week (P < .05) and six-month (P < .01) follow-ups.50 They concluded that these workshops can be important adjuncts to surgery as practitioners strive to provide a more comprehensive service, above and beyond that of surgical intervention, for people with disfiguring conditions.


   CONCLUSIONS
TOP
 ABSTRACT
 AMELOGENESIS IMPERFECTA
 METHODS AND MATERIALS
 RESULTS
 DISCUSSION
 CONCLUSIONS
REFERENCES
 
In our esthetics-driven society, with its high expectations for oral health and appearance, it is critical to identify and define oral characteristics that are important in the development of optimal psychosocial health. This study represents an important first step in characterizing how a specific developmental defect of teeth can influence a person’s psychosocial well-being. Although our focus is on a well-defined population with a severe developmental dental defect, this study has global implications for other populations with similar defects (such as tricho-dento-osseous syndrome, dentinogenesis imperfecta, some ectodermal dysplasias) and potentially for people with other deviations from normal teeth. This study provides new insight into the important relationship between oral and systemic health, and it raises issues related to the importance of managing oral conditions that negatively affect people’s psychosocial well-being. The findings of this study have important ramifications regarding dental insurance coverage for treatment of conditions such as AI, which typically is excluded as being not medically necessary or being solely for elective esthetic treatment.


   FOOTNOTES
 

Dr. Coffield is a resident, Department of Pediatric Dentistry, School of Dentistry, University of North Carolina, Chapel Hill.


Dr. Phillips is a professor, Department of Orthodontics, School of Dentistry, University of North Carolina, Chapel Hill.


Ms. Brady is a student, Department of Pediatric Dentistry, School of Dentistry, University of North Carolina, Chapel Hill.


Dr. Roberts is a professor, Department of Pediatric Dentistry, School of Dentistry, University of North Carolina, Chapel Hill.


Dr. Strauss is a professor and the chair, Department of Dental Ecology, School of Dentistry, University of North Carolina, Chapel Hill.


Dr. Wright is a professor and the chair, Department of Pediatric Dentistry, School of Dentistry, University of North Carolina, Chapel Hill, Campus Box 7450, Chapel Hill, N.C. 27599-7450, e-mail "tim_wright{at}dentistry.unc.edu". Address reprint requests to Dr. Wright.


The research described in this article was supported by National Institute of Dental and Craniofacial Research grant DE12879 and Maternal and Child Health grant 2T17 MC 00015-11.


The authors would like to thank Jamie DeGraw, Amy Gadol, Courtney Boyd and Debbie Price for their assistance with this project.


   REFERENCES
TOP
 ABSTRACT
 AMELOGENESIS IMPERFECTA
 METHODS AND MATERIALS
 RESULTS
 DISCUSSION
 CONCLUSIONS
 REFERENCES
 

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