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المؤلفDoaa, Taqi
المؤلفMoussa, Hanan
المؤلفSchwinghamer, Timothy
المؤلفVieira, Alexandre Rezende
المؤلفDagdeviren, Didem
المؤلفRetrouvey, Jean-Marc
المؤلفRauch, Frank
المؤلفTamimi, Faleh
تاريخ الإتاحة2021-09-30T09:20:09Z
تاريخ النشر2021-09-30
اسم المنشورBone
المعرّفhttp://dx.doi.org/10.1016/j.bone.2021.116011
الاقتباسTaqi 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
الرقم المعياري الدولي للكتاب87563282
معرّف المصادر الموحدhttps://www.sciencedirect.com/science/article/pii/S8756328221001733
معرّف المصادر الموحدhttp://hdl.handle.net/10576/23583
الملخصIntroductionOsteogenesis 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.
راعي المشروعNational Institutes of Health. Grant Number: 1U54AR068069-0.
اللغةen
الناشرElsevier
الموضوعOsteogenesis imperfecta
Rare disease
Missing teeth
Agenesis
Unerupted
Retained teeth
Tooth development
Tooth eruption
Bisphosphonate
العنوانMissing and unerupted teeth in osteogenesis imperfecta
النوعArticle
رقم المجلد150
dc.accessType Open Access


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