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Background: Traditional surgical approaches excluded septoplasty at primary cleft lip reconstruction due to concerns about restricted nasal and midfacial growth. Modern opinion in the treatment of cleft lip has increasingly employed primary septoplasty; this scoping review and historical perspective aims to chronicle the evolution of septoplasty in patients born with cleft lip and palate and discuss current evidence., Methods: The historical perspective explicitly contrasts American and European perceptions of septoplasty in cleft lip deformity and the competing anatomical theories of the role of the septum on midfacial and nasal growth. For the scoping review, articles were extracted from Embase, PubMed, and Medline, as well as manual searches of reference lists. Results were compiled, grouped, and appraised by date, outcomes, and historical significance. Inclusion criteria consisted of children who underwent primary septoplasty for any indication and were followed up on outcomes of facial growth and nasal function. Literature reviews, opinion articles, case reports, guidelines, or studies not available in English or online were excluded., Results: Evolving anatomical theories relating to midfacial growth in the mid-late 20th century underpinned a progressive ideological shift on the safety and efficacy of septoplasty in children. This is supported by our scoping review, which included 23 articles mutually selected for inclusion by 2 blinded assessors. Several competing methods have been employed to measure endpoints on facial growth and nasal function, but generally indicate primary septoplasty is successful in improving nasal function and preserving midfacial growth., Conclusion: Perceptions towards septoplasty on facial growth in the pediatric population have transformed significantly and suggest a growing acceptance of primary septoplasty techniques in patients born with a cleft lip., Competing Interests: The authors report no conflicts of interest., (Copyright © 2024 by Mutaz B. Habal, MD.)

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: Standardized and reliable medical photographs are crucial for preoperative and postoperative comparisons and academic communication in the medical field. There is limited research on photographic techniques in patients with cleft lip. Deformities of the lip and nose in patients with cleft lip are not only associated with morphological abnormalities but also with abnormalities of muscle function., Methods: Considering the morphology and function of the lips and nose in cleft lip patients, the study captured the deformity of cleft lip patients in 6 positions: frontal view, 45 degrees left-right tilted side view, 90 degrees left-right tilted side view, and basal view, and in 5 facial expressions: tightly closed lips, slightly open mouth, smiling, teeth bared, and pout., Results: In 6 different positions and 5 different expressions, we took pictures of lip and nasal deformities covering most of the common deformities in patients with cleft lip, such as white lip scarring, interruption of continuity of vermillion border, lip prolapse, asymmetric corners of the mouth, collapsed ala nasi, loss of the nasal base and deviated nasal septum., Conclusions: This paper suggests a set of effective, easy-to-follow, and precise photographic protocols to assist cleft lip surgeons in capturing suitable and informative, high-quality 2D digital photographs., Level of Evidence: Level-V., Competing Interests: The authors report no conflicts of interest., (Copyright © 2024 by Mutaz B. Habal, MD.)

This paper presents the findings of an observational study involving 38 patients to evaluate the application of a surgical technique utilizing an autologous costal cartilage scaffold for correcting nasal tip and alar asymmetry in unilateral cleft lip-nasal deformity. Nasal septum extension spreader grafts (SEG) and warped alar batten grafts, both made from autologous costal cartilage, were utilized in open rhinoplasty procedures. The warped alar batten graft was fixed to the caudal end of the SEG, with the lower lateral cartilage on the cleft side suspended to the free part of the newly created warped alar batten graft to lift the collapsed nasal alar further. Measurements of nasal tip height, nostril height, and the intersection angle of the nasal sill and alar (α) were taken before and after surgery, comparing the ratios between the normal and cleft sides. Patients were followed up for 2.5 to 5.5 years, with all cases showing successful healing and no complications. Postoperative improvements in nasal tip and nostril asymmetries were significant, with statistically significant changes observed in nasal tip height, nostril height, and the intersection angle of nasal sill and alar (α) ( P <0.05). The combined use of SEG and warped alar batten graft, both crafted from autologous costal cartilage, effectively corrected nasal tip and alar asymmetry in adult cleft lip nasal deformity cases., Competing Interests: The authors report no conflicts of interest., (Copyright © 2024 by Mutaz B. Habal, MD.)

Background: The primary surgical management of cleft lip (CL) or cleft lip and palate (CLP) aims to achieve harmonious lip and nasal symmetry while ensuring satisfactory ventilation. Postoperative nasal retainers are commonly used, though both duration of use and conformer type used vary widely. This study aimed to establish an inventory of current practices for primary cheilorhinoplasty and nasal retainer use in France., Methods: A survey was sent to surgeons within and outside the French National Clefts and Facial Malformations (MAFACE) network. Questions focused on age when primary cleft closure is performed, retainer types used, conformation duration, and estimated patient compliance. Responses were collected March-July 2023., Results: Thirty-two surgeons responded with substantial variations in practices. For isolated CL, the age for primary cleft closure was 1-6 months, with 28 % performing surgery at 3 months, 12.5 % between 3 and 6 months, 44 % at 6 months. In cases with CLP, 63 % performed simultaneous surgery at 6 months. Two surgeons (6 %) reported preoperative nasoalveolar molding and 30 surgeons (94 %) reported postoperative nasal retainer use. Retainer type used immediately after surgery varied, with equal use of commercial retainers (31 %), silicone sheets (31 %), and in-house retainers (31 %). Duration of retainer prescription was in majority 3-4 months. Notably, 44 % of surgeons reported <70 % adherence rates for the recommended conformation duration, while 25 % reported very good compliance., Conclusion: Primary cheilorhinoplasty and nasal conformation practices are highly diverse in France. Suboptimal patient compliance demonstrates the need for improved retainer design and strategies to enhance compliance., 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 The Author(s). Published by Elsevier Masson SAS.. All rights reserved.)

The objective of this study is to utilize the Nasometer to objectively assess velopharyngeal competence, specifically through the quantification of nasalance. Initial calibration of the nasometer was conducted on American adults. The objective of this study was to validate the use of the nasometer for the objective diagnosis of velopharyngeal insufficiency (VPI) in French children born with a total cleft lip and palate and to select relevant verbal stimuli for clinical practice., Material and Methods: The nasalance scores of 42 children aged 8 to 10 years old, born with a cleft lip and palate, were collected and compared with 50 control children. The scores were then analyzed in relation to 31 verbal stimuli from the French corpus created for this study (sentences and syllables). The most relevant threshold values were determined by receiver operating characteristic curves, which exhibited the highest sensitivity and specificity., Results: The results demonstrated statistically significant differences (p < 0.05) in the mean nasalance scores of the control and cleft groups for all verbal stimuli containing oral phonemes. Threshold values with good diagnostic accuracy were defined, and 15 verbal stimuli were selected for use in clinical practice., Conclusion: The nasalance threshold values defined in this study can be utilized for the objective diagnosis of velopharyngeal insufficiency (VPI) and the subsequent monitoring of French children aged 8 to 10 years old, born with a cleft lip and palate., 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 Masson SAS. All rights reserved.)

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.)

Background: Non-syndromic cleft lip with or without cleft palate (NSCL/P) is a prevalent craniofacial birth defect on a global scale. A number of candidate genes have been identified as having an impact on NSCL/P. However, the association between interferon regulatory factor 6 (IRF6) polymorphisms and NSCL/P has yielded inconsistent results, prompting the need for a meta-analysis to obtain more accurate estimates., Methods: We conducted a thorough screening of all relevant articles published up until November 15, 2023, in online bibliographic databases. The statistical analysis of the collected data was performed using the Comprehensive Meta-Analysis (Version 4.0) software., Results: A total of 79 case-control studies, comprising 14,003 cases and 19,905 controls, were included in our analysis. The combined data indicated that the IRF6 rs642961 and rs2235371 polymorphisms were associated with an increased risk of NSCL/P in the overall population. However, no significant association was found between the rs2013162 and rs2235375 polymorphisms and the risk of NSCL/P in the overall population. Furthermore, subgroup analyses revealed significant correlations between the IRF6 rs642961, rs2235371, and rs2235375 polymorphisms and the risk of NSCL/P based on ethnic background and country of origin. Nevertheless, the rs2013162 polymorphism plays a protective role in Caucasians and mixed populations., Conclusions: Our collective data indicates a significant association between the rs642961 and rs2235371 polymorphisms and the risk of NSCL/P in the overall population. The rs2235375 polymorphism could influence the susceptibility to NSCL/P based on ethnic background. Meanwhile, the rs2013162 polymorphism provides protective effects in Caucasian, mixed populations, and the Brazilian population., 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 Masson SAS. 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]

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: 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.)

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.

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.)

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.

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]

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.)

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: 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]

Cleft lip and/or cleft palate (CL/P) are the common birth defects that result when facial structures developing in an unborn baby do not close completely. To design for treatment schedule, some constraints including hospital eligibility constraints, capacity limitations, treatment age limitations, multi-hospital assignment, and multidisciplinary care team assignment should be determined. However, efficient treatment scheduling is difficult owing to the complicated conditions of specific treatment. This paper presents a multi-objective mathematical model of the CL/P patient treatment scheduling problem in order to minimize the makespan, travel distance, and total least preference assignment score. Since the problem is NP-hard, a solution method is developed based on differential evolution (DE) with particular encoding and decoding schemes for solving the CL/P patient treatment scheduling problem. The performance of DE is evaluated and compared the results with those obtained from the modified particle swarm optimization. The results show that DE is capable of finding high-quality solutions with fast convergence. To apply the proposed approach for a case study, the CL/P patient treatment scheduling program is formulated. The program can be a decision support system in helping the administrators to schedule the patients in order to identify a list of selected treatments, assign each operation of patients to the selected hospital, and intelligently identify the period of the selected treatments. [ABSTRACT FROM AUTHOR]

Introduction: One of the most prevalent congenital craniofacial abnormalities, cleft lip, and palate, adversely affecting infant's ability to take feed properly. A cleft or gap in the lip and/or palate is formed during embryonic development, which makes it difficult for a child to feed, speak, and also affects the esthetics. Improper seal of the oral cavity often results in choking and nasal regurgitation. Hence, feeding plate is a specially designed appliance which obturates the cleft between oral and nasal cavities until surgery, creating a rigid platform against which baby can press the nipple to extract the milk. Case Report: Presented cases are of cleft lip and palate of infants with a chief complaint of feeding difficulty. Palatal obturator was constructed according to age modifications. This paper will highlight various factors such as impression making, selection of material, and fabrication of appliance for cleft patients. Clinical Significance: The primary goals of these feeding appliances are to ensure adequate nutrition and hydration for the infant, promote proper weight gain and growth, and reduce the risk of complications associated with feeding difficulties. In addition, these appliances serve to support the infant's overall health and development while awaiting surgical intervention for cleft repair. [ABSTRACT FROM AUTHOR]

Introduction: Presurgical infant orthopedics has been employed since the 1950s as an adjunctive therapy for the correction of cleft lip and palate. Nasoalveolar molding represents a paradigm shift from the traditional methods of presurgical infant orthopedics. The traditional approach failed to address the deformity of the nasal cartilages in unilateral, as well as bilateral, clefts of the lip and palate and the deficiency of columella tissue in infants with bilateral clefts. The nasoalveolar molding (NAM) technique utilizes wire and acrylic nasal stents attached to an intraoral denture. This appliance is used to mold the nasal cartilages, premaxilla, and alveolar ridges into normal form and position during the neonatal period. In effect, this presurgical management of the cleft infant is intended to reduce the severity of the oronasal deformity before surgery. Case Report: This paper will discuss a case report of a presurgical NAM that has been done in our department. A 20-day-old neonate presented with unilateral cleft lip and alveolus. Wax impression was taken immediately, followed by NAM plate fabrication. The NAM plate was modified in the next 2-3 appointments by inducing selective bone deposition and resorption to approximate the segments. Soft relining material was used to modify the plate. After this, the nasal stent was added to the plate to correct the deviated nasal columella and ala of the nose. Clinical Significance: Significant improvement of the deformity was observed after 3 months of treatment. Now, the patient awaits surgical closure of the defect after she gains the appropriate weight necessary for surgery. [ABSTRACT FROM AUTHOR]

BACKGROUNDS: Nasoalveolar molding (NAM) application is among presurgical management (PSM) techniques used for infants with cleft lip and palate (CLP). It helps to approximate the palatal cleft and to reshape the nasoalveolar complex prior to primary lip repair. This study aimed to explore types of PSM and the dental speciality provision for infants with CLP in Baghdad. The status of NAM usage and surgeons' perceptions toward NAM usage were assessed. MATERIALS AND METHODS: This is a cross-sectional paper-based questionnaire study that collected responses of surgeons perform primary lip and nose repair regarding PSM. The questionnaire was distributed amongst public and private hospitals in Baghdad. Twenty surgeons were enrolled (only those surgeons who perform primary repair for infants with CLP); two females and eighteen males. RESULTS: The majority of participants' responses suggested that the majority of infants with CLP were provided with baby feeding plates and lip straps. Six surgeons reported that a percentage of their patients who have been provided with NAM. PSM in Baghdad was mostly supplied by orthodontists and plastic surgeons, and the next most likely providers were prosthodontists. 82.35% of the surgeons found that primary surgical repair procedures were easier with NAM than for the other infants. The rest have not perceived any differences. CONCLUSIONS: Orthodontists, surgeons and prosthodontists were involved in providing PSM. Baby feeding plates and lip straps were the most common PSM in Baghdad, although NAM is not uncommon. Most surgeons believe that using NAM made surgical procedures easier and permitted the prediction of surgical outcomes. [ABSTRACT FROM AUTHOR]

Background: Craniofacial clefts can form a significant defect within bone and cartilage, which can negatively affect tissue homeostasis and the remodeling process. Multiple proteins can affect supportive tissue growth, while also regulating local immune response and tissue protection. Some of these factors, like galectin-10 (Gal-10), nuclear factor kappa-light-chain-enhancer of activated B cells protein 65 (NF-κB p65), heat shock protein 60 (HSP60) and 70 (HSP70) and cathelicidin (LL-37), have not been well studied in cleft-affected supportive tissue, while more known tissue regeneration regulators like type I collagen (Col-I) and bone morphogenetic proteins 2 and 4 (BMP-2/4) have not been assessed jointly with immunomodulation and protective proteins. Information about the presence and interaction of these proteins in cleft-affected supportive tissue could be helpful in developing biomaterials and improving cleft treatment. Methods: Two control groups and two cleft patient groups for bone tissue and cartilage, respectively, were organized with five patients in each group. Immunohistochemistry with the semiquantitative counting method was implemented to determine Gal-10-, NF-κB p65-, HSP60-, HSP70-, LL-37-, Col-I- and BMP-2/4-positive cells within the tissue. Results: Factor-positive cells were identified in each study group. Multiple statistically significant correlations were identified. Conclusions: A significant increase in HSP70-positive chondrocytes in cleft patients could indicate that HSP70 might be reacting to stressors caused by the local tissue defect. A significant increase in Col-I-positive osteocytes in cleft patients might indicate increased bone remodeling and osteocyte activity due to the presence of a cleft. Correlations between factors indicate notable differences in molecular interactions within each group. [ABSTRACT FROM AUTHOR]

Background: Tranexamic acid (TXA) is commonly considered a safe drug to mitigate bleeding during and after various surgical settings among adults and children. In recent decades, anaphylaxis induced by TXA has been increasingly reported in adults. However, among pediatrics, there are fewer reported cases. Case presentation: We report a case of a 6-year-old female who experienced anaphylaxis after receiving intravenous TXA following unilateral cleft lip and palate repair surgery. She exhibited clinical symptoms involving the cardiovascular system, respiratory system, and skin. Following the administration of epinephrine, corticosteroid, and anti-histamine, the patient's symptoms were relieved. A few months after discharge, an intradermal test, yielded a positive result, confirming TXA as the culprit drug. Conclusion: Our report emphasizes the importance of considering anaphylaxis as a potential adverse reaction to TXA in pediatric patients, showing the criticality of rapid diagnosis and appropriate management for a successful outcome. [ABSTRACT FROM AUTHOR]

Fetal cleft lip is a common congenital defect. Considering the delicacy and difficulty of observing fetal lips, we have utilized deep learning technology to develop a new model aimed at quickly and accurately assessing the development of fetal lips during prenatal examinations. This model can detect ultrasound images of the fetal lips and classify them, aiming to provide a more objective prediction for the development of fetal lips. This study included 632 pregnant women in their mid-pregnancy stage, who underwent ultrasound examinations of the fetal lips, collecting both normal and abnormal fetal lip ultrasound images. To improve the accuracy of the detection and classification of fetal lips, we proposed and validated the Yolov5-ECA model. The experimental results show that, compared with the currently popular 10 models, our model achieved the best results in the detection and classification of fetal lips. In terms of the detection of fetal lips, the mean average precision (mAP) at 0.5 and mAP at 0.5:0.95 were 0.920 and 0.630, respectively. In the classification of fetal lip ultrasound images, the accuracy reached 0.925. The deep learning algorithm has accuracy consistent with manual evaluation in the detection and classification process of fetal lips. This automated recognition technology can provide a powerful tool for inexperienced young doctors, helping them to accurately conduct examinations and diagnoses of fetal lips. [ABSTRACT FROM AUTHOR]

Background and Objectives: Cleft lip alone or a combination of cleft lip and palate (CLP) is a common developmental abnormality in the craniofacial region. This umbrella review aims to identify promising avenues for treatment using stem cell therapy. Materials and Methods: Systematic reviews from 2014 to 2024 were searched among databases like PubMed, Medline, and Google Scholar. PRISMA guidelines were employed to ensure the thoroughness of the search. A quality assessment (ROBIS) of the included reviews was conducted to ensure the reliability and validity of the synthesized evidence. Results: Five systematic reviews were selected for this umbrella review. Results show that stem cell therapy, specifically using mesenchymal stem cells (MSCs) and adipocyte stem cells (ADSCs), promotes bone regeneration in CLP deformities. Although multiple studies have established the effectiveness of diverse types of stem cells in treating CLP, important considerations including safety concerns, methodological variability, and the need for standardization have been identified. The fact that the number of relevant systematic reviews that matched our inclusion criteria was limited could affect this research's robustness and may limit the breadth and depth of evidence synthesis. Definitive conclusions could not be reached due to variation among treatments and outcomes. Conclusions: The examined studies highlight the potential of stem cell therapy as a complementary approach to existing treatments for CLP. However, there are challenges that need to be addressed, including concerns regarding safety, variations in methodologies, and the need for standardization. Exploring the potential of other stem cell types may further enhance treatment outcomes for CLP patients. [ABSTRACT FROM AUTHOR]

This case report demonstrates a rare finding of a pediatric patient with Morning Glory anomaly and Moyamoya Disease with a palatal meningeal hamartoma discovered as a mass within a previously repaired incomplete cleft of the alveolus. Oral meningeal hamartomas are exceedingly rare with only two palatal cases described and none within a cleft palate or alveolus. These findings prompt a review of oral hamartomas with meningeal subclassification. Further discussion describes the relationship of the proposed origins of meningeal hamartomas within the setting of cleft palate development. [ABSTRACT FROM AUTHOR]

Objective: Our goal was to compare data collected from 3- and 7-day Infant with Clefts Observation Outcomes (iCOO) diaries. Design: Secondary data analysis of an observational longitudinal cohort study. Caregivers completed the daily iCOO for 7 days before cleft lip surgery (T0) and for 7 days after cleft lip repair (T1). We compared 3- and 7-day diaries collected at T0 and 3- and 7-day diaries collected at T1. Setting: United States. Participants: Primary caregivers of infants with cleft lip with and without cleft palate (N = 131) planning lip repair and enrolled in original iCOO study. Main Outcomes Measure(s): Mean differences and Pearson correlation coefficients. Results: Correlation coefficients were high for global impressions (>0.90) and scaled scores (0.80–0.98). Mean differences were small across iCOO domains at T0. T1 comparisons reflected the same pattern. Conclusions: Three-day diary data is comparable to 7-day diaries for measuring caregiver observations using iCOO across T0 and T1. [ABSTRACT FROM AUTHOR]

Background: Cleft lip and palate is an anomaly that affects both women and men. It is considered to be among the most frequent congenital abnormalities and is related to modifications in chromosomal DNA and multiple genetic alterations. This anomaly can also be associated with various environmental factors, such as tobacco and alcohol consumption, medication use, and exposure to different environmental and industrial toxic substances. The objective of this study was to document the frequency of risk factors related to cleft lip and palate through a systematic review of Mexican studies. Methods: In this systematic review, a bibliographic search was conducted following PRISMA guidelines in the databases Scielo, ScienceDirect, PubMed, and EBSCO. Keywords related to cleft lip and palate, epidemiology, and risk factors were used. In all, 3 independent reviewers (J.A.S.L., S.L.V., and N.M.F.) selected and evaluated a total of 17 articles included in this analysis, achieving a coefficient of κ = 0.84. Results: The analysis revealed that the highest frequency of conducted studies was in the State of Mexico. The most common risk factors identified were environmental, pharmacological, consumption habits, and gynecological factors. Conclusions: Identifying the main risk factors for cleft lip and palate in the Mexican population will enable the implementation of preventive measures aimed at reducing exposure to these factors. Additionally, early intervention can improve the quality of life for individuals affected by this condition. [ABSTRACT FROM AUTHOR]

Cleft lip and/or palate are prevalent congenital anomalies. Early and accurate diagnosis allows proper case management. The Objective: This retrospective cohort study aimed to investigate the association between cleft lip and palate and other congenital anomalies. Methods: This study analyzed 17 pregnancies prenatally diagnosed with cleft lip and palate. The investigations consisted of ultrasound examination, fetal karyotyping through amniocentesis, and family tree analysis. In the presence of an abnormal fetal karyotype, the parental karyotype was also indicated. Results: Of the 17 cases identified, 9 (52.94%) were syndromic and 8 (47.06%) were non-syndromic. The genetic syndromes identified in association with cleft lip and palate in this study included translocation syndrome (one case), Patau syndrome, trisomy 13 (seven cases), and Edwards syndrome, mosaic trisomy 18 (one case). Conclusions: A comprehensive approach ensures a thorough assessment and accurate diagnosis. Early detection and a multidisciplinary approach allow appropriate case management. [ABSTRACT FROM AUTHOR]

Background: Orofacial clefts are one of the most common congenital malformations of the fetal face and ultrasound is mainly responsible for its diagnosis. It is difficult to view the fetal palate, so there is currently no unified standard for fetal palate screening, and the diagnosis of cleft palate is not included in the relevant prenatal ultrasound screening guidelines. Many prenatal diagnoses for cleft palate are missed due to the lack of effective screening methods. Therefore, it is imperative to increase the display rate of the fetal palate, which would improve the detection rate and diagnostic accuracy for cleft palate. We aim to introduce a fetal palate screening software based on the "sequential sector scan though the oral fissure", an effective method for fetal palate screening which was verified by our follow up results and three-dimensional ultrasound and to evaluate its feasibility and clinical practicability. Methods: A software was designed and programmed based on "sequential sector scan through the oral fissure" and three-dimensional ultrasound. The three-dimensional ultrasound volume data of the fetal face were imported into the software. Then, the median sagittal plane was taken as the reference interface, the anterior upper margin of the mandibular alveolar bone was selected as the fulcrum, the interval angles, and the number of layers of the sector scan were set, after which the automatic scan was performed. Thus, the sector scan sequential planes of the mandibular alveolar bone, pharynx, soft palate, hard palate, and maxillary alveolar bone were obtained in sequence to display and evaluate the palate. In addition, the feasibility and accuracy of the software in fetal palate displaying and screening was evaluated by actual clinical cases. Results: Full views of the normal fetal palates and the defective parts of the cleft palates were displayed, and relatively clear sequential tomographic images and continuous dynamic videos were formed after the three-dimensional volume data of 10 normal fetal palates and 10 cleft palates were imported into the software. Conclusions: The software can display fetal palates more directly which might allow for a new method of fetal palate screening and cleft palate diagnosis. [ABSTRACT FROM AUTHOR]

This case report presents a virtual treatment simulation of the orthodontic treatment and surgery-first orthognathic surgery employed to treat a patient with a repaired unilateral cleft lip and alveolus with Class III malocclusion and lower third facial asymmetry. The patient exhibited a negative overjet of 9 mm, a missing lower right second premolar, and a 5 mm gap between the upper right central and lateral incisors with midline discrepancy. The three-dimensional virtual planning began with virtual pre-surgical orthodontics, followed by the positioning of the facial bones and teeth in their ideal aesthetic and functional positions. The sequence of steps needed to achieve this outcome was then reverse-engineered and recorded using multiplatform Nemostudio software (Nemotec, Madrid, Spain), which facilitated both surgical and orthodontic planning. The treatment included a two-piece segmental maxillary osteotomy for dental space closure, a LeFort I maxillary advancement, and a mandibular setback with bilateral sagittal split osteotomy to correct the skeletal underbite and asymmetry. A novel approach was employed by pre-treating the patient for orthognathic surgeries at age 11, seven years prior to the surgery. This early phase of orthodontic treatment aligned the patient's teeth and established the dental arch form. The positions of the teeth were maintained with retainers, eliminating the need for pre-surgical orthodontics later. This early phase of treatment significantly reduced the treatment time. The use of software to predict all the necessary steps for surgery and post-surgical orthodontic tooth movements made this approach possible. Multi-step virtual planning can be a powerful tool for analyzing complex craniofacial problems that require multidisciplinary care, such as cleft lip and/or palate. [ABSTRACT FROM AUTHOR]

Objective: Measuring disability as a concept of impaired global function enables beneficiaries of treatment, the impact of treatment, and targets of health system investment to be rigorously assessed. Measures of disability are not well established for cleft lip and palate. This study aims to systematically review disability weight (DW) studies pertaining to orofacial clefts (OFCs) and identify methodological strengths and shortcomings of each approach. Design: Systematic literature review of studies that met the following criteria: (1) peer-reviewed publication, (2) focus on disability valuation, (3) mention orofacial clefts, and (4) publication January 2001-December 2021. Setting: None. Patients/Participants: None. Interventions: None. Main Outcome Measure(s): Disability weight method of valuation and the value itself. Results: The final search strategy yielded 1,067 studies. Seven manuscripts were ultimately included for data extraction. The disability weights used in our studies, including those newly generated or taken from the Global Burden of Disease Studies (GBD), ranged widely for isolated cleft lip (0.0–0.100) and cleft palate with or without cleft lip (0.0−0.269). The GBD studies limited their consideration of cleft sequelae informing disability weights to impact on appearance and speech-related concerns, while other studies accounted for comorbidities such as pain and social stigma. Conclusions: Current measures of cleft disability are sparse, inadequately reflect the comprehensive impact of an OFC on function and socialization, and are limited in detail or supporting evidence. Use of a comprehensive health state description in evaluating disability weights offers a realistic means of accurately representing the diverse sequelae of an OFC. [ABSTRACT FROM AUTHOR]

Objective: To assess modelled facial development of infants with unilateral cleft lip (CL) and cleft lip and palate (UCLP) compared to controls up to two years of age. Design and Participants: A total of 209 facial images of children (CL: n = 37; UCLP: n = 39; controls: n = 137) were obtained in four age categories (T0 = 0.2–0.5; T1 = 0.6–1.0; T2 = 1.1–1.5; T3 = 1.6–2.0 years) and were evaluated using stereophotogrammetry and geometric morphometry. All patients underwent lip surgery before T0, patients with UCLP underwent palatoplasty (T0, T1 before palatoplasty; T2, T3 after palatoplasty). Results: In patients with CL, the forehead was significantly retracted (p ≤ 0.001), while the supraorbital and ocular regions were prominent (p ≤ 0.001). The oronasal region appeared convex (p ≤ 0.001). The lower lip and chin were non-significantly protruded. In patients with UCLP, a significantly retracted forehead and prominent supraorbital region were apparent (p ≤ 0.001). A retrusive oronasal region (p ≤ 0.001) was observed in the middle face. The chin was anteriorly protruded (p ≤ 0.01). No progression of deviations was found with increasing age. After the first year, a slight improvement in the morphological features became apparent. The shape variability of the clefts and controls overlapped, suggesting a comparable modelled facial development. Conclusions: The facial morphology of individuals with cleft was comparable to the norm. Shape deviation was apparent in the oronasal region, forehead, and chin, which minimised with increasing age even in complete clefts. [ABSTRACT FROM AUTHOR]

Background: Children born with Trisomy 13 or 18 (T13/18) often have multiple congenital anomalies, many of which drastically shorten their lifespan. Among these defects are cleft lip and palate, the repair of which presents an ethical dilemma to the surgeon given the underlying comorbidities associated with T13/18. The authors present an ethical discussion and institutional experience in navigating this dilemma. Methods: The authors analyzed existing literature on T13 and T18 surgery and mortality. A retrospective study over ten years was also conducted to identify pediatric patients who underwent surgical correction of cleft lip and/or palate secondary to a confirmed diagnosis of T13/18. The authors identified two patients and examined their treatment course. Results: The authors' review of literature coupled with their institution's experience builds on the published successes of correcting cleft lip and palate in the setting of T13/18. It was found that both patients identified in the case series underwent successful correction with no surgical complications. Conclusion: A careful balance must be struck between improved quality of life, benefits of treatment, and risks of surgery in children with T13/T18. Careful consideration should be given to the medical status of these complex patients. If the remaining medical comorbidities are well managed and under control, there is an ethical precedent for performing cleft lip and palate surgeries on these children. A diagnosis of T13/T18 alone is not enough to disqualify patients from cleft lip/palate surgery. [ABSTRACT FROM AUTHOR]

Non-syndromic orofacial cleft (OFC) is the most common facial developmental defect in the global population. The etiology of these birth defects is complex and multifactorial, involving both genetic and environmental factors. This study aimed to determine if SNPs in the WNT gene family (rs1533767, rs708111, rs3809857, rs7207916, rs12452064) are associated with OFCs in a Polish population. The study included 627 individuals: 209 children with OFCs and 418 healthy controls. DNA was extracted from saliva for the study group and from umbilical cord blood for the control group. Polymorphism genotyping was conducted using quantitative PCR. No statistically significant association was found between four variants and clefts, with odds ratios for rs708111 being 1.13 (CC genotype) and 0.99 (CT genotype), for rs3809857 being 1.05 (GT genotype) and 0.95 (TT genotype), for rs7207916 being 0.86 (AA genotype) and 1.29 (AG genotype) and for rs12452064 being 0.97 (AA genotype) and 1.24 (AG genotype). However, the rs1533767 polymorphism in WNT showed a statistically significant increase in OFC risk for the GG genotype (OR = 1.76, p < 0.001). This research shows that the rs1533767 polymorphism in the WNT gene is an important risk marker for OFC in the Polish population. [ABSTRACT FROM AUTHOR]

Establishing a successful perioperative technique for better outcomes remains a priority in cleft surgery. In the present review, the safety and efficacy of nerve blocks for cleft lip and cleft palate are evaluated. A systematic review of randomized controlled trials (RCTs) involving the perioperative nerve block was conducted. PubMed, Google Scholar databases, and manual searching were performed to screen the eligible RCTs. Dosage and the route of administration of anesthetics were considered for all the studies. A total of nine RCTs with 568 children were included in our review. Six out of nine studies reported cleft lip surgery, only two reported cleft palate surgery, and one study reported both cleft lip and palate surgery. Lignocaine, ropivacaine, or bupivacaine was used as anesthetics. Adrenaline or epinephrine was used as adjuvants. Post-operative pain was lower in the perioperative nerve block group compared to the comparison group, resulting in fewer emergency analgesic prescriptions. Physiological factors like heart rate, blood pressure, and respiratory rate did not differ between groups. There were fewer adverse outcomes linked with perioperative nerve blocks. Perioperative nerve blocks can be a useful adjunct to general anesthesia in cleft lip and palate surgeries to provide additional pain relief and reduce opioid use, but they should be used judiciously and with consideration of potential risks and complications. [ABSTRACT FROM AUTHOR]

Background/Objectives: Orofacial clefts (OFCs) are some of the most common congenital anomalies worldwide. The aim of this case–control study was to evaluate the association of OFCs with selected maternal characteristics. Methods: Data on isolated non-syndromic cases of OFCs were extracted from the population-based registry of congenital anomalies of Tuscany. A sample of live-born infants without any congenital anomaly was used as the control group. We investigated the association with sex and some maternal characteristics: age, body mass index, smoking, and education. Adjusted odds ratios (OR) were calculated using a logistic regression model. Analyses were performed for the total OFCs and separately for cleft lip (CL) and cleft palate (CP). Results: Data on 219 cases and 37,988 controls were analyzed. A higher proportion of males (57.9%) was observed, particularly for CL. A decreasing trend among the maternal age classes was observed (OR:0.81 (95%CI 0.70–0.94)). Underweight mothers had a higher prevalence of OFCs, in particular for CL (OR:1.88 (95%CI 1.08–3.26)). Conclusions: We found an association of OFCs with lower maternal age. The association with maternal age remains controversial and further epidemiological evidence is needed through multicenter studies. We observed that CL was more common in underweight mothers, suggesting actions of primary prevention. [ABSTRACT FROM AUTHOR]

Introduction and aim. This study aims to assess caries experience in children with cleft lip and palate (CLP). Material and methods. A total of 127 children (aged 3-12 years) with CLP and 141 non-cleft controls were assessed for dental caries using the decayed-extracted/missing-filled teeth (deft/DMFT) index. Logistic regression analysis was employed to identify factors associated with dental caries. Statistical tests, including t-test, ANOVA, Chi-square, and Fisher's exact test, were used to analyze differences between cleft and non-cleft populations. Results. In primary teeth, caries prevalence was significantly higher in children with CLP (63.8%) compared to non-cleft controls (40.4%, p<0.001). The deft scores were 3.30 for CLP and 1.63 for non-CLP group (p<0.01). No significant difference was observed in mean DMFT scores (p=0.02). Significant variations in caries prevalence (p=0.01) and mean DMFT values (p=0.001) were noted in permanent dentition among different cleft groups. Conclusion. Children with CLP show higher caries prevalence and experience, emphasizing the need for targeted dental care interventions in this population. Logistic regression analysis highlights an age-related increase in caries experience among individuals with CLP. [ABSTRACT FROM AUTHOR]

When considering the management of asymmetry, volume deficiency, scarring and vermillion notching following cleft lip repair, surgical techniques are commonly applied. However, surgery may not be suitable for all patients. Hyaluronic acid (HA) is a common, non-permanent filler widely used in non-surgical aesthetic practice. We present our unit's two-year experience of the regular use of HA filler in a dedicated Adult Cleft Facial Augmentation Clinic. Methods: Retrospective case note review was undertaken of all patients treated in the clinic over a two-year period. Results: 20 patients underwent HA filler injections to the upper lip 26 times. Most were female (F:M = 3:1) and patients were aged 18-58 years. Most patients had a unilateral cleft lip +/− palate (n = 13, 65%). The most common indication was to address upper lip volume (n = 13, 65%). Other indications included vermillion notch (n = 5, 25%), cupid bow peak height asymmetry (n = 4, 20%), scar asymmetry (n = 1, 5%) and nasal sill flattening (n = 1, 5%). Small volumes of filler were used with an average of 0.34 ml (range 0.05-1.2 ml). There were no complications and one patient reported pruritis post procedure. Conclusions: HA filler is a safe and reliable treatment for certain aspects of asymmetry following cleft lip repair. It can be used to address volume deficiency and asymmetry, cupid bow peak height discrepancies and a vermillion notch for patients who do not want surgery. Injection of HA to the lips can be performed easily, with appropriate training, in the outpatient setting. [ABSTRACT FROM AUTHOR]

Objective: : The current study examined stop consonant production in children with cleft lip and/or palate (CP ± L) 2-6 months following palatal surgery. Design: : Prospective comparative study. Setting: : Multisite institutional. Participants: : Participants included 113 children with repaired CP ± L (mean age = 16 months) who were participating in the multicenter CORNET study. Procedures: Parents of participants were asked to record approximately two hours of their child's vocalizations/words at home using a Language ENvironmental Analysis (LENATM) recorder. Four ten-minute audio-recorded samples of vocalizations were extracted from the original recording for each participant and analyzed for presence of oral stop consonants. A minimum of 100 vocalizations were required for analysis. Results: : Preliminary findings indicate that at least one oral stop was evident in the consonant inventory for 95 of the 113 children (84%) at the time of their post-surgery 16-month recording, and 80 of these children (71%) were producing two or more different stops. Approximately 50% of the children (57/113) produced the three voiced stops, and eight of the children (7%) were producing all six stop consonants. Conclusions: : The findings of this study suggest that the majority of children with repaired CP ± L from English-speaking homes are producing oral stops within six months following palatal surgery. Similar to same-age children without CL ± P, voiced stops were more frequently evident in the children's inventories than voiceless stops. In contrast to findings of previous reports suggesting place of articulation differences, a somewhat comparable percentage of children in this study produced voiced bilabial, alveolar, and velar stops. [ABSTRACT FROM AUTHOR]

Objective: This study aimed to determine whether increased raising of the back of the tongue is evident in children with repaired cleft palate with or without cleft lip (CP+/-CL). We hypothesized that children with CP+/-CL would show increased raising of the tongue dorsum, a compensatory pattern. Method: Secondary data analysis of mid-sagittal ultrasound tongue imaging data from 31 children with CP+/-CL and 29 typically developing children were used. We annotated the consonants /ʃ, t, s, k/ at the point of maximum constriction in an /aCa/ environment. Children with CP+/-CL said the tokens 10 times, typically developing children said them once. We automatically fitted splines to the tongue contour and extracted the Dorsum Excursion Index (DEI) for each consonant. This metric measures the relative use of the tongue dorsum, with more posterior consonants having higher values. We compared DEI values across groups and consonants using a linear mixed effects model. DEI was predicted by the interaction of consonant (baseline: /ʃ/) and speaker type (baseline: TD), including by-speaker random slopes for consonant and random intercepts for speaker. Results: Overall DEI was not higher in children with CP+/-CL compared to typically developing children. Between groups the only significant difference was the position of /k/ relative to /ʃ/, where the difference between these two consonants was smaller in the children with CP+/-CL. Conclusions: There was no support for the hypothesis that increased raising of the tongue dorsum is a common characteristic in children with repaired CP+/-CL. However, individual children may present with this pattern. [ABSTRACT FROM AUTHOR]

Structural variation is a source of genetic variation that, in some cases, may trigger pathogenicity. Here, we describe two cases, a mother and son, with the same partial inverted duplication of the long arm of chromosome 8 [invdup(8)(q24.21q24.21)] of 17.18 Mb, showing different clinical manifestations: microcephaly, dorsal hypertrichosis, seizures and neuropsychomotor development delay in the child, and a cleft lip/palate, down-slanted palpebral fissures and learning disabilities in the mother. The deleterious outcome, in general, is reflected by the gain or loss of genetic material. However, discrepancies among the clinical manifestations raise some concerns about the genomic configuration within the chromosome and other genetic modifiers. With that in mind, we also performed a literature review of research published in the last 20 years about the duplication of the same, or close, chromosome region, seeking the elucidation of at least some relevant clinical features. [ABSTRACT FROM AUTHOR]