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Objectives Hyperbaric oxygen therapy ( HBOT) is used clinically in irradiation-induced injury to healthy tissues, but the effectiveness and working mechanism remain unclear. This study examined the effects of HBOT on irradiated salivary glands and tongue in a mouse model. Materials and Methods Mice were irradiated with a single dose (15 Gy) in the head and neck region and subjected to HBOT, either before or after irradiation. During the course of the treatments, salivary flow rates were measured and at different time points after radiation (2, 6, 10 and 24 weeks), salivary glands and tongue were harvested and (immuno) histochemically analysed. Results Proliferation and blood vessel density in salivary glands were enhanced by HBOT in the medium term (10 weeks after irradiation), while salivary flow rates were not influenced. In the long term, irradiation-induced proliferation in the muscle tissue of the tongue was decreased by HBOT. Conclusion Hyperbaric oxygen therapy ( HBOT) appears to stimulate regeneration or protection of salivary gland tissue following radiation therapy. Possible implications of the effect of HBOT on muscle tissue of the tongue for the prevention of dysphagia and trismus are discussed. This study provides insights on the cellular changes after HBOT and encourages further research on this topic to achieve a better implementation of the therapy in humans. [ABSTRACT FROM AUTHOR]

Objectives: To investigate whether timing, accumulation and trajectories of poverty are associated with dental caries in young adolescents., Methods: The study was conducted within the Generation R Study, which is an ongoing population-based prospective cohort study conducted in Rotterdam, the Netherlands. This study included 2653 children. Information about household income and number of children and adults living in a household at six time points from pregnancy to 13 years old was retrieved from parental questionnaires to construct the poverty variable. Dental caries was assessed with the decayed, missing and filled teeth index through intraoral photographs at the age of 13 years. Sociodemographic and oral health-related characteristics were included as possible confounders. The association between poverty and dental caries was analysed on the basis of the three lifecourse theories, that is, critical period, cumulative risk and social mobility model. For the latter, we used latent class growth analysis (LCGA) to identify poverty trajectories over time. Next, the associations were studied with Hurdle Negative Binomial Models., Results: Poverty at birth and intermittent poverty up to the age of 13 were significantly associated with dental caries at 13 years of age (OR 1.41, 95% CI 1.01-1.99; OR 1.36, 95% CI 1.01-1.83 respectively) and with an increased mean number of decayed teeth by 34% (95% CI 1.02-1.76; 95% CI 1.05-1.71, respectively). LCGA showed four trajectories for the probabilities of poverty. All trajectories were significantly associated with dental caries at 13 years of age, with the 'downward mobility' trajectory showing the strongest association with dental caries (OR 1.55, 95% CI 1.05-2.29) and an increasing mean number of decayed teeth by 58% (95% CI 1.18-2.12) than the 'stable absent' trajectory., Conclusion: Poverty at birth, intermittent poverty and downward poverty trajectory were associated with higher odds and higher mean number of decayed teeth at 13 years of age. The three lifecourse models influenced dental caries status during adolescence, hence strategies and policies targeted to improve socioeconomic conditions on deprived children should be implemented., (© 2024 The Author(s). Community Dentistry and Oral Epidemiology published by John Wiley & Sons Ltd.)

To determine the skeletal changes after midface surgery in patients with syndromic craniosynostosis who underwent Le Fort III (LFIII), monobloc (MB), or facial bipartition (FB). This was a retrospective study including 75 patients: 33 treated by LFIII, 29 by MB, and 13 by FB. Twenty-five had a diagnosis of Apert, 39 Crouzon, and 11 craniofrontonasal syndrome. A three-dimensional mesh was created from the preoperative scan and registered to the postoperative scan to visualise the advancement. LFIII at age 7-12 years effectuated a higher mean advancement in the maxillary (15.5 mm) and zygomatic (7.6 mm) regions when compared to ≥13 years (10.2 mm and 5.5 mm). After MB, mean advancement of the fronto-orbital region was higher at <7 years (16.4 mm), and similarly lower at ages 7-12 (13.8 mm) and ≥13 (12.5 mm). The mean preoperative inter-dacryon distance (34.4 ± 4.4 mm) was reduced by 8.7 ± 4.2 mm after FB without distraction (n = 10). More advancement was seen when midface surgery was performed at a younger age, due to more severe cases and a desire for overcorrection. The highest mean advancement was observed in the fronto-orbital region. Antero-inferior rotational movement was seen after all three techniques., (Copyright © 2024 The Author(s). Published by Elsevier Inc. All rights reserved.)

Background: Craniosynostosis, a congenital condition characterized by the premature fusion of cranial sutures, necessitates objective methods for evaluating cranial morphology to enhance patient treatment. Current subjective assessments often lead to inconsistent outcomes. This study introduces a novel, quantitative approach to classify craniosynostosis and measure its severity., Methods: An artificial neural network was trained to classify normocephalic, trigonocephalic, and scaphocephalic head shapes based on a publicly available dataset of synthetic 3D head models. Each 3D model was converted into a low-dimensional shape representation based on the distribution of normal vectors, which served as the input for the neural network, ensuring complete patient anonymity and invariance to geometric size and orientation. Explainable AI methods were utilized to highlight significant features when making predictions. Additionally, the Feature Prominence (FP) score was introduced, a novel metric that captures the prominence of distinct shape characteristics associated with a given class. Its relationship with clinical severity scores was examined using the Spearman Rank Correlation Coefficient., Results: The final model achieved excellent test accuracy in classifying the different cranial shapes from their low-dimensional representation. Attention maps indicated that the network's attention was predominantly directed toward the parietal and temporal regions, as well as toward the region signifying vertex depression in scaphocephaly. In trigonocephaly, features around the temples were most pronounced. The FP score showed a strong positive monotonic relationship with clinical severity scores in both scaphocephalic (ρ = 0.83, p < 0.001) and trigonocephalic (ρ = 0.64, p < 0.001) models. Visual assessments further confirmed that as FP values rose, phenotypic severity became increasingly evident., Conclusion: This study presents an innovative and accessible AI-based method for quantifying cranial shape that mitigates the need for adjustments due to age-specific size variations or differences in the spatial orientation of the 3D images, while ensuring complete patient privacy. The proposed FP score strongly correlates with clinical severity scores and has the potential to aid in clinical decision-making and facilitate multi-center collaborations. Future work will focus on validating the model with larger patient datasets and exploring the potential of the FP score for broader applications. The publicly available source code facilitates easy implementation, aiming to advance craniofacial care and research., (© 2024 The Authors. Journal of Anatomy published by John Wiley & Sons Ltd on behalf of Anatomical Society.)

This study was performed to evaluate the efficacy of outcome measures for the orofacial domain included in the International Consortium for Health Outcomes Measurement Standard Set for Cleft Lip and Palate (ICHOM-SCS). In this multicentre study involving two cleft centres, suggestions to optimize the type and timing of outcome measures were made based on data and clinical experience. Patient-reported outcome measures (PROMs) (CLEFT-Q Jaw, Teeth, Eating/Drinking; Child Oral Health Impact Profile-Oral Symptoms Scale (COHIP-OSS)) and clinical outcome measures (caries experience and dental occlusion) data were collected retrospectively for age 5, 8, 10, 12, 19, and 22 years. The data were categorized by cleft type and analysed within and between age groups using Spearman correlation, the distribution of responses per item, a two-sample test for equality of proportions, and effect plots. Most correlations between PROMs and clinical outcome measures were weak (r < 0.5), suggesting PROMs and clinical outcome measures complement each other. The COHIP-OSS and CLEFT-Q Eating/Drinking barely detected problems in any patient category and are no longer recommended. A suitable alternative appears complex to find; outcomes of this study and the recent literature doubt an added value. Similar problems were found in the CLEFT-Q Jaw at time-point 12 years. Therefore, time-points 15 and 17 years are currently suggested., (Copyright © 2024 The Author(s). Published by Elsevier Inc. All rights reserved.)

Background  The best timing of closure of the hard palate in individuals with cleft lip, alveolus, and palate (CLAP) to reach the optimal speech outcomes and maxillary growth is still a subject of debate. This study evaluates changes in compensatory articulatory patterns and resonance in patients with unilateral and bilateral CLAP who underwent simultaneous closure of the hard palate and secondary alveolar bone grafting (ABG). Methods  A retrospective study of patients with nonsyndromic unilateral and bilateral CLAP who underwent delayed hard palate closure (DHPC) simultaneously with ABG at 9 to 12 years of age from 2013 to 2018. The articulatory patterns, nasality, degree of hypernasality, facial grimacing, and speech intelligibility were assessed pre- and postoperatively. Results  Forty-eight patients were included. DHPC and ABG were performed at the mean age of 10.5 years. Postoperatively hypernasal speech was still present in 54% of patients; however, the degree of hypernasality decreased in 67% ( p  < 0.001). Grimacing decreased in 27% ( p  = 0.015). Articulation disorders remained present in 85% ( p  = 0.375). Intelligible speech (grade 1 or 2) was observed in 71 compared with 35% of patients preoperatively ( p  < 0.001). Conclusion  This study showed an improved resonance and intelligibility following DHPC at the mean age of 10.5 years, however compensatory articulation errors persisted. Sequential treatments such as speech therapy play a key role in improvement of speech and may reduce remaining compensatory mechanisms following DHPC., Competing Interests: Conflict of Interest None declared., (The Author(s). This is an open access article published by Thieme under the terms of the Creative Commons Attribution License, permitting unrestricted use, distribution, and reproduction so long as the original work is properly cited. ( https://creativecommons.org/licenses/by/4.0/ ).)

Purpose: To evaluate the self-perceived oral health and aesthetics of the dentition and jaw in patients with different types of oral cleft, measured by patient-reported outcome measures (PROMs). Additionally, to compare the results of the PROMs between cleft lip and or/palate (CL/P) patients and non-affected controls., Methods: 420 CL/P patients treated at the cleft team of the Erasmus Medical Center, Rotterdam, The Netherlands, were included, and 138 non-cleft patients were recruited as control-group. Patient's perceptions were retrospectively evaluated using the CLEFT-Q Teeth for dental aesthetics at ages 8, 12 and 22, CLEFT-Q Jaw for jaw aesthetics at ages 12 and 22, and the Child Oral Health Impact Profile-Oral Symptoms Subscale (COHIP-OSS) for oral health at ages 8 and 12. One-way ANOVA was used to compare differences in oral health and aesthetic perceptions among age-groups, cleft types, as well as between cases and controls., Results: CL/P patients were significantly less satisfied than controls with their dental aesthetics (p = 0.001). CL/P patients reported significantly lower satisfaction on CLEFT-Q Teeth scores at ages 8 and 12, than at 22 years (p < 0.001). Patients with the most extensive cleft phenotype, Cleft Lip and Palate (CLAP), reported lowest satisfaction on the CLEFT-Q Teeth. No differences in perceptions of oral health nor in aesthetics of the jaw were found in the different cleft types, ages, nor in study versus control group., Conclusion: This study found differences in self-perceived dental aesthetics: CL/P patients are less satisfied than non-affected controls. CLAP patients are least satisfied, but satisfaction increases with age., (© 2024. The Author(s).)

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., (© 2024. The Author(s).)

To determine the effect of midface surgery on soft tissue changes and their relationship to hard tissue changes in patients with syndromic craniosynostosis. A retrospective analysis of patients who had undergone Le Fort III (LFIII), monobloc (MB), or facial bipartition (FB) was conducted. A 3D soft tissue mesh was generated from the preoperative scan and registered to the postoperative scan, after which the advancement was visualised. A total of 68 patients were included: 28 had undergone LFIII, 27 MB, and 13 FB. The included diagnoses were Apert (n = 23), Crouzon (n = 34), and craniofrontonasal syndrome (n = 11). After LFIII, most soft tissue advancement was seen around subnasale and pronasale (mean 15.1 ± 5.9 mm and 14.7 ± 5.7 mm, at age 7-12 years). After MB, a greater hard tissue than soft tissue advancement was seen for most landmarks, showing a high positive correlation. In patients undergoing FB without distraction (n = 10), mean preoperative inter-canthal distance was 48.9 mm, this reduced by 6.9 mm postoperatively. This study provides a comprehensive overview of the outcomes after midface surgery using 3D quantification for a better understanding of the soft tissue changes and their relationship to hard tissue changes., Competing Interests: Competing interests None., (Copyright © 2024 The Authors. Published by Elsevier Inc. All rights reserved.)

Bone Health Index (BHI) has been proposed as a useful instrument for assessing bone health in children. However, its relationship with fracture risk remains unknown. We aimed to investigate whether BHI is associated with bone mineral density (BMD) and prevalent fracture odds in children from the Generation R Study. We also implemented genome-wide association study (GWAS) and polygenic score (PGS) approaches to improve our understanding of BHI and its potential. In total, 4150 children (49.4 % boys; aged 9.8 years) with genotyped data and bone assessments were included in this study. BMD was measured across the total body (less head following ISCD guidelines) using a GE-Lunar iDXA densitometer; and BHI was determined from the hand DXA scans using BoneXpert®. Fractures were self-reported collected with home questionnaires. The association of BHI with BMD and fractures was evaluated using linear models corrected for age, sex, ethnicity, height, and weight. We observed a positive correlation between BHI and BMD (ρ = 0.32, p-value<0.0001). Further, every SD decrease in BHI was associated with an 11 % increased risk of prevalent fractures (OR:1.11, 95 % CI 1.00-1.24, p-value = 0.05). Our BHI GWAS identified variants (lead SNP rs1404264-A, p-value = 2.61 × 10 -14 ) mapping to the ING3/CPED1/WNT16 locus. Children in the extreme tails of the BMD PGS presented a difference in BHI values of -0.10 standard deviations (95% CI -0.14 to -0.07; p-value<0.0001). On top of the demonstrated epidemiological association of BHI with both BMD and fracture risk, our results reveal a partially shared biological background between BHI and BMD. These findings highlight the potential value of using BHI to screen children at risk of fracture., Competing Interests: Declaration of competing interest None., (Copyright © 2024 The Authors. Published by Elsevier Inc. All rights reserved.)

Allogeneic stem-cell based regenerative medicine is a promising approach for bone defect repair. The use of chondrogenically differentiated human marrow stromal cells (MSCs) has been shown to lead to bone formation by endochondral ossification in immunodeficient pre-clinical models. However, an insight into the interactions between the allogeneic immune system and the human MSC-derived bone grafts has not been fully achieved yet. The choice of a potent source of MSCs isolated from pediatric donors with consistent differentiation and high proliferation abilities, as well as low immunogenicity, could increase the chance of success for bone allografts. In this study, we employed an immunodeficient animal model humanised with allogeneic immune cells to study the immune responses towards chondrogenically differentiated human pediatric MSCs (ch-pMSCs). We show that ch-differentiated pMSCs remained non-immunogenic to allogeneic CD4 and CD8 T cells in an in vitro co-culture model. After subcutaneous implantation in mice, ch-pMSC-derived grafts were able to initiate bone mineralisation in the presence of an allogeneic immune system for 3 weeks without the onset of immune responses. Re-exposing the splenocytes of the humanised animals to pMSCs did not trigger further T cell proliferation, suggesting an absence of secondary immune responses. Moreover, ch-pMSCs generated mature bone after 8 weeks of implantation that persisted for up to 6 more weeks in the presence of an allogeneic immune system. These data collectively show that human allogeneic chondrogenically differentiated pediatric MSCs might be a safe and potent option for bone defect repair in the tissue engineering and regenerative medicine setting., Competing Interests: Declaration of competing interest The authors declare the following financial interests/personal relationships which may be considered as potential competing interests: Eric Farrell reports financial support was provided by AO Foundation., (Copyright © 2024 The Author(s). Published by Elsevier Ltd.. All rights reserved.)

This study was performed to describe observed healthcare utilization and medical costs for patients with a cleft, compare these costs to the expected costs based on the treatment protocol, and explore the additional costs of implementing the International Consortium for Health Outcomes Measurement (ICHOM) Standard Set for Cleft Lip and Palate (CL/P). Forty patients with unilateral CL/P between 0 and 24 years of age, treated between 2012 and 2019 at Erasmus University Medical Center, were included. Healthcare services (consultations, diagnostic and surgical procedures) were counted and costs were calculated. Expected costs based on the treatment protocol were calculated by multiplying healthcare products by the product prices. Correspondingly, the additional expected costs after implementing the ICHOM Standard Set (protocol + ICHOM) were calculated. Observed costs were compared with protocol costs, and the additional expected protocol + ICHOM costs were described. The total mean costs were highest in the first year after birth (€5596), mainly due to surgeries. The mean observed total costs (€40,859) for the complete treatment (0-24 years) were 1.6 times the expected protocol costs (€25,198) due to optional, non-protocolized procedures. Hospital admissions including surgery were the main cost drivers, accounting for 42% of observed costs and 70% of expected protocol costs. Implementing the ICHOM Standard Set increased protocol-based costs by 7%., Competing Interests: Competing interests None., (Copyright © 2023 The Authors. Published by Elsevier Inc. All rights reserved.)

Background: Efficient identification of individuals at high risk of skin cancer is crucial for implementing personalized screening strategies and subsequent care. While Artificial Intelligence holds promising potential for predictive analysis using image data, its application for skin cancer risk prediction utilizing facial images remains unexplored. We present a neural network-based explainable artificial intelligence (XAI) approach for skin cancer risk prediction based on 2D facial images and compare its efficacy to 18 established skin cancer risk factors using data from the Rotterdam Study., Methods: The study employed data from the Rotterdam population-based study in which both skin cancer risk factors and 2D facial images and the occurrence of skin cancer were collected from 2010 to 2018. We conducted a deep-learning survival analysis based on 2D facial images using our developed XAI approach. We subsequently compared these results with survival analysis based on skin cancer risk factors using cox proportional hazard regression., Findings: Among the 2810 participants (mean Age = 68.5 ± 9.3 years, average Follow-up = 5.0 years), 228 participants were diagnosed with skin cancer after photo acquisition. Our XAI approach achieved superior predictive accuracy based on 2D facial images (c-index = 0.72, 95% CI: 0.70-0.74), outperforming that of the known risk factors (c-index = 0.59, 95% CI 0.57-0.61)., Interpretation: This proof-of-concept study underscores the high potential of harnessing facial images and a tailored XAI approach as an easily accessible alternative over known risk factors for identifying individuals at high risk of skin cancer., Funding: The Rotterdam Study is funded through unrestricted research grants from Erasmus Medical Center and Erasmus University, Rotterdam, Netherlands Organization for the Health Research and Development (ZonMw), the Research Institute for Diseases in the Elderly (RIDE), the Ministry of Education, Culture and Science, the Ministry for Health, Welfare and Sports, the European Commission (DG XII), and the Municipality of Rotterdam. G.V. Roshchupkin is supported by the ZonMw Veni grant (Veni, 549 1936320)., Competing Interests: The authors declare the following financial interests which may be considered potential competing interests: The Erasmus MC Department of Dermatology has received an unrestricted research grant from SkinVision B.V. None of the authors received any direct fees for consulting or salary from the company. T.E. Sangers reports receiving personal consultancy fees from Mylan B.V. and speakers honoraria from Pfizer, Janssen-Cilag, UCB, and Eli Lilly. There are no other declarations of interest., (© 2024 The Author(s).)

Objectives: Patients with head and neck cancer are routinely screened for dental foci prior to radiotherapy (RT) to prevent post- RT tooth extractions associated with an increased risk of osteoradionecrosis. We evaluated the risk factors for post-RT tooth extraction to personalise dental screening and prevention protocols prior to RT., Materials and Methods: This retrospective cohort study included dentulous patients diagnosed with oropharyngeal cancer who had undergone radiation therapy at doses 60-70 Gy and achieved a disease-free survival of ≥ 1 year (N = 174). Risk factors were assessed using Cox regression models., Results: The cumulative incidence of post-RT tooth extraction was 30.7 % at 5 years. Main indications for extraction (n = 62) were radiation caries (n = 20) and periodontal disease (n = 27). Risk factors associated (p < 0.05) with radiation caries-related extractions included active smoking, alcohol abuse, poor oral hygiene, parotid gland irradiation, and mandibular irradiation. A high-dose volume in the mandible was associated with periodontal disease events., Conclusion: Post-RT extractions due to radiation caries were influenced by lifestyle factors and RT dose in the mandible and parotid glands. Periodontal disease-related extractions were primarily associated with the mandibular dose. During dental screening these post-RT risk factors should be taken into account to prevent osteoradionecrosis., Competing Interests: The authors declare that they have no known competing financial interests or personal relationships that could have appeared to influence the work reported in this paper., (© 2024 The Author(s).)

Background: Abnormalities of the hyoid bone are associated with impairment of oropharyngeal functions including feeding, swallowing, and breathing. Few studies have characterized anatomic abnormalities of the hyoid in patients with Robin sequence (RS), e.g. a less mineralized and voluminous hyoid. The purpose of this study was to compare normal hyoid bone morphology and hyoid bone morphology in children with isolated RS., Methods: Three-dimensional (3D) reconstructions of the hyoid bone were obtained from CT-imaging of children with RS and unaffected controls. A 3D morphable model was constructed using Principal Component Analysis (PCA). Partial least squares - Discriminant Analysis (PLS-DA) and multivariate analysis of variance (MANOVA) were used to characterize and compare hyoid shape differences between patients with RS and an age-matched control group., Results: The study included 23 subjects with RS (mean age 9.8 ± 10.3 months) and 46 age-matched control samples. A less voluminous hyoid was observed for the RS group with a larger lateral divergence of the greater horns compared to controls (MANOVA, p -value<0.001). The first shape variable from the PLS-DA model showed a significant correlation for the observed variance between the two groups (Spearman R  = -0.56, p -value<0.001). The control samples and 151 CT-scans of subjects up to age 4 years were used to create a 3D morphable model of normal hyoid shape variation ( n  = 197, mean age 22.1 ± 13.1 months). For the normal 3D morphable model, a high degree of allometric shape variation was observed along the first principal component., Conclusions: The 3D morphable models provide a comprehensive and quantitative description of variation in normal hyoid bone morphology, and allow detection of distinct differences between patients with isolated RS and controls., Competing Interests: The authors declare no potential conflicts of interest with respect to the research, authorship, and/or publication of this article., (© 2024 The Authors. Published by Elsevier Inc.)

Craniosynostosis, characterized by premature fusion of one or more cranial sutures, results in a distorted skull shape. Only three studies have assessed facial asymmetry manually in unicoronal synostosis patients. It is therefore important to understand how uni- and bicoronal synostosis affect facial asymmetry with a minimum risk of human bias. An automated algorithm was developed to quantify facial asymmetry from three-dimensional images, generating a mean facial asymmetry (MFA) value in millimeters to reflect the degree of asymmetry. The framework was applied to analyze postoperative 3D images of syndromic patients (N = 35) diagnosed with Muenke syndrome, Saethre-Chotzen syndrome, and TCF12-related craniosynostosis with respect to MFA values from a healthy control group (N = 89). Patients demonstrated substantially higher MFA values than controls: Muenke syndrome (unicoronal 1.74 ± 0.40 mm, bicoronal 0.77 ± 0.21 mm), Saethre-Chotzen syndrome (unicoronal 1.15 ± 0.20 mm, bicoronal 0.69 ± 0.16 mm), and TCF12-related craniosynostosis (unicoronal 1.40 ± 0.51 mm, bicoronal 0.66 ± 0.05 mm), compared with controls (0.49 ± 0.12 mm). Longitudinal analysis identified an increasing MFA trend in unicoronal synostosis patients. Our study revealed higher MFA in syndromic patients with uni- and bicoronal synostosis compared with controls, with the most pronounced MFA in Muenke syndrome patients with unilateral synostosis. Bicoronal synostosis patients demonstrated higher facial asymmetry than expected given the condition's symmetrical presentation., (Copyright © 2023 The Authors. Published by Elsevier Ltd.. All rights reserved.)

Maxillofacial injury is a common injury resulting from bicycle (including e-bike) and scooter accidents. With 80,000 admissions to emergency departments in 2019, bicycle accidents account for more than half of all traffic-related emergency department visits in the Netherlands. The United States reports approximately 130,000 injuries and 1000 fatalities related to cycling annually. This systematic review and meta-analysis was performed to examine the protective effect of helmets against maxillofacial injuries resulting from bicycle and scooter (including e-bike and e-scooter) accidents. After a systematic literature search, 14 studies were found to be eligible for this systematic review. Of these, 11 were included in the meta-analysis. None of the included studies focused on vehicles with motors (e-bikes and e-scooters); all focused only on non-motorized vehicles. All included studies were non-randomized, which could have led to bias in the pooled results. Data from the included studies were tested for heterogeneity using the binary random-effects model (DerSimonian-Laird method), and the odds ratio for the occurrence of maxillofacial injury in cyclists wearing a helmet versus those not wearing a helmet was calculated by random-effects meta-analysis. Patients who had worn a helmet suffered significantly fewer maxillofacial injuries than patients who had not, in bicycle accidents (odds ratio 0.682). In conclusion, wearing a helmet has a significant protective effect against maxillofacial injury, indicating the need for strict helmet legislation., Competing Interests: Competing interests None., (Copyright © 2023 The Authors. Published by Elsevier Inc. All rights reserved.)

The caries prevalence in patients with cleft lip and/or palate is higher in both the deciduous and permanent dentition, compared to non-cleft patients. Inadequate oral hygiene is one of the main causes. Additional, individualized advice concerning diet and oral hygiene, such as the additional advice of the Ivoren Kruis (Dutch association of dental hygienists), is of great importance. During curative treatment, attention should be paid to any dental anxiety and possible reduced compliance.

Objective: Mandibular reconstruction using patient-specific cage implants is a promising alternative to the vascularized free flap reconstruction for nonirradiated patients with adequate soft tissues, or for patients whose clinical condition is not conducive to microsurgical reconstruction. This study aimed to assess the biomechanical performance of 3D printed patient-specific cage implants designed with a semi-automated workflow in a combined cadaveric and retrospective case series study., Methods: We designed cage implants for two human cadaveric mandibles using our previously developed design workflow. The biomechanical performance of the implants was assessed with the finite element analysis (FEA) and quasi-static biomechanical testing. Digital image correlation (DIC) was used to measure the full-field strains and validate the FE models by comparing the distribution of maximum principal strains within the bone. The retrospective study of a case series involved three patients, each of whom was treated with a cage implant of similar design. The biomechanical performance of these implants was evaluated using the experimentally validated FEA under the scenarios of both mandibular union and nonunion., Results: No implant or screw failure was observed prior to contralateral bone fracture during the quasi-static testing of both cadaveric mandibles. The FEA and DIC strain contour plots indicated a strong linear correlation (r = 0.92) and a low standard error (SE=29.32με), with computational models yielding higher strain values by a factor of 2.7. The overall stresses acting on the case series' implants stayed well below the yield strength of additively manufactured (AM) commercially pure titanium, when simulated under highly strenuous chewing conditions. Simulating a full union between the graft and remnant mandible yielded a substantial reduction (72.7±1.5%) in local peak stresses within the implants as compared to a non-bonded graft., Conclusions: This study shows the suitability of the developed semi-automated workflow in designing patient-specific cage implants with satisfactory mechanical functioning under demanding chewing conditions. The proposed workflow can aid clinical engineers in creating reconstruction systems and streamlining pre-surgical planning. Nevertheless, more research is still needed to evaluate the osteogenic potential of bone graft insertions., Competing Interests: Declaration of competing interest The authors declare that they have no known competing financial interests or personal relationships that could have appeared to influence the work reported in this paper., (Copyright © 2023. Published by Elsevier Ltd.)

Introduction: Dental caries remains one of the most prevalent but preventable diseases among children worldwide and especially affects children with a lower socioeconomic status or ethnic minority background. It is important that all groups of children are reached by preventive interventions to reduce oral health inequalities. So far, it is unknown whether children from different social and ethnic groups benefit equally from potentially effective oral health interventions., Objectives: This scoping review aimed to identify European public health interventions that report their effect on dental caries across different social groups., Methods: Four databases were searched for studies evaluating the effect of oral health interventions on dental caries among children from 0 to 12 y, and studies were included when results were presented by children of different social groups separately., Results: A total of 14 studies were included, representing 4 different countries: 3 randomized and 11 nonrandomized studies. Most studies were performed at schools. Six studies showed results indicative of a reduction in oral health inequalities, 4 studies showed results that potentially widen oral health inequalities, and 5 studies showed results that were indicative of no impact on oral health inequalities. Interventions that contain early approaches, with a high frequency, approaching multiple levels of influence, and including at least the broader organizational or public policy level, may have the potential to reduce oral health inequalities among children from birth to young adolescence., Conclusion: We recommend researchers to perform high-quality intervention studies and to evaluate the effectiveness of oral health intervention always in different socioeconomic or ethnic groups separately, to better understand their contribution toward oral health (in)equalities., Knowledge Transfer Statement: This review offers insight in the differential effects that oral health interventions might have across different social groups. Its results can be used to develop interventions that might reduce oral health inequalities among children. Also, we recommend future researchers to always evaluate the effects of any preventive oral health measure in different social groups separately.

Objective: To reflect upon our non-surgical respiratory management by evaluating clinical outcomes regarding airway, feeding, and growth during the first year of life in patients with Robin Sequence., Design: Prospective study., Setting: Sophia Children's Hospital, Rotterdam, the Netherlands., Patients/ Participants: 36 patients with Robin Sequence who were treated between 2011 and 2021., Interventions: Positional therapy and respiratory support ., Main Outcome Measure(s): Data on respiratory outcomes included polysomnography characteristics and capillary blood gas values. Feeding outcomes were based on the requirement of additional tube feeding. Outcomes on growth were expressed as standard-deviation-scores (SDS) for weight-for-age (WFA) and height-for-age (HFA)., Results: Twenty patients were treated with positional therapy (PT), whilst the other 16 patients required respiratory support. Twenty-two patients presented with non-isolated Robin Sequence (RS). During the first year of life, obstructive apnea hypopnea index decreased, oxygen levels enhanced, and capillary blood gas values improved. Eighty-six percent (31/36) experienced feeding difficulties, which completely resolved in 71% (22/31) during their first year of life. From start treatment, to stop treatment, to the age of 1 year, the SDS WFA worsened from -0.40 to -0.33 to -1.03, respectively., Conclusions: Non-surgical respiratory treatment resulted in an improvement of respiratory outcomes to near normal during the first year of life in patients with RS. These patients often experience feeding difficulties and endure impaired weight gain up to 1 year of age, despite near normalization of breathing. The high prevalence of feeding difficulties and impaired weight for age indicate the urgency for early recognition and adequate treatment to support optimal growth.

Characteristics of patients with craniofacial microsomia (CFM) vary in type and severity. The diagnosis is based on phenotypical assessment and no consensus on standardized clinical diagnostic criteria is available. The use of diagnostic criteria could improve research and communication among patients and healthcare professionals. Two sets of phenotypic criteria for research were independently developed and based on multidisciplinary consensus: the FACIAL and ICHOM criteria. This study aimed to assess the sensitivity of both criteria with an existing global multicenter database of patients with CFM and study the characteristics of patients that do not meet the criteria. A total of 730 patients with CFM from were included. Characteristics of the patients were extracted, and severity was graded using the O.M.E.N.S. and Pruzansky-Kaban classification. The sensitivity of the FACIAL and ICHOM was respectively 99.6% and 94.4%. The Cohen's kappa of 0.38 indicated a fair agreement between both criteria. Patients that did not fulfill the FACIAL criteria had facial asymmetry without additional features. It can be concluded that the FACIAL and ICHOM criteria are accurate criteria to describe patients with CFM. Both criteria could be useful for future studies on CFM to create comparable and reproducible outcomes., Competing Interests: The authors report no conflicts of interest., (Copyright © 2023 The Author(s). Published by Wolters Kluwer Health, Inc. on behalf of Mutaz B. Habal, MD.)

Background: Even though dietary sugars are the most important nutrient for caries development, the disease process is dependent on other dietary practices. The intake of individual nutrient components cannot be evaluated separately from the overall diet which includes other nutrients, foods and habits. Therefore, the aim of this study was to investigate the association between adherence to dietary guidelines and dental caries., Methods: This study was embedded in the Generation R Study, conducted in Rotterdam, the Netherlands. In total, 2911 children were included in the present analyses. Dietary intake at the age of 8 years was assessed using food-frequency questionnaires. Diet quality scores were estimated, reflecting adherence to Dutch dietary guidelines. Dental caries was assessed at the age of 13 years using intra-oral photographs. Associations were estimated using multinomial logistic regression analyses, adjusted for sociodemographic characteristics and oral hygiene practices., Results: The prevalence of dental caries at the age of 13 years was 33% (n = 969). Better diet quality was associated with a lower occurrence of severe dental caries after adjustments for sociodemographic factors [e.g. highest vs. lowest quartile of diet quality: odds ratio (OR) 0.62, 95% confidence interval (CI) 0.39-0.98]. After additional adjustments for oral hygiene practices, this association was not statistically significant (OR 0.65, 95% CI 0.41-1.03)., Conclusion: Adherence to dietary guidelines has the potential to reduce dental caries in children; however, with proper oral hygiene practices, this relationship might be attenuated. To understand the role of dietary patterns and dental caries, the contributing role of daily eating occasions needs to be studied further., (© The Author(s) 2023. Published by Oxford University Press on behalf of the European Public Health Association.)

Objectives: Oral conditions are of high prevalence and chronic character within the general population. Identifying the risk factors and determinants of oral disease is important, not only to reduce the burden of oral diseases, but also to improve (equal access to) oral health care systems, and to develop effective oral health promotion programs. Longitudinal population-based (birth-)cohort studies are very suitable to study risk factors on common oral diseases and have the potential to emphasize the importance of a healthy start for oral health. In this paper, we provide an overview of the comprehensive oral and craniofacial dataset that has been collected in the Generation R study: a population-based prospective birth cohort in the Netherlands that was designed to identify causes of health from fetal life until adulthood., Methods: Within the multidisciplinary context of the Generation R study, oral and craniofacial data has been collected from the age of 3 years onwards, and continued at the age of six, nine, and thirteen. Data collection is continuing in 17-year-old participants., Research Outcomes: In total, the cohort population comprised 9749 children at birth, and 7405 eligible participants at the age of seventeen. Based on questionnaires, the dataset contains information on oral hygiene, dental visits, oral habits, oral health-related quality of life, orthodontic treatment, and obstructive sleep apnea. Based on direct measurements, the dataset contains information on dental caries, developmental defects of enamel, objective orthodontic treatment need, dental development, craniofacial characteristics, mandibular cortical thickness, and 3D facial measurements., Conclusions: Several research lines have been set up using the oral and craniofacial data linked with the extensive data collection that exists within the Generation R study., Clinical Relevance: Being embedded in a multidisciplinary and longitudinal birth cohort study allows researchers to study several determinants of oral and craniofacial health, and to provide answers and insight into unknown etiologies and oral health problems in the general population., (© 2023. The Author(s).)

It was the aim of the study to provide a three-dimensional evaluation of dento-skeletal effects following bone-borne vs tooth-borne mandibular midline distraction (MMD) and tooth-borne surgically assisted rapid maxillary expansion (SARME). A retrospective observational study was conducted. Cone beam computed tomography (CBCT) records were taken pre-operatively (T1), immediately post-distraction (T2) and 1 year post-operatively (T3). All included 30 patients had undergone MMD (20 bone-borne MMD; 10 tooth-borne MMD). A total of 20 bone-borne MMD and 8 tooth-borne MMD patients had simultaneously undergone tooth-borne SARME. At T1 vs T3, canine (p = 0.007; 26.0 ± 2.09 vs 29.2 ± 2.02) and first premolar (p = 0.005; 33.8 ± 2.70 vs 37.0 ± 2.43) showed significant expansion on the tip level for tooth-borne MMD. This was no significant on the apex level, indicating tipping. Bone-borne MMD showed a parallel distraction gap, whereas tooth-borne MMD showed a V-shape. There was a significant (p = 0.017; 138 ± 17.8 vs 141 ± 18.2) inter-condylar axes increase for bone-borne MMD. In conclusion, bone-borne vs tooth-borne MMD and tooth-borne SARME showed stable dento-skeletal effects at 1 year post-operatively. Bone-borne and tooth-borne MMD seemed not to be superior to each other. The choice of distractor type therefore depends more on anatomical and comfort factors., (Copyright © 2023 The Authors. Published by Elsevier Ltd.. All rights reserved.)

Study Question: Is there an association between low-to-moderate levels of prenatal alcohol exposure (PAE) and children's facial shape?, Summary Answer: PAE before and during pregnancy, even at low level (<12 g of alcohol per week), was found associated with the facial shape of children, and these associations were found attenuated as children grow older., What Is Known Already: High levels of PAE during pregnancy can have significant adverse associations with a child's health development resulting in recognizably abnormal facial development., Study Design, Size, Duration: This study was based on the Generation R Study, a prospective cohort from fetal life onwards with maternal and offspring data. We analyzed children 3-dimensional (3D) facial images taken at ages 9 (n = 3149) and 13 years (n = 2477) together with the data of maternal alcohol consumption., Participants/materials, Setting, Methods: We defined six levels of PAE based on the frequency and dose of alcohol consumption and defined three tiers based on the timing of alcohol exposure of the unborn child. For the image analysis, we used 3D graph convolutional networks for non-linear dimensionality reduction, which compressed the high-dimensional images into 200 traits representing facial morphology. These 200 traits were used for statistical analysis to search for associations with PAE. Finally, we generated heatmaps to display the facial phenotypes associated with PAE., Main Results and the Role of Chance: The results of the linear regression in the 9-year-old children survived correction for multiple testing with false discovery rate (FDR). In Tier 1 where we examined PAE only before pregnancy (exposed N = 278, unexposed N = 760), we found three traits survived FDR correction. The lowest FDR-P is 1.7e-05 (beta = 0.021, SE = 0.0040) in Trait #29; In Tier 2b where we examine any PAE during first trimester (exposed N = 756; unexposed N = 760), we found eight traits survived FDR correction. The lowest FDR-P is 9.0e-03 (beta = -0.013, SE = 0.0033) in Trait #139. Moreover, more statistically significant facial traits were found in higher levels of PAE. No FDR-significant results were found in the 13-year-old children. We map these significant traits back to the face, and found the most common detected facial phenotypes included turned-up nose tip, shortened nose, turned-out chin, and turned-in lower-eyelid-related regions., Limitations, Reasons for Caution: We had no data for alcohol consumption more than three months prior to pregnancy and thus do not know if maternal drinking had chronic effects. The self-reported questionnaire might not reflect accurate alcohol measurements because mothers may have denied their alcohol consumption., Wider Implications of the Findings: Our results imply that facial morphology, such as quantified by the approach we proposed here, can be used as a biomarker in further investigations. Furthermore, our study suggests that for women who are pregnant or want to become pregnant soon, should quit alcohol consumption several months before conception and completely during pregnancy to avoid adverse health outcomes in the offspring., Study Funding/competing Interest(s): This work was supported by Erasmus Medical Centre, Rotterdam, the Erasmus University Rotterdam, and the Netherlands Organization for Health Research. V.W.V.J. reports receipt of funding from the Netherlands Organization for Health Research (ZonMw 90700303). W.J.N. is a founder, a scientific lead, and a shareholder of Quantib BV., Trial Registration Number: N/A., (© The Author(s) 2023. Published by Oxford University Press on behalf of European Society of Human Reproduction and Embryology.)

Dental occlusion requires harmonious development of teeth, jaws, and other elements of the craniofacial complex, which are regulated by environmental and genetic factors. We performed the first genome-wide association study (GWAS) on dental development (DD) using the Demirjian radiographic method. Radiographic assessments from participants of the Generation R Study (primary study population, N 1  = 2,793; mean age of 9.8 y) were correlated with ~30 million genetic variants while adjusting for age, sex, and genomic principal components (proxy for population stratification). Variants associated with DD at genome-wide significant level ( P  < 5 × 10 -8 ) mapped to 16q12.2 ( IRX5 ) (lead variant rs3922616, B  = 0.16; P  = 2.2 × 10 -8 ). We used Fisher's combined probability tests weighted by sample size to perform a meta-analysis ( N  = 14,805) combining radiographic DD at a mean age of 9.8 y from Generation R with data from a previous GWAS ( N 2  = 12,012) on number of teeth (NT) in infants used as proxy of DD at a mean age of 9.8 y (including the ALSPAC and NFBC1966). This GWAS meta-analysis revealed 3 novel loci mapping to 7p15.3 ( IGF2BP3: P  = 3.2 × 10 -8 ), 14q13.3 ( PAX9: P  = 1.9 × 10 -8 ), and 16q12.2 ( IRX5: P  = 1.2 × 10 -9 ) and validated 8 previously reported NT loci. A polygenic allele score constructed from these 11 loci was associated with radiographic DD in an independent Generation R set of children ( N  = 703; B  = 0.05, P  = 0.004). Furthermore, profiling of the identified genes across an atlas of murine and human stem cells observed expression in the cells involved in the formation of bone and/or dental tissues (>0.3 frequency per kilobase of transcript per million mapped reads), likely reflecting functional specialization. Our findings provide biological insight into the polygenic architecture of the pediatric dental maturation process.

Augmented reality (AR) has shown potential in computer-aided surgery. It allows for the visualization of hidden anatomical structures as well as assists in navigating and locating surgical instruments at the surgical site. Various modalities (devices and/or visualizations) have been used in the literature, but few studies investigated the adequacy/superiority of one modality over the other. For instance, the use of optical see-through (OST) HMDs has not always been scientifically justified. Our goal is to compare various visualization modalities for catheter insertion in external ventricular drain and ventricular shunt procedures. We investigate two AR approaches: (1) 2D approaches consisting of a smartphone and a 2D window visualized through an OST (Microsoft HoloLens 2), and (2) 3D approaches consisting of a fully aligned patient model and a model that is adjacent to the patient and is rotationally aligned using an OST. 32 participants joined this study. For each visualization approach, participants were asked to perform five insertions after which they filled NASA-TLX and SUS forms. Moreover, the position and orientation of the needle with respect to the planning during the insertion task were collected. The results show that participants achieved a better insertion performance significantly under 3D visualizations, and the NASA-TLX and SUS forms reflected the preference of participants for these approaches compared to 2D approaches.

Background: To ensure the feasibility of implementing PROMs in clinical practice, they must be continually appraised for undue burden placed on patients and clinicians and their usefulness for decision-making. This study assesses correlations between the CLEFT-Q psychosocial scales in the International Consortium for Health Outcomes Measurement Standard Set for cleft and explores their associations with patient characteristics and psychosocial care referral., Methods: Spearman correlation coefficients were calculated for CLEFT-Q psychological function, social function, school function, face, speech function, and speech-related distress scales. Logistic regressions were used to assess the association of cleft phenotype, syndrome, sex, and adoption status on scale scores and clinical referral to psychosocial care for further evaluation and management., Results: Data were obtained from 3067 patients with cleft lip and/or palate at three centers. Strong correlations were observed between social function and psychological function (r > 0.69) and school function (r > 0.78) scales. Correlation between school function and psychological function scales was lower (r = 0.59 to 0.68). Genetic syndrome (OR, 2.37; 95% CI, 1.04 to 5.41), psychological function (OR, 0.92; 95% CI, 0.88 to 0.97), school function (OR, 0.94; 95% CI, 0.90 to 0.98), and face (OR, 0.96; 95% CI, 0.94 to 0.98) were significant predictors for referral to psychosocial care., Conclusions: Because social function as measured by the CLEFT-Q showed strong correlations with both school and psychological function, its additional value for measuring psychosocial function within the Standard Set is limited, and it is reasonable to consider removing this scale from the International Consortium for Health Outcomes Measurement Standard Set for cleft., (Copyright © 2022 The Authors. Published by Wolters Kluwer Health, Inc. on behalf of the American Society of Plastic Surgeons. All rights reserved.)

Objective: Osteoradionecrosis (ORN) is a severe late complication after radiotherapy but current knowledge on ORN risks in the setting of postoperative radiotherapy (PORT) is limited. We studied the incidence and risk factors of ORN in patients with oral cavity cancers (OCC, treated with PORT., Patients and Methods: A retrospective cohort study was conducted including OCC patients (mainly squamous cell) treated with postoperative intensity modulated radiotherapy between 2010 and 2018 with > 1 year disease-free survival. Cumulative incidences of ORN were computed using the Kaplan Meier method. Clinical and dosimetric risk factors for mandibular ORN were evaluated using Cox regression models., Results: Within our cohort (N = 227, median follow-up 49 months) we observed 46 cases of ORN, mainly in the mandible (n = 41). The cumulative incidence of mandibular ORN was 15.9 % (SE 2.5 %) at three years and 19.8 % (SE 3.0 %) at five years. At univariable analysis, smoking, mandibular mandibulotomy or segment resection, mean dose to the mandible, and mandible volume (%) ≥ 60 Gy (V60) were significantly associated with increased ORN risks. At multivariable analysis, smoking (HR 2.13, 95 %CI 1.12-4.06) and V60 (HR 1.02 per 1 % increase, 95 %CI 1.01-1.04) remained predictive factors. For active smokers with a high V60 ≥ 40 % we observed rapid ORN development with a 1-year incidence of 29 % vs 6 % for others (p < 0.01)., Conclusion: OCC Patients treated with PORT are at high risk for mandibular ORN. We identified the mandibular volume receiving ≥ 60 Gy as the dominant risk factor, especially in active smokers. Limiting high-dose volumes at treatment planning may decrease ORN risks., (Copyright © 2022 The Authors. Published by Elsevier Ltd.. All rights reserved.)

Mucopolysaccharidoses (MPSs) and mucolipidosis II and III (ML II and III) often manifest with orofacial (progressive) abnormalities, which may have a major impact on quality of life. However, because these patients have multiple somatic health issues, orofacial problems are easily overlooked in clinical practice and available literature on this topic solely consists of case reports, small case series, and small cohort studies. The aim of this systematic review was to gain more insight in the nature and extent of orofacial abnormalities in MPS, ML II, and III. A systematic review of all previously published articles addressing orofacial abnormalities in MPS, ML II, and III was performed. Both clinical studies and case reports were included. Outcome was the described orofacial abnormalities, subdivided into abnormalities of the face, maxilla, mandible, soft tissues, teeth, and occlusion. The search resulted in 57 articles, describing orofacial features in 340 patients. Orofacial abnormalities were present in all subtypes of MPS, ML II, and III, and consisted of thickened lips, a hypoplastic midface, a high-arched palate, hypoplastic condyles, coronoid hyperplasia, macroglossia, gingival hyperplasia, thick dental follicles, dentigerous cysts, misshapen teeth, enamel defects, and open bite. Orofacial abnormalities are present in all subtypes of MPS, ML II, and III. As orofacial abnormalities may cause complaints, evaluation of orofacial health should be part of routine clinical care., Competing Interests: Chiel J. de Bode, Emma J. Dogterom, Antoinette V. J. Rozeboom, Janneke J. Langendonk, Eppo B. Wolvius, Ans T. van der Ploeg, Esmée Oussoren, and Margreet A. E. M. Wagenmakers declare that they have no conflict of interest., (© 2022 The Authors. JIMD Reports published by John Wiley & Sons Ltd on behalf of SSIEM.)

The main objective of this study was to provide an overview of the current practice for transverse mandibular and maxillary discrepancies in the Netherlands using a web-based survey. Orthodontists (ORTHO) and Oral and Maxillofacial Surgeons (OMFS) in the Netherlands were invited to the web-based survey via their professional association. Three cases were presented which could be treated non-surgically and surgically. Participants were asked what treatment they preferred: no treatment, orthodontic treatment with optional extractions or surgically assisted orthodontic treatment. The web-based survey ended with questions on various technical aspects and any experienced complication. Invitation was sent to all 303 members of professional association for ORTHO and to all 379 members of professional association for OMFS. Overall response number was 276 (response rate of 40.5%), including 127 incomplete responses. Generally, ORTHO prefer orthodontic treatment with optional extractions and OMFS lean towards surgically assisted orthodontic treatment. Mandibular Midline Distraction appears to be less preferred, possibly due to lack of clinical experience or knowledge by both professions despite being proven clinical stable surgical technique with stable long-term outcomes. There seems to be consensus on technical aspects by both professions, however, there are various thoughts on duration of consolidation period. Complications are mostly minor and manageable., Competing Interests: Declaration of Conflicting Interests: The author(s) declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article., (© The Author(s) 2021.)

Purpose: In minimally invasive spring-assisted craniectomy, surgeons plan the surgery by manually locating the cranial sutures. However, this approach is prone to error. Augmented reality (AR) could be used to visualize the cranial sutures and assist in the surgery planning. The purpose of our work is to develop an AR-based system to visualize cranial sutures, and to assess the accuracy and usability of using AR-based navigation for surgical guidance in minimally invasive spring-assisted craniectomy., Methods: An AR system was developed that consists of an electromagnetic tracking system linked with a Microsoft HoloLens. The system was used to conduct a study with two skull phantoms. For each phantom, five sutures were annotated and visualized on the skull surface. Twelve participants assessed the system. For each participant, model alignment using six anatomical landmarks was performed, followed by the participant delineation of the visualized sutures. At the end, the participants filled a system usability scale (SUS) questionnaire. For evaluation, an independent optical tracking system was used and the delineated sutures were digitized and compared to the CT-annotated sutures., Results: For a total of 120 delineated sutures, the distance of the annotated sutures to the planning reference was [Formula: see text] mm. The average delineation time per suture was [Formula: see text] s. For the system usability questionnaire, an average SUS score of 73 was obtained., Conclusion: The developed AR-system has good accuracy (average 2.4 mm distance) and could be used in the OR. The system can assist in the pre-planning of minimally invasive craniosynostosis surgeries to locate cranial sutures accurately instead of the traditional approach of manual palpation. Although the conducted phantom study was designed to closely reflect the clinical setup in the OR, further clinical validation of the developed system is needed and will be addressed in a future work., (© 2022. The Author(s).)

The reconstruction of large mandibular defects with optimal aesthetic and functional outcomes remains a major challenge for maxillofacial surgeons. The aim of this study was to design patient-specific mandibular reconstruction implants through a semi-automated digital workflow and to assess the effects of topology optimization on the biomechanical performance of the designed implants. By using the proposed workflow, a fully porous implant (LA-implant) and a topology-optimized implant (TO-implant) both made of Ti-6Al-4V ELI were designed and additively manufactured using selective laser melting. The mechanical performance of the implants was predicted by performing finite element analysis (FEA) and was experimentally assessed by conducting quasi-static and cyclic biomechanical tests. Digital image correlation (DIC) was used to validate the FE model by comparing the principal strains predicted by the FEM model with the measured distribution of the same type of strain. The numerical predictions were in good agreement with the DIC measurements and the predicted locations of specimen failure matched the actual ones. No statistically significant differences (p < 0.05) in the mean stiffness, mean ultimate load, or mean ultimate displacement were detected between the LA- and TO-implant groups. No implant failures were observed during quasi-static or cyclic testing under masticatory loads that were substantially higher (>1000 N) than the average maximum biting force of healthy individuals. Given its relatively lower weight (16.5%), higher porosity (17.4%), and much shorter design time (633.3%), the LA-implant is preferred for clinical application. This study clearly demonstrates the capability of the proposed workflow to develop patient-specific implants with high precision and superior mechanical performance, which will greatly facilitate cost- and time-effective pre-surgical planning and is expected to improve the surgical outcome., (Copyright © 2022 The Authors. Published by Elsevier Ltd.. All rights reserved.)

Patients with mandibular hypoplasia and upper airway obstruction are at an increased risk of feeding and swallowing difficulties. Little has been described regarding these outcomes following mandibular distraction. The aim of this study was to evaluate the effect of mandibular distraction on feeding and swallowing function. A retrospective study was performed on 22 patients with non-isolated mandibular hypoplasia and severe upper airway obstruction treated with mandibular distraction. Median age at first mandibular distraction was 3.1 years (interquartile range 2.3-6.0 years) and the median follow-up time was 3.5 years (interquartile range 2.0-9.4 years). Prior to mandibular distraction, feeding difficulties were present in 18 patients. Swallowing difficulties were present in 20 patients, all of whom had problems in the oral phase of swallowing, while 11 patients had additional problems in the pharyngeal phase. Following mandibular distraction, at the time of follow-up, feeding difficulties persisted in 13 patients. Swallowing difficulties in the oral phase remained present in all 20 patients, while pharyngeal phase problems persisted in seven patients. In conclusion, feeding and swallowing difficulties are highly prevalent in non-isolated patients and often persist following mandibular distraction. Moreover, these can be the reason that decannulation cannot be accomplished. Hence, awareness and close follow-up by a specialized speech therapist is of paramount importance., (Copyright © 2021 The Authors. Published by Elsevier Inc. All rights reserved.)

Objectives: To determine whether dental maturity (dental development) was delayed in patients with Muenke syndrome, Saethre-Chotzen syndrome, and TCF12-related craniosynostosis, compared with a Dutch control group without syndromes., Materials and Methods: This study included 60 patients (38 patients with Muenke syndrome, 17 patients with Saethre-Chotzen syndrome, and 5 with TCF12-related craniosynostosis), aged 5.8-16.8 years that were treated at the Department of Oral Maxillofacial Surgery, Special Dental Care, and Orthodontics, in Sophia Children's Hospital, Erasmus University Medical Center, Rotterdam, the Netherlands. Dental age was calculated according to Demirjian's index of dental maturity. The control group included 451 children without a syndrome., Results: Compared with the control group, dental development was delayed by an average of one year in 5- to 8-year-old patients with Muenke syndrome (P = 0.007) and in 8- to 10-year-old patients with Saethre-Chotzen syndrome (P = 0.044), but not in patients with TCF12-related craniosynostosis., Conclusions: Our results indicated that dental development was delayed by one year, on average, in patients with Muenke syndrome and Saethre-Chotzen syndrome, compared with a Dutch control group without syndromes., Implications: Our findings have improved the understanding of dental development in patients with Muenke and Saethre-Chotzen syndrome. These results can provide guidance on whether the orthodontist needs to consider growth disturbances related to dental development., (© The Author(s) 2021. Published by Oxford University Press on behalf of the European Orthodontic Society.)

Objectives: To determine whether the midface of patients with Muenke syndrome, Saethre-Chotzen syndrome, or TCF12-related craniosynostosis is hypoplastic compared to skeletal facial proportions of a Dutch control group., Material and Methods: We included seventy-four patients (43 patients with Muenke syndrome, 22 patients with Saethre-Chotzen syndrome, and 9 patients with TCF12-related craniosynostosis) who were referred between 1990 and 2020 (age range 4.84 to 16.83 years) and were treated at the Department of Oral Maxillofacial Surgery, Special Dental Care and Orthodontics, Children's Hospital Erasmus University Medical Center, Sophia, Rotterdam, the Netherlands. The control group consisted of 208 healthy children., Results: Cephalometric values comprising the midface were decreased in Muenke syndrome (ANB: β = -1.87, p = 0.001; and PC1: p < 0,001), Saethre-Chotzen syndrome (ANB: β = -1.76, p = 0.001; and PC1: p < 0.001), and TCF12-related craniosynostosis (ANB: β = -1.70, p = 0.015; and PC1: p < 0.033)., Conclusions: In this study, we showed that the midface is hypoplastic in Muenke syndrome, Saethre-Chotzen syndrome, and TCF12-related craniosynostosis compared to the Dutch control group. Furthermore, the rotation of the maxilla and the typical craniofacial buildup is significantly different in these three craniosynostosis syndromes compared to the controls., Clinical Relevance: The maxillary growth in patients with Muenke syndrome, Saethre-Chotzen syndrome, or TCF12-related craniosynostosis is impaired, leading to a deviant dental development. Therefore, timely orthodontic follow-up is recommended. In order to increase expertise and support treatment planning by medical and dental specialists for these patients, and also because of the specific differences between the syndromes, we recommend the management of patients with Muenke syndrome, Saethre-Chotzen syndrome, or TCF12-related craniosynostosis in specialized multidisciplinary teams., (© 2021. The Author(s).)

Study Design: Retrospective Cohort Study., Objective: Yearly, bicycle and scooter accidents in the Netherlands amount to 90 thousand emergency department visits. Maxillofacial injuries are common after cycling or scooter accidents. To prevent these injuries, helmet use might be beneficial. However, the effect of helmet use on maxillofacial injuries remains unclear. The aim of this retrospective study is to assess the effect of helmet use on maxillofacial injuries in patients who had a bicycle or scooter accident., Methods: We collected data from the emergency department of the Erasmus Medical Center in the period from October 2017 to October 2019. Patients that were involved in a bicycle or scooter accident and subsequently received a CT scan of the head were included. We compared the incidence of maxillofacial injuries in helmeted cyclists and scooter users to non-helmeted users. Descriptive and analytic statistics were computed. Level of statistical significance was set at p < 0.05., Results: Helmet use among scooter users was associated with a significant reduction in maxillofacial fractures (p < 0.001) and soft tissue injuries (p < 0.001). Helmet use among cyclists was not associated with a reduction in maxillofacial fractures (p = 0.17) or soft tissue injuries (p = 0.30). Helmet use was not associated with a reduction in soft tissue injuries of the lower face in both cyclists (p = 0.47) and scooter users (p = 0.24)., Conclusions: Helmet use should be considered among cyclists and scooter users to prevent maxillofacial injuries. Especially unhelmeted scooter users might benefit from helmet use as this is associated with a lower incidence of maxillofacial injuries., Competing Interests: Declaration of Conflicting Interests: The author(s) declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article., (© The Author(s) 2021.)

Objectives: In patients with mandibular hypoplasia, mandibular distraction osteogenesis (MDO) aims to relieve tongue-based airway obstruction. Drug-induced sleep endoscopy (DISE) provides a dynamic assessment of the upper airway and visualizes anatomical site and cause of airway obstruction. The aim of this study was to evaluate the effect of MDO on tongue-based airway obstruction found by DISE within a non-isolated patient population with severe upper airway obstruction (UAO). Furthermore, we aimed to assess the additional value of DISE in clinical decision making by correlating DISE findings to functional airway outcomes after MDO., Methods: Findings on DISE in children who underwent MDO were retrospectively gathered and evaluated. According to DISE findings, severity of tongue-based obstruction was scored using a 4-step classification similar to the one that is used by Bravo et al.. Intubation conditions were scored according to the Cormack Lehane score (CLS). Pre-and postoperative DISE findings were compared and correlated with functional airway outcomes following MDO., Results: In 19 out of 28 MDO procedures, both a pre-and postoperative DISE was available. Tongue-based obstruction scores improved in 13 procedures, which correlated to a functional improvement in seven. Postoperative tongue-based obstruction differed significantly between patients with successful MDO and patients treated unsuccessfully (2.00 ((Interquartile range (IQR) 1.00-2.00) vs. 3.00 (IQR 2.00-4.00), p = 0.028), whereas this difference was not significant for the CLS (1.00 (IQR 1.00-1.50) vs. 2.00 (IQR 1.00-4.00), p = 0.066). If no improvement of tongue-based obstruction was seen, MDO is very unlikely to be successful on the functional airway., Conclusions: DISE provides information on the site and nature of airway obstruction and can visualize the effect of MDO on the severity of tongue-based airway obstruction. Therefore, it can be of additional value in understanding the differences in functional airway outcomes after MDO and aids in deciding appropriate and targeted treatment. Hence, standardized use of DISE, in addition to the clinical assessment of mandibular position and a polysomnography, during MDO management is highly recommended., (Copyright © 2021 The Authors. Published by Elsevier B.V. All rights reserved.)

In recent decades, an increasing number of tissue engineered bone grafts have been developed. However, expensive and laborious screenings in vivo are necessary to assess the safety and efficacy of their formulations. Rodents are the first choice for initial in vivo screens but their size limits the dimensions and number of the bone grafts that can be tested in orthotopic locations. Here, we report the development of a refined murine subcutaneous model for semi-orthotopic bone formation that allows the testing of up to four grafts per mouse one order of magnitude greater in volume than currently possible in mice. Crucially, these defects are also "critical size" and unable to heal within the timeframe of the study without intervention. The model is based on four bovine bone implants, ring-shaped, where the bone healing potential of distinct grafts can be evaluated in vivo. In this study we demonstrate that promotion and prevention of ossification can be assessed in our model. For this, we used a semi-automatic algorithm for longitudinal micro-CT image registration followed by histological analyses. Taken together, our data supports that this model is suitable as a platform for the real-time screening of bone formation, and provides the possibility to study bone resorption, osseointegration and vascularisation., (Copyright © 2021 The Authors. Published by Elsevier Ltd.. All rights reserved.)

We performed genome-wide association study meta-analysis to identify genetic determinants of skeletal age (SA) deviating in multiple growth disorders. The joint meta-analysis (N = 4557) in two multiethnic cohorts of school-aged children identified one locus, CYP11B1 (expression confined to the adrenal gland), robustly associated with SA (rs6471570-A; β = 0.14; P = 6.2 × 10 -12 ). rs6410 (a synonymous variant in the first exon of CYP11B1 in high LD with rs6471570), was prioritized for functional follow-up being second most significant and the one closest to the first intron-exon boundary. In 208 adrenal RNA-seq samples from GTEx, C-allele of rs6410 was associated with intron 3 retention (P = 8.11 × 10 -40 ), exon 4 inclusion (P = 4.29 × 10 -34 ), and decreased exon 3 and 5 splicing (P = 7.85 × 10 -43 ), replicated using RT-PCR in 15 adrenal samples. As CYP11B1 encodes 11-β-hydroxylase, involved in adrenal glucocorticoid and mineralocorticoid biosynthesis, our findings highlight the role of adrenal steroidogenesis in SA in healthy children, suggesting alternative splicing as a likely underlying mechanism., (© 2021. The Author(s).)

Objectives Up to 68% of field hockey players have experienced at least one orodental injury in their sport career. Therefore, the Royal Dutch Hockey Association (KNHB) made mouthguard use mandatory for field hockey players during competition and training from August 2015 onwards.This study evaluates the effects of the new regulations on mouthguard use and the occurrence of injuries in Dutch field hockey. Methods A 35-item online questionnaire about mouthguard use and orodental injuries was sent to 13 field hockey clubs in the Netherlands. Absolute numbers and percentages of mouthguard ownership, mouthguard use, number and type of injuries were assessed. The results were related to comparable data before mandatory mouthguard use. Associations of gender and training frequency with the number of injuries were analysed with logistic regression. Results In total, 1169 hockey players were included in the study and almost all owned a mouthguard (females:99.6%, males:93.7%), which significantly increased after implementation (p < 0.001). 90.6% of the respondents wore a mouthguard during matches and 70.1% during training. Of the 1169 players, 68(5.8%) experienced at least one orodental injury after the implementation with a total of 100 injuries. Injuries happened more often during matches (63.2%) than during training (36.8%). Lip cuts account for most of the injuries, the number of broken (p = 0.116) and knocked out teeth (p = 0.026) decreased. Conclusion Although mouthguard use already increased in recent years, the new regulations led to an additional increase and a successful change of attitude towards mouthguard use. Most importantly, the severity of orodental injuries decreased measurable.