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. 2022 Dec;23(6):885-895.
doi: 10.1007/s40368-022-00737-3. Epub 2022 Jul 27.

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

S B Appelstrand  1 , A Robertson  2 , N Sabel  3
Affiliations

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

S B Appelstrand et al. Eur Arch Paediatr Dent. 2022 Dec.

Abstract

Purpose: Amelogenesis imperfecta (AI) is a hereditary condition which affects the composition and structure of enamel in terms of hypoplasia and/or hypomineralization. The condition severely affects patients facing such difficulties as hypersensibility, loss of tooth substance and poor aesthetics. The objective is to perform a systematic review of patient-reported outcome measures (PROMs) in patients with amelogenesis imperfecta.

Methods: Inclusion criteria were articles written in English, including PROMs from patients with amelogenesis imperfecta. The databases PubMed, Scopus and Web of Science were searched on April 27, 2022, and eligible articles were screened. Exclusion criteria were articles based on proxy reports and single case reports.

Results: 405 studies were screened in terms of title and abstract, with 31 articles eligible for full-text screening, resulting in a total of 11 articles eligible for inclusion, (articles including 4-82 patients). The content was analyzed, resulting in the outcome divided into seven domains: Oral Health-Related Quality of Life (OHRQoL), Dental fear, Esthetics, Psychosocial factors, Function, Dental hypersensitivity, and Treatment outcome.

Conclusion: The limited quantity of research on PROMS from patients with AI indicates a significant impact of OHRQoL and daily life. A large variety of approaches have been presented in the articles. Patients report concerns of esthetics, hypersensitivity, function, and a general impact on well-being and social interaction. This highlights the importance for the need of early dental treatment.

Prospero registration number: 256875.

Keywords: Amelogenesis imperfecta; Esthetics; Oral health-related quality of life; Patient-reported outcome measures; Self-esteem; Sensibility.

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Conflict of interest statement

The authors confirm no conflict of interest in relation to this manuscript.

Figures

Fig. 1
Fig. 1
PRISMA flow diagram describing the flow of identification and selection of articles from the databases toward inclusion in the systematic review. From: Page et al. (2021)
Fig. 2
Fig. 2
Search strategy in PubMed, Scopus and Web of Science Core Collection 2021-04-12, with the two combined blocks; Amelogenesis imperfecta and patient reports. The search strategy was re-ran April 2022. The updated search resulted in 38 new records and after screening 1 article was additionally included
See this image and copyright information in PMC

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