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OBJECTIVE: To summarize studies published between 2017 and 2023 examining the clinical diagnosis and restorative management of amelogenesis imperfecta (AI) in children and adolescents. DATA: The review incorporated publications on clinical diagnosis, patient-reported outcomes, clinical trials, cohort studies, and case reports that included individuals below 19 years of age with non-syndromic AI. SOURCES: A literature search was conducted across electronic databases, PubMed, Web of Science, and CINAHL, including papers published between 2017 and 2023. The search yielded 335 unique results, of which 38 were eligible for inclusion. RESULTS: New evidence on the genetic background of AI makes it now advisable to recommend genetic testing to supplement a clinical AI diagnosis. The discussions of the dental profession and the public on social media do not always incorporate recent scientific evidence. Interview studies are finding that the impact of AI on quality of life is more severe than previously appreciated. New evidence suggests that single-tooth ceramic crowns should be the first choice of treatment. Due to incomplete reporting, case reports have been of limited value. CONCLUSION: In young patients with AI symptoms of pain and hypersensitivity decreased, and aesthetics were improved following all types of restorative therapy. Resin composite restorations were mainly performed in cases with hypoplastic AI and mild symptoms. Single tooth ceramic crown restorations have a high success rate in all types of AI and can be used in young individuals with AI. CLINICAL SIGNIFICANCE: Prosthetic rehabilitation in adolescents with severe AI is cost effective, improves esthetics, reduces tooth sensitivity, and improves oral health-related quality of life.
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Amelogénesis Imperfecta , Calidad de Vida , Humanos , Amelogénesis Imperfecta/terapia , Amelogénesis Imperfecta/diagnóstico , Adolescente , Niño , Coronas , Restauración Dental Permanente/métodos , Estética Dental , Resinas Compuestas , Pruebas GenéticasRESUMEN
OBJECTIVES: Children and adolescents with amelogenesis imperfecta (AI) have extensive treatment needs, but costs have not been explored previously. We calculated the costs of prosthetic rehabilitation and analyzed whether costs of treatment begun in early adolescence are lower compared with in early adulthood. METHODS: Data from the dental records of 25 patients with AI and 25 age- and sex-matched controls were analyzed. Patients were followed for a mean period of 12.3 ± 1.5 years. Number of dental visits, dental treatment costs, and indirect costs were calculated during three time periods, before, during and after prosthetic therapy. Swedish national reference prices for general and specialist dental care were used. RESULTS: The mean number of visits was significantly higher in the AI group: 43.3 ± 19.7 (controls: 17.5 ± 19.8; p < .001). Mean total costs were 8.5 times higher for patients with AI: 16,257 ± 5,595 (controls: 1,904 ± 993). Mean number of crowns made in patients with AI was 12 ± 7 (range 1 - 31). Indirect costs were significantly higher in the AI group and constituted 22% of the total costs. After crown therapy, costs between groups ceased to differ significantly. Calculations of total costs in the hypothetical scenario (discount rate at 3% annually) were 18,475 for prosthetic rehabilitation began at age 12 years compared with 20,227 if treatment began at 20 years of age. CONCLUSIONS: Prosthetic rehabilitation costs for children and adolescents with AI can be high. Early crown therapy is associated with lower costs and a lower number of dental visits. CLINICAL IMPLICATIONS: Early crown therapy in children and adolescents with severe AI is a cost-reducing treatment associated with few complications and reduced need of dental care during adolescence compared with treatment begun at age 20.
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Amelogénesis Imperfecta , Adolescente , Adulto , Amelogénesis Imperfecta/complicaciones , Niño , Costos y Análisis de Costo , Coronas , Humanos , Suecia , Adulto JovenRESUMEN
Amelogenesis imperfecta (AI) is a hereditary developmental disorder affecting the enamel of teeth. Affected patients present with tooth hypersensitivity, rapid tooth wear, or fractures of enamel as well as alterations in color and shape, all of which compromise esthetic appearance and masticatory function. Chronic conditions in childhood severely impact the whole family, affecting normal family routines and/or increasing the family's financial burden. The aim of this study was to explore experiences and the impact on daily life of being a parent to a child with severe forms of amelogenesis imperfecta. Parents of children and adolescents with AI participated in an interview with a psychologist. The transcribed interviews were analyzed using thematic analysis. The parents talked about several concerns about having a child with AI. Four main themes emerged from the interviews: Feelings associated with passing on a hereditary disorder, knowledge decreases stress, unfamiliarity with the diagnosis, and psychosocial stress. In these main categories we identified several subthemes. Feelings associated with passing on a hereditary disorder included the subtheme of guilt/shame; knowledge decreases stress included knowledge about diagnosis in the family and support from dental health care professionals; Unfamiliarity with diagnosis included missed diagnosis, fear of not getting correct treatment, and insufficient pain control; finally, the subtheme Psychosocial stress included fear of child being bullied and emergency dental visits. The findings show that parents of children with severe amelogenesis imperfecta report similar experiences as do parents of children with other chronic and rare diseases.
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Patients with Amelogenesis imperfecta (AI) can present with rapid tooth loss or fractures of enamel as well as alterations in enamel thickness, color, and shape; factors that may compromise aesthetic appearance and masticatory function. The aim was to explore the experiences and perceptions of adolescents and young adults living with AI and receiving early prosthetic therapy. Seven patients with severe AI aged 16 to 23 years who underwent porcelain crown therapy participated in one-to-one individual interviews. The interviews followed a topic guide consisting of open-ended questions related to experiences of having AI. Transcripts from the interviews were analyzed using thematic analysis. The analysis process identified three main themes: Disturbances in daily life, Managing disturbances, and Normalization of daily life. These themes explain the experiences of patients living with enamel disturbances caused by AI and receiving early crown therapy. Experiences include severe pain and sensitivity problems, feelings of embarrassment, and dealing with dental staff that lack knowledge and understanding of their condition. The patients described ways to manage their disturbances and to reduce pain when eating or drinking, and strategies for meeting other people. After definitive treatment with porcelain crown therapy, they described feeling like a normal patient. In conclusion the results showed that adolescents and young adults describe a profound effect of AI on several aspects of their daily life.
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Amelogénesis Imperfecta/cirugía , Coronas , Satisfacción del Paciente , Adolescente , Esmalte Dental , Femenino , Humanos , Entrevistas como Asunto , Masculino , Resultado del Tratamiento , Adulto JovenRESUMEN
BACKGROUND: Amelogenesis imperfecta (AI) is a rare, genetically determined defect in enamel mineralization associated with poor esthetics and dental sensitivity. Because the condition is associated with negative social outcomes, this study evaluated oral health-related quality of life (OHRQoL), dental fear, and dental beliefs before and after early prosthetic crown therapy for AI during adolescence. METHODS: The study included 69 patients with AI, aged 6-25 yr: 33 males and 36 females (mean age 14.5 ± 4.3); healthy controls (n = 80), patients with cleft lip and palate (CLP; n = 30), and patients with molar incisor hypomineralization (MIH; n = 39). All matched in age and gender, and all but the CLP group insocioeconomic area. Patients completed three questionnaires measuring OHRQoL (OHIP-14), dental fear (CFSS-DS), and dental beliefs (DBS-R). Twenty-six patients with severe AI between ages 9 and 22 yr received crown therapy and completed the questionnaires twice: before and after therapy. RESULTS: OHIP-14 scores were significantly higher among patients with AI (7.0 ± 6.7), MIH (6.8 ± 7.6) and CLP (13.6 ± 12.1) than healthy controls (1.4 ± 2.4) (p < 0.001). After crown therapy, quality of life problems in the 26 patients with severe AI decreased significantly, from 7.8 ± 6.1 to 3.0 ± 4.8 (p < 0.001). Early prosthetic therapy did not increase dental fear or negative attitudes toward dental treatment. CONCLUSIONS: OHRQoL increased after early crown therapy in patients with severe AI. Therapy did not increase dental fear or negative attitudes toward dental treatment.