Machine learning-based decision support system for orthognathic diagnosis and treatment planning.

BACKGROUND: Dento-maxillofacial deformities are common problems. Orthodontic-orthognathic surgery is the primary treatment but accurate diagnosis and careful surgical planning are essential for optimum outcomes. This study aimed to establish and verify a machine learning-based decision support system for treatment of dento-maxillofacial malformations. METHODS: Patients (n = 574) with dento-maxillofacial deformities undergoing spiral CT during January 2015 to August 2020 were enrolled to train diagnostic models based on five different machine learning algorithms; the diagnostic performances were compared with expert diagnoses. Accuracy, sensitivity, specificity, and area under the curve (AUC) were calculated. The adaptive artificial bee colony algorithm was employed to formulate the orthognathic surgical plan, and subsequently evaluated by maxillofacial surgeons in a cohort of 50 patients. The objective evaluation included the difference in bone position between the artificial intelligence (AI) generated and actual surgical plans for the patient, along with discrepancies in postoperative cephalometric analysis outcomes. RESULTS: The binary relevance extreme gradient boosting model performed best, with diagnostic success rates > 90% for six different kinds of dento-maxillofacial deformities; the exception was maxillary overdevelopment (89.27%). AUC was > 0.88 for all diagnostic types. Median score for the surgical plans was 9, and was improved after human-computer interaction. There was no statistically significant difference between the actual and AI- groups. CONCLUSIONS: Machine learning algorithms are effective for diagnosis and surgical planning of dento-maxillofacial deformities and help improve diagnostic efficiency, especially in lower medical centers.

Orofacial Deformities in 3 Related Rhesus Monkeys (Macaca mulatta) Resembling Human Binder's Syndrome.

Binder's syndrome is a rare congenital deformity characterized by midface hypoplasia, particularly around the nasomaxillary area. Genetic etiology or developmental failure caused by prenatal exposure to teratological agents has been considered. In this article, we present 3 related rhesus monkeys born with orofacial deformities similar to those found in infants with the Binder phenotype. For the first time, a primate biomodel for this condition is presented. The clinical description and association with management and environmental factors are discussed. These findings reinforce the knowledge about the relationship between possible vitamin K metabolism interference and Binder's syndrome.

Manejo odontológico en paciente con síndrome de Cornelia de Lange / Dental treatment of a patient with Cornelia de Lange syndrome

El síndrome de Cornelia de Lange (SCdL) es un trastorno genético poco frecuente y se atribuye principalmente a mutaciones en los genes NIPBL, SMC3 y SMC1A. Sus principales características clínicas son múltiples anomalías congénitas, dimorfismo facial, hirsutismo, hipertricosis, retraso psicomotor, discapacidad intelectual, restricción del crecimiento prenatal y postnatal, anomalías de manos y pies, así como malformaciones congénitas que afectan a distintos órganos. En pacientes con SCdL es necesario hacer hincapié en la higiene oral debido a la discapacidad intelectual que puede presentarse y asegurarse de que se realiza una adecuada valoración y saneamiento dental de forma periódica con el fin de prevenir enfermedades bucodentales. El objetivo de este reporte de caso es describir el manejo odontológico de un paciente de 10 años con SCdL y revisar las características clínicas y hallazgos radiológicos presentes en la cavidad oral (AU)

Cornelia de Lange syndrome (CdLS) is a rare genetic disorder and is principally attributed to mutations in the NIPBL, SMC3 and SMC1A genes. The main clinical characteristics are multiple congenital anomalies, facial dimorphism, hirsutism, hypertrichosis, psychomotor retardation, intellectual disability, prenatal and postnatal growth restriction, hand and foot anomalies, as well as congenital malformations affecting different organs. In patients with CDLS, it is necessary to focus on oral hygiene due to the intellectual disability that may be present and to ensure that adequate dental valuation and hygiene is routinely performed in order to prevent oral diseases. The aim of this case report is to describe the dental management of a 10-year-old patient with CDLS and review the clinical characteristics and radiological findings that are present in the oral cavity (AU)

Binder's syndrome: A narrative review.

AIM: The classical features of Binder's syndrome (BS) have been widely reported, yet there is a lack of information on diagnostic and treatment challenges. Therefore, we aimed to review the literature on various aspects, including the role of a multidisciplinary approach in the management of BS. METHODS: A thorough literature search was performed on PubMed, Scopus, Embase, Ovid, Web of Science, EBSCOhost, and Google Scholar using the keywords"Maxillonasal dysplasia", "Maxillonasal dysostosis", "nasomaxillary hypoplasia", "Binder type", "Binder syndrome", "Binder phenotype", and "OMIM 155050". In addition, the articles published in the English language from the inception of the database until June 2021 were considered. RESULTS: The search of different databases yielded 958 publications, out of which 145 relevant articles were reviewed. The studies were categorised by year of publication, study design, and theme. Most of the studies were case reports (42%) followed by case series (19%). The most reported topic was based on different surgical techniques (42%). Finally, the results were synthesised into a narrative review. CONCLUSIONS: The need for a multidisciplinary approach involving clinicians from different specialities is highlighted. Future research is warranted to develop concrete clinical guidelines for the management of this syndrome.

Surgical Treatment for Patients With Binder Syndrome, Clinical Features and Associated Symptoms: A Systematic Review.

BACKGROUND: Binder syndrome is a rare congenital malformation of the midface. Since 1990, no systematic review has been conducted on this condition. Our study aims to review and collect the surgical treatment options available for correcting nasomaxillary dysplasia and the information available in the international literature on Binder syndrome and associated symptoms. METHODS: This systematic literature review was registered in the PROSPERO (International Prospective Register of Systematic Reviews) database. Systematic literature searches were performed in Medline, Scopus, the Cochrane Library, and Web of Science databases. Study quality assessment was performed according to recommendations from the Cochrane Handbook for Systematic Reviews of Diagnostic Test Accuracy. RESULTS: The authors identified 200 articles from our database research. One hundred sixty-nine articles were excluded after the title and abstract screening. Seven articles were excluded after screening the full text. In total, 24 clinical studies were included: 14 studies aim to describe a surgical option and 10 articles treat information about Binder syndrome or associated features. Most of the studies (12/14 studies) performed rhinoplasties to treat nasomaxillary dysplasia. The 2 other articles evaluated maxillary osteotomy techniques. Only 3 articles compared the results obtained from different surgical techniques. CONCLUSIONS: No optimal surgical treatment plan has been developed, even if numerous surgical options are available. Etiology is still uncertain.

Early surgical intervention for nasal deformity in Binder's syndrome.

OBJECTIVE: To assess benefits of surgical intervention at an early age and focus on surgical techniques using exclusively autologous cartilage grafts. METHODS: Five children aged 8-15 years, treated between March 2016 and 2020, underwent augmentation rhinoplasty using autologous cartilage, with post-operative follow up ranging from 1.5 to 2 years. Photographs and Rhinoplasty Outcome Evaluation questionnaire scores were examined. RESULTS: Augmentation was achieved without complications. No restricted skin or mucosal pockets were encountered. Patients showed improved confidence and perspective with regard to self-image, and were less socially self-conscious. There was significant improvement in all Rhinoplasty Outcome Evaluation questionnaire scores. CONCLUSION: Early surgery allows augmentation with easier release of skin and mucosal pockets, and without the tension otherwise encountered if primary augmentation is performed at a later age. It offers a chance to improve confidence and self-perception in a growing child conscious about their appearance, which may make them socially withdrawn. Autologous cartilage gives better results than bone grafts, and is comparable with silicone but without its complications.

Our Strategy in Management of Maxillonasal Dysplasia in Pediatric Patients.

ABSTRACT: Maxillonasal dysplasia or Binder syndrome is an uncommon condition. It is a congenital disease characterized by undergrowth of the central face and may include elements of the nose and upper jaw. The hallmark of the deformity is a retruded mid-face and an extremely flat nose. The timing and the surgical approaches for management of such deformity are still controversial. In this paper, we are going to present our experience in management of Binder syndrome in children. Seven children ranging in age from 6 to 13 years with classic features of Binder syndrome were operated upon in this study. The surgical approach was done at an early age and included 3 main components; nasal dorsum augmentation by costal cartilage graft, maxillary augmentation by rib grafts and columellar reconstruction by VY-plasty and strut grafts. Patients were followed for up to 6 years. Excellent results were obtained in all patients with this surgical procedure. Half of our patients required more than 1 surgery but none of them required any secondary orthognathic surgery. Therefore, we recommend that children with Binder syndrome should be managed at an early age at least for correction of their nasal deformities. Maxillary augmentation can also be done simultaneously or delayed for a second stage. During the second stage, further nasal augmentation can be accomplished. Definitive orthognathic surgeries have to be postponed to adolescence. This strategy can dramatically improve the patient aesthetic and alleviate the psychological upset without much disturbance of the facial growth.

Dinámica relacionada con la edad de parámetros antropométricos craneofaciales y su asociación con el tipo de oclusión dental en seres humanos de ambos géneros de: 17 a 24 años / Age-related dynamics of craniofacial anthropometric parameters and their association with the type of dental occlusion in humans of both genders aged 17­24 years

El propósito del estudio fue analizar la dinámica de los cambios relacionados con la edad de las estructuras craneofaciales de acuerdo con los parámetros antropométricos en hombres y mujeres de 17 a 24 años, y determinar la correlación, definida por análisis antropométrico, entre las formas de morfología craneofacial y los tipos de anomalías dento-maxilares. Materiales y métodos: Se realizó un examen antropométrico de 851 individuos (418 hombres y 433 mujeres) de 17 a 24 años y la evaluación de 45 mediciones entre puntos de referencia de tejidos blandos. Además, se realizó una valoración odontológica general, que incluyó la clasificación de las anomalías dentomaxilares. Resultados: Se detectó que el ancho de la cabeza, el largo de la cabeza, así como el ancho de la mandíbula y el ancho morfológico facial en las hembras alcanzan valores máximos de crecimiento a la edad de 20 años. Además, se obtuvieron datos que indicaron la continuación del crecimiento de los parámetros antropométricos craneofaciales mencionados anteriormente en los hombres durante el período de transición de la adolescencia a la edad adulta. Conclusión: la mordida profunda distal es la anomalía dento-maxilar (DMAn) más común en hombres y mujeres bielorrusos de 17 a 24 años. Junto con esto, la frecuencia de ocurrencia de maloclusión de mordida profunda distal en individuos con morfología craneofacial dolicocefálica es mayor en hombres y mujeres en comparación con otras formas de morfología craneofacial. La mordida profunda distal se acompaña de la disminución de los parámetros antropométricos de la altura facial, en comparación con la mordida mesial y abierta en la que hay un aumento estadístico. Además, se observa una disminución estadística en el valor de la profundidad facial inferior en individuos diagnosticados con mordida distal, en comparación con individuos con mordida mesial y normal. Estos resultados obtenidos del análisis antropométrico podrían ser útiles en el diagnóstico de anomalías dentofaciales y dento-maxilares, y en la simulación de resultados de tratamiento estéticamente estables.

The purpose of the study was to analyze the dynamics of age-related changes of craniofacial structures according to anthropometric parameters in males and females aged 17­24 years, and to determine the correlation, defined by anthropometric analysis, between forms of craniofacial morphology and the type of dento-maxillary anomalies. Materials and Methods: An anthropometric examination of 851 individuals (418 males and 433 females) aged 17­24 years and the evaluation of 45 measurements between soft tissue landmarks was performed. In addition, general dental assessment was conducted, which included the classification of the dento-maxillary anomalies. Results: It was detected that the head width, head length, as well as the mandible width, and the morphological facial width in females reach growth peak values at the age of 20 years. Furthermore, data was obtained which indicated growth continuation of the stated above craniofacial anthropometric parameters in males during the transitional period from adolescence to adulthood. Conclusion: Distal deep bite is the most commonly prevailed dento-maxillary anomalies (DMAn) in Belarusian men and women aged 17­24 years. Along with this, the frequency of distal deep bite malocclusion occurrence in individuals with dolichocephalic craniofacial morphology is higher in men and women when compared to other forms of craniofacial morphology. Distal deep bite is accompanied by the decrease of facial height anthropometric parameters, when compared to mesial and open bite in which there is a statistical increase. Moreover, a statistical decrease in the value of the inferior facial depth is observed in individuals diagnosed with distal bite, when compared to individuals with mesial and normal bite. These obtained results from anthropometric analysis could be useful in diagnosis of dentofacial and dento-maxillary anomalies, and in the simulation of aesthetically stable treatment results

Alternativas terapéuticas para el síndrome de apnea e hipoapnea obstructiva del sueño en niños con anomalías intermaxilares sagitales o transversales: Revisión narrativa / Therapeutic alternatives for obstructive sleep apnea syndrome in children with sagittal and transverse intermaxillary anomalies: Narrative review

RESUMEN: Introducción: El síndrome de apnea e hipoapnea obstructiva del sueño corresponde a un trastorno respiratorio del sueño muy prevalente en niños. Muchas alternativas terapéuticas buscan controlar los factores desencadenantes y la progresión de los signos y síntomas. El objetivo de esta revisión es describir los efectos de los tratamientos para síndrome de apnea e hipoapnea del sueño en niños con anomalías intermaxilares sagitales o transversales. Material y método: Se realizó una búsqueda electrónica en las bases de datos: PubMed, Epistemónikos, EBSCO, TripDataBase y The Cochrane Library, con las palabras clave: "Obstructive sleep apnea", "Obstructive sleep apnoea", "Sleep apnea syndrome", "Sleep apnoea syndrome", "Sleep apnea", "Sleep apnoea", "hypoapnea", "OSA", "Treatment", "Therapy", "Children", "Pediatric", "Craniofacial abnormalities", "Craniofacial anomalies", "Craniofacial abnormality", "Orthodontic" y una búsqueda retrógrada en los textos seleccionados. Resultados: Se incluyeron 23 artículos; 12 ensayos clínicos, 6 revisiones sistemáticas, 1 estudio observacional y 4 revisiones narrativas. Conclusión: Los tratamientos ortopédicos para el síndrome de apnea e hipoapnea son la expansión rápida maxilar y dispositivos de avance mandibular. No se encontró suficiente evidencia para determinar que estos dispositivos resuelvan completamente el síndrome, pero disminuyen el índice de apnea e hipoapnea y sus signos y síntomas.

ABSTRACT: Introduction: Obstructive sleep apnea and hypoapnea syndrome refers to a respiratory sleep disorder with an increased prevalence among children. There are many therapeutic alternatives, focused on controlling trigger factors and the signs and symptoms progression. The objective of this review is to describe the effects of the available treatments for sleep apnea and hypoapnea syndrome in children with sagittal and transverse intermaxillary anomalies. Materials and Method: An electronic search was performed in PubMed database, Epistemonikos, EBSCO, Tripdatabase, and The Cochrane Library, using the keywords "Obstructive sleep apnea", "Obstructive sleep apnoea", "Sleep apnea syndrome", "Sleep apnoea syndrome", "Sleep apnea", "Sleep apnoea", "hypoapnea", "OSA", "Treatment", "Therapy", "Children", "Pediatric", "Craniofacial abnormalities", "Craniofacial anomalies", "Craniofacial abnormality" and "Orthodontic". Also, a retrieval search in the selected articles references was performed. Results: 23 articles were included; 12 clinical trials, 6 systematic reviews, 1 observational study and 4 narrative reviews. Conclusion: There are two types of orthopedic treatment for sleep apnea and hypoapnea syndrome in children with sagittal and transverse intermaxillary anomalies: rapid maxillary expansion and mandibular advancement devices. There was not enough evidence to determine that these devices achieve the syndrome's complete resolution. An important decrease in the apnea and hypoapnea index and signs and symptoms were observed.

Comprehensive correction of maxillofacial bone deformity-consideration and combined application of orthognathic surgery and facial contouring surgery.

The maxillofacial skeleton is the basis of the contour of the face. Orthognathic surgery and facial contouring surgery change jaw tissue and affect facial appearance in different manners. Orthognathic surgery is the main method to correct dental and maxillofacial deformities. It changes the shape of the jaw and improves the occlusal relationship by changing the three-dimensional position of the jaw. Facial contouring surgery mainly adopts the method of "bone reduction", which changes the "amount"of the jawbone by cutting a part of the bone tissue to improve the facial appearance, generally without changing oral function. The combined use of orthognathic surgery and facial contouring surgery is becoming increasingly common in clinical practice. This also requires oral and maxillofacial surgeons to have a holistic consideration of the comprehensive correction of maxillofacial bone deformity, and to perform comprehensive analysis of jaw deformities and jaw plastic surgery to achieve the most ideal results. The author's team has been engaged in the clinical work of orthognathic surgery and facial contouring surgery and accumulated rich clinical experience in the comprehensive correction of maxillofacial bone deformity. In this article, the indications, treatment goals, treatment modes, treatment methods, and key points in the surgical operations of comprehensive maxillofacial bone surgery were summarized.

[Obstructive sleep apnea in microtia children with maxillofacial dysostosis].

Children with microtia are often associated with maxillofacial dysostosis, such as Treacher Collins syndrome, Goldenhar syndrome, and Nager syndrome, and they are prone to suffer from obstructive sleep apnea（OSA）. Obstruction widely occurred in the upper airway is the main mechanism of OSA in these children, and dysplasia of the pharynx and neurodevelopmental abnormalities may also participate. Early diagnosis requires symptom screening and polysomnography. Imaging techniques and endoscopy can be adopted to fully assess the upper airway status to guide further treatment. According to the child's condition and the main obstruction site, treatment methods include maxillofacial deformity correction, continuous positive pressure ventilation and tracheotomy. OSA in microtia children with maxillofacial dysostosis needs to be identified and treated in time to reduce the adverse effects on the growth and development of children.

Analytical study of the psychosocial impact of malocclusion and maxillofacial deformity in patients undergoing orthodontic treatment.

Patients whose with facial appearance involves dental anomalies and malocclusion face an increased prevalence of various psychosocial problems such as a high level of social anxiety, social avoidance, and low quality of life. This study investigates the patients with craniofacial anomalies and their psychological adjustment concerning the facial and dental appearance. It also evaluates the expectations of this patient group from the orthodontic treatment. Two steps were done in this study. In the first step, translation and validation of the Derriford Appearance Scale (DAS59), The Psychosocial Impact of Dental Aesthetics Questionnaire (PIDAQ), and Patient Expectation from the Orthodontic Treatment (PEOTQ) questionnaires into Maithili were done, and then the main study was conducted using these valid questionnaires. This was a cross-sectional study conducted on the patients with congenital craniofacial anomalies visiting the orthodontics department of Patna Dental College and Hospital, Patna (Bihar). All the patients received the Maithili DAS, Maithili PIDAQ and Patients' Expectation from the orthodontic treatment questionnaires. The Maithili version of DAS59, PIDAQ and PEOTQ were developed with outstanding reliability and validity. A significant difference between PIDAQ (p<0.001) and DAS59 scores (p<0.001) was found. In females, the total PIDAQ score was significantly higher as compared to males, but there was no association of DAS scores with gender. Place of residence showed no association with PIDAQ and DAS59 scores in patients. Patients and controls had significant differences between various items, and a comparison was made in terms of expectation from the orthodontic treatment. Altered facial and dental appearance in patients with craniofacial anomalies showed a significant psychological impact.

Long-term maxillary anteroposterior changes following maxillary protraction with or without expansion: A meta-analysis and meta-regression.

BACKGROUND: Maxillary protraction with or without expansion is a widely known orthopedic treatment modality in growing skeletal Class III patients. However, limited data are available regarding the outcomes of long-term changes in the maxilla. Aim of this meta-analysis was to assess the effectiveness of the long-term maxillary anteroposterior changes following a facemask therapy with or without rapid maxillary expansion in growing skeletal Class III patients. METHODS: A comprehensive literature search was conducted using the databases of PubMed, Science Direct, Web of Science, and Embase. Randomized controlled trials and cohort studies, published up to Sep. 2020, with maxillary protraction and/or expansion as keywords were included in this meta-analysis. Risk of bias within and across studies were assessed using the Cochrane tools (RoB2.0 and ROBINS-I) and GRADE approach. Overall and subgroup comparisons with the random-effect model were performed in this meta-analysis. Meta-regression models were designed to determine potential heterogeneity. RESULTS: There was a statistically significant increase (Mean difference, 2.29°; 95% confidence interval, 1.86-2.73; and p < 0.001 after facemask (FM) protraction. Mean difference, 1.73°; 95% confidence interval, 1.36-2.11; and p < 0.001 after rapid maxillary expansion(RME) and facemask protraction) in the Sella-Nasion-A point (SNA) angle in the treatment groups as compared with the control groups, when measured during the less than 3-year follow-up period. However, no statistically significant changes (Mean difference, 0.28°; 95% confidence interval, -0.57-1.13; and p = 0.52 after facemask protraction. Mean difference, 0.34°; 95% confidence interval, -0.64-1.33; and p = 0.50 after rapid maxillary expansion and facemask protraction) were observed in the SNA angle in the groups, when measured after 3 years of follow-up. Meta-regression analysis also showed that with increased follow-up duration, the effectiveness of maxillary protraction decreased. CONCLUSION: This meta-analysis revealed that maxillary protraction therapy could be effective for a short-term in correcting maxillary hypoplasia and the treatment result was not affected by mean age and sex. However, with increased follow-up duration, the sagittal maxillary changes gradually decreased. Limitations on this review were only the SNA angle was used and clinical heterogeneity was not discussed. The quality of evidence was moderate. Further long-term observational studies are necessary for a comprehensive evaluation of the effects on maxillary skeletal changes.

Lacrimal drainage anomalies in Tessier cleft 3 with unilateral anophthalmos.

Bilateral Tessier cleft types 3 and 4 are rare and commonly involve the lacrimal drainage system owing to their anatomical location. Such clefts commonly present with associated ocular anomalies and include colobomatous eyelids, hypertelorism, microphthalmia, punctal or canalicular agenesis, and nasolacrimal duct obstruction or exstrophy. The current report presents an 18-month-old baby with bilateral Tessier cleft 3 with a unilateral anophthalmos, symmetrical eyelid colobomas, and lacrimal drainage anomalies. The lacrimal anomalies noted include small lacrimal sac with inferior canaliculus on the right side and upper and lower punctal and canalicular agenesis on the left side. Computed tomographic dacryocystography demonstrated unilateral lacrimal sac and bilateral maldevelopment of the bony nasolacrimal duct.

Secondary Rhinoplasty in Binder Syndrome: Considerations and Management of Complex Problem With Heterologous Bone Graft.

ABSTRACT: Binder Syndrome occurs in less than 1 per 10,000 live births. Mean features of the syndrome include arhinoid face, abnormal position of the nasal bones, intermaxillary hypoplasia with associated malocclusion, reduced or absent anterior nasal spine, atrophy of the nasal mucosa and absence of the frontal sinus. Treatment of these facial deformities is obviously surgical. In the present work, the authors describe, step by step, their technique in secondary rhinoplasty in a 36-years-old patient affected by Binder Syndrome. In this case, the authors used autologous cartilage graft and heterologous bone graft.Satisfying results are achieved in 12 months of follow-up: graft resorption is acceptable, position of the graft is stable, the authors have no signs of infection and the patient is satisfied with the aesthetical and functional results. The authors believe that the first option, in complex nose's reconstruction, is the use of autologous grafts but, the use of cartilage heterologous bone graft should be taken in account, in the future, as a secondary option in Binder Syndrome and in malformed patients.

Interpositional collagenated cancellous bone blocks for nasal dorsum augmentation: A new technique for nasomaxillary hypoplasia treatment.

Nasomaxillary hypoplasia is a rare congenital malformation involving the middle third of the face. The present paper describes a novel technique for restoring the nasal projection in a patient with nasomaxillary hypoplasia, analyses its advantages and limitations, and discusses its potential applicability in other similar contexts. After orthognathic surgery, lateral osteotomies of the nasal bones were performed integrally with a piezoelectric device using a long cutting saw tip through the intraoral approach. The nasal bones were then projected by interpositioning two triangular-shaped collagenated cancellous bone graft blocks on each side in the osteotomies between the nasal and the frontal processes of the maxillary bones. Cone-beam computed tomography (CBCT) data was used to perform a morphometric analysis at one and 12 months of follow-up through image superimposition, which revealed a stable increased projection of the nasal dorsum and an anterior nasal spine (ANS) of 5.18 mm and 5.52 mm, respectively. The results of this case suggest that the technique affords satisfactory nasal dorsum augmentation while avoiding the use of permanent foreign materials, with minimal morbidity, no unsightly and visible scars, great patient satisfaction, and adequate stability at 12 months of follow-up.