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AuthorDoaa, Taqi
AuthorMoussa, Hanan
AuthorSchwinghamer, Timothy
AuthorVieira, Alexandre Rezende
AuthorDagdeviren, Didem
AuthorRetrouvey, Jean-Marc
AuthorRauch, Frank
AuthorTamimi, Faleh
Available date2021-09-30T09:20:09Z
Publication Date2021-09-30
Publication NameBone
Identifierhttp://dx.doi.org/10.1016/j.bone.2021.116011
CitationTaqi D, Moussa H, Schwinghamer T, Vieira AR, Dagdeviren D, Retrouvey JM, Rauch F, Tamimi F; Members of the BBDC. Missing and unerupted teeth in osteogenesis imperfecta. Bone. 2021 Sep;150:116011. doi: 10.1016/j.bone.2021.116011
ISSN87563282
URIhttps://www.sciencedirect.com/science/article/pii/S8756328221001733
URIhttp://hdl.handle.net/10576/23583
AbstractIntroductionOsteogenesis imperfecta (OI) is a genetic disorder characterized by bone fragility and craniofacial and dental abnormalities such as congenitally missing teeth and teeth that failed to erupt which are believed to be doubled in OI patients than normal populations and were associated with low oral health quality of life. However, the etiology of these abnormalities remains unclear. To understand the factors influencing missing and unerupted teeth, we investigated their prevalence in a cohort of OI patients as a function of the clinical phenotype (OI type), the genetic variant type, the tooth type and the onset of bisphosphonate treatment. MethodA total of 144 OI patients were recruited from The Shriners Hospital, Montreal, Canada, between 2016 and 2017. Patients were evaluated using intraoral photographs and panoramic radiographs. Missing teeth were evaluated in all patients, and unerupted teeth were assessed only in patients ≥15 years old (n = 82). ResultsOn average, each OI patient had 2.4 missing teeth and 0.8 unerupted teeth, and the most common missing and unerupted teeth were the premolars and the upper second molars, respectively. These phenomena were more prominent in OI type III and IV than in OI type I, and were not sex or age-related. Missing teeth were significantly more common in patients with C-propeptide variants than all other variants (p-value <0.05). Unerupted teeth were significantly more common in patients with α1 and α2 glycine variants or substitutions than in those with haploinsufficiency variants. Early-onset of bisphosphonate treatment would significantly increase the risk of unerupted teeth in patients with OI types III and IV (OR = 1.68, 95% CI (1.15–1.53)). ConclusionThe prevalence of missing and unerupted teeth at the tooth type level in OI patients varies according to the nature of the collagen variants and the OI type. These findings highlight the role of collagen in tooth development and eruption.
SponsorNational Institutes of Health. Grant Number: 1U54AR068069-0.
Languageen
PublisherElsevier
SubjectOsteogenesis imperfecta
Rare disease
Missing teeth
Agenesis
Unerupted
Retained teeth
Tooth development
Tooth eruption
Bisphosphonate
TitleMissing and unerupted teeth in osteogenesis imperfecta
TypeArticle
Volume Number150
dc.accessType Open Access


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