Effect of Model Body Type and Print Angle on the Accuracy of 3D-Printed Orthodontic Models.

The assortment of low-cost 3D printers for "in-practice" use, e.g., for clear aligner therapies, is ever increasing. To address concerns about the accuracy of orthodontic models produced on such printers when cost-efficient modes of 3D printing are employed, this study examined the effect of print model body type and print angulation on accuracy. Six printing-configuration groups were included: two model types (solid or hollow shell) combined with three print angles (0°, 70°, or 90°) with 10 models/group; all models were printed with 100 µm layer thickness using a digital light processing-based three-dimensional printer. Eleven selected structures and distances were measured on the printed models with a digital microscope and compared to the same measures on a digitized master model. The clinically acceptable range was set at ±0.25 mm difference from the master model for single tooth measurements (intra-tooth) and ±0.5 mm for cross-arch measurements (inter-tooth). For individual measurements across all models, 98% fell within clinical acceptability. For mean measurements within each model group, only canine height for the shell-0° model had a mean difference (-0.26 mm ± 0.03) outside the clinically acceptable range for intra-tooth measurements. Standard deviations for all intra-tooth measurements were within 0.07 mm. While none of the mean inter-tooth measurements exceeded the acceptability range, the standard deviations were larger (0.04 to 0.30 mm). The accuracy of the orthodontic models for clear aligner therapies was not impacted beyond the clinically acceptable range when altering model body type and print angulation to improve efficiency of 3D printing. These findings suggest greater flexibility of the practitioner to alter print settings to address time and cost efficiency in various clinical scenarios and still maintain clinically acceptable model accuracy.

Cranio-cervical abnormalities in moderate-to-severe osteogenesis imperfecta - Genotypic and phenotypic determinants.

INTRODUCTION: Cranio-cervical anomalies are significant complications of osteogenesis imperfecta (OI), a rare bone fragility disorder that is usually caused by mutations in collagen type I encoding genes. OBJECTIVE: To assess cranio-cervical anomalies and associated clinical findings in patients with moderate-to-severe OI using 3D cone beam computed tomography (CBCT) scans. METHODS: Cross-sectional analysis of CBCT scans in 52 individuals with OI (age 10-37 years; 32 females) and 40 healthy controls (age 10-32 years; 26 females). Individuals with a diagnosis of OI type III (severe, n = 11), type IV (moderate, n = 33) and non-collagen OI (n = 8) were recruited through the Brittle Bone Disorders Consortium. Controls were recruited through the orthodontic clinic of the University of Missouri-Kansas City (UMKC). RESULTS: OI and control groups were similar in mean age (OI: 18.4 [SD: 7.2] years, controls: 18.1 [SD: 6.3] years). The cranial base angle was increased in the OI group (OI: mean 148.6° [SD: 19.3], controls: mean 130.4° [SD: 5.7], P = .001), indicating a flatter cranial base. Protrusion of the odontoid process into the foramen magnum (n = 7, 14%) and abnormally located odontoid process (n = 19, 37%) were observed in the OI group but not in controls. Low stature, expressed as height z-score (P = .01), presence of DI (P = .04) and being male (P = .04) were strong predictors of platybasia, whereas height z-score (P = .049) alone was found as positive predictor for basilar impression as per the Chamberlain measurement. CONCLUSION: The severity of the phenotype in OI, as expressed by the height z-score, correlates with the severity of cranial base anomalies such as platybasia and basilar impression in moderate-to-severe OI. Screening for cranial base anomalies is advisable in individuals with moderate-to-severe OI, with special regards to the individuals with a shorter stature and DI.

Craniofacial features of POLR3-related leukodystrophy caused by biallelic variants in POLR3A, POLR3B and POLR1C.

BACKGROUND: RNA polymerase III-related or 4H leukodystrophy (POLR3-HLD) is an autosomal recessive hypomyelinating leukodystrophy characterized by neurological dysfunction, hypodontia and hypogonadotropic hypogonadism. The disease is caused by biallelic pathogenic variants in POLR3A, POLR3B, POLR1C or POLR3K. Craniofacial abnormalities reminiscent of Treacher Collins syndrome have been originally described in patients with POLR3-HLD caused by biallelic pathogenic variants in POLR1C. To date, no published studies have appraised in detail the craniofacial features of patients with POLR3-HLD. In this work, the specific craniofacial characteristics of patients with POLR3-HLD associated with biallelic pathogenic variants in POLR3A, POLR3B and POLR1C are described. METHODS: The craniofacial features of 31 patients with POLR3-HLD were evaluated, and potential genotype-phenotype associations were evaluated. RESULTS: Various craniofacial abnormalities were recognized in this patient cohort, with each individual presenting at least one craniofacial abnormality. The most frequently identified features included a flat midface (61.3%), a smooth philtrum (58.0%) and a pointed chin (51.6%). In patients with POLR3B biallelic variants, a thin upper lip was frequent. Craniofacial anomalies involving the forehead were most commonly associated with biallelic variants in POLR3A and POLR3B while a higher proportion of patients with POLR1C biallelic variants demonstrated bitemporal narrowing. CONCLUSION: Through this study, we demonstrated that craniofacial abnormalities are common in patients with POLR3-HLD. This report describes in detail the dysmorphic features of POLR3-HLD associated with biallelic variants in POLR3A, POLR3B and POLR1C.

Decoding Deep Learning applications for diagnosis and treatment planning.

INTRODUCTION: Artificial Intelligence (AI), Machine Learning and Deep Learning are playing an increasingly significant role in the medical field in the 21st century. These recent technologies are based on the concept of creating machines that have the potential to function as a human brain. It necessitates the gathering of large quantity of data to be processed. Once processed with AI machines, these data have the potential to streamline and improve the capabilities of the medical field in diagnosis and treatment planning, as well as in the prediction and recognition of diseases. These concepts are new to Orthodontics and are currently limited to image processing and pattern recognition. OBJECTIVE: This article exposes and describes the different methods by which orthodontics may benefit from a more widespread adoption of these technologies.

Craniofacial and dental phenotype of two girls with osteogenesis imperfecta due to mutations in CRTAP.

Mutations in CRTAP lead to an extremely rare form of recessive osteogenesis imperfecta (OI). CRTAP deficient mice have a brachycephalic skull, fusion of facial bones, midface retrusion and class III dental malocclusion, but in humans, the craniofacial and dental phenotype has not been reported in detail. Here, we describe craniofacial and dental findings in two 11-year-old girls with biallelic CRTAP mutations. Patient 1 has a homozygous c.472-1021C>G variant in CRTAP intron 1 and a moderately severe OI phenotype. The variant is known to create a cryptic splice site, leading to a frameshift and nonsense-mediated RNA decay. Patient 1 started intravenous bisphosphonate treatment at 2 years of age. At age 11 years, height Z-score was +0.6. She had a short and wide face, concave profile and class III malocclusion, with a prognathic mandible and an antero-posterior crossbite. A panoramic radiograph showed a poor angulation of the second upper right premolar, and no dentinogenesis imperfecta or dental agenesis. Cone-beam computed tomography confirmed these findings and did not reveal any other abnormalities. Patient 2 has a homozygous CRTAP deletion of two amino acids (c.804_809del, p.Glu269_Val270del) and a severe OI phenotype. As previously established, the variant leads to instability of CRTAP protein. Intravenous bisphosphonate treatment was started at the age of 15 months. At 11 years of age her height Z-score was -9.7. She had a long and narrow face and convex profile, maxillary retrusion leading to a class III malocclusion, an edge-to-edge overjet and lateral open bite. Panoramic radiographs showed no dental abnormalities. Cone-beam computed tomography showed occipital bossing, platybasia and wormian bones. In these two girls with CRTAP mutations, the severity of the skeletal phenotype was mirrored in the severity of the craniofacial phenotype. Class III malocclusion and antero-posterior crossbite were a common trait, while dental agenesis or dentinogenesis imperfecta were not detected.

Machine learning in orthodontics: Automated facial analysis of vertical dimension for increased precision and efficiency.

INTRODUCTION: The digitization of dentistry has brought many opportunities to the specialty of orthodontics. Advances in computing power and artificial intelligence are set to significantly impact the specialty. In this article, the accuracy of automated facial analysis for vertical dimensions using machine learning is evaluated. METHODS: Automated facial analysis of 45 patients (20 female, 25 male) was conducted. The subjects' ages were between 15 and 25 years (mean, 18.7; standard deviation, 3.2). A python program was written by the authors to detect the faces, annotate them and compute vertical dimensions. The accuracy of the manual annotation of digital images was compared with the proposed model. Intrarater and interrater reliability were evaluated for the manual method, whereas intraclass correlation and the Bland-Altman analysis were compared with manual and automated methods. RESULTS: The authors found acceptable intrarater reliability and moderate to poor interrater reliability for the manual method. The agreement was found between manual and automated methods of facial analysis. The 95% confidence interval limit of agreements was <10% for the metrics assessing vertical dimension. CONCLUSIONS: Machine learning offers the ability to conduct reliable and easily reproducible analyses on large datasets of images. This new tool presents opportunities for further advances in research and clinical orthodontics.

Decoding Deep Learning applications for diagnosis and treatment planning

ABSTRACT Introduction: Artificial Intelligence (AI), Machine Learning and Deep Learning are playing an increasingly significant role in the medical field in the 21st century. These recent technologies are based on the concept of creating machines that have the potential to function as a human brain. It necessitates the gathering of large quantity of data to be processed. Once processed with AI machines, these data have the potential to streamline and improve the capabilities of the medical field in diagnosis and treatment planning, as well as in the prediction and recognition of diseases. These concepts are new to Orthodontics and are currently limited to image processing and pattern recognition. Objective: This article exposes and describes the different methods by which orthodontics may benefit from a more widespread adoption of these technologies.

RESUMO Introdução: Inteligência Artificial (AI, de Artificial Intelligence), Machine Learning (Aprendizado de máquinas) e Deep Learning (Aprendizado Profundo) possuem um papel significativo e crescente na área médica do século 21. Essas tecnologias recentes são baseadas no conceito de criar máquinas com potencial de funcionar como um cérebro humano. Isso demanda que uma grande quantidade de dados seja reunida para ser processada. Uma vez processados em máquinas com AI, esses dados têm o potencial de agilizar e potencializar as capacidades de diagnóstico e planejamento do tratamento nas áreas médicas, assim como no diagnóstico e prognóstico de doenças. Esses são conceitos novos na Ortodontia, que atualmente são subutilizados, limitando-se ao processamento de imagens e reconhecimento de padrões. Objetivo: O presente artigo expõe e descreve os diferentes métodos pelos quais os ortodontistas podem se beneficiar com o uso mais abrangente dessas tecnologias.

Missing and unerupted teeth in osteogenesis imperfecta.

INTRODUCTION: Osteogenesis 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. METHOD: A 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). RESULTS: On 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)). CONCLUSION: The 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.

Osteogenesis imperfecta tooth level phenotype analysis: Cross-sectional study.

INTRODUCTION: 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. METHOD: 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. RESULTS: 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. CONCLUSION: 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.

Malocclusion traits and oral health-related quality of life in children with osteogenesis imperfecta: A cross-sectional study.

BACKGROUND: The incidence of malocclusion is higher among people with osteogenesis imperfecta (OI) than the general population, and treatment options are limited due to the weak structure of bones and teeth. Focusing on those malocclusion traits that might have a high impact on a patient's oral health-related quality of life (OHRQoL) is warranted. METHODS: A total of 138 children and adolescents with OI were examined for malocclusion traits. OHRQoL was measured using age-specific versions (8 through 10 years and 11 through 14 years) of the Child Perceptions Questionnaire (CPQ), considering the following domains: oral symptoms, functional limitation, emotional well-being, and social well-being. Higher scores implied worse OHRQoL. Multivariable ordinal logistic regression was used to estimate the association between malocclusion traits and OHRQoL. RESULTS: Among children aged 8 through 10 years (n = 56), the CPQ and its constituent domain scores were relatively similar between those with malocclusion (higher scores) and those without. In the adolescent (n = 82) group aged 11 through 14 years; however, those with posterior crossbite (odds ratio, 5.01; 95% confidence interval, 1.40 to 12.41) or open bite (odds ratio, 3.21; 95% confidence interval, 1.21 to 10.23) experienced statistically significantly higher degrees of functional limitations (a higher functional limitation score) than those without. CONCLUSIONS: Adolescents with OI and posterior open bites or crossbites have substantial self-reported functional limitations and worse oral symptoms, which warrants additional investigation and therapeutic trials in an attempt to improve the malocclusion. In addition, the authors found that the CPQ can be a useful tool in a clinical trial of orthodontic interventions in OI.

Fabrication of Nasoalveolar Molding Devices for the Treatment of Cleft Lip and Palate, Using Stereolithography Additive Manufacturing Processes and Computer-Aided Design Manipulation Software.

INTRODUCTION: Nasoalveolar molding (NAM) is a presurgical orthopedic treatment modality that attempts to reorient misaligned bony and soft tissue structures in patients with clefting of the lip and palate. The NAM devices are implemented prior to surgical intervention in order to minimize the gap across the cleft and thereby reduce tension across the eventual repair. Currently, NAM devices are fabricated in a laboratory and then refined chairside by the provider. The present article describes the potential of three-dimensional (3D) printing and computer-aided design (CAD) software for the fabrication of NAM devices. MATERIAL AND METHODS: A workflow was developed to demonstrate the use of 3D printing and CAD software to design NAM devices. This workflow encompasses scanning an impression into CAD software, performing a series of manipulations, and then printing the digital model. RESULTS: To test the workflow, a cleft palate plaster model was scanned into CAD software. Through a series of linear and angular freeform manipulations of the body, the model was modified to display a cleft with a reduced alveolar gap. Sequential molding devices were produced which would gradually apply pressure to targeted areas of hard and soft tissue until the cleft is minimized. The resulting devices are printed using a stereolithography printer. CONCLUSIONS: The use of 3D printing and CAD software shows promise in improving the accuracy, speed, and cost-effectiveness of designing NAM devices. The accuracy and flexibility from digitally visualizing the manipulations made to an appliance before its creation can result in a more personalized device for the patient.

Cone-Beam Computed Tomography of Osteogenesis Imperfecta Types III and IV: Three-Dimensional Evaluation of Craniofacial Features and Upper Airways.

This cross-sectional study investigated the natural history of craniofacial deformities in osteogenesis imperfecta (OI) and determined the impact of three-dimensional (3D) analysis on diagnosis and treatment planning in orthodontics and orthognathic surgery in comparison to conventional two-dimensional (2D) cephalometric examination. 3D images of the craniofacial complex were acquired during 1 calendar year using cone-beam computed tomography (CBCT) from a cohort of 41 individuals (aged 11 to 35 years; 28 females) with OI type III (n = 13) or IV (n = 28). 3D evaluation of the craniocervical junction and upper airways was conducted using InVivoTM. 2D lateral cephalogram was constructed, traced, and examined using the University of Western Ontario analysis (DolphinTM). Quantitative and qualitative parameters were compared between OI type III and type IV groups (unpaired t test) and the unaffected population (Z-score). 3D evaluation revealed a high prevalence of craniocervical abnormalities, craniofacial asymmetries, and nasal septum deviation in both OI groups. Mean airway dimensions were comparable to the non-affected population norms, except for 5 individuals who had insufficient airway dimensions. In 2D, the maxilla was retrognathic and hypoplastic, and the mandibular position was convergent with respect to the face, resulting in mandibular prognathism and face height reduction. The 2D trends were more pronounced in OI type III, whereas the 3D craniocervical and airway abnormalities were common in both types. This study illustrates the prevalence of craniofacial and airway anomalies in OI that occur along with facial deformities are not associated with postcranial phenotype and OI type, are apparent only in 3D evaluation, and are likely to influence treatment strategy. For OI patients, a team effort involving a dentist, orthodontist, neurologist, and ear-nose-throat (ENT) practitioner is recommended for successful management of craniofacial deformities.

Caries prevalence and experience in individuals with osteogenesis imperfecta: A cross-sectional multicenter study.

OBJECTIVE: Dentinogenesis Imperfecta (DI) forms a group of dental abnormalities frequently found associated with Osteogenesis Imperfecta (OI), a hereditary disease characterized by bone fragility. The objectives of this study were to quantify the dental caries prevalence and experience among different OI-types in the sample population and quantify how much these values change for the subset with DI. METHODS: To determine which clinical characteristics were associated with increased Caries Prevalence and Experience (CPE) in patients with OI, the adjusted DFT scores were used to account for frequent hypodontia, impacted teeth and retained teeth in OI population. For each variable measured, frequency distributions, means, proportions and standard deviations were generated. Groups means were analyzed by the unpaired t-test or ANOVA as appropriate. For multivariate analysis, subjects with caries experience of zero were compared with those with caries experience greater than zero using logistic regression. RESULTS: The stepwise regression analysis while controlling for all other variables demonstrated the presence of DI (OR 2.43; CI 1.37-4.32; P = 0.002) as the significant independent predictor of CPE in the final model. CONCLUSION: This study found no evidence that CPE of OI subjects differs between the types of OI. The presence of DI when controlled for other factors was found to be the significant predictor of CPE.

Dental and craniofacial characteristics caused by the p.Ser40Leu mutation in IFITM5.

Severe forms of osteogenesis imperfecta (OI) are usually caused by mutations in genes that code for collagen Type I and frequently are associated with craniofacial abnormalities. However, the dental and craniofacial characteristics of OI caused by the p.Ser40Leu mutation in the IFITM5 gene have not been reported. We investigated a 15-year-old girl with severe OI caused by this mutation. She had marked deformations of extremity long bones. There were no clinical or radiological signs of dentinogenesis imperfecta, but one tooth was missing and several teeth were impacted. Cone beam computed tomography revealed a generalized osteopenic appearance of the craniofacial skeleton, bilateral enlargement of mandibular bodies, and areas of cortical erosions. The cranial base and skull showed a generalized granular bone pattern with a mixture of osteosclerosis and osteolysis. Sphenoid and frontal sinuses were congenitally missing. Cephalometric analysis indicated a Class III growth pattern. In this case, the IFITM5 p.Ser40Leu mutation did not affect tooth structure but was associated with deformities in craniofacial bones that resemble those in the other parts of the skeleton.

Oro-dental and cranio-facial characteristics of osteogenesis imperfecta type V.

Osteogenesis imperfecta (OI) type V is an ultrarare heritable bone disorder caused by the heterozygous c.-14C > T mutation in IFITM5. The oro-dental and craniofacial phenotype has not been described in detail, which we therefore undertook to evaluate in a multicenter study (Brittle Bone Disease Consortium). Fourteen individuals with OI type V (age 3-50 years; 10 females, 4 males) underwent dental and craniofacial assessment. None of the individuals had dentinogenesis imperfecta. Six of the 9 study participants (66%) for whom panoramic radiographs were obtained had at least one missing tooth (range 1-9). Class II molar occlusion was present in 8 (57%) of the 14 study participants. The facial profile was retrusive and lower face height was decreased in 8 (57%) individuals. Cephalometry, performed in three study participants, revealed a severely retrusive maxilla and mandible, and moderately to severly retroclined incisors in a 14-year old girl, a protrusive maxilla and a retrusive mandible in a 14-year old boy. Cone beam computed tomograpy scans were obtained from two study participants and demonstrated intervertebral disc calcification at the C2-C3 level in one individual. Our study observed that OI type V is associated with missing permanent teeth, especially permanent premolar, but not with dentinogenesis imperfecta. The pattern of craniofacial abnormalities in OI type V thus differs from that in other severe OI types, such as OI type III and IV, and could be described as a bimaxillary retrusive malocclusion with reduced lower face height and multiple missing teeth.

Oral health-related quality of life in children and adolescents with osteogenesis imperfecta: cross-sectional study.

BACKGROUND: Osteogenesis imperfecta (OI) affects dental and craniofacial development and may therefore impair Oral Health-Related Quality of Life (OHRQoL). However, little is known about OHRQoL in children and adolescents with OI. The aim of this study was to explore the influence of OI severity on oral health-related quality of life in children and adolescents. METHODS: Children and adolescents aged 8-14 years were recruited in the context of a multicenter longitudinal study (Brittle Bone Disease Consortium) that enrolls individuals with OI in 10 centers across North America. OHRQoL was assessed using the Child Perceptions Questionnaire (CPQ) versions for 8 to 10-year-olds (CPQ8-10) and for 11 to 14-year-olds (CPQ11-14). RESULTS: A total of 138 children and adolescents (62% girls) diagnosed with OI types I, III, IV, V and VI (n = 65, 30, 37, 4 and 2, respectively) participated in the study. CPQ8-10 scores were similar between OI types in children aged 8 to 10 years. In the 11 to 14-year-old group, CPQ11-14-scores were significantly higher (i.e. worse) for OI types III (24.7 [SD 12.5]) and IV (23.1 [SD 14.8]) than for OI type I (16.5 [SD 12.8]) (P < 0.05). The difference between OI types was due to the association between OI types and the functional limitations domain, as OI types III and IV were associated with significantly higher grade of functional limitations compared to OI type I. CONCLUSION: The severity of OI impacts OHRQoL in adolescents aged 11 to 14 years, but not in children age 8 to 10 years.

Osteogenesis imperfecta: potential therapeutic approaches.

Osteogenesis imperfecta (OI) is a genetic disorder that is usually caused by disturbed production of collagen type I. Depending on its severity in the patient, this disorder may create difficulties and challenges for the dental practitioner. The goal of this article is to provide guidelines based on scientific evidence found in the current literature for practitioners who are or will be involved in the care of these patients. A prudent approach is recommended, as individuals affected by OI present with specific dentoalveolar problems that may prove very difficult to address. Recommended treatments for damaged/decayed teeth in the primary dentition are full-coverage restorations, including stainless steel crowns or zirconia crowns. Full-coverage restorations are also recommended in the permanent dentition. Intracoronal restorations should be avoided, as they promote structural tooth loss. Simple extractions can also be performed, but not immediately before or after intravenous bisphosphonate infusions. Clear aligners are a promising option for orthodontic treatment. In severe OI types, such as III or IV, orthognathic surgery is discouraged, despite the significant skeletal dysplasia present. Given the great variations in the severity of OI and the limited quantity of information available, the best treatment option relies heavily on the practitioner's preliminary examination and judgment. A multidisciplinary team approach is encouraged and favored in more severe cases, in order to optimize diagnosis and treatment.

Correlation between Nasoalveolar Molding and Surgical, Aesthetic, Functional and Socioeconomic Outcomes Following Primary Repair Surgery: a Systematic Review.

OBJECTIVES: The authors performed a systematic review to evaluate the potential beneficial effects of the nasoalveolar molding appliance on nonsyndromic unilateral clefts of the lip and/or palate prior to primary lip repair. MATERIAL AND METHODS: A literature search was performed using three electronic databases (PubMed, Embase, Web of Science) and three journals ("Cleft Palate-Craniofacial Journal", "Plastic and Reconstructive Surgery Journal" and "American Journal of Orthodontics and Dentofacial Orthopaedic") from January 1980 to April 2017. Data extraction was performed with tables treating different subjects: surgical, aesthetical, functional, socio-economical effects of nasoalveolar molding (NAM) appliances and the evolution of NAM appliances, especially three-dimensional technology. RESULTS: Of the 145 articles retrieved in the literature surveys, 28 were qualified for the final analysis and 20 studies were excluded because of their small sample size (less than 10 patients) and/or too long follow-up (exceeded 18 months). Four randomized controlled trials were available. Although literature allowed discussing the short-term benefits of NAM appliance and the three-dimensional technology, scientific evidence is lacking. CONCLUSIONS: Based on the results, nasoalveolar molding appliances have positive surgical, aesthetical, functional and socio-economical effects on unilateral clefts of the lip and/or palate treatment before the primary repair surgeries. Three-dimensional technology results in a more efficient and predictable nasoalveolar molding appliance treatment. However, nasoalveolar molding appliance effect in a short term remains unclear with the available literature. Further studies that integrate three-dimensional technology in a large scale are still needed.