Osteogenesis imperfecta tooth level phenotype analysis: Cross-sectional study.
Connective tissue
Dentin
Dentinogenesis
Oral medicine
Osteogenesis Imperfecta
Tooth abnormalities
Journal
Bone
ISSN: 1873-2763
Titre abrégé: Bone
Pays: United States
ID NLM: 8504048
Informations de publication
Date de publication:
06 2021
06 2021
Historique:
received:
06
11
2020
revised:
16
02
2021
accepted:
11
03
2021
pubmed:
21
3
2021
medline:
10
7
2021
entrez:
20
3
2021
Statut:
ppublish
Résumé
Dental anomalies in Osteogenesis imperfecta (OI), such as tooth discoloration, pulp obliteration (calcified dental pulp space), and taurodontism (enlarged dental pulp space) vary between and within patients. To better understand the associations and variations in these anomalies, a cross-sectional study was designed to analyze the dental phenotype in OI patients at the individual tooth type. A cohort of 171 individuals with OI type I, III and IV, aged 3-55 years, were recruited and evaluated for tooth discoloration, pulp obliteration, and taurodontism at the individual tooth level, using intraoral photographs and panoramic radiographs. Genetic variants were identified in 154 of the participants. Patients with Helical α1 and α2 glycine substitutions presented the highest prevalence of tooth discoloration, while those with α1 Haploinsufficiency had the lowest (<10%). C-propeptide variants did not cause discoloration but resulted in the highest pulp obliteration prevalence (~%20). The prevalence of tooth discoloration and pulp obliteration was higher in OI types III and IV and increased with age. Tooth discoloration was mainly observed in teeth known to have thinner enamel (i.e. lower anterior), while pulp obliteration was most prevalent in the first molars. A significant association was observed between pulp obliteration and tooth discoloration, and both were associated with a lack of occlusal contact. Taurodontism was only found in permanent teeth and affected mostly first molars, and its prevalence decreased with age. The dental phenotype evaluation at the tooth level revealed that different genetic variants and associated clinical phenotypes affect each tooth type differently, and genetic variants are better predictors of the dental phenotype than the type of OI. Our results also suggest that tooth discoloration is most likely an optical phenomenon inversely proportional to enamel thickness, and highly associated with pulp obliteration. In turn, pulp obliteration is proportional to patient age, it is associated with malocclusion and likely related to immature progressive dentin deposition. Taurodontism is an isolated phenomenon that is probably associated with delayed pulpal maturation.
Identifiants
pubmed: 33741542
pii: S8756-3282(21)00079-X
doi: 10.1016/j.bone.2021.115917
pmc: PMC8278321
mid: NIHMS1689306
pii:
doi:
Types de publication
Journal Article
Research Support, N.I.H., Extramural
Langues
eng
Sous-ensembles de citation
IM
Pagination
115917Subventions
Organisme : NIAMS NIH HHS
ID : U54 AR068069
Pays : United States
Investigateurs
Brendan Lee
(B)
V Reid Sutton
(VR)
Sandesh C S Nagamani
(SCS)
Francis Glorieux
(F)
Janice Lee
(J)
Paul Esposito
(P)
Maegen Wallace
(M)
Michael Bober
(M)
David Eyre
(D)
Danielle Gomez
(D)
Gerald Harris
(G)
Tracy Hart
(T)
Mahim Jain
(M)
Deborah Krakow
(D)
Jeffrey Krischer
(J)
Eric Orwoll
(E)
Lindsey Nicol
(L)
Cathleen Raggio
(C)
Peter Smith
(P)
Laura Tosi
(L)
Informations de copyright
Copyright © 2021. Published by Elsevier Inc.
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