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OBJECTIVE: To evaluate the role of psychosocial well-being on perioperative pain and opioid use among patients with cleft lip and palate (CLP) undergoing alveolar bone grafting (ABG). DESIGN: Retrospective review. SETTING: Tertiary level craniofacial clinic. PARTICIPANTS: 34 patients with CLP (median age: 11.7 years), including 25 (73.5%) unilateral CLP and 9 (26.5%) bilateral CLP, who underwent ABG from 2015 to 2022. INTERVENTIONS: ABG using iliac crest bone graft. Patients were prospectively administered four patient-reported psychosocial instruments from the Patient-Reported Outcomes Measurement Information System. MAIN OUTCOME MEASURES: Perioperative opioid use in morphine equivalent dosage/kilogram, patient-reported pain scores, and length of hospital stay after ABG. RESULTS: Patient-reported anxiety (r = 0.41, p = 0.02) and depressive symptoms (r = 0.35, p = 0.04) correlated to higher perioperative opioid usage. Multivariable regression models including psychosocial scores, total acetaminophen usage, length of surgery, and other simultaneous surgeries were developed for total opioid usage, patient-reported pain, and length of hospital stay. Patient-reported anxiety was independently predictive of higher perioperative opioid use (β=0.36, p = 0.01) and higher pain scores (β=0.39, p = 0.02), but not length of hospital stay. CONCLUSIONS: We identified an association for patient-reported anxiety and perioperative opioid use and pain in a CLP cohort undergoing ABG. Future considerations in preoperative patient and family consultation may be indicated in patients self-reporting higher anxiety in an effort to minimize perioperative opioid usage.

PURPOSE OF REVIEW: Patients with cleft lip -palate (CLP) experience morbidity and social stigma, particularly in low-income and middle-income countries (LMICs) such as those of sub-Saharan Africa (SSA). Delays in treatment secondary either to lack of awareness, skills, equipment and consumables; poor health infrastructure, limited resources or a combination of them, has led to SSA having the highest rates of death and second highest rates of disability-adjusted life years in patients with CLP globally. Here we review current perspectives on the state of comprehensive cleft lip and palate repair in Africa. RECENT FINDINGS: To bridge gaps in government health services, nongovernmental organizations (NGOs) have emerged to provide care through short-term surgical interventions (STSIs). These groups can effect change through direct provision of care, whereas others strengthen internal system. However, sustainability is lacking as there continue to be barriers to achieving comprehensive and longitudinal cleft care in SSA, including a lack of awareness of CLP as a treatable condition, prohibitive costs, poor follow-up, and insufficient surgical infrastructure. With dedicated local champions, a comprehensive approach, and reliable partners, establishing sustainable CLP services is possible in countries with limited resources. SUMMARY: The replacement of CLP missions with locally initiated, internationally supported capacity building initiatives, integrated into local healthcare systems will prove sustainable in the long-term.

Objectives: Addressing a single-tooth gap in the anterior region, resulting from aplasia or trauma, poses both esthetic and functional challenges. This case report presents the restoration ofa young adult with a cleft, exhibiting anterior hypoplasia and aplasia in the canine and incisor regions, using all-ceramic cantilever resin-bonded fixed dental prostheses. Method and materials: After verification of esthetic and functional considerations through a diagnostic wax-up and an intraoral mock-up, three anterior all-ceramic cantilever resin-bonded fixed dental prostheses made of veneered zirconium dioxide were planned in the region ofthe maxillary right lateral incisor and maxillary left canine. The impression was made with an intraoral scanner. The framework fit was evaluated. Glaze bring and full adhesive cementation under rubber dam followed. Results: The final restoration met the patients' expectations and restored facial esthetics and function. Conclusions: All-ceramic cantilever resin-bonded fixed dental prostheses offer a promising minimally invasive therapeutic option for cleft patients. [ABSTRACT FROM AUTHOR]

ObjectiveThe purpose of this study was to evaluate long-term outcomes of sphincter pharyngoplasties, including speech outcomes, revision surgeries, and postoperative incidence of obstructive sleep apnea (OSA).DesignRetrospective matched-cohort study SETTING: Two craniofacial centers in Los Angeles, CA PATIENTS: Patients (n = 166) with cleft lip and palate (CLP) or isolated cleft palate (iCP) who underwent sphincter pharyngoplasty from 1992 to 2022 were identified. An age- and diagnosis-matched control group of 67 patients with CLP/iCP without velopharyngeal insufficiency (VPI) was also identified.InterventionsThe pharyngoplasty group underwent sphincter pharyngoplasty, whereas the non-VPI group had no history of VPI surgery or sphincter pharyngoplasty.Main outcome measuresPostoperative speech outcomes, revision surgeries, and incidence of OSA were evaluated. Multivariable regression was used to evaluate independent predictors of OSA.ResultsAmong the patients in the pharyngoplasty cohort, 63.9% demonstrated improved and sustained speech outcomes after a single pharyngoplasty, with a median postoperative follow-up of 8.8 years (interquartile range [IQR], 3.6-12.0 years). One-third of the patients who underwent pharyngoplasty required a revision surgery, with a median time to primary revision of 3.9 years (IQR, 1.9-7.0 years). OSA rates increased significantly among the pharyngoplasty cohort, from 3% before surgery to 14.5% after surgery (p

Objective(s): It was the first study to apply and compare two CT methods to assess the validity and clinical significance of structural alterations of the nasal valve in patients with cleft lip nose for assessing nasal ventilation disturbance. Methods: The study collected data from the NOSE score, as well as internal nasal valve area, internal nasal valve angle, external nasal valve area, and septal deviation angle, to evaluate the differences and correlations between those factors in patients with cleft lip and nose. Results: There were significant differences among INV transverse and coronal area and INV angle on different axial standardized planes between clefted side and non-clefted side. There were statistically significant negative correlations between NOSE scores and those indicators of standard plane and acoustic-axis standardized coronal plane. NOSE score and NSD angle were the indicators of significant differences in the measured data of different complications groups (p = 0.002, p = 0.017). The correlation comparison showed that two standardized CT imaging transverse planes have similar correlations in NOSE score, NSD angle, and complications. Conclusion: The results of the two CT evaluation methods showed that there was a significant difference in nasal valve area on the cleft and non-cleft sides, which was significantly associated with nasal ventilation disturbance. The CT evaluation method based on standard axial 3D reconstruction is more convenient to use in the clinic, can be used for pre-surgical evaluation of nasal repair in patients with secondary nasal deformities of unilateral cleft lip, and is valuable for treatment. Level of Evidence IV: This journal requires that authors assign a level of evidence to each article. For a full description of these Evidence-Based Medicine ratings, please refer to the Table of Contents or the online Instructions to Authors www.springer.com/00266. [ABSTRACT FROM AUTHOR]

Background: This study evaluates a three-dimensional (3D) visualisation design combined with customized surgical guides to assist anterior maxillary segmental distraction osteogenesis (AMSDO) in correcting maxillary hypoplasia in adolescents with cleft lip and palate (CLP), focusing on treatment outcomes, satisfaction and the validity of 3D planning. Methods: This retrospective cohort study was conducted at a single hospital in China. Between January 2020 and December 2023, 12 adolescents with CLP with maxillary hypoplasia were included. An advanced 3D simulation was used to convey the treatment strategy to the patients and their families. A customized surgical guide and distraction osteogenesis device were designed. Cephalometric analysis evaluated AMSDO changes and long-term stability. Patient satisfaction was assessed. The Chinese version of the Child Oral Health Impact Profile was used to evaluate the children's oral health-related quality of life before and after treatment. The postoperative outcomes were compared with the planned outcomes by superimposing the actual postoperative data onto the simulated soft tissue models and calculating the linear and angular differences between them. Results: One patient experienced postoperative gingivitis, yielding an 8.33% complication rate. Most patients (83.33%) were highly satisfied with the target position, with the rest content. Cephalometric analysis showed significant improvements in various indices post-traction. Quality-of-life scores significantly improved post-treatment. The discrepancies in facial soft tissue between the simulated and actual results were within clinically satisfactory ranges. Conclusions: Digitally designed surgical guides effectively treat maxillary hypoplasia in adolescents with CLP, ensuring stability, reducing complications, reducing dependency on operator experience, and enhancing satisfaction and health outcomes. Although the simulated results were clinically acceptable, it is important to inform patients of potential variations in the predicted soft tissue. [ABSTRACT FROM AUTHOR]

Objective: The aim of this study was to present optimized device‐specific low‐dose cone‐beam computed tomography (CBCT) protocols with sufficient image quality for pre‐surgical diagnostics and three‐dimensional (3D) modelling of cleft defects. Methods: Six paediatric skulls were acquired, and an artificial bony cleft was created. A high‐resolution CBCT scan acted as a reference standard (Accuitomo 170, Morita, Kyoto, Japan) for comparing eight low‐dose protocols of Newtom VGi‐evo (QR Verona, Cefla, Verona, Italy), which included Eco and Regular protocols with different field of views (FOVs). Delineation of lamina dura, cementoenamel junction (CEJ), trabecular bone and bony bridge were assessed. A 3D model of the defect was also evaluated. Result: The dose area product of low‐dose protocols ranged from 31 to 254 mGy*cm2. Despite the dose difference of up to eight times between applied protocols, trabecular bone and CEJ exhibited appropriate image quality in all scans. However, Regular small FOV protocols (5 × 5 and 8 × 5 cm2), for both lamina dura and bony bridge, demonstrated a significant improvement in image quality compared to Eco FOV counterparts. Based on 3D defect analysis, no significant difference existed between low‐dose protocols and the reference standard. Conclusion: The findings highlight the possibility of achieving a considerable reduction (up to eight times) in the radiation dose using low‐dose CBCT protocols while maintaining sufficient image quality for assessing anatomical structures and 3D modelling in cleft cases. [ABSTRACT FROM AUTHOR]

This study aimed to document the prevalence, severity, and risk factors of velopharyngeal dysfunction (VPD) in craniofacial microsomia (CFM) and to analyse differences in VPD-related speech characteristics between CFM patients without cleft lip and/or palate (CL/P), CFM patients with CL/P, and CL/P patients without CFM (control). A total of 223 patients with CFM were included, of whom 59 had a CL/P. Thirty-four CFM patients had VPD, including 20 with a CL/P. VPD was significantly more prevalent in CFM with CL/P than in CFM without CL/P (odds ratio (OR) 4.1, 95% confidence interval (CI) 1.9–8.7; P < 0.001). Multivariate logistic regression showed a significant association between CL/P and VPD in CFM patients (OR 7.4, 95% CI 2.1–26.3; P = 0.002). The presence of VPD was not associated with sex, the laterality or severity of CFM. Speech problems related to VPD appeared to be similar among the different groups (CFM without CL/P, CFM with CL/P, CL/P without CFM). As 15.2% of all CFM patients and 8.5% of CFM patients without CL/P had VPD, it is proposed that all patients with CFM, with or without CL/P, should be assessed by a speech and language therapist for the potential risk of VPD. [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: The study primarily aimed to compare satisfaction with lip appearance among adults treated for unilateral cleft lip and palate (UCLP) with Skoog's primary lip repair procedure to those without clefts. The secondary aim was to determine whether satisfaction with lip appearance and the desire to change the lip/face appearance correlated with the number of secondary lip revisions performed. Design: Long-term follow-up Patients/settings: All UCLP patients treated at the Uppsala University Hospital born between 1960- and 1987 (n = 109) were invited. At an average of 37 years following the primary lip repair, the participation rate was 76% (n = 83). A control group of adults without cleft (n = 67) completed the same study protocol for comparison. Main Outcome Measures: Satisfaction with appearance was measured with The Satisfaction with Appearance Questionnaire (SWA) and a modified version of the Body Cathexis -Scale was used to assess the desire to change the lip and facial appearance. Results: UCLP patients were less satisfied with their lip, face, and overall appearance and reported a greater desire to change the appearance of their lips and face than non-cleft controls (p < 0.001). Dissatisfaction with lip appearance correlated to a greater willingness to change the appearance of the lip and face. No correlation was found between satisfaction with appearance and the number of the previously performed secondary lip revisions. Conclusion: Adults treated for UCLP are less satisfied with the appearance of their lips compared to the non-cleft population. The number of secondary revisions does not necessarily correlate to greater satisfaction with lip appearance. [ABSTRACT FROM AUTHOR]

Objective: To evaluate a method of measuring the change in palatal length and shape following maxillary advancement using synchronous lateral videofluoroscopy and voice recording in order to understand how movement of the maxilla may affect VPI risk in patients with cleft lip and/or palate (CL/P). Design: Retrospective cohort study of children with cleft lip and/or palate. Setting: Single center, tertiary children's hospital. Participants: Patients with cleft lip and/or palate who underwent maxillary advancement between 2016–21 inclusive. Interventions: Maxillary advancement surgery, including those who underwent concurrent mandibular procedures. Main outcome measures: The length of the soft palate and the genu angle were measured throughout palatal dynamic range. Pre- and post-operative measurements were compared using a one sided T-test, with subgroup analysis for patients with clinical VPI. Results: Ten patients were examined. The mean distance of maxillary advancement was 10.5 mm. The average increase in pre-genu soft palate length was 2.8 mm in the resting position and 2.9 mm in the closed position. The genu angle decreased in the closed position by 16.3 degrees. Conclusions: The soft palate showed limited ability to lengthen following maxillary advancement and this may explain the risk of VPI. There was partial compensation by the muscle sling of the palate as demonstrated by a more acute post-operative genu angle and this suggests one reason for the variability of VPI reported. Future research is required to investigate how length and shape changes measured using this method can predict VPI risk. [ABSTRACT FROM AUTHOR]

Objective: To compare the soft tissue measurements of the upper lip and the nose on the cleft side and the noncleft side before surgery, immediately after cheiloplasty and two months after surgery. Design: Descriptive clinical study with one group, prospective. Setting: Department of Odonto-Stomatology, Children's Hospital 1, at Ho Chi Minh City - Vietnam. Patients: 31 patients with a unilateral complete cleft lip participated in this study, 30 patients were evaluated at the time two months after surgery. Interventions: Interventions include PNAM and cheiloplasty by modified Millard technique. Main Outcome Measures: Patients proceed to acquire 3D images of lips and nose, define landmarks and measure dimensions. There are 11 evaluators to be compared, p < 0.05 was considered statictical significance. Results: After two months of surgery, on cleft side and noncleft side, the lengths of the upper lip were 10.87 ± 0.80 and 11.92 ± 0.78 (mm), the widths of the upper lip were 16.06 ± 1.10 and 16.40 ± 1.02 (mm), the heights of the nostril were 4.85 ± 0.44 and 5.93 ± 0.43 (mm), the lengths of columella were 4.08 ± 0.37 and 4.93 ± 0.38 (mm), and the widths of the nostril were 9.07 ± 0.37 and 8.37 ± 0.40 (mm). Conclusion: Cheiloplasty by modified Millard technique on the patients who used PNAM showed that after two months of surgery, the morphology of the upper lip and nose has a slight disproportion with the nasolabial measurements of the cleft side being smaller than on the noncleft side. [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]

Objective : Evaluate periodontal bone support of maxillary central incisors (MCI) in patients with bilateral complete cleft lip and palate (BCCLP). Determine if syndromic diagnosis, age at time of alveolar bone graft (ABG), presence of maxillary lateral incisor (MLI), history of dentofacial orthopedics, maxillary expansion, and pre-maxillary osteotomy are associated with the periodontal bone support of MCI. Design : Retrospective radiographic study. Setting : Tertiary care children's hospital Patients : One hundred seventy-nine patients with BCCLP (22 syndromic) who had post-operative ABG cone beam computed tomography (CBCT) scans taken between 2002-2018. Main Outcome Measures: Crown to root (C/R) ratio of MCI measured on CBCT scans. Results : The C/R ratio in 65% of MCI indicated periodontally compromised teeth. Presence of a MLI improved bone support on adjacent MCI when compared to those missing a MLI (51.4% vs 28.4%, P =.010). There was no significant difference in C/R ratios for syndromic diagnosis, age at ABG, history of dentofacial orthopedics, maxillary expansion, and pre-maxillary osteotomy. Conclusions : The majority of MCI in patients with BCCLP are periodontally compromised but bone support is improved when cleft adjacent lateral incisors are present. [ABSTRACT FROM AUTHOR]

Objective: Taking advantage of the broad coverage of Wireless Application Protocol (WAP), we developed a Content Management System (CMS)-programmed mobile learning application. This application can help the undergraduate to obtain a comprehensive understanding of concepts in Cleft lip and palate Phenotype, and Embryonic development (CPE). The present study aims to evaluate the feasibility and efficacy of the concept acquisition teaching model on the basis of WAP in a practical undergraduate course of CPE. Design: The concept acquisition teaching model based on WAP was programmed by CMS, covering definitions of various cleft lip and palate, the mechanisms underlying the phenotypes, practical medical cases, as well as corresponding tests after learning. Setting: The CPE concept acquisition teaching model was distributed to a total of 524 undergraduate students and 46 tutors participated in CPE teaching from seven highly ranked schools of stomatology in China since April 2022. Participants: 524 undergraduate students and 46 tutors from seven highly ranked schools of stomatology in China. Interventions: The CPE concept acquisition teaching model. Main outcome measures: The effectiveness of the CPE teaching model. Results: The response rate to the survey was 100%. The grading of the questionnaires indicated that the students were satisfied with the usability, practicality, and outcome, whereas the tutors were more positive with the contents, cooperation, and outcome. Conclusions: The present study demonstrated the feasibility and efficacy of the WAP-based concept acquisition teaching model of CPE and a high level of satisfaction among undergraduate students and tutors who major in Stomatology. [ABSTRACT FROM AUTHOR]

Objective: To evaluate the outcomes of Secondary Alveolar Bone Grafting (SABG) in patients treated either in daycare or with multiple day hospitalization (MDH) in relation to costs and complication rates. Design: Retrospective comparative cohort study. Setting: The data was collected from two settings: Postoperative daycare or MDH after oral cleft surgery in an Academic Medical Center in The Netherlands. Patients: Data of 137 patients with unilateral Cleft lip, alveolus, and palate (CLAP) treated between 2006-2018 were evaluated. Registered clinical variables: age, gender, cleft subtype, bone donor site, type of hospitalization, length of stay, additional surgery, complications, surgeons, and costs. Interventions: Closure of the alveolar cleft with/without closure of the anterior palate. Main outcome measures: Univariate analyses. Results: Of the 137 patients, 46.7% were treated in MDH, and 53.3% in daycare. Total costs for daycare were significantly lower (P <.001). All patients treated in daycare received mandibular symphysis bone, whereas in MDH, 46.9% received iliac crest bone instead. Bone donor site was associated with postoperative care type. Complication rates were slightly but not significantly higher in daycare (26%) vs. MDH (14.1%) (P =.09). Most were Grade I (minor) according to Clavien Dindo classification. Conclusions: Daycare after alveolar cleft surgery is about as safe as MDH, but significantly cheaper. [ABSTRACT FROM AUTHOR]

Objective: Significant evidence links epigenetic processes governing the dynamics of DNA methylation and demethylation to an increased risk of syndromic and nonsyndromic cleft lip and/or cleft palate (CL/P). Previously, we characterized mesenchymal stem/stromal cells (MSCs) at different stages of osteogenic differentiation in the mouse incisor dental pulp. The main objective of this research was to characterize the transcriptional landscape of regulatory genes associated with DNA methylation and demethylation at a single-cell resolution. Design: We used single-cell RNA sequencing (scRNA-seq) data to characterize transcriptome in individual subpopulations of MSCs in the mouse incisor dental pulp. Settings: The biomedical research institution. Patients/Participants: This study did not include patients. Interventions: This study collected and analyzed data on the single-cell RNA expssion in the mouse incisor dental pulp. Main outcome measure(s): Molecular regulators of DNA methylation/demethylation exhibit differential transcriptional landscape in different subpopulations of osteogenic progenitor cells. Results: scRNA-seq analysis revealed that genes encoding DNA methylation and demethylation enzymes (DNA methyltransferases and members of the ten-eleven translocation family of methylcytosine dioxygenases), methyl-DNA binding domain proteins, as well as transcription factors and chromatin remodeling proteins that cooperate with DNA methylation machinery are differentially expressed within distinct subpopulations of MSCs that undergo different stages of osteogenic differentiation. Conclusions: These findings suggest some mechanistic insights into a potential link between epigenetic alterations and multifactorial causes of CL/P phenotypes. [ABSTRACT FROM AUTHOR]

Background: Microform cleft lip is the mildest type of cleft lip without obvious defects of the upper lip. The nose deformities of microform cleft lip include flattened nostril rim, alar base asymmetry, and septal deviation. A hidden skin incision with nasal base muscle reconstruction was introduced in nose deformities of microform cleft lip. Method: To investigate the operative effect, we reviewed 21 patients with microform cleft lip treated with a hidden skin incision with nasal base muscle reconstruction from May 2020 to October 2022. Photogrammetric nasal morphometric measurements were compared preoperatively and six months postoperatively. The proportional value was obtained from the cleft and the noncleft sides, and paired t-test analysis was used to evaluate the surgical outcome. Result: Significant differences were found in all nasal morphologic measurements at 6 months postoperatively compared to preoperatively (p < 0.05). After surgery, the alar base and nostril were narrowed, while the lateral lip height was increased on the affected side. The height of the nostrils on the affected side was increased, and the nasal columella deviation was released. In addition, the ratio of the cleft-to-noncleft nostril area was closer to 1.0 after surgery. Conclusion: The unilateral microform cleft lip nasal deformity can be repaired through a small hidden incision. Level of Evidence III: This journal requires that authors assign a level of evidence to each article. For a full description of these evidence-based medicine ratings, please refer to the Table of Contents or the online Instructions to Authors www.springer.com/00266. [ABSTRACT FROM AUTHOR]

Background: Cleft lip and palate are often accompanied by maxillary asymmetry and midface hypoplasia. However, there has been limited examination of the symmetry of the zygoma in these patients. In this study, computed tomography scans prior to alveolar bone grafting are utilized to examine zygomatic symmetry through width, projection, and volume. Methods: Retrospective review was conducted of patients with nonsyndromic unilateral cleft lip and palate with eligible scans prior to alveolar bone grafting at our institution. Anterior zygomatic width, posterior zygomatic width, and zygomatic projection were measured on axial images oriented to the Frankfort horizontal, and three-dimensional digital models of each zygoma were created from the computed tomography data to estimate zygoma volume. These measurements were then compared on cleft and noncleft sides. Results: Twenty-two patients were included in this study. The mean (95% CI) zygoma volume of the cleft side and non-cleft side was 4219 mm3 (3858-4574 mm3) and 4286 mm3 (3932-4660 mm3), respectively, with a mean difference of −67 mm3 ([−402-266 mm3]; P =.704). The zygomatic projection of the cleft side was 43 mm (41-45 mm) in comparison to 47 cm (45-49 mm) for the non-cleft side. Mean difference in zygomatic projection was −4 mm (−5 to −2 mm, P <.001). Mean anterior zygomatic width was greater on the cleft side (42 mm [41-44 mm]) than the non-cleft side (41 mm [40-42 mm]; P =.037). No significant difference was found between posterior zygomatic width of the cleft side (59 mm [58-60 mm]) and non-cleft side (58 mm [57-59 mm]; P =.118). Conclusions: Upon preoperative image analysis in patients with non-syndromic, unilateral lip and palate clefts, zygomatic projection was significantly different between cleft and non-cleft sides. Evaluation of zygoma asymmetry should be included as a component of the physical exam when assessing a patient with unilateral cleft lip and palate, as improved awareness of existing zygomatic asymmetry could aid in setting patient expectations, improving satisfaction/outcomes, and in planning for potential surgical correction. [ABSTRACT FROM AUTHOR]

Congenital midline cervical cleft (CMCC) is a rare congenital abnormality of the ventral neck that is often misdiagnosed. We present a case of a 12-year-old male with a history of incomplete CMCC excision and limited neck extension. We highlight the need for increased awareness of this anomaly and propose a clinical algorithm for clinicians when faced with this rare congenital condition. CMCC typically presents at birth and involves a defect along the midline of the neck, characterized by a nipple-like protrusion of the skin and a fibrous cord with an underlying sinus. Micrognathia and mandibular growth restriction are anticipated consequences of CMCC, requiring early intervention. Current treatment guidelines recommend surgical excision prior to age two using a Z-plasty technique. This report emphasizes the rarity and diagnostic challenges of CMCC. It underscores the significance of accurate identification and appropriate surgical planning in a timely manner. Based on the authors' experience with this case and a review of the literature, we propose a clinical algorithm aimed to enhance CMCC diagnosis and management. [ABSTRACT FROM AUTHOR]

Background: Tubercle aesthetics are under-emphasized in unilateral cleft lip repair. Teenagers and adults with repaired cleft lip often display a central whistle deformity medial to the cleft lip scar. Lateral segment vermillion elements that are often discarded at the time of the initial repair can be used for tubercle reconstruction. Methods: We obtained anthropometric measurements of the dry vermilion around the central tubercle in neonates with unilateral cleft lip to determine the extent and morphology of deficiencies. A modification of the Noordhoff vermillion flap is proposed with a trapezoidal configuration to precisely match the measured, dry vermillion deficiency. Results: Photographic documentation illustrates the surgical technique and results. Conclusions: Tubercle reconstruction is described using a modification of the Noordhoff flap. Four-year results are encouraging. Observations through maturity are needed. [ABSTRACT FROM AUTHOR]

Backgroud: A systematic analysis was conducted to investigate the molecular etiology of fetal cleft lip and/or palate (CL/P) and the association between various types of CL/P and copy number variations (CNVs), as well as their impact on birth outcomes. Methods: In this retrospective study conducted between January 2016 and July 2022, a cohort of pregnancies diagnosed with fetal CL/P was enrolled and comprehensive clinical data for all cases were extracted from our medical record database, including demographic data about the pregnancies, ultrasound findings, results of Chromosomal microarray (CMA), as well as relevant pregnant and perinatal outcomes. Results: Among the 358 cases, 32 clinically significant variants in 29 (8.1%) fetuses with CL/P were detected by CMA. In 338 singleton pregnancies, the diagnostic yield of CMA in the context of CL/P fetuses was determined to be 7.7% (26/338). CP cases exhibited a relatively higher prevalence of pathogenic/likely pathogenic CNVs at a rate of 25% (3/12), followed by CLP cases at 8.0% (23/288). Notably, the CL group did not demonstrate any pathogenic/likely pathogenic CNV findings among the examined cases (0/38). The diagnostic rate of clinically significant variants was notably higher in the non-isolated CL/P group than in the isolated CL/P group (11/33, 33.3% vs. 15/305, 4.9%, p < 0.001). Within the remaining 20 twin pregnancies, three clinically significant variants (15%) were observed. Conclusions: This study provides powerful evidence supporting the efficacy of CMA as a valuable tool for facilitating the prenatal genetic diagnosis of fetal CL/P. The presence of CP and CLP in fetal cases demonstrated a relatively higher incidence of pathogenic/likely pathogenic CNVs. Moreover, when these cases were accompanied by additional ultrasound abnormalities, the likelihood of identifying diagnostic CNVs significantly increased. Conversely, cases of CL alone might not be associated with positive CNVs. The present data may significantly enhance prenatal diagnosis accuracy and facilitate informed genetic counseling for cases of fetal CL/P. [ABSTRACT FROM AUTHOR]

Aim Methods Results Conclusions Cleft lip is a common congenital disease, accompanied by a complicated etiology. Medical exposure in women is one of the reasons leading to cleft lip. Phenobarbital is a major drug used for the treatment of epilepsy and has been reported to induce cleft lip, with or without cleft palate. This study aimed to explore the protective effects of Sasa veitchii extract (SE) against phenobarbital‐induced inhibition of human lip fibroblast (JCRB9103) cell proliferation.We evaluated the cell viability of JCRB9103 cells after 48 h of phenobarbital treatment. We examined the rescue experiment using SE, sodium copper chlorophyllin, or recombinant transforming growth factor‐beta 1. Western blotting was performed to identify key proteins involved in lip formation in JCRB9103 cells.Phenobarbital treatment decreased the cell viability of JCRB9103 cells in a dose‐dependent manner. Cotreatment with SE or sodium copper chlorophyllin alleviated phenobarbital‐induced inhibition of cell viability in JCRB9103 cells. SE treatment upregulated the transforming growth factor‐beta 1 level, and transforming growth factor‐beta 1 protected against phenobarbital‐induced inhibition of cell proliferation.These results suggest that phenobarbital‐induced inhibition of cell proliferation was restored by sodium copper chlorophyllin present in SE via the regulation of transforming growth factor‐beta 1. [ABSTRACT FROM AUTHOR]

Background: Pattern of dental anomalies encountered in cleft patients shows subtle signs of genetic involvement. This study aimed to evaluate the prevalence and pattern of tooth agenesis and supernumerary teeth in Thai cleft population according to the cleft type. Methods: Data collected from patients with cleft lip and palate, who had been treated at Tawanchai Cleft Center, Khon Kaen University, Thailand, available during year 2012–2022, were investigated. Records from 194 patients with non-syndromic clefts met the inclusion criteria. Standard dental records, and at least either orthopantomogram (OPG) or cone beam computed tomography (CBCT), were examined. Statistical analysis was performed using chi-square and binominal test (p ≤ 0.05). Results: Prevalence of tooth agenesis was higher (77.3%) than that of supernumerary teeth (5.7%) and was more common in bilateral cleft lip and palate (BCLP) (88.1%) than in unilateral cleft lip and palate (UCLP) (72.6%) (p = 0.017). The upper lateral incisor was more frequently affected (46.4%), followed by the upper second premolar. The number of missing teeth observed on the left side was significantly higher. Patients with left UCLP (ULCLP) had the highest prevalence of tooth agenesis. A total of 41 tooth agenesis code (TAC) patterns was found. The prevalence of supernumerary teeth was comparable with 6.6% of ULCLP, 5.1% of BCLP, and 4.5% of URCLP. Tooth-number anomalies were observed more often in the BCLP and were most likely to occur on the left side of the maxilla. Both types of anomalies could be featured in a small proportion of cleft patients. Conclusions: More than half of the patients with non-syndromic cleft lip and palate in this study, presented with tooth-number anomalies. Tooth agenesis was approximately 10-time more prevalent than supernumerary teeth. Tooth agenesis was likely to appear on the left-side of the maxilla regardless of the laterality of the cleft. [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/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]

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]

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]

Comprehensive treatment is crucial for patients with a cleft lip/palate. While studies have investigated its impact on children's quality of life, few have examined the effects on primary caregivers. The aim of the study was to compare the quality of life of caregivers of children with cleft lip/palate to a control group at the National School for Higher Studies, National Autonomous University of Mexico, Guanajuato. A cross-sectional study was conducted at a teaching dental clinic of the National School of Higher Studies, National Autonomous University of Mexico, Guanajuato, México, from May to December 2021 involving 140 caregivers (70 in each group). The WHOQoL Bref instrument assessed the quality of life. In addition to the descriptive analysis, a binary logistic regression analysis was carried out, taking dichotomized reported quality of life as the dependent variable. Among the caregivers, 88.6% were female (p > 0.05), and 45 (64.8%) from the study group reported poor quality of life (p < 0.05). A multivariate analysis indicated that caring for a child with cleft lip/palate raised the likelihood of poor quality of life (p < 0.05). The findings emphasize the need for comprehensive support for both patients and caregivers, as their well-being affects patient outcomes. [ABSTRACT FROM AUTHOR]

Background: Morphological differences in the temporomandibular joint (TMJ) are crucial for the treatment of patients with cleft lip and palate (CLP). This study aims to evaluate and compare the TMJ parameters in patients with unilateral and bilateral CLP across growing and non-growing age groups using cone-beam computed tomography (CBCT). Methods: CBCT records from 57 patients (23 males and 34 females) aged 6–50 years with a diagnosed unilateral or bilateral CLP were analyzed. Patients were categorized into four groups: growing unilateral (UGCLP), growing bilateral (BGCLP), non-growing unilateral (UNGCLP), and non-growing bilateral (BNGCLP). Measurements of TMJ parameters, including the mandibular fossa, articular eminence inclination, joint spaces, and roof thickness of the glenoid fossa, were conducted using CBCT images. Results: Significant differences were observed in the anterior joint space (AJS) and the roof of the glenoid fossa (RGF) between growing and non-growing unilateral cleft patients. Additionally, significant discrepancies were found in the articular eminence angle when comparing the cleft and non-cleft sides within the unilateral growing group. No significant differences were observed in TMJ parameters between the right and left sides among bilateral cleft patients. Conclusions: The study highlights distinct TMJ morphological differences between growing and non-growing patients with CLP, emphasizing the importance of age-specific considerations in the treatment planning and growth monitoring of these patients. [ABSTRACT FROM AUTHOR]

Background: Ectrodactyly-ectodermal dysplasia-cleft lip/palate (EEC) syndrome mainly affects ectodermal and mesodermal tissues. It is usually manifested as split hands and feet, ectodermal dysplasia, and orofacial clefting, along with other signs and symptoms. A multidisciplinary approach to treatment is required, in which dentists play an important role in identifying and treating various oral conditions that may be genetically linked to or may be the result of EEC syndrome. Case presentation: The present case describes the oral condition of a young child suffering from EEC syndrome and presenting with peripheral giant cell granuloma (PGCG) in the mandibular anterior region. After obtaining a thorough medical and family history and a clinical examination, the lesion was surgically excised under local anesthesia. The patient was followed up at periodic intervals for the next twenty four months, during which no recurrence of the lesion was observed. Conclusion: This report highlights the role of a dentist in the management of the oral conditions of patients suffering from EEC syndrome. [ABSTRACT FROM AUTHOR]

Background: Cleft lip and palate are congenital craniofacial anomalies that significantly impact individuals and their families, both medically and psychosocially. The Qatari population has unique characteristics that are suggestive of a high prevalence of congenital anomalies: high consanguinity rate, large family size, advanced paternal age and high prevalence of certain genetic disorders. The lack of existing data concerning the epidemiology of cleft lip and/or palate in Qatar warrants a descriptive study addressing this topic. Methods: A retrospective nationwide hospital-based cross-sectional study conducted to determine the prevalence of orofacial clefts in Qatar from 2016 to 2021. Data were extracted from the corporation's Cerner database and electronic health records. Information concerning the cleft's characteristics, type, affected side, patient's gender, presence of associated syndromes, nationality, and maternal age were collected. Results: Out of the 147,727 live births, 148 had an orofacial cleft. The prevalence of cleft lip and/or palate was determined to be 1 per 1000 livebirths (95% CI: 0.85, 1.18). The prevalence of cleft lip was 0.18 (95% CI: 0.12, 0.27), cleft palate 0.39 (95% CI: 0.30, 0.51), and cleft lip and palate 0.43 (95% CI: 0.33, 0.55). Qataris had a prevalence of CL 0.25, CP 0.40, and CLP 0.56, compared to 0.16, 0.39, 0.39 for non-Qataris (p-value 0.186). Unilateral clefts predominated over bilateral (74.4% and 25.6%, respectively). Among the unilateral cases, 70.2% occurred on the left side. Most cases were isolated clefts, with only 10.2% having associated syndromes. Conclusions: The prevalence of orofacial clefts in Qatar is consistent with the globally reported prevalence. Most cases were unilateral and on the left side. Associated syndromes were infrequent and more common with cleft palate alone. Intriguing patterns were revealed between Qatari nationals and non-Qatari residents, with specific subtypes of orofacial clefts showing higher prevalence among nationals. [ABSTRACT FROM AUTHOR]

The mechanisms utilized by neurons to regulate the efficacy of phasic and tonic inhibition and their impacts on synaptic plasticity and behavior are incompletely understood. Cleft lip and palate transmembrane protein 1 (Clptm1) is a membrane-spanning protein that interacts with multiple γ-aminobutyric acid type A receptor (GABAAR) subunits, trapping them in the endoplasmic reticulum and Golgi network. Overexpression and knock-down studies suggest that Clptm1 modulates GABAAR-mediated phasic inhibition and tonic inhibition as well as activity-induced inhibitory synaptic homeostasis in cultured hippocampal neurons. To investigate the role of Clptm1 in the modulation of GABAARs in vivo, we generated Clptm1 knock-out (KO) mice. Here, we show that genetic KO of Clptm1 elevated phasic and tonic inhibitory transmission in both male and female heterozygous mice. Although basal excitatory synaptic transmission was not affected, Clptm1 haploinsufficiency significantly blocked high-frequency stimulation-induced long–term potentiation (LTP) in hippocampal CA3→CA1 synapses. In the hippocampus-dependent contextual fear-conditioning behavior task, both male and female Clptm1 heterozygous KO mice exhibited impairment in contextual fear memory. In addition, LTP and contextual fear memory were rescued by application of L-655,708, a negative allosteric modulator of the extrasynaptic GABAAR α5 subunit. These results suggest that haploinsufficiency of Clptm1 contributes to cognitive deficits through altered synaptic transmission and plasticity by elevation of inhibitory neurotransmission, with tonic inhibition playing a major role. [ABSTRACT FROM AUTHOR]

Aim Methods Results Conclusion Speech difficulties are common in children with cleft palate, but research on foreign‐born children is limited. This study aimed to compare speech outcomes, surgery and speech intervention in 5‐year‐old foreign‐born and Swedish‐born children with cleft palate with or without cleft lip.This retrospective study analysed data from the Swedish cleft lip and palate registry for children born between 2009 and 2016 using Pearson's Chi‐squared test and binary logistic regression.Among 160 foreign‐born (106 boys, 54 girls) and 847 Swedish‐born (479 boys, 368 girls) 5‐year‐olds, foreign‐born children had significantly lower rates of sufficient velopharyngeal competence (77% vs. 86%), age‐appropriate consonant production (28% vs. 60%), and speech without non‐oral speech errors (70% vs. 86%). Differences remained after adjustment for cleft type, gender and additional diagnosed conditions. After further adjustments for age at completed primary palatal surgery, differences in age‐appropriate consonant production and speech without non‐oral speech errors remained significant. Foreign‐born children underwent completed primary palatal surgery at older ages and received more secondary palatal surgery and speech intervention than Swedish‐born peers.Foreign‐born children showed poorer speech outcomes than Swedish‐born peers, despite more secondary palatal surgery and speech intervention. Age at completed primary palatal surgery could partly explain these differences. [ABSTRACT FROM AUTHOR]

To use the bibliographic data of publications regarding the association between orofacial cleft (OC) and cancer to examine the implications of publication growth, co-citation, co-words, and authorship networks using bibliometric indicators and network visualization. Bibliometric study analyzed documents related to the association between OC and cancer. Data were obtained in October 2023 from the Scopus, Pubmed, Web of Science. The search strategy was developed, and data obtained were imported into R ("bibliometrix") for analysis. Results: 70 documents were found from 1977 to 2023. Most were journal articles (90.0%) designed as case–control studies (42.8%). American Journal of Epidemiology was the most relevant source. The most cited document was Frebourg T, et al. (2006). Vieira A, Martelli-Junior were the most cited author. Most authors were affiliated to Brazil (University of Montes Claros) and United States (University of Pittsburgh). Research in this thematic has included children and adults of both sexes. Research trend points to gastric cancer, leukemia and breast cancer as the most investigated cancers in association to OC. This bibliometric analysis helps fill research gaps regarding the knowledge of the relationship between OC and cancer, providing some clues for selecting future research in this topic. Analysis of publications regarding the association between orofacial cleft and cancer highlighted: The majority of publications as journal articles (90.0%) designed as case–control studies (42.8%). American Journal of Epidemiology was the most relevant source. The most global cited documents were Frebourg T, et al. (2006). Vieira A, Martelli-Junior was the top author of the published literature and the most local cited author. Most of the authors were mainly affiliated to Universities from Brazil (University of Montes Claros) and United States (University of Pittsburgh). Motor themes of study include genetic association studies, investigating single nucleotides polymorphisms shared by OC and cancer types. Research in this thematic has included children and adults of both sexes. Research trend points to gastric cancer, leukemia and breast cancer as the most investigated cancers in association to OC. [ABSTRACT FROM AUTHOR]