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Objectives: A prospective parallel cohort trial was conducted to compare outcomes of patients treated with maxillary protraction vs LeFort 1 maxillary advancement surgery., Setting and Sample Population: The primary site for the clinical trial is Children's Hospital Los Angeles; the satellite test site is Seattle Children's Hospital. All patients have isolated cleft lip and palate and a skeletal Class III malocclusion., Material and Methods: A total of 50 patients, ages 11-14, will be recruited for the maxillary protraction cohort. The maxillary surgery cohort consists of 50 patients, ages 16-21, who will undergo LeFort 1 maxillary advancement surgery. Patients with additional medical or cognitive handicaps were excluded from the study., Results: Current recruitment of patients is on track to complete the study within the proposed recruitment period., Conclusion: This observational trial is collecting information that will examine dental, skeletal, financial and quality-of-life issues from both research cohorts., (© 2017 John Wiley & Sons A/S. Published by John Wiley & Sons Ltd.)

Citation analysis is an established bibliometric method which catalogues papers according to the number of times they have been referenced. It is believed that the total number of citations an article receives reflects its importance among its peers. Never before has a bibliometric analysis been performed in the area of Cleft Lip and Palate. Our citation analysis creates a comprehensive list of the 50 most influential papers in this field. Journals specializing in Cleft Palate, Craniofacial, Plastic Surgery, Maxillofacial Surgery, Aesthetics and Radiology were searched to establish which articles most enriched the specialty over the past 70 years. The results show an interesting collection of papers which reveal developing trends in surgical techniques. These landmark papers mould and influence management and decision-making today.

Background: A longitudinal cohort study was established to investigate the well-being of children born with cleft lip and/or palate (CL/P) during the COVID-19 pandemic, in Victoria, Australia., Materials and Methods: The Royal Children's Hospital cleft service database was used to identify children aged between 4 and 17 years old born with an isolated CL/P. Families of eligible children who consented to participate were asked to complete the Strengths and Difficulties Questionnaire (SDQ) between October and December 2020 and again 6-month later. SDQ results from typically developing Australian children during the COVID-19 pandemic were utilized from a previously published study., Results: 63 parents completed the baseline questionnaire, with 44 completing the 6-month follow-up. For participants at baseline, the mean age was 8.9 years, with 55% male. All outcome domains of the SDQ improved between baseline and timepoint 2, with the difference in total difficulties scores being statistically significant, indicating a reduction in total difficulties at timepoint 2, associated with the easing of COVID-19 restrictions. When compared with the Australian population during the COVID-19 pandemic, Victorian children born with CL/P had lower SDQ scores for all difficulties outcome domains, with statistically significant results for conduct problems, hyperactivity, peer problems and total difficulties, indicating fewer difficulties for children born with CL/P., Conclusions: Children born with CL/P experienced fewer difficulties when compared with the typically developing Australian population during the COVID-19 pandemic. The level of restrictions imposed because of the pandemic also had little influence on the well-being of these children., (© 2023 John Wiley & Sons A/S. Published by John Wiley & Sons Ltd.)

Background: Congenital anomalies are a leading cause of morbidity and mortality worldwide. We aimed to review the common surgically correctable congenital anomalies with recent updates on the global disease burden and identify the factors affecting morbidity and mortality., Method: A literature review was done to assess the burden of surgical congenital anomalies with emphasis on those that present within the first 8000 days of life. The various patterns of diseases were analyzed in both low- and middle-income countries (LMIC) and high-income countries (HIC)., Results: Surgical problems such as digestive congenital anomalies, congenital heart disease and neural tube defects are now seen more frequently. The burden of disease weighs more heavily on LMIC. Cleft lip and palate has gained attention and appropriate treatment within many countries, and its care has been strengthened by global surgical partnerships. Antenatal scans and timely diagnosis are important factors affecting morbidity and mortality. The frequency of pregnancy termination following prenatal diagnosis of a congenital anomaly is lower in many LMIC than in HIC., Conclusion: Congenital heart disease and neural tube defects are the most common congenital surgical diseases; however, easily treatable gastrointestinal anomalies are underdiagnosed due to the invisible nature of the condition. Current healthcare systems in most LMICs are still unprepared to tackle the burden of disease caused by congenital anomalies. Increased investment in surgical services is needed., (© 2023. The Author(s) under exclusive licence to Société Internationale de Chirurgie.)

The purpose of this study was to classify the skeletal phenotypes of adult patients with skeletal class III (C-III) malocclusion and unilateral or bilateral cleft lip and palate using principal component analysis and cluster analysis. The samples consisted of 81 adult C-III patients with cleft lip and palate (CLP) who underwent orthognathic surgery (OGS) or distraction osteogenesis (59 males and 22 females; 50 unilateral cleft lip and palate and 31 bilateral cleft lip and palate; mean age when lateral cephalograms were taken, 22.2±4.6 y). Thirteen angular and one ratio cephalometric variables were measured. Using 4 representative variables obtained from principal component analysis (SNA, SNB, Gonial angle, and Bjork sum), K-means cluster analysis was performed to classify the phenotypes. Then, statistical analysis was conducted to characterize the differences in the variables among the clusters. Five clusters were obtained from 3 groups: severely retrusive maxilla and moderately retrusive mandible group: cluster-1 (23.5%, severely hyperdivergent pattern), cluster-4 (27.2%, moderately hyperdivergent pattern), and cluster-5 (11.1%, normodivergent pattern); moderately retrusive maxilla and normal mandible group: cluster-2 (30.9%, normodivergent pattern); normal maxilla and moderately protrusive mandible group: cluster-3 (7.4%, normodivergent pattern). Although skeletal phenotypes were diverse, distribution of sex and cleft type did not differ among 5 clusters ( P >0.05). Sixty-two percent of cleft patients showed a severely retrusive maxilla and moderately retrusive mandible (cluster-1, cluster-4, and cluster-5), which indicated that these are the main cause of skeletal C-III malocclusion in CLP patients who were treated with OGS. Therefore, it is necessary to consider presurgical orthodontic treatment and surgical planning based on the skeletal phenotypes of CLP patients., Competing Interests: The authors report no conflicts of interest., (Copyright © 2023 by Mutaz B. Habal, MD.)

This study aimed to classify the skeletal phenotypes of preadolescent patients with isolated cleft palate using principal component analysis and cluster analysis. Sixty-four preadolescent female patients with isolated cleft palate (incomplete hard palate and complete soft palate cleft group, n=51; complete cleft of the hard and soft palate group, n=13; the mean age when lateral cephalograms were taken, 7.08±0.76 y) were included. Ten angular and 2 ratio cephalometric variables were measured on a lateral cephalogram. Cluster analysis was performed using 3 representative variables obtained from principal component analysis (SN-GoMe, SNA, and SNB). The differences in the variables among the clusters were characterized using the Kruskal-Wallis test. As a result of the analysis, 6 clusters were obtained from 3 groups: the retrusive maxilla and mandible group: cluster 3 (14.1%, moderately hyperdivergent pattern), cluster 5 (17.2%, severely hyperdivergent pattern); the normal maxilla and mandible group: cluster 1 (23.4%, normodivergent pattern), cluster 4 (12.5%, moderately hyperdivergent pattern), cluster 6 (20.3%, severely hyperdivergent pattern); the normal maxilla and protrusive mandible group: cluster 2 (12.5%, normodivergent pattern). The distribution of isolated cleft palate types did not differ among the 6 clusters ( P >0.05). Two thirds of the patients (68.7%, clusters 1, 2, 4, and 6) had a normal anteroposterior position of the maxilla, while one third of the patients (31.3%, clusters 3 and 5) showed a retrusive mandible. These results indicate that isolated cleft palate patients have diverse maxillo-mandibular growth patterns compared with patients with cleft lip and palate., Competing Interests: The authors report no conflicts of interest., (Copyright © 2023 by Mutaz B. Habal, MD.)

Background: The authors performed a bibliometric and visualized analysis of the top 100 highly cited articles in rhinoplasty published between 2001 and 2020, to provide researchers with the present research status and the potential directions., Methods: The authors obtained data from the Web of Science Core Collection on April 7, 2021. Quantitative and qualitative analyses were performed using the Web of Science. Collaboration and keyword analyses were performed using VOSviewer software., Results: The top 100 highly cited articles included 86 original research articles and 14 reviews. The average total citations was 77 (range, 50 to 202). The article with 202 citations was published by Toriumi in 2006 and was the most influential reference. The most common level of evidence was level IV ( n = 24). Only five articles had a level of evidence of I. The top 100 articles were published in 24 journals. Plastic and Reconstructive Surgery published 49 articles with 4013 citations. The United States published the most highly cited articles. The University of Texas System published 16 articles with 1166 citations. R. J. Rohrich published 14 articles with 1025 total citations. "Cleft lip and palate," "FACE-Q," "Functional Rhinoplasty Outcome Inventory 17," "NOSE scale" (Nasal Obstruction Symptom Evaluation scale), "quality-of-life," "questionnaire," and "RHINO scale" (Rhinoplasty Health Inventory and Nasal Outcomes scale) were important keywords., Conclusions: Plastic and Reconstructive Surgery was the most influential journal, most research was reported from the United States, and the University of Texas System published the most highly cited article. R. J. Rohrich, R. K. Daniel, and B. Guyuron were the most prolific and influential authors. Global cooperation in rhinoplasty requires further strengthening., (Copyright © 2022 by the American Society of Plastic Surgeons.)

Abstract: Crowd sourcing has been used in multiple disciplines to quickly generate large amounts of diverse data. The objective of this study was to use crowdsourcing to grade preoperative severity of unilateral cleft lip phenotype in a multiethnic cohort with the hypothesis that crowdsourcing could efficiently achieve similar rankings compared to expert surgeons. Deidentified preoperative photos were collected for patients with primary, unilateral cleft lip with or without cleft palate (CL ± P). A platform was developed with C-SATS for pairwise comparisons utilizing Elo rankings by crowdsource workers through Amazon Mechanical Turk. Images were independently ranked by 2 senior surgeons for comparison. Seventy-six patients with varying severity of unilateral (CL ± P) phenotype were chosen from Operation Smile missions in Bolivia, Madagascar, Vietnam, and Morocco. Patients were an average of 1.2 years' old, ranging from 3 months to 3.3 years. Each image was compared with 10 others, for a total of 380 unique pairwise comparisons. A total of 4627 total raters participated with a median of 12 raters per pair. Data collection was completed in <20 hours. The crowdsourcing ranking and expert surgeon rankings were highly correlated with Pearson correlation coefficient of R = 0.77 (P = 0.0001). Crowdsourcing provides a rapid and convenient method of obtaining preoperative severity ratings, comparable to expert surgeon assessment, across multiple ethnicities. The method serves as a potential solution to the current lack of rating systems for preoperative severity and overcomes the difficulty of acquiring large-scale assessment from expert surgeons., Competing Interests: The authors report no conflicts of interest., (Copyright © 2020 by Mutaz B. Habal, MD.)

Background: Management of cleft lip and palate has been well characterized in pediatric patients, but limited data exist regarding the long-term functional outcomes of cleft patients once they reach adulthood., Materials and Methods: An institutional, cross-sectional survey of adult patients with a history of cleft lip and/or palate was performed. The survey recorded patient characteristics, concerns, and barriers to care. Patient-reported outcome measures were assessed using the Nasal Obstruction Symptom Evaluation Scale, Epworth Sleepiness Scale, Mandibular Function Impairment Questionnaire, and the CLEFT-Q Speech Modules., Results: A total of 63 patients (18.2%) participated in the survey. The mean patient age was 43.7 years (median: 41 y, range: 19-93 y), and the most common diagnosis was cleft lip and palate (51%) followed by isolated cleft palate (35%) and isolated cleft lip (14%). A subset of patients scored with moderate to severe dysfunction on each outcome measure including the Nasal Obstruction Symptom Evaluation Instrument (59%), Epworth Sleepiness Scale (7%), and Mandibular Function Impairment Questionnaire (13%). Respondent scores on the CLEFT-Q Speech modules demonstrated a bimodal distribution with lower scores in a significant subset of patients with cleft palate and cleft lip and palate. Many respondents (41%) were interested in clinical evaluation but cited barriers to seeking treatment including financial barriers (35%) or lack awareness of clinical options (27%)., Conclusions: Many cleft patients have persistent needs or concerns in adulthood, especially regarding speech and nasal breathing. Systemic barriers pose challenges to these patients undergoing clinical evaluation., Competing Interests: The authors report no conflicts of interest., (Copyright © 2022 by Mutaz B. Habal, MD.)

Cleft lip and palate is one of the most frequent congenital developmental defects. Autophagy is a highly conserved process of cell self-degradation in eukaryotes, involving multiple biological processes in which chloroquine (CQ) is the most common inhibitor. However, whether CQ affects and how it affects palate development is unknown. Mouse embryonic palatal cells (MEPCs) were treated with CQ to observe cell viability, apoptosis, migration, osteogenic differentiation by cell proliferation assay, flow cytometric analysis, scratch assay, and alizarin red staining. PI staining was used to measure cell cycle distribution. Immunofluorescence (IF) assay and transmission electron microscopy were used to detect autophagosomes. The autophagy-related factors (LC3 and P62), apoptosis-related markers (P53, caspase-3 cleaved caspase-3, BAX, and BCL-2), and cell cycle-related proteins (P21, CDK2, CDK4, cyclin D1, and cyclin E) were all measured by western blot. CQ inhibited the proliferation of MEPCs by arresting the G0/G1 phase of the cell cycle in a concentration- and time-dependent manner with cell cycle-related proteins P21 upregulated and CDK2, CDK4, cyclin D1, and cyclin E downregulated. Then we detected CQ also induced cell apoptosis in a dose-dependent manner by decreasing the BCL-2/BAX ratio and increasing cleaved caspase-3. Next, it was investigated that migration and osteogenesis of MEPCs decreased with CQ treatment in a dose-dependent manner. Meanwhile, CQ blocked the autophagy pathway by upregulating LC3II and P62 expressions which activated the P53 pathway. CQ activates P53 which affects MEPC biological characteristics by changing the proliferation and apoptosis of MEPCs through inhibiting autophagy., (© 2022. The Society for In Vitro Biology.)

Objective: To provide caregivers with all the resources needed to care for a surgical site following a primary cleft lip repair and evaluate its efficacy on postoperative care., Setting/participants: Caregivers of infants ages 3 to 6 months with a cleft lip and/or palate undergoing a primary repair at the Texas Children's Hospital., Methods: Packages were given to caregivers at discharge following repair. Packages included instructions and supplies needed for surgical site care. At discharge an advanced practice provider obtained informed consent and a questionnaire that established baseline knowledge of surgical site care. Following the questionnaire, the advanced practice provider demonstrated how to care for the site using the package provided. Assessment of scar healing, nasal stent compliance, and ease of care was evaluated at postoperative follow up., Results: Thirty-two families were enrolled in this study. Our data supports that caregivers who are provided resources to care for the site had increased comfort level, preparedness, and compliance rates following a primary cleft lip repair. Eighty-four percent of respondents strongly agreed that the package provided aided in preparedness for site care with 100% of respondents recommending the resources to future families undergoing a cleft lip repair., Conclusions: Caregivers feel comfortable and equipped with their ability to care for their child's repaired cleft lip when given the appropriate instructions and supplies. In addition, they would recommend the packages to future families following a repair. Empowering families to be proactive in postoperative care will potentially lead to better outcomes in cleft care., Competing Interests: The authors report no conflicts of interest., (Copyright © 2021 by Mutaz B. Habal, MD.)

Background: Orofacial clefts are one of the most common congenital anomalies, but this disease burden is unevenly distributed worldwide. The authors hypothesize that this burden falls disproportionately on the countries with the smallest surgical workforce or lowest Socio-Demographic Index, rather than those with the highest prevalence of disease., Methods: The authors estimated the prevalence and disease burden of orofacial clefting from 1990 to 2017 in 195 countries using the Global Burden of Disease methodology. Prevalence and disability-adjusted life-years were compared geographically, temporally, and against the size of the national surgical workforce, Socio-Demographic Index, and income status. Linear and logarithmic regressions were performed., Results: In 2017, the prevalence of orofacial clefting was estimated to be 10.8 million people, representing a disease burden of 652,084 disability-adjusted life-years, with most of this disease burden experienced by low- and middle-income countries (94.1%). From 1990 to 2017, there was a decrease in disease burden (-70.2%) and prevalence (-4.9%). There was negative logarithmic association between surgical workforce size and disease burden, with a surgical workforce of greater than six providers per 100,000 population (3.6 disability-adjusted life-years versus 22.4 disability-adjusted life-years per 100,000 population; p < 0.0001)., Conclusions: Burden of orofacial clefting has a strong negative association with the size of the surgical workforce, suggesting that strengthening the surgical workforce will help alleviate this burden. Epidemiologic data on countries and regions with inadequate surgical workforces and high disease burden should guide future research efforts and allocation of resources, and guide the treatment and educational goals of international charitable organizations., (Copyright © 2021 by the American Society of Plastic Surgeons.)

Background: The status of the velopharyngeal mechanism can be inferred from perceptual ratings of specified speech parameters. Several studies have proposed the measure of an overall velopharyngeal composite score based on these perceptual ratings and have reported good validity. The Cleft Audit Protocol for Speech-Augmented (CAPS-A) is a validated and reliable perceptual framework for the assessment of cleft speech and velopharyngeal function used by all Regional Cleft Services in the UK and Ireland. An overall velopharyngeal composite summary score based on the CAPS-A would serve as an important surgical outcome measure of speech., Aims: To develop and validate a velopharyngeal composite summary score based on perceptual ratings made on the CAPS-A (CAPS-A VPC-Sum) using data from a maxillary osteotomy (MO) study., Methods & Procedures: There were two surgical groups: a cleft lip and palate (CLP) (N = 20) group and a non-CLP group (N = 10), and a normal control group (N = 20). Participants in groups 1 and 2 were seen for perceptual and instrumental assessments of speech and velopharyngeal function preoperatively (T1), 3 months (T2) and 12 months (T3) postoperatively. Perceptual speech data were collected and rated by independent listeners using CAPS-A., Outcomes & Results: Moderate to strong interrater reliability for perceptual data (r s = 0.503-1.000, all p < 0.01) and strong to very strong reliability for videofluoroscopic measurements (r s = 0.746-0.947) were found. Construct validity of the CAPS-A VPC-Sum was shown by an increase in postoperative scores for the CLP group only ϰ 2 (2) = 9.769, p = 0.008 and significant differences between the CLP and the other two groups at T2 and T3 using independent t-tests. Convergent and divergent validity was indicated by a positive moderate correlation with related parameters (e.g., hypernasality r s = 0.869, p < 0.01) and a weak correlation with unrelated parameters (e.g., amount of forward advancement r s = 0.160, p = 0.526). Criterion validity was found by a moderate correlation between closure ratio r s = -0.541, p = 0.020 and CAPS-A VPC-Sum., Conclusions & Implications: A velopharyngeal composite score based on perceptually rated parameters serves as an important surgical speech outcome measure. The CAPS-A VPC-Sum is a useful, reliable and valid outcome measure of velopharyngeal function. There are added positive implications for other clinicians using geographically and language-specific adapted versions of the CAPS-A internationally., What This Paper Adds: What is already known on this subject Velopharyngeal composite scores based on perceptually rated speech parameters have been shown to have both clinical and research utility, serving as a useful surgical outcome measure. However, such a composite score must be specifically validated on the perceptual speech framework upon which it is based, as there are differences in measurement methods and terminology across cleft speech perceptual frameworks internationally. What this paper adds to existing knowledge The CAPS-A is a nationally used tool in the UK and Ireland for audit and research purposes with validated and adapted international versions. This paper reports on the validation of the derived velopharyngeal composite score measure based on the CAPS-A and an English-speaking sample, providing evidence of its validity through a speech osteotomy study. What are the potential or actual clinical implications of this work? This work provides CLP teams who use CAPS-A with a validated surgical speech outcome measure of velopharyngeal function. It has positive implications also for adapted versions of the CAPS-A internationally., (© 2021 Royal College of Speech and Language Therapists.)

Background: Maxillary osteotomy is typically undertaken to correct abnormal facial growth in cleft lip and palate. The surgery can cause velopharyngeal insufficiency resulting in hypernasality. This study aims to identify valid predictors of acquired velopharyngeal insufficiency following maxillary osteotomy by using a range of perceptual and instrumental speech investigations and multiple regression., Methods: A prospective study was undertaken consisting of a consecutive series of patients with cleft lip and palate (N = 20) undergoing maxillary osteotomy by a single surgeon. Participants were seen at: 0 to 3 months pre-surgery (T1), 3-months (T2), and 12-months (T3) post-surgery. Hypernasality was rated using the cleft audit protocol for speech-augmented (CAPS-A) and visual analog scales, and nasalance was measured on the Nasometer II 6400. For lateral videofluorosopic and nasendoscopic images, visual perceptual ratings and quantitative ratiometric measurements were undertaken. Multiple regression analyses were undertaken to identify predictors., Results: T3 models with hypernasality as the dependent variable were found to be a good fit and significant (eg, CAPS-A: R2 = 0.920, F(11,7) = 7.303, P = 0.007). Closure ratio (a quantitative ratiometric measurement) and proportion of palate contacting the posterior pharyngeal wall (a visual perceptual rating) were identified as significant predictors for the CAPS-A model (P = 0.030, P = 0.002).

The surgical techniques and execution of primary cleft lip and palate repair are no longer the greatest challenge to achieving successful rehabilitation for those born with facial clefting (i.e., bilateral and unilateral cleft lip and palate). Despite a surgeon's best efforts, when cleft palate repair is carried out during infancy, by the mixed dentition, a majority will demonstrate nasomaxillary deficiency. The cleft team's commitment to a family under their care is to ensure that the newborn reaches adulthood reconstructed without need for special regard to their original birth malformation. Guiding principles are provided for the accurate diagnosis and reliable reconstruction of the bilateral and unilateral cleft lip and palate adolescent/adult who presents with nasomaxillary deficiency and any residual oronasal fistula, bony defects, cleft dental gap(s), nasal obstructions, and associated facial dysmorphology. Successful orthognathic surgery provides a stable foundation on which any remaining soft-tissue cleft lip or cleft nasal deformities can be accurately assessed and then reconstructed.

Learning Objectives: After reading this article, the participant should be able to: 1. Understand the components of unilateral and bilateral cleft lip nasal deformity. 2. Assess the deformity. 3. Design a treatment plan for secondary correction of cleft lip nasal deformity. 4. Discuss methods for managing suboptimal outcomes., Summary: Correction of cleft lip nasal deformity has been addressed in this Maintenance of Certification/Continuing Medical Education series a number of times-a testament to the complexity of the topic. In this addition to the series, the authors provide a principle-based approach toward management of unilateral and bilateral cleft lip nasal deformity with an emphasis on timing intervention, role for intermediate correction, and methods for cleft rhinoplasty after completion of nasal growth.

Cleft lip and palate is a congenital maxillofacial deformity. Unilateral complete cleft lip and palate is one of the most common clinical types. Nasal alveolar molding (PNAM) is a recognized strategy for the treatment of cleft lip and palate. However, the current design of PNAM devices mainly relies on the subjective experience of doctors. The purpose of this paper is to describe the design and manufacture of a new computer-aided design appliance, which can be applied to the presurgical nasoalveolar molding of unilateral complete cleft lip and palate, eliminate individual differences, and improve production efficiency. In this paper, seven feature points on the healthy side and the affected side are extracted by the method of Gaussian curvature and ridge line extraction, and the healthy side rotation and built-in model are designed by using these seven feature points, which can quickly generate eight treatment stages of PNAM. The correction effects of the PNAM appliance designed in this paper were compared with the original maxillary model and the clinical PNAM appliance (hand-made by subjective experience) from the aspects of alveolar fissure width and symmetry. The PNAM appliance designed in this paper can effectively improve the symmetry of patients with unilateral complete cleft lip and palate (morphological similarity: t = 3.250, p ≤ 0.01; length similarity: t = 1.559, p = 0.150) and reduce the width of alveolar cleft (t = 8.330, p < 0.01). This can fully achieve the therapeutic effect of PNAM appliances prepared by experienced doctors and is more efficient. The method based on complex 3D surface feature point extraction can provide the basis for the design and evaluation of a unilateral complete PNAM correction model, improve the design and production efficiency of unilateral complete cleft lip and palate appliance, eliminate the design problems caused by individual differences, and reduce the burden of doctors. [ABSTRACT FROM AUTHOR]

The secondary alveolar bone grafting procedure is typically recommended during the late mixed dentition phase, prior to the eruption of the permanent canine, in patients with cleft lip and palate. The anatomical and functional adaptations observed in the grafted area allow spontaneous migration and eruption of the adjacent maxillary canine. An alveolar bone graft can be performed using autogenous bone or recombinant human bone morphogenetic protein-2 (rhBMP-2). Employing rhBMP-2 in a collagen membrane eliminates the need for a donor site, thus reducing surgical morbidity. This paper aims to present a case involving complete orthodontic rehabilitation with a three-year follow-up of a male patient with a unilateral complete cleft lip and palate, posterior and anterior crossbite, where grafting was performed with rhBMP-2 at a single centre. Orthodontic intervention began at 8 years of age with rapid maxillary expansion, followed by facemask therapy. The alveolar bone grafting procedure was performed using rhBMP-2 in a collagen membrane, according to the surgical protocol developed by the Oslo team. Comprehensive orthodontic treatment started 15 months post bone grafting, during which the maxillary permanent lateral incisor distal to the alveolar cleft was successfully moved mesially into the grafted region. This intervention resulted in adequate occlusal and periodontal outcomes. The alveolar graft with rhBMP-2 produced adequate and stable alveolar bone formation, facilitating tooth eruption, orthodontic movement, and stability at the cleft site., (Copyright © 2024 CEO. All rights reserved.)

Objective: The present investigation examined how factors such as cleft type, age of primary palatal surgery, diagnosed syndromes, hearing problems, and malocclusions could predict persistent speech difficulties and the need for speech services in school-aged children with cleft palate., Methods: Participants included 100 school-aged children with cleft palate. Americleft speech protocol was used to assess the perceptual aspects of speech production. The logistic regression was performed to evaluate the impact of independent variables (IV) on the dependent variables (DV): intelligibility, posterior oral CSCs, audible nasal emission, hypernasality, anterior oral CSCs, and speech therapy required., Results: Sixty-five percent of the children were enrolled in (or had received) speech therapy. The logistic regression model shows a good fit to the data for the need for speech therapy (Hosmer and Lemeshow's χ 2 (8)=9.647,p=.291). No IVs were found to have a significant impact on the need for speech therapy. A diagnosed syndrome was associated with poorer intelligibility (Pulkstenis-Robinson's χ 2 (11)=7.120,p=.789). Children with diagnosed syndromes have about six times the odds of a higher hypernasality rating (Odds Ratio = 5.703) than others. The cleft type was significantly associated with audible nasal emission (Fisher'sexactp=.006). At the same time, malocclusion had a significant association with anterior oral CSCs (Fisher'sexactp=.005)., Conclusions: According to the latest data in the Cleft Registry and Audit Network Annual Report for the UK, the majority of children with cleft palate attain typical speech by age five. However, it is crucial to delve into the factors that may influence the continuation of speech disorders beyond this age. This understanding is vital for formulating intervention strategies aimed at mitigating the long-term effects of speech disorders as individuals grow older., Competing Interests: Declaration of competing interest There's no financial/personal interest or belief that could affect the objectivity of this paper., (Copyright © 2024 Elsevier B.V. All rights reserved.)

Background : Patients affected by cleft lip and palate have a characteristic nasal deformity; however, the treatment timeline varies amongst providers. There has been a shift from a more conservative approach to earlier intervention in order to allow for more normal development of the nose. Form, function, and future development all must be considered. For this reason, this investigation was undertaken to present the current literature available on the effects to all aspects of primary septoplasty in the cleft nasal deformity. Design : An initial list of 222 papers was identified, and it was determined that 16 papers fit the inclusion criteria. Studies were included in which the initial age of operation for the majority of patients was between 3 and 12 months and in which patients underwent septal repositioning at the time of cleft lip repair. These papers were all reviewed by a single author initially, and the results recorded. All results were then verified by a second author for accuracy and completeness. Results : Symmetry was found to be improved by primary septoplasty. Growth was not found to be impaired in any study; data was insufficient to indicate that growth was improved. Obstruction was improved as determined both by imaging, endoscopy, and patient survey. Finally, reoperation rates occurred at an acceptable rate not exceeding that of primary rhinoplasty without septoplasty. Conclusion : Primary septoplasty leads to better aesthetic symmetry and function of the cleft nose without impairing growth. This change is maintained into adulthood often without the need for revisionary surgery. [ABSTRACT FROM AUTHOR]

Background: Cleft lip with or without cleft palate (CL/CP) is a prevalent congenital malformation. Approximately 16 candidate loci for CL/CP have been identified in both animal models and humans through association or genetic linkage studies. One of these loci is the platelet-derived growth factor-C (PDGFC) gene. In animal models, a mutation in the PDGFC gene has been shown to lead to CL/CP, with PDGF-C protein serving as a growth factor for mesenchymal cells, playing a crucial role in embryogenesis during the induction of neural crest cells. In this study, we present the identification of a novel frameshift mutation in the PDGFC gene, which we hypothesize to be associated with CL/CP, within a consanguineous Iranian family., Case Presentation: The proband was a 3-year-old girl with non-syndromic CL/CP. A history of craniofacial clefts was present in her family. Following genetic counseling, karyotype analysis and whole-exome sequencing (WES) were performed. Cytogenetic analysis revealed normal results, while WES analysis showed that the proband carried a homozygous c.546dupA (p.L183fs) mutation in the PDGFC gene. Sanger sequencing confirmed that her parents were carriers of the mutation., Conclusion: The c.546dupA (p.L183fs) mutation of PDGFC has not been previously reported and was not found in human genome databases. We speculate that the c.546dupA mutation of the PDGFC gene, identified in the Iranian patient, may be responsible for the phenotype of non-syndromic CL/CP (ns-CL/CP). Further studies are warranted to explore the specific pathogenesis of the PDGFC mutation in ns-CL/CP., 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 © 2024. Published by Elsevier B.V.)

Treatment outcome measures are critical in the decision making of best practices in the OFC field. OFC consortium working groups provided standardization of outcome measures based on previous treatment outcome studies. However, the implementation of such standardization in OFC centres worldwide is unknown. This study presented mapped outcome measures in cleft care using a structured review method complemented by quantitative overview of the relevant published research to provide initial guidelines for the implementation of treatment outcome standardization. A scoping review of the literature of treatment outcomes in cleft care following Preferred Reporting Items for Systematic reviews and Meta-Analyses extension for Scoping Reviews guidelines was performed. The selected indexed paper in outcome measures mapped following the international consortium in standard set of outcome measures in cleft care. Three hundred and sixty-five articles were filtered. The most discussed domains of cleft care were dental and oral health, appearance and speech/communication. Overall, the majority of publications were produced in high-income countries. The current review indicates that there are inequalities of treatment outcome studies among the domain of cleft care. In addition, there are also inequalities of published articles from HIC versus LMIC in treatment outcomes. This information can be used to develop targeted interventions aimed at encouraging cleft centres worldwide to adapt standardized outcome measures., (© 2023 The Authors. Orthodontics & Craniofacial Research published by John Wiley & Sons Ltd.)

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.

Orofacial clefts (OFCs) represent the most prevalent congenital craniofacial anomalies, significantly impacting patients' appearance, oral function, and psychological well-being. Among these, non-syndromic OFCs (NSOFCs) are the most predominant type, with the etiology attributed to a combination of genetic and environmental factors. Rare variants of key genes involved in craniofacial development-related signaling pathway are crucial in the occurrence of NSOFCs, and our recent studies have identified PTCH1, a receptor-coding gene in the Hedgehog signaling pathway, as a causative gene for NSOFCs. However, the role of PTCH2, the paralog of PTCH1, in pathogenesis of NSOFCs remains unclear. Here, we perform whole-exome sequencing to explore the genetic basis of 144 sporadic NSOFC patients. We identify five heterozygous variants of PTCH2 in four patients: p.L104P, p.A131G, p.R557H, p.I927S, and p.V978D, with the latter two co-occurring in a single patient. These variants, all proven to be rare through multiple genomic databases, with p.I927S and p.V978D being novel variants and previously unreported. Sequence alignment suggests that these affected amino acids are evolutionarily conserved across vertebrates. Utilizing predictive structural modeling tools such as AlphaFold and SWISS-MODEL, we propose that these variants may disrupt the protein's structure and function. In summary, our findings suggest that PTCH2 may be a novel candidate gene predicted to be associated with NSOFCs, thereby broadening the spectrum of causative genes implicated in the craniofacial anomalies., Competing Interests: Declaration of competing interest The authors declare the following financial interests/personal relationships which may be considered as potential competing interests: [Yue Zhang reports financial support was provided by Natural Science Foundation of Xinjiang Uygur Autonomous Region. Huaxiang Zhao reports financial support was provided by National Natural Science Foundation of China. Yuxia Hou reports financial support was provided by National Natural Science Foundation of China. Wenbin Huang reports financial support was provided by Shenzhen Clinical Research Center for Oral Diseases. If there are other authors, they 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 © 2024 The Author(s). Published by Elsevier B.V. All rights reserved.)

Congenital malformations can affect almost 7% of canine newborns. The increase of commercial dog breeding and inbreeding used to maintain the striking characteristics of each breed, the appearance of malformations has become increasingly common, especially in brachycephalic dogs. The causes are diverse, and include genetic, nutritional, iatrogenic, and infectious factors, often making it difficult to establish a cause-consequence relationship. The high mortality associated with malformations comes not only from the fact that some are incompatible with life, but also because even if many undergo surgical treatment or correction, they require specific management, monitoring, and clinical treatment for an indefinite period of time. The most common malformations such as cleft lip and palate, hydrocephalus and anasarca have been studied for a long time, and it is currently known that brachycephalic dogs have a greater predisposition, however, for other less common conditions as gastroschisis and hypospadias, there is only a few case reports. The appearance of congenital defects in a litter leads to financial losses for the breeder, emotional losses for the owner and the veterinarian and harms the well-being of that individual. For this reason, the aim of this review article is to gather relevant information on the characteristics, diagnosis, and management of the main malformations in puppies. It is essential that the veterinarian is prepared to diagnose and treat these conditions, reducing negative impacts on animals and owners., 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 © 2024 Elsevier Ltd. All rights reserved.)

The aim of this paper is to describe a technique that can be utilised for the targeted correction of the Whistle Deformity and accompanying upper lip subunits. All patients were post-primary cleft lip repair, either unilateral or bilateral cleft lips, with a resulting Whistle Deformity post-operatively. They all elected to undergo surgical correction of the Whistle Deformity using bilateral philtral dermofascial flaps to correct their Whistle Deformity. Post-operative clinical photographs demonstrate the results of the technique. We assessed for the resolution of the Whistle Deformity and improvement of the appearance of the upper lip subunits, including; philtral, columella, and nostril sill elements. The patients treated with this technique, with Whistle Deformities following unilateral and bilateral cleft lip repair, had satisfactory results from the procedure and recovered with no complications. We propose this is an effective method for treating up to moderate-severe Whistle deformities following both unilateral and bilateral primary cleft lip repairs., Competing Interests: Declaration of Conflicting InterestsThe author(s) declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.

Introduction: A cleft palate is a common type of facial malformation. Compensatory articulation errors are one of the important causes of unclear speech in children with cleft palate. Tele-practice (TP) helps to connect therapists and clients for assessment and therapy. Our goal is to investigate the effectiveness of articulation therapy through tele-practice on cleft palate children in Khuzestan Province during the COVID-19 pandemic., Materials & Methods: Before starting the treatment, a 20-min speech sample was recorded individually from all the children. Speech intelligibility and the percentage of correct consonants were assessed for each speech sample. The control group received treatment sessions in person at the cleft palate center, and the other group received treatment via tele-practice using the ZOOM platform. Treatment sessions were provided in the form of 45-60-min group sessions, twice a week, for 5 weeks (10 sessions in total). After 10 treatment sessions, the speech sample was recorded again. The level of parental satisfaction was measured using a Likert 5-level survey., Results: The mean score of intelligibility of the two groups decreased (-1.4400 and 0.7200). The two groups' mean percentage of correct consonants increased. (26.09 and 17.90). In both groups, the mean score of parents' satisfaction with the treatment was high (3.44 and 3.84). The mean of difference before and after the speech intelligibility and the percentage of correct consonants variables in both groups was statistically significant (P = 0.001 and P = 0.002, respectively). In both groups, the satisfaction variable was not associated with a statistically significant difference (P = 0.067)., Conclusion: The effectiveness of in-person therapy over a certain period of time is higher than tele-practice. Nevertheless, the results demonstrated an increase in the intelligibility of speech and the percentage of correct consonants in both groups, thus proving the effectiveness of articulation therapy in correcting compensatory articulation errors in children with cleft palate through in-person and tele-practice., 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 © 2024 Elsevier B.V. All rights reserved.)

Background/Objectives: Three-dimensional (3D) analysis of maxillofacial structures in dysmorphic patients offers clinical advantages over 2D analysis due to its high accuracy and precision in measuring many morphological parameters. Currently, no reliable gold standard exists for calculating 3D volumetric measurements of maxillofacial structures when captured by 3D surface imaging techniques. The aim of this scoping review is to provide an overview of the scientific literature related to 3D surface imaging methods used for volumetric analysis of the dysmorphic maxillofacial structures of patients affected by CL/P or other syndromes and to provide an update on the existing protocols, methods, and, when available, reference data. Methods: A total of 17 papers selected according to strict inclusion and exclusion criteria were reviewed for the qualitative analysis out of more than 4500 articles published between 2002 and 2024 that were retrieved from the main electronic scientific databases according to the PRISMA-ScR guidelines. A qualitative synthesis of the protocols used for the selection of the anatomical areas of interest and details on the methods used for the calculation of their volume was completed. Results: The results suggest a great degree of heterogeneity between the reviewed studies in all the aspects analysed (patient population, anatomical structure, area selection, and volume calculation), which prevents any chance of direct comparison between the reported volumetric data. Conclusions: Our qualitative analysis revealed dissimilarities in the procedures specified in the studies, highlighting the need to develop uniform methods and protocols and the need for comparative studies to verify the validity of methods in order to achieve high levels of scientific evidence, homogeneity of volumetric data, and clinical consensus on the methods to use for 3D volumetric surface-based analysis. [ABSTRACT FROM AUTHOR]

Objective: Cleft lip and palate (CLP) is the most common human cranial and maxillofacial birth defect. The aim of this bibliometric analysis was to provide an overview of the development of CLP-related research. Method: Cleft lip and palate-related studies published from 2000 to 2017 were retrieved from the Science Citation Index Expanded core database. Publication date, journal, authors, first authors, keywords, and citations were extracted and quantitatively analyzed using Bibliographic Item Co-Occurrence Matrix Builder software. The word matrix and co-occurrence matrix were established, and the co-citation analysis, keyword clustering, and social network analysis (SNA) of highly cited papers were completed. Results: A total of 9040 articles were retrieved from the 18 years of publications that were searched. The number of documents steadily increased over the period of interest, with a slight decrease in 2016 and 2017. This article separately examined the top most cited papers and high-frequency keywords from 3 time periods: 2000 to 2005, 2006 to 2011, and 2011 to 2017. The strategy coordinates of citation reflect TGF-β3, MSX1 gene, technique for cleft lip repair, TTF2, P63, IRF6 gene, FGF signaling, PVRL1, TGFBR2, and BMP4 gene as areas of research interest in the field. Moreover, the SNA of keywords highlighted new research topics: meta-analysis, cone beam computed tomography, tooth agenesis, case–control study, association study, micrognathia, DiGeorge syndrome, NSCL/P, UCLP, GWAS, MTHFR, and CLPTM1L. Conclusion: We conducted bibliometric research of CLP across an 18-year span. The results help to define an overall command of the latest topics in CLP and provide insight for launching new projects. [ABSTRACT FROM AUTHOR]

Background: Upper lip wounds that lie perpendicular to the relaxed skin tension lines are subjected to repetitive dynamic tension caused by the orbicularis oris muscle and are susceptible to unsatisfactory scarring., Methods: In this double-blind, randomized, vehicle-controlled, prospective trial, 60 consecutive patients with unilateral cleft lip undergoing primary cheiloplasties between August of 2011 and June of 2012 were randomized to receive botulinum toxin type A or vehicle injections into the subjacent orbicularis oris muscle immediately after wound closure. Scars were assessed after 6 months using the Vancouver Scar Scale, photographic visual analogue scale, and photographic scar width measurements., Results: Fifty-nine patients completed the trial. Measurements of scar widths at two defined points revealed significantly better visual analogue scale scores and narrower scars in the experimental group. However, Vancouver Scar Scale assessments were similar between groups., Conclusions: Botulinum toxin injections into the subjacent orbicularis oris muscle produced better appearing and narrower cheiloplasty scars, but provided no additional benefits in terms of scar pigmentation, vascularity, pliability, or height., Clinical Question/level of Evidence: Therapeutic, II.

The aim of this paper was to describe a modification to an old method to enhance the vermillion in adult cleft patients. We present ten consecutive patients who requested enhancement of the upper lip vermillion. The technique involves a continuous V plasty within the non-visible mucosa to elevate the vermillion. Each V incision is of a different size to match the defect. Then the V flaps are sutured to one another but the donor defect is left open to epithelialise. Adjunctive procedures are possible at the same time. There were no major complications but one patient was over corrected and needed reduction of mucosa. The technique offers a permanent enhancement of the vermillion and is a safe alternative to other methods of lip augmentation including fillers., (Copyright © 2023 European Association for Cranio-Maxillo-Facial Surgery. Published by Elsevier Ltd. All rights reserved.)

Patients affected by cleft lip and palate have a characteristic nasal deformity; however, the treatment timeline varies amongst providers. There has been a shift from a more conservative approach to earlier intervention in order to allow for more normal development of the nose. Form, function, and future development all must be considered. For this reason, this investigation was undertaken to present the current literature available on the effects to all aspects of primary septoplasty in the cleft nasal deformity., An initial list of 222 papers was identified, and it was determined that 16 papers fit the inclusion criteria. Studies were included in which the initial age of operation for the majority of patients was between 3 and 12 months and in which patients underwent septal repositioning at the time of cleft lip repair. These papers were all reviewed by a single author initially, and the results recorded. All results were then verified by a second author for accuracy and completeness., Symmetry was found to be improved by primary septoplasty. Growth was not found to be impaired in any study; data was insufficient to indicate that growth was improved. Obstruction was improved as determined both by imaging, endoscopy, and patient survey. Finally, reoperation rates occurred at an acceptable rate not exceeding that of primary rhinoplasty without septoplasty., Primary septoplasty leads to better aesthetic symmetry and function of the cleft nose without impairing growth. This change is maintained into adulthood often without the need for revisionary surgery., Competing Interests: Declaration of Conflicting InterestsThe authors declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.

The aim of this paper is to describe a technique that can be utilised for the targeted correction of the Whistle Deformity and accompanying upper lip subunits. All patients were post-primary cleft lip repair, either unilateral or bilateral cleft lips, with a resulting Whistle Deformity post-operatively. They all elected to undergo surgical correction of the Whistle Deformity using bilateral philtral dermofascial flaps to correct their Whistle Deformity. Post-operative clinical photographs demonstrate the results of the technique. We assessed for the resolution of the Whistle Deformity and improvement of the appearance of the upper lip subunits, including; philtral, columella, and nostril sill elements. The patients treated with this technique, with Whistle Deformities following unilateral and bilateral cleft lip repair, had satisfactory results from the procedure and recovered with no complications. We propose this is an effective method for treating up to moderate-severe Whistle deformities following both unilateral and bilateral primary cleft lip repairs. [ABSTRACT FROM AUTHOR]

Objective: To critically appraise the body of scientific literature supporting the risks and efficacy of nasoalveolar molding (NAM), specifically in contrast to alternative methods of presurgical infant orthopedics (PSIO) or to treatment without PSIO. Main Outcome Measures: Five outcome domains were considered: nasolabial aesthetics; dentoalveolar relationship; midfacial growth; cost and burden of care; and number of anesthetic events. Design: MEDLINE, Embase, and Scopus were queried for articles from the first description of the Grayson-Santiago NAM technique (1993) through December 13, 2021. After the application of inclusionary and exclusionary criteria, selected articles were critically appraised using a systematic framework that included risk of bias assessment using the Cochrane RoB 2.0 and ROBINS-I tools. Results: A total of 88 studies were included. Level-I and -II evidence showed on par or better approximation and alveolar alignment achieved by NAM compared to other PSIO. Level-II and -III evidence showed improved nasolabial aesthetics compared to other PSIOs. Level-II and -III evidence supported no harm to maxillofacial skeletal growth through age 12. Sparse level-III evidence supported a reduced number of labial or nasal revisions following NAM. Level-II and -III evidence showed NAM requiring upfront cost and frequent appointments but reducing caregiver psychosocial burden and reducing long-term costs compared to select alternatives. Many studies carried a high risk of bias. Conclusions: Current evidence supports the overall efficacy of NAM regarding short/mid-term outcomes, with a low risk of negative effects on midfacial growth or dental development. The high risk of bias discovered in many papers underscores the need for robust study design in future research. [ABSTRACT FROM AUTHOR]

Background: It is necessary to analyze and monitor the facial growth of orofacial cleft patients. The documentation should therefore begin before and after primary surgeries. Technological evolution has transformed plaster models into 3D images through scanners that allow rational storage, manipulation, and rotation without the possibility of breakage or damage. Based on this fact, this narrative review aims to provide a feature on the three-dimensional tools available for the assessment of dental arches in children with orofacial cleft and mixed dentition. Material and Methods: Three databases were chosen (PubMed, ScienceDirect, and Scopus) and keywords were used to select papers. Results: During the database screening, 292 potentially relevant papers were found. After removing duplicates, titles, and abstracts, 32 papers presented qualifications for analysis. Through evaluating each document by reading it one by one, 24 papers fulfilled the eligibility criteria. Conclusions: It was concluded that digital tools—i.e., benchtop scanners which evaluate the dental arches of children with cleft lip, palate, and mixed dentition—are reproducible and reliable, without the use of ionizing radiation, allow storage, manipulation with sustainability, and help preserve the environment. [ABSTRACT FROM AUTHOR]

2 Lachmann R, Schilling U, Brückmann D, Weichert A, Brückmann A. Isolated cleft lip and palate: maxillary gap sign and palatino-maxillary diameter at 11-13 weeks. We have modified the image to do so herein (Figure 1), in which the posterior border of the palate is indicated by the red asterisk and our white line indicates the nasal septum and vomer. We thank Drs Lachmann and Brückmann for their comments regarding measurement of the palatomaxillary diameter (PMD) in our recent paper. [Extracted from the article]

Background: At the beginning of the 21st century, international adoptions of children with cleft lip and/or palate increased dramatically in Sweden. Many children arrived partially or totally unoperated, despite being at an age when palatoplasty has usually been performed. To date, the speech development of internationally adopted (IA) children has been described up to age 7–8 years, but later development remains unstudied. Aims: To investigate speech development between ages 5 and 10 years in children born with cleft lip and palate (CLP) adopted from China and to compare them with non‐adopted (NA) children with CLP. A secondary aim was to compare the frequencies of secondary palatal surgery and number of visits to a speech and language pathologist (SLP) between the groups. Methods & Procedures: In a longitudinal study, 23 IA children from China were included and matched with 23 NA children born in Sweden. Experienced SLPs blindly reassessed audio recordings from routine follow‐ups at ages 5 and 10 years. Velopharyngeal function (VPF) was assessed with the composite score for velopharyngeal competence (VPC‐Sum) for single words and rated on a three‐point scale (VPC‐Rate) in sentence repetition. Target sounds in words and sentences were phonetically transcribed. Per cent correct consonants (PCC) were calculated at word and sentence levels. For in‐depth analyses, articulation errors were divided into cleft speech characteristics (CSCs), developmental speech characteristics (DSCs) and s‐errors. Information on secondary palatal surgery and number of visits to an SLP was collected. Outcomes & Results: VPF differed significantly between the groups at both ages when assessed with VPC‐Sum, but not with VPC‐Rate. Regardless of the method for assessing VPF, a similar proportion in both groups had incompetent VPF but fewer IA than NA children had competent VPF at both ages. IA children had lower PCC at both ages at both word and sentence levels. More IA children had CSCs, DSCs and s‐errors at age 5 years, and CSCs and s‐errors at age 10. The development of PCC was significant in both groups between ages 5 and 10 years. The proportion of children receiving secondary palatal surgery did not differ significantly between the groups, nor did number of SLP visits. Conclusions & Implications: CSCs were more persistent in IA children than in NA children at age 10 years. Interventions should target both cleft and DSCs, be comprehensive and continue past the pre‐school years. WHAT THIS PAPER ADDS: What is already known on this subject: At the beginning of the 21st century, IA children with cleft lip and/or palate arrived in Sweden partially or totally unoperated, despite being at an age when palatoplasty has usually been performed. Studies up to age 7–8 years show that adopted children, compared with NA peers, have poorer articulation skills, demonstrate both cleft‐related and developmental articulation errors, and are more likely to have velopharyngeal incompetence. Several studies also report that adopted children more often require secondary palatal surgery due to fistulas, dehiscence or velopharyngeal incompetence compared with NA peers. What this paper adds to existing knowledge: This longitudinal study provides additional knowledge based on longer follow‐ups than previous studies. It shows that the proportion of children assessed to have incompetent VPF was similar among IA and NA children. It was no significant difference between the groups regarding the proportion that received secondary palatal surgery. However, fewer IA children were assessed to have a competent VPF. Developmental articulation errors have ceased in most IA and all NA children at age 10 years, but significantly more adopted children than NA children still have cleft‐related articulation errors. What are the potential or actual clinical implications of this work?: Speech and language therapy should target both cleft‐related and developmental articulation errors. When needed, treatment must be initiated early, comprehensive, and continued past the pre‐school years, not least for adopted children. [ABSTRACT FROM AUTHOR]

Birth defects have always been one of the most important diseases in medical research as they affect the quality of the birth population. Orofacial clefts (OFCs) are common birth defects that place a huge burden on families and society. Early screening and prevention of OFCs can promote better natal and prenatal care and help to solve the problem of birth defects. OFCs are the result of genetic and environmental interactions; many genes are involved, but the current research has not clarified the specific pathogenesis. The mouse animal model is commonly used for research into OFCs; common methods of constructing OFC mouse models include transgenic, chemical induction, gene knockout, gene knock-in and conditional gene knockout models. Several main signal pathways are involved in the pathogenesis of OFCs, including the Sonic hedgehog (SHH) and transforming growth factor (TGF)-β pathways. The genes and proteins in each molecular pathway form a complex network to jointly regulate the formation and development of the lip and palate. When one or more genes, proteins or interactions is abnormal, OFCs will form. This paper summarises the mouse models of OFCs formed by different modelling methods, as well as the key pathogenic genes from the SHH and TGF-β pathways, to help to clarify the pathogenesis of OFCs and develop targets for early screening and prevention.

Objective: The objective of this paper is to conduct a systematic review that summarizes the cost-effectiveness of cleft lip and/or palate (CL/P) care in low- and middle-income countries (LMICs) based on existing literature., Design: We searched eleven electronic databases for articles from January 1, 2000 to December 29, 2020. This study is registered in PROSPERO (CRD42020148402). Two reviewers independently conducted primary and secondary screening, and data extraction., Setting: All CL/P cost-effectiveness analyses in LMIC settings., Patients, Participants: In total, 2883 citations were screened. Eleven articles encompassing 1,001,675 patients from 86 LMICs were included., Main Outcome Measures: We used cost-effectiveness thresholds of 1% to 51% of a country's gross domestic product per capita (GDP/capita), a conservative threshold recommended for LMICs. Quality appraisal was conducted using the Joanna Briggs Institute (JBI) checklist., Results: Primary CL/P repair was cost-effective at the threshold of 51% of a country's GDP/capita across all studies. However, only 1 study met at least 70% of the JBI criteria. There is a need for context-specific cost and health outcome data for primary CL/P repair, complications, and existing multidisciplinary management in LMICs., Conclusions: Existing economic evaluations suggest primary CL/P repair is cost-effective, however context-specific local data will make future cost-effectiveness analyses more relevant to local decision-makers and lead to better-informed resource allocation decisions in LMICs., Competing Interests: Declaration of Conflicting InterestsThe authors declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.

The maxillary permanent central incisor (MPCI) has been reported to be rotated in up to 96% of cleft patients. A severely rotated MPCI is highly visible when smiling, drawing attention to a repaired cleft lip, and is often the main presenting complaint of a patient with cleft lip and palate attending for orthodontic treatment. The benefit of interceptive orthodontic treatment to alleviate these concerns must be weighed up against the consequences of adding to the already substantial orthodontic burden likely to be faced by these patients.Despite the high prevalence of this dental abnormality among cleft patients, there is little published in the literature regarding this type of malocclusion. This paper explores the aetiology, presentation and management of this condition, using examples from cases treated within our department. In addition, the commonly found pre-existing anomaly of a short root length is discussed, in combination with the implications this may have upon orthodontic treatment planning for a rotated MPCI., Competing Interests: Declaration of conflicting interestsThe author(s) declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.

Background: The occurrence of orofacial Clefts (OFCs) is a congenital disease caused by many factors. According to recent studies, air pollution has a strong correlation with the occurrence of OFCs. However, there are still some controversies about the current research results, and there is no relevant research to review the latest results in recent years., Objective: In this paper, the authors conducted a systematic review and meta-analysis to explore the correlation between ambient air pollution and the occurrence of neonatal OFCs deformity., Methods: We searched Pubmed, Web of science, and Embase databases from the establishment of the database to May 2023. We included observational studies on the relationship between prenatal exposure to fine particulate matter 2.5 (PM2.5), fine particulate matter 10 (PM10), sulfur dioxide (SO2), nitrogen dioxide (NO2), ozone (O3), carbon monoxide (CO) and the risk of cleft lip (CL), cleft palate (CP), cleft lip with or without palate (CL/P). the Newcastle-Ottawa quality assessment scale (NOS) was used to evaluate the quality of the literature. Funnel plot and Egger's regression were used to verify the publication bias. Random effect model or fixed effect model was used to estimate the combined relative risk (RR) and 95% confidence interval (95%CI)., Results: A total of eleven studies were included in this study, including four cohort studies and seven case-control studies, including 22,453 cases of OFCs. Ten studies had low risk of bias and only one study had high risk of bias. Three studies reported that PM 2.5 was positively correlated with CL and CP, with a combined RR and 95%CI of 1.287(1.174,1.411) and 1.267 (1.105,1.454). Two studies reported a positive correlation between O 3 and CL, with a combined RR and 95%CI of 1.132(1.047,1.225). Two studies reported a positive correlation between PM 10 and CL, with a combined RR and 95%CI of 1.108 (1.017,1.206). No association was found between SO 2 , CO, NO 2 exposure during pregnancy and the risk of OFCs., Conclusion: The results of this study showed that there was a significant statistical correlation between exposure to PM 10 , PM 2.5 , O 3 and the risk of OFCs in the second month of pregnancy. Exposure assessment, research methods and mechanisms need to be further explored., (© 2023. The Author(s).)

Cleft lip secondary deformity is a common oral and maxillofacial deformity, usually accompanied by the upper lip and nasal cleft. The main treatment method for cleft lip secondary deformity is surgical repair. Among them, upper lip flap repair is a common method; the principle is to transplant the healthy tissue to the lesion site to achieve the purpose of repair. However, during the surgical process, each key point's stress distribution and strain conditions are quite different. Therefore, special attention should be paid to the mechanical properties of each key point during the repair operation to ensure the surgical effect. This paper aims to establish a 3-dimensional finite element model and perform finite element analysis to simulate the mechanical properties and key point displacement of the upper lip flap repair operation for cleft lip secondary deformity to improve the operation's success rate and safety., Competing Interests: The authors report no conflicts of interest., (Copyright © 2023 by Mutaz B. Habal, MD.)

Unilateral complete cleft lip and palate (UCCLP) is one of the most severe clinical subphenotypes among nonsyndromic cleft lip and/or palate (NSCL/P), that complicates surgical repair operations. Presurgical nasoalveolar molding (PNAM) is a technique used to reshape the nose, lip and alveolar bone of infants with UCCLP before surgery (the modified Mohler rotation advancement cheiloplasty and two flap palatoplasty), with the potential to facilitate surgical repair. However, the effectiveness of PNAM treatment is still a matter of debate. In this paper, the 3Shape scanning system and 3dMD stereophotography were used to assess the short-term and long-term effects of PNAM treatment on the dental arch morphology and nasolabial features of patients with UCCLP, respectively. The findings indicated that PNAM treatment negatively affects both short-term and long-term dental arch shape compared to the treatment without PNAM, particularly in terms of limiting the transverse width of the maxillary canine-to-midline. Regarding the nasal and labial symmetry, PNAM improves the symmetry of the nasal alae in patients over 7 years old and the symmetry of the lip in patients under 7 years old. Moreover, UCCLP patients who received PNAM treatment exhibited a shorter and wider shape of the nostril on the cleft side compared to those without PNAM treatment. In clinical practice, the multidisciplinary team should carefully consider the advantages and disadvantages of the outcomes of PNAM treatment when treating infants with cleft lip and palate., Competing Interests: The authors declare no conflict of interest., (©2023 The Author(s). Published by MRE Press.)

Background: The pathogenesis of orofacial cleft (OFC) is multifactorial, involving both genetic and non-genetic factors, the latter of which play a key role in the development of these anomalies. This paper addresses the incidence of OFC in Indonesia, with a focus on identifying and examining the distribution of contributory factors, including parental medical history, pregnancy history, and environmental influences. Methods: The study was conducted through the collection of primary data. An interdisciplinary research team from Indonesia administered a standardized questionnaire to parents who had children with OFC and who had provided informed consent. The case group comprised 133 children born with cleft lip and/or palate, and the control was 133 noncleft children born full-term. The risk factors associated with OFC anomalies were analyzed using the chi-square test and logistic regression. All statistical analyses were performed using SPSS version 25. A p-value of 0.05 or less was considered to indicate statistical significance. Results: The study comprised 138 children, of whom 82 were boys (59.4%) and 56 were girls (40.6%). Among them, 45 patients (32.6%) presented with both cleft lip and cleft palate, 25 individuals (18.1%) had a cleft palate only, and 28 patients (20.3%) had a cleft lip only. OFC was found to be significantly associated with a maternal family history of congenital birth defects (p< 0.05), complications during the first trimester (p< 0.05), consumption of local fish (p< 0.05), caffeine intake (p< 0.05), prolonged medication use (p< 0.05), immunization history (p< 0.05), passive smoking (p< 0.05), and X-ray exposure during pregnancy (p< 0.05). Conclusion: The findings indicate close relationships between the incidence of OFC and maternal medical history, prenatal factors, and environmental influences. [ABSTRACT FROM AUTHOR]

Background: Presurgical orthopedics (PSO) can be used to treat patients with cleft lip and palate. PSO is a powerful tool for changing nasolabial and palate structures during the 1st months of life, helping improve the restoration of form and essential functions. Materials and Methods: This preliminary retrospective pilot study evaluated treatment efficacy with a nasal PSO protocol using the Rhinoplasty Appliance System (RAS) in seven centers in Mexico that specialize in managing children born with cleft lip and palate. Twenty‑five unilateral cleft lip and palate patients (UCLP) (9 females and 16 males) were treated by an orthodontist trained in the RAS system. The severity of the cleft, treatment time, the number of devices used during treatment, and clinical complications were documented using a survey. The results were measured using standardized submental view two‑dimensional photographs. Two independent investigators evaluated one angular variable and two lineal variables before and after treatment. The columella deviation angle was measured. On the affected side, between the line in the middle of the columella from anthropometric points Sn-Prn and reference line between Sn-Sbal. The lineal variables that compare the healthy and cleft sides were measured as ratios. The lineal variables were nostril width (this measurement is taken from all are anthropometric points to the columella on the horizontal plane) and nostril height (from the nasal base plane to the upper part of the nostril). All relevant data are within the paper and its supplementary information files. Results: The resulting data were analyzed using a paired t‑test (P < 0.05). The results of nasal symmetry showed a statistically significant decrease in the nostrils’ midline deviation and horizontal symmetry and a statistically significant increase in unilateral patients’ vertical symmetry and tip‑base angle. Of the reported complications, 36% of orthodontists did not express any concerns. Meanwhile, 32% experienced skin irritation on the cheeks, 16% faced challenges with treatment attachment, 12% encountered difficulties in applying the correct tractions on the rubber bands, and 4% required a larger device from the system. Conclusions: In these preliminary results, the Rhinoplasty Appliance System (RAS) streamlines procedures, enhances reproducibility, and offers a practical solution for nasal protocols in managing cleft lip and palate in infants. RAS improves nasal symmetry, displaying promising outcomes in holistic care for infants with UCLP conditions. This research highlights the potential of three‑dimensional printing and innovative digital approaches to revolutionize presurgical interventions for pediatric patients, particularly in infant nasal PSO. [ABSTRACT FROM AUTHOR]