Nonsyndromic Cleft Palate Research Articles

Prevalence of upper cervical vertebral anomalies in children with non-syndromic cleft lip and/or palate in comparison with children without cleft in Iranian population

BackgroundTo evaluate the prevalence of upper cervical vertebral anomalies (CVA) in Iranian children with cleft lip and/or palate (CL/P) and compare it with children without a cleft.MethodsA case-control study on lateral cephalograms from Orthodontics Research Center, Shiraz University of Medical Sciences, Shiraz, Iran of 92 subjects (41 females and 51 males) with non-syndromic CL/P with a mean age of 13.54 ± 4.63 years, and 184 age- and sex-matched individuals (82 females and 102 males) with no craniofacial anomalies or skeletal malocclusion as the control group. Upper cervical vertebrae (C1-C3) were examined regarding the following CVA: (1) posterior arch deficiencies: spina bifida and dehiscence; (2) Fusion Anomalies (FAs): fusion and occipitalization; (3) accessory ossicles. Vertebral artery canal morphology was also evaluated.ResultsThe prevalence of CVA was significantly higher in the cleft group (62%) than in the control group (25%) (P < 0.001). FAs, fusion, accessory ossicle, and deviation of artery canal type 2 were the anomalies with significantly higher prevalence in the cleft group compared to the noncleft group (all P < 0.05). 11 individuals (11.9%) of the cleft group and five (2.7%) of the control group had more than one CVA. When considering the subgroups of the CL/P, the prevalence of CVA was significantly higher in almost all the CL/P subgroups compared to the control group (all P < 0.05).ConclusionsUpper CVA, especially fusion anomalies, were significantly more prevalent in children with non-syndromic CL/P compared to the children without cleft in an Iranian population. A female predilection for CVA was also noted in both the general population and the cleft group.

Targeted Resequencing Identifies Novel MAFB Variants Associated With Nonsyndromic Cleft Lip With or Without Cleft Palate.

ObjectivesNonsyndromic cleft lip with or without palate (NSCL/P) is one of the most prevalent congenital orofacial defects. It arises from a combination of genetic and environmental factors. This study aims to identify new risk loci around the musculoaponeurotic fibrosarcoma oncogene family, protein B (MAFB) gene in NSCL/P patients from the Western Han Chinese population.DesignA targeted region sequencing approach was employed to examine the MAFB gene in 159 NSCL/P cases. We conducted both single-variant association and gene-based burden analyses.SettingThe study was conducted in a stomatological hospital.Patients, participantsOne hundred and fifty-nine NSCL/P cases were analyzed.InterventionsBlood samples were collected.Main outcome measuresTo explore the association analysis between variants at MAFB and NSCL/P in Western Han Chinese population.ResultsWe identified a cluster of significant common variants near the 3' end of MAFB. Notably, rs6029223 showed significantly associated with NSCL/P (P = 3.82E-09, odds ration [OR] = 0.29, 95% confidence interval [CI]: 0.18-0.46), nonsyndromic cleft lip and palate (NSCLP) (P = 5.31E-08, OR = 0.25, 95% CI: 0.14-0.44) and nonsyndromic cleft lip only (NSCLO) (P = 1.3E-04, OR = 0.34, 95%CI: 0.19-0.62). In Addition, rs79836852 and rs200392238 were significantly associated with both NSCL/P and NSCLP.ConclusionsOur study revealed that single nucleotide polymorphisms near the 3' end of the MAFB gene are risk factors for NSCL/P and NSCLP in the Western Han Chinese population. Our findings reinforce the notion that MAFB is a susceptibility gene for NSCL/P.

A Large Multicenter Brazilian Case-Control Study Exploring Genetic Variations in Interferon Regulatory Factor 6 and the Risk of Nonsyndromic Cleft Lip With or Without Cleft Palate.

Nonsyndromic cleft lip with or without cleft palate (NSCL ± P) is strongly associated with both environmental and genetic risk factors, but its genetic underpinnings remain partially known. While variants in interferon regulatory factor 6 (IRF6) are linked to NSCL ± P risk in populations from Asia and Europe, studies on the highly admixed Brazilian population are scarce and have produced ambiguous results. This study aimed to investigate the contribution of IRF6 variants to the risk of NSCL ± P. Five tag-single nucleotide polymorphisms (rs599021, rs2073485, rs2235375, rs7552506, and rs642961) were analyzed in a large multicenter cohort composed of 1006 patients with NSCL ± P and 942 healthy controls. Statistical analyses involved multiple logistic regression tests consideration the tri-hybrid genetic origin of the Brazilian population, under a Bonferroni p value correcting for multiple comparisons. The A allele (OR: 1.43, 95% CI: 1.22-1.67, p < 0.0001) and AA genotype (OR: 2.04, 95% CI: 1.46-2.86, p < 0.0001) frequencies of rs642961 were significantly associated with NSCL ± P risk. Stratified analyses indicated that the variant is associated with susceptibility to both nonsyndromic cleft lip only (NSCLO) and nonsyndromic cleft lip and palate (NSCLP). However, the association with NSCLO was primarily observed in patients with high African ancestry, whereas the association with NSCLP was predominantly seen in patients with high European ancestry. No significant associations were found for the other investigated variants. Our results support the notion that the IRF6 rs642961 variant represents a marker of susceptibility to NSCL ± P in the Brazilian population, and that genetic ancestry composition plays a central role in the association with the cleft type.

Cephalometric Assessment of Sella Turcica Morphology and Dimensions in Patients with Non-Syndromic Cleft Lip and Palate: A Systematic Review and Meta-Analysis.

ObjectiveThis review aimed to analyze the size and morphology of the Sella Turcica (ST) in 2-dimensional cephalometric radiographs of individuals with non-syndromic cleft lip and or palate (CLP/CP) compared with non-cleft individuals.MethodsA comprehensive search of multiple electronic databases was conducted without language and date restrictions until October 12, 2024. Studies were included that assessed ST dimensions in lateral cephalograms and compared the patients with non-syndromic CLP or CP and non-cleft individuals. The studies' quality assessment was assessed using the JBI case-control guidelines and the Anatomical Quality Assurance checklist. Quantitative analysis was performed using the random-effects meta-analysis model. The GRADE approach was employed to evaluate the certainty of evidence.ResultsOut of 1087 studies, 10 met the inclusion criteria, and 6 were included in the quantitative analysis. Of all, 7 studies were rated moderate, 2 low, and 1 high quality. Unilateral CLP (UCLP) cohort showed statistically significant smaller dimensions for length (MD = -1.22; 95% CI: -2.21, -0.24), depth (MD = -0.65; 95% CI: -1.41, -0.11), and area (MD = -4.75; 95% CI: -7.22, -2.28) and the odds of ST bridging were more than twice compared to non-cleft control. No significant differences were observed in ST dimensions in bilateral CLP versus non-Cleft and UCLP.ConclusionThe evidence of differences in ST dimensions between non-cleft individuals and patients with UCLP is weak, owing to a greater risk of bias resulting from inadequate reporting of design variability, participant demographics, and measurement methods. Future studies with better methodology and larger sample sizes are needed.

A Review and Meta-Analysis on Altered Brain Structure in Patients Born with Non-Syndromic Cleft Lip and/or Palate.

ObjectiveTo determine if there is evidence for a relationship between the presence of a non-syndromic cleft lip and/or palate (NSCL/P) and altered brain structure in cleft affected individuals.DesignElectronic database (MEDLINE; EMBASE; Cochrane library) and manual searches were performed and were limited to English language texts published between 1st of January 1969 until 1st of June 2024. Data extraction and risk of bias assessment were carried out independently by two reviewers. A meta-analysis on 9 publications was performed using a random effects model.Main outcome measureComparison of brain structure between patients born with cleft lip and/or palate and unaffected healthy individuals.ResultsThe review identified 11 studies, of which 9 were included in the meta-analysis. The latter comprised 398 individuals with NSCL/P compared to 458 unaffected controls. There was statistical evidence to suggest a reduction in the overall intracranial volume and the total cerebellar volume in the NSCL/P cases compared to the controls (Hedges' g and 95% confidence intervals: -0.36 [95% CI: -0.65, -0.08] and -0.69 [95% CI: -0.84, -0.53], respectively). For the cortical gray matter, straight gyrus, and total cerebral volume, there was no statistical evidence to suggest a difference between the NSCL/P cases and the controls.ConclusionIn individuals with NSCL/P, the overall intracranial volume and the total cerebellar volume were both smaller than in unaffected controls.

Prevalence and Risk Factors of Obstructive Sleep Apnea in Patients With Complete Cleft Lip and Palate in the Northeastern Thailand.

ObjectiveTo evaluate the prevalence of obstructive sleep apnea (OSA) in patients with nonsyndromic unilateral complete cleft lip and palate (UCLP) and to study the associations between contributing factors and OSA.DesignProspective study adopting pediatric sleep questionnaire (PSQ) and portable polysomnography (PSG) in patients at high risk of OSA.SettingTawanchai Cleft Center, Khon Kaen University.PatientsPatients aged 7 to 15 years with nonsyndromic UCLP.Main Outcome MeasureA PSQ score ≥8 was positive for OSA risk. Potential contributing factors included sex, weight for height, Friedman tongue position, tonsil grading, dental occlusion, transverse discrepancy, daytime sleepiness, quality of life, and pharyngeal airway.ResultsThe study population comprised 102 participants (mean age, 9.67 years). Nine (8.8%) patients had a high risk of OSA, based on PSQ results. On PSG, 7 (77.8%) patients had mild OSA and 2 (22.2%) patients had moderate OSA. The mean respiratory event index (REI) was 3.47 ± 1.65 events/h. OSA severity and contributing factors were not correlated. The minimal cross-sectional area and total airway volume showed a moderate negative correlation, with significant alterations observed at the oropharyngeal level. Reduced dimensions correlated with increased OSA severity at the oropharyngeal level, as demonstrated by a moderate negative correlation with REI.ConclusionsOSA risk among patients with UCLP was 8.8%. While this rate may seem moderate relative to that of the general population, it indicates that patients with UCLP have an inherent predisposition to OSA. This highlights the importance of comprehensive management, including PSQ screening, evidence-based counseling, and appropriate specialist referrals.

Effectiveness of nasoalveolar molding treatment for babies with nonsyndromic complete unilateral cleft lip, alveolus, and palate: A systematic review and meta-analysis.

Effectiveness of nasoalveolar molding treatment for babies with nonsyndromic complete unilateral cleft lip, alveolus, and palate: A systematic review and meta-analysis.

Distinguishing syndromic and nonsyndromic cleft palate through analysis of protein-altering de novo variants in 816 trios.

De novo variants (DNs) are sporadically occurring variants that most commonly arise in the germline and are present in offspring but absent in both parents. As they are not under selective pressure, they may be enriched for disease-causing alleles and have been implicated in multiple rare genetic disorders. Cleft palate (CP) is a common craniofacial congenital anomaly occurring in ∼1 in 1700 live births. Genome-wide association studies for CP have found fewer than a dozen loci, while exome and targeted sequencing studies in family-based and case-control cohorts often lack statistical power to conclusively identify causal genes. Based on previous work by our group and others, deciphering the genetic architecture of CP and gene discovery efforts are complicated by the heterogeneous nature of the disorder. We aggregated sequence data for 816 case-parent trios with CP, representing all subtypes of CP and roughly evenly split between isolated and syndromic presentations. We hypothesized there would be a burden of DNs in CP probands and tested this hypothesis in the full cohort and various phenotypic subgroupings. We identified global enrichment of protein-altering DNs (1.36, p=2.39×10 -22 ), and exome-wide significant (p<1.3×10 -6 ) gene-specific enrichment for SATB2 , MEIS2 , COL2A1 , ZC4H2 , EFTUD2 , KAT6B , and ANKRD11. We found a statistically significant higher enrichment of loss-of-function and missense DNs in syndromic (1.49, p=2.84×10 -19 ) versus nonsyndromic probands (1.25, p=4.01×10 -7 ) but no differences between CP subtypes. We also evaluated biological differences, identifying distinct enrichments across two single cell RNA sequencing datasets: mouse palate at the time of palate fusion and human embryos at post-conceptional weeks 3-5. Altogether, we show DNs are a contributor to CP risk, and that combined analysis can enhance our ability to find genetic associations that would otherwise be undetected.

Association of MSX1 gene polymorphisms and maxillary lateral incisor agenesis in Non-syndromic cleft lip and/or palate individuals.

Association of MSX1 gene polymorphisms and maxillary lateral incisor agenesis in Non-syndromic cleft lip and/or palate individuals.

Prediction of Palatoplasty Timing for Infants With Cleft Lip and Palate Using Machine Learning Algorithm.

This study aims to predict the timing of palatoplasty in infants with cleft lip and palate (CLP) using a machine learning (ML) algorithm. The study included 111 patients with nonsyndromic CLP. Patient subgroups were classified based on birth weight (criterion: -1 SD, low birth weight versus normal birth weight) and cheiloplasty timing (criterion: 5mo, early cheiloplasty versus late cheiloplasty). Growth trajectories at T2 were compared using T0-weight z-scores between the birth weight subgroups. Changes in the z-scores of weight and height from T1 to T2 were compared between the cheiloplasty timing subgroups. After training the tree-based ML models using cleft type, age, height, and weight at T0, T1, and T2, the palatoplasty timing was predicted with cleft type, weight at T0, and age, height, and weight at T1. The low-birth weight subgroup showed significant catch-up growth during T0-T1 and T0-T2 (all P<0.0001), resulting in no significant difference from the normal birth weight subgroup at T2. Compared with the late cheiloplasty subgroup, the early cheiloplasty subgroup underwent palatoplasty earlier (13.1 versus 14.3mo; P<0.0001) and showed higher growth rates of weight and height and a greater increase in weight z-scores from T1 to T2 (all P<0.001). The CatBoost algorithm, with a root mean square error of 1.6 months, accurately predicted the palatoplasty timing (mean: actual, 12.8±1.8mo versus prediction, 12.8±1.0mo). Use of ML-assisted prediction method may help clinicians decide the timing of personalized palatoplasty in infants with CLP.

Functional variant at 19q13.3 confers nonsyndromic cleft palate susceptibility by regulating HIF3A.

This study investigates the genetic factors associated with nonsyndromic cleft palate only (NSCPO) through a two-stage genome-wide association study (GWAS) and functional experiments. A total of 311 NSCPO cases and 1,136 controls were included, and we identified rs3826795 in HIF3A significantly associated with NSCPO risk (p= 4.27E-09). Functional assays showed that the A allele of rs3826795 reduced VEZF1 binding to the HIF3A promoter, thereby enhancing HIF3A expression. Additionally, we demonstrated that HIF3A significantly influenced cellular processes, including apoptosis, proliferation, and migration, in both invitro and invivo models. Pathway enrichment analysis indicated that HIF3A suppressed the GABAergic synapse pathway, which plays a crucial role in embryonic development. These findings provide valuable insights into the genetic mechanisms underlying NSCPO susceptibility and suggest potential targets for future therapeutic interventions.

Association analysis between forkhead box E1 gene and non-syndromic cleft lip with or without cleft palate in Han Chinese population.

This study aims to explore the association between single nucleotide polymorphisms (SNPs) loci near the haplotype region hg19 chr9:100560865-100660865 of the forkhead box E1 (FOXE1) gene and the occurrence of non-syndromic cleft lip with or without cleft palate (NSCL/P) in western Han Chinese population. In the first stage, our study recruited 159 NSCL/P patients and performed targeted region sequencing to screen SNPs loci near the haplotype region of the FOXE1 gene associated with NSCL/P. In the second stage, we selected 21 common SNPs and re-enrolled 1 000 non-syndromic cleft lip only (NSCLO) patients, 1 000 non-syndromic cleft palate only (NSCPO) patients, and 1 000 normal controls to verify the association. PLINK software was used to perform Hardy-Weinberg equilibrium (HWE) test. Association analysis for common variants, gene burden analysis for rare mutations, and function prediction of SNPs with non-synonymous mutations were performed using Mutation Taster and other software programs. In the first stage, 126 variants, including 76 single nucleotide variants and 50 insertion-deletions were identified. All the included SNPs confirmed to HWE, and the results of gene burden analysis and prediction of functional harmfulness for rare variants were not statistically significant. Association analysis showed that rs13292899 of the FOXE1 gene was significantly associated with NSCL/P (P=1.85E-27) and was also correlated with NSCLO (P=6.41E-23) and non-syndromic cleft lip with cleft palate (NSCLP) (P=2.36E-15) subtypes. In the validation phase, rs79268293 (P=0.013, P=0.022), rs10983951 (P=0.009 2, P=0.007 6), rs117227387 (P=0.009 2, P=0.007 6), rs3758250 (P=0.009 2, P=0.007 6), and rs116899397 (P=0.009 2, P=0.007 6) were significantly associated with NSCLO and NSCPO; rs13292899 (P=0.008 5), rs74606599 (P=0.008 3), rs143226042 (P=0.008 3), and rs117236550 (P=0.01) were associated with the occurrence of NSCLO; and rs12343182 (P=0.008 7), rs10119760 (P=0.012), rs10113907 (P=0.012), and rs13299924 (P=0.012) were associated with the occurrence of NSCPO. This study found a new susceptible SNP rs13292899 of the FOXE1 gene that is closely associated with NSCL/P and NSCLO subtype and 13 other SNPs associated with NSCLO or NSCPO.

Robin Sequence and Isolated Cleft Palate are Associated With a High Prevalence of Obstructive Sleep Apnea in School-Aged Children.

To analyze the prevalence of obstructive sleep apnea (OSA) in children aged 6 to 12 years with nonsyndromic Robin sequence (NSRS) and in those with nonsyndromic cleft palate (NSCP). All patients presented complete cleft palate (Veau II). Cross-sectional study. Tertiary public hospital. A total of 146 children divided into 2 groups: (1) NSRS (n = 69), (2) NSCP (n = 77). (1) Anthropometric assessment, dentoskeletal, and facial analysis. (2) Clinical interview with "Sleep Disturbance Scale for Children" and "Congestion Quantifier Five-Item" (CQ5); and (3) 48 patients: Type IV polysomnography. Frequency of OSA in children with NSRS and NSCP, assessed by Type IV polysomnography. Positive scores for OSA were found in 59.42% of children with NSRS and 46.75% of those with NSCP (P > .05). Excessive daytime sleepiness was observed in 23.19% of the NSRS group and 9.01% of the NSCP group (P > .05). Positive scores for nasal obstruction were noted in 14.49% with NSRS and 20.78% of those with NSCP (P > .05). In polysomnography IV subgroups, an Oxygen Desaturation Index compatible with mild to moderate OSA was observed in 89.65% of the NSRS group and 78,94% of the NSCP group (P > .05). Also, facial and pharyngeal alterations, such as Angle Class II malocclusion, Mallampati classifications III and IV, and deep crossbite, were associated with OSA. Both children with NSRS and NSCP have a high frequency of mild to moderate OSA, highlighting the need for systematic evaluation of the presence of sleep-disordered breathing in this population.

Polymorphisms and dental age in non-syndromic cleft lip and palate: a cross-sectional study.

Children with non-syndromic cleft lip with or without palate (CL ± P) may present alterations in dental development. The purpose of this cross-sectional study was to compare the dental age (DA) between children with and without CL ± P, and whether single nucleotide polymorphisms (SNPs) in genes encoding growth factors are associated with DA variations. Children aged between 5 and 14 years with and without CL ± P were recruited to participate in this study. DA was evaluated by calibrated examiners (kappa > 0.80) using the method proposed by Demirjian et al. (1973). Genomic DNA was extracted from buccal cells, and SNPs in Epidermal Growth Factor (EGF) - rs4444903 and rs2237051, Epidermal Growth Factor Receptor (EGFR) - rs2227983 -, Transforming Growth Factor Beta 1 (TGFB1) - rs1800470 and rs4803455 -, and Transforming Growth Factor Beta Receptor 2 (TGFBR2) - rs3087465 - were genotyped by real-time polymerase chain reactions using the TaqMan assay. The Student T-test was used to compare the variations in DA between the phenotypes "with CL ± P" and "without CL ± P", and the ANOVA two-way test was performed to compare the variations in DA among the genotypes (α = 0.05). A post-hoc analysis was performed using Bonferroni correction. Two hundred and nine (n = 209) children (100 with CL ± P and 109 without CL ± P) with a mean chronological age of 8.66 years - standard deviation (SD) = 1.92 - were included. The group with CL ± P demonstrated a significantly delayed DA (mean=-0.23; SD = 0.71) compared to the group without CL ± P (mean=-0.01; SD = 0.88) (p = 0.049). Genotype distributions were in Hardy-Weinberg equilibrium. The SNP rs4803455 in TGFB1 was significantly associated with DA variations in children without CL ± P (p < 0.01). In the group with CL ± P, no significant differences in DA were observed among the genotypes. Children with CL ± P presented delayed DA compared with children without CL ± P. The SNP rs4803455 in TGFB1 is associated with variations in DA in children without CL ± P.

Alveolar Bone Quality in Individuals With Cleft Lip and Palate With Missing Lateral Incisors: Orthodontic Space Closure Versus Space Opening.

The purpose of this study was to quantitatively assess the alveolar bone support of teeth adjacent to the cleft site in individuals with nonsyndromic cleft lip and palate (CLP) who have undergone either orthodontic space closure or space opening for missing lateral incisors. A cross-sectional retrospective study. University orthodontic clinic serving individuals with CLP. Twenty-eight individuals with nonsyndromic CLP who were missing lateral incisors divided into 2 groups: space closure (21 subjects) and space opening (7 subjects). Orthodontic space closure or space opening for replacement of missing lateral incisors in individuals with nonsyndromic CLP. Buccal and palatal alveolar bone thickness were measured at 5 mm and 10 mm above the cementoenamel junction (CEJ) for cleft-adjacent central incisors and canines. Additionally, buccal, lingual, and proximal alveolar bony coverage ratio on cleft-side central incisors and canines were recorded. No significant differences were observed in alveolar bone thickness and bony coverage between the space closure group and the space opening group, except for the buccal thickness 5 mm above the CEJ, which was thinner in the space closure group. The overall alveolar bone support in the grafted alveolus in both the space closure and space opening groups were comparable.

Identifying New Susceptibility Genes of Non-Syndromic Orofacial Cleft Based on Syndromes Accompanied With Craniosynostosis.

Orofacial cleft (OC) can be classified into syndromic orofacial cleft (SOC) and non-syndromic orofacial cleft (NSOC), depending on whether there are other congenital deformities. Craniosynostosis, the premature closure of cranial sutures, is a common phenotype of SOC resulting in abnormal ossification of skull and brain development disorders. Its correlation with OC offers a promising approach to identify susceptibility genes for NSOC by examining causative genes of SOCs with craniosynostosis. This study included 2556 patients with NSOC and 2255 normal controls from western Han Chinese with their genomic DNA samples. We selected 31 causative genes of 34 syndromes with both craniosynostosis and OC as candidate genes and performed quality control. Allelic and genotypic association analyses and haplotype analysis were performed to identify statistically significant single nucleotide polymorphisms (SNPs). In allelic association analysis performed with 1265 qualified SNPs in 20 genes, only rs2239936, located in MYH3 gene, was statistically associated with non-syndromic cleft lip only (NSCLO) (P = 1.70×10-07, OR = 1.33, 95%CI: 1.17-1.52) and non-syndromic cleft palate only (NSCPO) (P = 6.43×10-05, OR = 1.33, 95%CI: 1.16-1.52). The higher frequency of allele G in NSCPO suggesting that minor allele G at rs2239936 will result in an elevated risk of NSCPO. However, rs2239936 only exhibited a statistical association with NSCPO in genotypic association analysis (P = 8.06×10-06) and haplotype analysis (P = 1.43×10-05). This study identified that allele G at rs2239936 in MYH3 gene was significantly associated with NSCPO as a risk factor and MYH3 was a new susceptibility gene for NSCPO in western Han Chinese population.

Does Palatoplasty Technique Impact Resolution of Eustachian Tube Dysfunction?

To determine whether palatoplasty technique affects the resolution of eustachian tube dysfunction and postoperative speech outcomes in children with cleft palate (CP). Retrospective cohort. Multidisciplinary cleft and craniofacial clinic at a tertiary care center. Seventy-three patients with nonsyndromic CP who underwent palatoplasty between 2005 and 2019. Inclusion criteria included soft palate repair with Furlow technique or intravelar veloplasty (IVV) and Veau classifications I-III. Either Furlow or IVV repair was performed based on the surgeon's clinical judgment. All patients had bilateral ear tubes placed prior to or at the time of palate repair, with postpalatoplasty ear tubes placed at the ENT surgeon's discretion. Patients received routine follow-up care for over 5 years. Data were analyzed with Wilcoxon tests, χ2 tests, and negative binomial regression. Number of postpalatoplasty ear tubes placed, rates of velopharyngeal insufficiency, and speech surgery after palatoplasty in each group. Furlow repair patients required a similar number of postpalatoplasty ear tubes (P = .321) and underwent additional sets at similar rates compared to those who underwent IVV repair. Negative binomial regression found no covariates (age, race, Veau, repair type, speech surgery, fistula repair) that predicted additional ear tube requirements. Furlow repair patients required postpalatoplasty ear tubes at a similar rate compared to IVV repair patients. While the palatoplasty techniques differ, patients may still need the same amount of time for resolution of their eustachian tube dysfunction.

Tooth Anomalies in Patients With Nonsyndromic Orofacial Cleft: A Systematic Review and Meta-Analysis.

To evaluate the frequency of tooth anomalies (TA) in the deciduous and permanent dentition of patients with nonsyndromic orofacial clefts (NSOC), both inside and outside the cleft area. The following databases were searched for the relevant literature: Cochrane, OVID, SciELO, Embase, Livivo, PubMed, Scopus, and Web of Science. The risk of bias was analyzed using the Joanna Briggs Institute. Fixed and random-effects meta-analysis was performed comparing the presence and absence of NSOC subtypes. The certainty of evidence was evaluated using the GRADE approach. Out of 1939 articles identified, after applying the inclusion and exclusion criteria, a total of 75 articles were included (46 cohort and 29 case-control), including 27,703 patients (16,450 with NSOC and 11,253 healthy controls) from 34 countries. The meta-analyses revealed higher odds for tooth agenesis (ORNSOC: 3.72; p = 0.001) and macrodontia (ORNSOC: 8.04; p = 0.04) across the different cleft subtypes outside the cleft area compared with the control group in the permanent dentition, whereas the frequency of root dilaceration was significantly lower in nonsyndromic cleft lip only (NSCLO) (ORNSCLO: 0.38; p < 0.0001) and in nonsyndromic cleft lip and palate (NSCLP) (ORNSCLP: 0.44; 95% p < 0.0001). The results also demonstrated a higher risk of tooth agenesis (ORNSOC: 16.49; p < 0.0001), microdontia (ORNSOC: 17.14; p < 0.0001), macrodontia (ORNSOC: 10.41; p = 0.02), supernumerary tooth (ORNSOC: 10.03; p < 0.0001), and enamel hypoplasia (ORNSOC: 5.62; p < 0.0001) in the permanent dentition inside the cleft area of patients with NSOC. However, for the deciduous dentition, outside the cleft area, microdontia was the only TA significantly more frequent in patients with NSOC (ORNSOC: 6.24; p = 0.006) and nonsyndromic cleft palate only (NSCPO) (ORNSCPO: 8.45; p = 0.02) compared with the control group. TA associations varied across populations. In Europe, no significant associations were found for NSOC, while in Asia, strong associations were observed for NSCLP and NSCL ± P (ORNSCLP and NSCL±P: 139.19; p < 0.0001). In South America, significant associations were identified for NSCLP (ORNSCLP: 2.16; p < 0.0001), NSCL ± P (ORNSCL±P: 2.48; p < 0.0001), and NSOC (ORNSOC: 2.72; p < 0.0001). In North America, tooth agenesis was more frequent in NSCL ± P (ORNSCL±P: 4.75; p < 0.0001), though no significant associations were found for NSCLP or NSOC. In the cleft area, significant associations were observed in European populations for NSOC, including increased frequencies of tooth agenesis (ORNSOC: 19.57; p = 0.003) and supernumerary teeth (ORNSOC: 9.77; p < 0.0001). Asian populations showed similar patterns (ORNSOC: 19.23; p = 0.002), while no significant associations were noted in South America due to limited data. Root dilaceration remained less frequent in NSCLO (ORNSCLO: 0.38; p < 0.0001) and NSCLP (ORNSCLP: 0.44; p < 0.0001), with no associations identified for microdontia, taurodontism, supernumerary tooth, impacted tooth, or transposition. The results confirm a higher frequency of TA in the permanent dentition of patients with NSOC compared to controls, regardless of cleft subtype. However, significant differences were observed depending on whether the TA occurred inside or outside the cleft area. Although only a limited number of studies were included, microdontia was the only TA significantly more prevalent in the cleft area of patients with NSOC in the deciduous dentition. Variations in the frequency of TA across populations highlight the complex interplay of genetic, environmental, and methodological factors influencing these associations. Despite these findings, the quality of the existing evidence is moderate, with limitations stemming from small sample sizes, methodological variations, and study heterogeneity. These results emphasize the importance of tailored dental management and early intervention strategies for individuals with different cleft subtypes to effectively address and mitigate the impact of these tooth anomalies on oral health and development.

Obstructive Sleep Apnea Following Secondary Velopharyngeal Insufficiency in Children with Non-Syndromic Cleft Palate: A Systematic Review

Obstructive Sleep Apnea Following Secondary Velopharyngeal Insufficiency in Children with Non-Syndromic Cleft Palate: A Systematic Review

Correlation between oral health knowledge and awareness and periodontal status in non-syndromic cleft lip and palate patients undergoing orthodontic treatment: a cross-sectional study

Introduction: Children and adolescents with non-syndromic cleft lip and palate (CLP) experience a higher prevalence of caries and periodontal disease compared to those without CLP. This increased prevalence is often attributed to greater plaque accumulation, which can lead to gingival inflammation and periodontal disease. This study aims to analyze the correlation between oral health knowledge and awareness and the periodontal health status of orthodontic patients with cleft lip and palate (CLP).Method: This study used a cross-sectional approach. The study population consisted of orthodontic patients with non-syndromic CLP who met the inclusion criteria. The research was conducted at the UNPAD Dental Teaching Hospital (RSGM Unpad) from March to May 2024. Twenty-seven participants with non-syndromic CLP were included in the study. A non-probability, purposive sampling technique was employed. oral health knowledge and awareness were assessed using questionnaires, while periodontal health examinations were evaluated using the Community Periodontal Index (CPI). Correlation analyses were performed using Kendall's Concordance Correlation (W). Additionally, Spearman Rank Correlation Analysis was conducted to identify the dominant variables. Results: The analysis revealed a Kendall's Concordance Correlation (W) of 0.544, which is statistically significant (p &lt; 0.0001), indicating a 54.4% relationship between the variables. The correlation between knowledge and periodontal health status in orthodontic patients with non-syndromic CLP was 11.16%, with a p-value of 0.0443 (p &lt; 0.05) and an r-value of 0.33. The correlation between awareness of oral health and periodontal health status in these patients was 19.85%, with a p-value of 0.0099 (p&lt;0.05) and an r-value of 0.45. Conclusion: There is a weak correlation between knowledge and awareness of oral health and the periodontal health status in non-syndromic CLP patients undergoing fixed orthodontic treatment.