Ectopic Activation of Fgf8 in Dental Mesenchyme Causes Incisor Agenesis and Molar Microdontia.

Putatively, tooth agenesis was attributed to the initiation failure of tooth germs, though little is known about the histological and molecular alterations. To address if constitutively active FGF signaling is associated with tooth agenesis, we activated Fgf8 in dental mesenchyme with Osr-cre knock-in allele in mice (Osr2-creKI; Rosa26R-Fgf8) and found incisor agenesis and molar microdontia. The cell survival assay showed tremendous apoptosis in both the Osr2-creKI; Rosa26R-Fgf8 incisor epithelium and mesenchyme, which initiated incisor regression from cap stage. In situ hybridization displayed vanished Shh transcription, and immunostaining exhibited reduced Runx2 expression and enlarged mesenchymal Lef1 domain in Osr2-creKI; Rosa26R-Fgf8 incisors, both of which were suggested to enhance apoptosis. In contrast, Osr2-creKI; Rosa26R-Fgf8 molar germs displayed mildly suppressed Shh transcription, and the increased expression of Ectodin, Runx2 and Lef1. Although mildly smaller than WT controls prenatally, the Osr2-creKI; Rosa26R-Fgf8 molar germs produced a miniature tooth with impaired mineralization after a 6-week sub-renal culture. Intriguingly, the implanted Osr2-creKI; Rosa26R-Fgf8 molar germs exhibited delayed odontoblast differentiation and accelerated ameloblast maturation. Collectively, the ectopically activated Fgf8 in dental mesenchyme caused incisor agenesis by triggering incisor regression and postnatal molar microdontia. Our findings reported tooth agenesis resulting from the regression from the early bell stage and implicated a correlation between tooth agenesis and microdontia.

Globe-shaped central incisors in a patient with otodental syndrome.

Hearing impairments and dental anomalies are found in many genetic syndromes. Otodental syndrome is a rare combination of hearing loss and the presence of a pathognomonic dental phenotype known as globodontia, in which the tooth exhibits an abnormal globe shape. There is no histologic evidence of structural anomalies in the enamel, dentin, or pulp. This report describes the case of a 12-year-old boy who had hearing loss and 2 supernumerary globe-shaped teeth in the sites of the permanent maxillary central incisors. The diagnosis of otodental syndrome was established based on the clinical, radiographic, and histologic features, but other conditions, including dens evaginatus, talon cusp, dens invaginatus, and compound odontoma, should be included in the differential diagnosis. Dental treatment consisted of the extraction of both anomalous teeth, allowing spontaneous eruption of the impacted permanent central incisors. Early diagnosis of otodental syndrome permits a multidisciplinary approach to prevent other pathologic conditions, reduce functional damage, and avoid social problems.

Application of personalized templates in minimally invasive management of coronal dens invaginatus: a report of two cases.

BACKGROUND: Treating the coronal dens invaginatus (CDI) with pulp infection commonly involves the removal of invagination, which increases the risk of perforation and fracture, and compromises the tooth structure. Minimally invasive endodontic management of CDI is highly recommended. This report describes two cases of type II CDI with the application of personalized templates. CASE PRESENTATION: Two cases of type II CDI, affecting the main root canal in a maxillary canine and a lateral incisor, were diagnosed. A guided endodontics (GE) approach was applied. Cone-beam computed tomography and intraoral scans were imported and aligned in a virtual planning software to design debridement routes and templates. The MICRO principle (which involves the aspects of Mechanical (M) debridement, Irrigation (I), Access cavities (C), Rectilinear routes (R), and Obstruction (O)) was proposed for designing optimal debridement routes for future applications. The templates were innovatively personalized and designed to preserve the tooth structure maximally while effectively debriding the root canal. Root canal treatment with supplementary disinfection was then performed. The follow-up of the two patients revealed favorable clinical and radiographic outcomes. CONCLUSIONS: The GE approach could be a feasible method for preserving healthy dental structure while effectively debriding the root canal, thereby achieving successful and minimally invasive endodontic treatment for CDI.

Autotransplantation of an inverse impacted dilacerated incisor: a case report with 11-year follow-up.

BACKGROUND: Dilaceration can lead to impaction of maxillary incisors, resulting in both aesthetic and functional problems. This case report presents the multidisciplinary approach to managing an inverse impacted dilacerated left lateral incisor in a 9-year-old male patient. CASE REPORT: The orthodontic alignment of the remaining three incisors was achieved within six months. After using the diode laser to remove the gingiva covering the right lateral incisor during alignment process, a lingual button was bonded. The primary left canine and the impacted permanent left lateral incisor were extracted by raising the full-thickness mucoperiosteal flap, followed by the transplantation of the dilacerated lateral incisor into its correct position, splinted to the adjacent central incisors using composite resin. Root canal treatment was performed after the apical plug was created with mineral trioxide aggregate. The composite splint was removed after three weeks, and a new bracket was bonded to the left lateral incisor. It was left passively for 18 months until the permanent canines started to erupt. Light orthodontic forces were then applied for six months, and a passive eruption was expected over three months to properly position the canines within the dental arch. After an 11-year follow-up, the incisor displayed no clinical symptoms, although radiographic examination revealed external resorption in the long-term. CONCLUSION: This case demonstrates the successful and fast autotransplantation of an immature inverse impacted dilacerated incisor, highlighting the long-term clinical success and favorable aesthetic outcomes.

Evaluation of the aesthetics and clinical findings in patients with missing maxillary lateral incisors treated with a 10-year interval.

INTRODUCTION: The most common treatment approaches for patients missing maxillary lateral incisors are implant replacement (IT) and orthodontic space closure (SC). Treatment techniques change and improve over time, and it is of interest to know if improvements differ between the methods. AIM: To compare the aesthetic outcome and other clinical findings in patients with one or two missing maxillary lateral incisors who were treated with a 10-year difference in time, with either orthodontic space closure or implant replacement. MATERIAL AND METHODS: A total of 88 patients were included in the study. Forty-four patients treated between 2011 and 2018 were included as the latter cohort (LC). The LC was compared to the early cohort (EC; n = 44), treated between 2001 and 2008. A total of 132 teeth was analysed: 62 teeth in the EC (28 teeth in IT cases and 34 teeth in SC cases) and 70 teeth in the LC (34 teeth in IT cases and 36 teeth in SC cases). Long-term clinical and aesthetic outcomes were evaluated. RESULTS: An improvement over time was found in crown length, BoP, papilla, the inclination of incisors, and overall appearance in IT cases and in crown colour and overbite in SC cases. A deterioration over time was found in crown length and BoP among the SC cases. CONCLUSION: Among the IT cases, an improvement in outcomes was noted over time. When comparing SC cases the colour of the crown and overbite had improved, while crown length and BoP had deteriorated over time.

Regenerative Endodontic Procedures in Immature Teeth Affected by Regional Odontodysplasia.

INTRODUCTION: Regional odontodysplasia (ROD) is a rare developmental disorder characterized by hypo-mineralization and hypoplasia of enamel and dentin. Symptoms include poorly developed tooth buds, delayed eruption of permanent teeth in affected quadrants, and ghost teeth. The affected teeth often become necrotic due to abnormal enamel and dentin development, making them susceptible to caries and infection. The aim of this case report is to describe the treatment of ROD through pulp revascularization. CASE REPORT: A 13-year-old girl was referred for endodontic treatment. The mandibular left incisors and first premolar, which were affected by regional odontodysplasia, lost their vitality because of the impaired structure of the enamel. Due to the teeth's early developmental stage, a regenerative endodontic treatment was attempted. All 3 teeth were treated using the same protocol following the AAE guidelines. After 4 weeks, treatment of the premolar was completed, whereas the incisor teeth remained symptomatic and were and therefore, intracanal dressing with calcium hydroxide was repeated and left in place for 5 months. Finally, the regenerative procedure was completed, and the crowns were restored. The patient was scheduled for follow-up examinations after 6 months, and then yearly for the next 3 years. After 1 year, the periapical lesion around the central incisor and premolar had resolved, the lesion around the apex of the lateral incisor was healing, and the roots had continued to develop. After 3 years, complete healing and pulp canal obliteration were observed in the central incisor and in the premolar. However, the root of the lateral incisor tooth was split, and it was recommended to extract this tooth. CONCLUSION: The positive outcomes of regenerative endodontics in the central incisor and premolar suggest that revascularization of the pulp may be optional for the treatment of immature necrotic teeth affected by developmental disorders, such as ROD, amelogenesis imperfecta, or dentinogenesis imperfecta.

DISP1 deficiency: Monoallelic and biallelic variants cause a spectrum of midline craniofacial malformations.

PURPOSE: DISP1 encodes a transmembrane protein that regulates the secretion of the morphogen, Sonic hedgehog, a deficiency of which is a major cause of holoprosencephaly (HPE). This disorder covers a spectrum of brain and midline craniofacial malformations. The objective of the present study was to better delineate the clinical phenotypes associated with division transporter dispatched-1 (DISP1) variants. METHODS: This study was based on the identification of at least 1 pathogenic variant of the DISP1 gene in individuals for whom detailed clinical data were available. RESULTS: A total of 23 DISP1 variants were identified in heterozygous, compound heterozygous or homozygous states in 25 individuals with midline craniofacial defects. Most cases were minor forms of HPE, with craniofacial features such as orofacial cleft, solitary median maxillary central incisor, and congenital nasal pyriform aperture stenosis. These individuals had either monoallelic loss-of-function variants or biallelic missense variants in DISP1. In individuals with severe HPE, the DISP1 variants were commonly found associated with a variant in another HPE-linked gene (ie, oligogenic inheritance). CONCLUSION: The genetic findings we have acquired demonstrate a significant involvement of DISP1 variants in the phenotypic spectrum of midline defects. This underlines its importance as a crucial element in the efficient secretion of Sonic hedgehog. We also demonstrated that the very rare solitary median maxillary central incisor and congenital nasal pyriform aperture stenosis combination is part of the DISP1-related phenotype. The present study highlights the clinical risks to be flagged up during genetic counseling after the discovery of a pathogenic DISP1 variant.

Developmental characteristics of the permanent upper lateral incisor in unilateral cleft lip and palate.

OBJECTIVES: This study aims to provide insights into the developmental characteristics of the upper lateral incisor in individuals with unilateral clefts. MATERIALS AND METHODS: Panoramic radiographs of a consistent group of Caucasian children taken over time (ages 6, 9, and 12) were extensively reviewed. The study assessed the distribution pattern, eruption path, tooth development, and crown size of the upper lateral incisor within the cleft region. RESULTS: The most commonly observed distribution pattern was the lateral incisor located distal to the cleft, accounting for 49.2% of cases. Furthermore, a significant delay in tooth development of the upper lateral incisor on the cleft side was noted at ages 6 and 9 (p > 0.001). Compared with the non-cleft side, these incisors often erupted along the alveolar cleft and exhibited microdontia (88.3%, p < 0.041). CONCLUSION: Lateral incisors on the cleft side display unique distribution patterns, microdontia, and delayed tooth development. Careful monitoring of the cuspid eruption is essential, as it can influence the eruption of the lateral incisor. CLINICAL RELEVANCE: A comprehensive understanding of the development of the upper lateral incisor relative to the cleft is vital for determining its prognosis over time. The position of the upper lateral incisor can also influence the timing and prognosis of secondary alveolar bone grafting. Preserving the upper lateral incisor favors arch length, perimeter, and symmetry in individuals with unilateral clefts.

Selective excavation and ozone therapy: new frontier of mini-invasive caries treatment in MIH paediatric patients. A case report.

BACKGROUND: The term hypomineralisation of molars and incisors (MIH), introduced in 2001 by Weerheijm et al., describes a clinical state of hypomineralisation of permanent molars with frequent involvement of the incisors. MIH is considered a global dental problem with a prevalence ranging from 2.4% to 40.2% in the entire world paediatric population. The continuous increase in the prevalence of enamel anomalies, including MIH, indicates the need to define new intervention protocols based on the technological advances that are revolutionising paediatric dentistry. The use of ozone associated with the selective and minimally invasive excavation of the dental tissue combines the antibacterial properties of the gas with an ultra-conservative approach aimed at the maximum conservation of the dental tissue. The operative protocol described can be an important tool in the prevention and treatment of MIH. The aim of this work is to illustrate an operative clinical protocol based on the combined use of selective excavation and ozone for the treatment of carious lesions in paediatric patients with MIH.

Alveolar morphometry in young adults with and without agenesis of the upper lateral incisor: A retrospective study.

INTRODUCTION: We aimed to compare alveolar morphometry in young adults with agenesis of the upper lateral incisor versus the side without agenesis and versus matched controls. MATERIAL AND METHODS: In this observational retrospective study, cone beam computed tomography scans were obtained of 36 upper hemiarches from young adults aged 15 to 30 years. The hemiarches were distributed into three groups: group 1: 12 upper hemiarches presenting agenesis of the upper lateral incisor; group 2: 12 upper hemiarches from the opposite side without agenesis of the upper lateral incisor (control group 1); and group 3: 12 upper hemiarches without agenesis of the upper lateral incisor matched for age and sex with respect to the affected group (control group 2). A trained and calibrated investigator performed all the alveolar measurements at two different times, including sagittal, coronal and axial slices of each hemiarch. Paired Student's t-tests, Chi-square and repeated measures ANOVA with Bonferroni correction were used, (P<0.05). RESULTS: Apical mesial evaluation of group 1 (4.22±1.19mm) was significantly lower (P<0.001) than that of groups 2 (6.72±1.17mm) and 3 (7.58±1.67mm). Apical distal evaluation also showed differences (P<0.001) among the three groups, with the dimension being smaller in group 1 with agenesis (4.53±1.14mm), followed by group 2 without agenesis (6.23±1.55mm) and the healthy control group 3 (7.73±1.71mm). CONCLUSIONS: Lateral incisor agenesis significantly reduces the alveolar dimensions of the affected area. In cases of unilateral agenesis, the unaffected side also shows sequelae, with decreased dimensions compared to cases without agenesis. This condition should be taken into account when making therapeutic decisions regarding rehabilitation with implants or canine replacement.

Management options and long-term follow-ups for permanent double incisors: six to eleven-year case reports.

BACKGROUND: Double teeth are dental anomalies that can lead to aesthetic and orthodontic problems. CASE PRESENTATION: This report discusses two cases involving the multidisciplinary management of permanent maxillary left lateral incisors fused with a supernumerary tooth in two girls aged 9 and 10. Following intraoral and radiographic examinations, one was diagnosed with fusion, and the other was diagnosed with concrescence. The crown of the fused incisor was separated using a burs and extracted intraorally. The concrescent incisor was separated along its length using a laser and intentionally replanted extraorally. After a 6-year follow-up, no pathological signs were observed in the fused incisor. However, after an 11-year follow-up, external resorption was observed in the concrescent incisor. CONCLUSIONS: Both incisors remained asymptomatic throughout the observation period. This case report highlights two different and effective methods employed to preserve the natural function, form, and aesthetics of double incisors.

Patterns and sexual dimorphism of non-syndromic hypodontia among a French orthodontic population.

OBJECTIVES: This retrospective study aimed to estimate the prevalence of non-syndromic congenitally missing teeth (CMT) and to explore the frequency of CMT patterns in a French orthodontic population. In addition, the study sought to assess sex-based differences in CMT patterns. DESIGN: Panoramic radiographs of 4569 orthodontic patients between 9 and 21 years-old performed over a 16-year period (2006-2022) were examined to identify non-syndromic tooth agenesis, excluding third molars. A chi-square test or a Fisher exact test were used to determine the difference in the prevalence of tooth agenesis between sex and between arches. RESULTS: Tooth agenesis was observed in 7.3% of the sample (7.9% for females and 6.6% for males). Approximately 86% of the included subjects presented 1 or 2 missing teeth. Single tooth agenesis was significantly more frequent in females than males (p = 0.002, χ2). In total, 23 of the 67 different patterns of CMT observed, were present more than once. 75.5% of male patients and 79.5% of female patients presented one or both missing lateral incisors or second premolars, rarely affected at the same time. This study showed no sex difference in the patterns of tooth agenesis. LIMITATIONS: This study has limitations due to its retrospective nature and our findings apply solely to an orthodontic population from a white ethnic background. CONCLUSIONS: Clinicians should be aware of this particular incisor/premolar phenotype regardless of biological sex. Issues associated with congenitally missing teeth can be managed more effectively with early teenage diagnosis.

Root Canal Treatment of Oehlers Type III Dens Invaginatus in Maxillary Lateral Incisor and Remote Sinus Tract Using Dental Surgical Microscope and Cone-Beam Computed Tomography.

Dens invaginatus is a morphological abnormality of the tooth that results from a developmental anomaly during tooth formation, in which part of the enamel and dentin of the crown invaginates into the pulp cavity. This report describes a case of a maxillary lateral incisor with apical periodontitis apparently caused by Oehlers Type III dens invaginatus. The patient was a 69-year-old man who visited our clinic complaining of discomfort in the maxillary right lateral incisor. Cone-beam computed tomography (CBCT) revealed dens invaginatus of the maxillary lateral incisor and a sinus tract in the maxillary central incisor region, which was derived from apical periodontitis of the maxillary lateral incisor. The dens invaginatus was accompanied by a complex root canal morphology. Treatment, which was performed using a dental surgical microscope, had a favorable outcome. The patient remains in good condition at 1 year postoperatively.

Root canal treatment of type â ¡ and â ¢A double dens invaginatus in maxillary lateral incisor: a case report.

Dens invaginatus is a rare developmental anomaly of the teeth that is caused by the infolding of enamel organs or the penetration of their proliferations into dental papillae before calcification has occurred. The presence of double dens invaginatus is extremely rare. This paper describes the use of cone beam computed tomography in the evaluation of a maxillary lateral incisor with double dens invaginatus and periapical periodontitis. The tooth was treated through microscopic root canal therapy. The tooth was free of clinical symptoms, and the periradicular lesion narrowed during the follow-up period of 1 year.

Closure of maxillary lateral incisor agenesis space in unilateral cleft lip and palate: a digital model assessment.

OBJECTIVE: To compare dental positional and gingival parameters of maxillary anterior teeth in unilateral cleft lip and palate (UCLP) after orthodontic treatment with canine substitution due to lateral incisor agenesis. METHODS: This split-mouth study comprised 57 subjects with UCLP (31 male, 26 female) and agenesis of maxillary lateral incisor at the cleft side, from a single center. Canine substitution was completed after the secondary alveolar bone graft. Dental models were taken between 2 to 6 months after debonding (mean age: 20.4 years). The following variables were measured in the maxillary anterior teeth: crown height, width, proportion, and symmetry, as well as steps between incisal edges, gingival margins, tooth mesiodistal angulation and labiolingual inclination. Paired t-tests with Bonferroni post-hoc correction was used for comparisons between cleft and non-cleft sides (p<0.05). RESULTS: At the cleft side, canines replacing missing lateral incisors had a higher crown height (0.77mm) and an increased width (0.67mm), and first premolars showed a shorter crown height (1.39mm). Asymmetries were observed in the gingival level of central and lateral incisors, with a greater clinical crown at the cleft side (0.61 and 0.81mm, respectively). Cleft side central incisors were more upright than their contralaterals (2.12º). CONCLUSIONS: Maxillary anterior teeth demonstrated positional, size and gingival height differences between cleft and non-cleft side after space closure of maxillary lateral incisor agenesis. Slight asymmetries in tooth position and gingival margin in the maxillary anterior teeth should be expected after orthodontic treatment in UCLP patients.

The design and clinical application of a new appliance to treat impacted maxillary central incisors.

Maxillary central incisor impaction is one of the most common types of dental anomalies in children. Treatment of impacted central incisors is complicated and challenging given the position of the impacted central incisors, root development, and the complexity of the crown eruption direction. This study aimed to describe the use of a new multifunctional appliance for the treatment of impacted maxillary central incisors. This article reports the use of a novel appliance for the treatment of impacted maxillary central incisors. We describe the cases of two young patients with labial horizontally impacted maxillary central incisors. Both patients were treated using this novel appliance. Therapeutic effects were evaluated by comparing the pretreatment results, posttreatment cone-beam computed tomography images, and posttreatment clinical examination results. At the end of the treatment period using the novel appliance, the impacted central incisors had successfully been properly aligned in the dental arch, and the tooth roots had not resorbed. Both patients exhibited good dental alignment, with restored function and acceptable aesthetics. This article demonstrates that the new appliance was comfortable, convenient, safe, and effective in the treatment of impacted maxillary central incisors and that its clinical use should be promoted in the future.

Solitary median maxillary central incisor in Kabuki syndrome 2 with novel missense mutation of KDM6A and ABCC8 genes.

Kabuki syndrome (KS) is an epigenetic machinery multisystem disorder with peculiar facial gestalt and dental-oral anomalies. This report describes the case of a KS patient with congenital hyperinsulinism, growth hormone deficiency and novel heterogenous missense mutations in exon 25 of the KDM6A (c.3715T>G, p.Trp1239Gly) and exon 1 of the ABCC8 (c.94A>G, p.Asn32Asp) genes. She presented with solitary median maxillary central incisor (SMMCI) and mandibular incisor hypodontia, which could be a unique dental manifestation in KS 2.

Associations among the occurrence or types of maxillary canine impaction, labial frenum attachment types, lateral incisor anomalies, and midline diastema in patients with and without impaction: A case-control study.

INTRODUCTION: We tested whether the labial frenum attachment types may affect the occurrence or type of maxillary canine impaction. METHODS: This case-control study was performed on 96 subjects (48 with maxillary canine impaction [20.50±4.99 years, 32 females], 48 controls [21.94±3.79, 31 females]). Types of canine impaction (buccal/palatal, bilateral/unilateral, left/right), types of upper lip middle frenums (mucosal, gingival, papillary), the anatomy of lateral incisors (impacted, peg shaped, small, normal), and the midline diastema were evaluated clinically and radiographically. Associations were examined using multiple binary logistic regression, 3-way repeated-measures ANCOVA, and Fisher tests (α=0.05, ß=0.2). RESULTS: The numbers of mucosal, gingival, and papillary frenums in the case group were '29, 14, and 5', respectively; in the control group, they were '25, 21, and 2' (Fisher, P=0.248). The numbers of mucosal, gingival, and papillary frenums were respectively '25, 11, and 1' in the palatally impacted canine subgroup; they were '4, 3, and 4' in the buccally impacted subgroup (Fisher, P=0.010). Abnormal laterals were present in 12 cases and 4 controls; normal laterals were observed in 36 cases and 44 controls (Fisher, P=0.053). Abnormal laterals were seen in 10 cases with palatally impacted canines and 2 cases with buccally impacted canines; normal laterals existed in 27 cases with palatally impacted canines and 9 cases with buccally impacted canines (Fisher, P=0.705). Lateral anatomy was not associated with frenum attachment (Fisher, P=0.827). Greater midline diastemas were seen in cases with maxillary canine impaction, papillary frenums, and abnormal laterals (ANCOVA, P≤0.016). CONCLUSIONS: Buccal type of canine impaction is positively associated with papillary labial frenums. Canine impaction is 3.6 folds more prevalent in cases with lateral abnormality. Midline diastema is positively and independently associated with canine impaction, papillary frenums, and abnormal laterals.