Primary failure of eruption: Clinical and genetic findings in the mixed dentition

Angle Orthod. 2018 May;88(3):275-282. doi: 10.2319/062717-430.1. Epub 2018 Jan 29.

Abstract

Objective: To test the hypothesis that mutations in the parathyroid hormone 1 receptor ( PTH1R) include effects in both primary and permanent teeth.

Materials and methods: DNA was extracted from saliva samples of 29 patients (8 familial and 21 sporadic) who presented with clinical evidence of infraoccluded teeth, and their unaffected relatives (N = 22). Sequencing followed by mutational analysis of the coding regions of PTH1R gene was completed for all individuals (N = 29).

Results: Eight of 29 cases revealed a heterozygous pathogenic variant in the PTH1R gene; five of eight variants represented distinct mutations based on comparison with the dbSNP, HGMD, and ESP databases. One mutation (c.1765 T>C p.Trp89Arg) was found to segregate within a family (n = 3). In silico analyses for all variants revealed a putative pathogenic effect. A genotype-phenotype correlation was reported as defined by a functional mutation in PTH1R and corresponding effects on one or more posterior teeth only; unilateral or bilateral involvement, infraoccluded primary teeth.

Conclusions: Novel mutations were reported in the PTH1R gene that included PFE-affected primary molars, thus providing the basis for using a genetic diagnostic tool for early diagnosis leading to proper management.

Keywords: Dental eruption; PTH1R; Primary failure of eruption (PFE).

MeSH terms

  • Adolescent
  • Adult
  • Child
  • Computer Simulation
  • DNA Mutational Analysis
  • Dentition, Mixed*
  • Early Diagnosis
  • Female
  • Heterozygote
  • Humans
  • Male
  • Middle Aged
  • Mutation*
  • Pedigree
  • Receptor, Parathyroid Hormone, Type 1 / genetics*
  • Tooth, Deciduous
  • Tooth, Unerupted / diagnosis
  • Tooth, Unerupted / genetics*
  • Young Adult

Substances

  • PTH1R protein, human
  • Receptor, Parathyroid Hormone, Type 1