INTRODUCTION: The primary aim of this study was to assess the amount and long-term stability of orthodontically created bone in patients with agenesis of maxillary lateral incisors after canine distalization. The secondary aim was to explore the impact of patient age on the process of alveolar bone resorption. METHODS: A group of patients with agenesis of the maxillary permanent lateral incisor was examined at 4 time points: the beginning of orthodontic treatment (T1, n = 80), the end of treatment (T2, n = 80), 2-5 years after treatment (T3, n = 79), and 12-15 years after treatment (T4, n = 32). The width of the edentulous alveolar bone was measured from study casts at the level of the bone ridge (point A) and 5 mm apically from the alveolar ridge (point B). Alveolar ridge height was also recorded using panoramic radiographs at all time points. Paired t tests, 2-sample t tests, Friedman test with Bonferroni correction, Spearman`s correlation, and linear regression tests were used to analyze the data. RESULTS: The alveolar ridge width was reduced by an average of 0.44 mm at point A and 0.47 mm at point B during the 12-15 years after treatment (T2-T4) and by 0.21 mm and 0.19 mm during the last 10 years (T3-T4). The alveolar ridge height was reduced by 0.59 mm between T2 and T4 and by 0.05 mm between T3 and T4. All reductions in ridge width and height were statistically significant (P <0.05). However, no significant correlation was observed between patient age and changes in alveolar bone parameters (P >0.05). CONCLUSIONS: Although the reductions in alveolar ridge dimensions were statistically significant, the orthodontically created bone after canine distalization remained stable 12-15 years after treatment in both the horizontal and vertical dimensions. Patient age did not significantly influence alveolar bone changes.

• MeSH
• Anodontia * therapy   diagnostic imaging   MeSH
• Jaw, Edentulous MeSH
• Child MeSH
• Adult MeSH
• Humans MeSH
• Maxilla * MeSH
• Adolescent MeSH
• Young Adult MeSH
• Follow-Up Studies MeSH
• Osteogenesis physiology   MeSH
• Tooth Movement Techniques * methods   MeSH
• Alveolar Process * diagnostic imaging   pathology   abnormalities   MeSH
• Radiography, Panoramic MeSH
• Alveolar Bone Loss diagnostic imaging   MeSH
• Incisor * abnormalities   diagnostic imaging   MeSH
• Age Factors MeSH
• Check Tag
• Child MeSH
• Adult MeSH
• Humans MeSH
• Adolescent MeSH
• Young Adult MeSH
• Male MeSH
• Female MeSH
• Publication type
• Journal Article MeSH

OBJECTIVES: Identification of early presenting signs of the Basal Cell Nevus (BCNS; synonyme Gorlin-Goltz) syndrome, which is associated with a principal triad of multiple basal cell nevi, jaw odontogenic keratocysts, and skeletal anomalies, in stomatological and neurological practices. Proposal of multidisciplinary diagnostic algorithm comprising other medical specialists, including pathology, imaging, laboratory and molecular analyses based on the study outcomes. DESIGN: Case report of a male patient reporting paresthesia of their lower jaw, with right facial asymmetry (maxilla and mandible) and radiological detection of large osteolytic lesions in both jaws, including a retrospective analysis of a representative Czech cohort with BCNS from within the last decade. SETTING: Clinical, imaging and laboratory analyses were carried out at a national tertiary centre. RESULTS: A multidisciplinary clinical approach followed by surgical management lead to the identification of odontogenic cysts, which were substantiated by histological examination. DNA sequencing of the PTCH1 gene detected a c.2929dupT resulting in p. Tyr977Leufs*16 pathogenic variant. This finding confirmed the clinical and laboraoty diagnosis of BCNS. Parental DNA analysis showed that this causal genetic defect arose de novo. Surgical management and orthodontic therapy were successful. CONCLUSIONS: Analysis of the reported case and retrospective data analysis provided evidence that paresthesia of the lower jaw should be considered as one of the early presenting signs of this rare disorder in stomatological and neurological practice. Obtained results allowed us to formulate recommendations for diagnostic practice in stomatology and neurology.

• MeSH
• Anodontia diagnostic imaging   etiology   MeSH
• Early Diagnosis MeSH
• Jaw diagnostic imaging   MeSH
• Child MeSH
• Gene Duplication MeSH
• Cohort Studies MeSH
• Infant MeSH
• Humans MeSH
• Adolescent MeSH
• Odontogenic Cysts diagnostic imaging   etiology   MeSH
• Paresthesia etiology   MeSH
• Tomography, X-Ray Computed MeSH
• Child, Preschool MeSH
• Patched-1 Receptor genetics   MeSH
• Radiography, Panoramic MeSH
• Retrospective Studies MeSH
• Sequence Analysis, DNA MeSH
• Practice Guidelines as Topic MeSH
• Basal Cell Nevus Syndrome complications   diagnosis   genetics   MeSH
• Imaging, Three-Dimensional MeSH
• Check Tag
• Child MeSH
• Infant MeSH
• Humans MeSH
• Adolescent MeSH
• Male MeSH
• Child, Preschool MeSH
• Female MeSH
• Publication type
• Journal Article MeSH
• Case Reports MeSH
• Geographicals
• Czech Republic MeSH