Abnormalities in Tooth Formation after Early Bisphosphonate Treatment in Children with Osteogenesis Imperfecta

Treatment with intravenous bisphosphonate (BP) in children and adolescents with osteogenesis imperfecta (OI) started in Sweden in 1991. No human studies on the role of BP therapy in development of disturbances in tooth mineralization or tooth morphology have been published. The study cohort comprised 219 individuals who were divided into four groups: group 1, BP treatment onset before 2 years of age (n = 22); group 2, BP treatment onset between 2 and 6 years of age (n = 20); group 3, BP treatment onset between 6 and 10 years of age (n = 13); and a control group of patients with OI who had not received BP therapy (n = 164). The chi-square test was used in between-group comparisons of the prevalence of tooth agenesis. The prevalence of tooth agenesis was significantly higher in children who began BP treatment before the age of 2 years (group 1; 59%,) compared to the controls (10%; p < 0.001) and to children who had begun BP therapy between ages 2 and 6 years (group 2; 10%; p = 0.009) or between ages 6 and 10 years (group 3; 8%; p = 0.003). Different types of disturbances in the enamel formation were seen in 52 premolars, where 51 were seen in those who began BP treatment before the age of 2 years. To conclude, starting BP treatment before the age of 2 years increases the risk of abnormalities in tooth formation manifesting as morphological aberrations, tooth agenesis, and enamel defects.Peer reviewe

Cost analysis of prosthetic rehabilitation in young patients with Amelogenesis imperfecta

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 &amp;lt; .001). Mean total costs were 8.5 times higher for patients with AI: (sic)16,257 +/- 5,595 (controls: (sic)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 (sic)18,475 for prosthetic rehabilitation began at age 12 years compared with (sic)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

Amelogenesis Imperfecta and Early Restorative Crown Therapy: An Interview Study with Adolescents and Young Adults on Their Experiences.

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

ReadMefile

This read me file describes the data file and supplementary file accompanying the above publication and contact for correspondenc

1) 2016-11-26 Data set of the trial

The submitted file include data from the Stop Caries Stockholm study. The database contains data on data caries, oral microorganisms and patient background factor