Patient-reported outcome measure for children and young people with amelogenesis imperfecta

Abstract

Background Amelogenesis imperfecta (AI) is a genetic enamel defect that can affect both the primary and permanent dentition. It has a range of clinical phenotypes, and children and young people often present with challenging oral health needs. Patient-reported outcome measures (PROMs) can identify key patient concerns. Methods This was a multi-centre service evaluation across several specialist paediatric dentistry services in the UK. A PROM questionnaire was created with clinician and patient input, through peer review with the national AI Clinical Excellence Network, as well as piloting the PROM with ten children and young people with AI. The final PROM questionnaire was distributed to all patients with AI attending each unit between January and March 2020. Results Sixty children and young people (aged 5-17 years) across four specialist units participated, with 72% reporting that they 'often' or 'sometimes' experienced pain or sensitivity and 76% reporting that they 'often' or 'sometimes' felt unhappy with the way their teeth look. Of the patients who were post-treatment, 81% indicated that they were happy with their teeth, compared to just 41% of patients who were mid-treatment and 33% of patients who were pre-treatment. Conclusion Children and young people with AI experience a range of issues related to their function and psychosocial wellbeing. This simple PROM demonstrates the range of issues this group of patients face, and could be used to monitor an individual's progress to ensure that treatment is planned to address the patient's individual concerns and needs.

Patient-reported outcome measures for children and adolescents having dental bleaching in the UK

“…Furthermore, bleaching-related sensitivity in children and adolescents seems to be reduced when compared to adults (Donly et al 2005). It is also of note that as a baseline, 72% of young people with a diagnosis of AI report experiencing pain or sensitivity from their teeth (Lyne et al 2021). As AI was the most common diagnosis leading to dental bleaching in this study, it must be remembered that they are likely to have had a degree of baseline sensitivity prior to bleaching.…”

Patient-reported outcome measures in individuals with amelogenesis imperfecta: a systematic review

“…Patients reported experiences of teasing due to their dental appearance (Coffield et al 2005 ; Lyne et al 2021 ). Through interviews, many of the participants reported a fear of the teeth falling apart and a feeling of embarrassment over their appearance (Pousette Lundgren et al 2016 ).…”

“…Before prosthetic treatment, patients were dissatisfied with their teeth with the discolouration of teeth being a major factor (Lindunger and Smedberg 2005 ). One article, Lyne et al ( 2021 ) used an AI-specific questionnaire and found appearance and confidence to smile as the most common concerns (76%).…”

“…Social interaction anxiety, measured by Social Anxiety Disorder (SAD) and Fear of Negative Evaluation (FNE), showed patients with AI scoring higher, indicating more social avoidance and higher social distress (Coffield et al 2005 ). Few patients reported teasing from peers, but not necessarily linked to the individuals perception of appearance (Lyne et al 2021 ).…”

“…More than 70% of the patients who answered an AI-questionnaire, reported to experience pain or hypersensitivity “often” or “sometimes” Lyne et al ( 2021 ). The dental hypersensitivity was found to be higher in patients with AI compared to the control group (Lundgren and Dahllof 2014 ; Quandalle et al 2020 ).…”