Cleft Type Research Articles

Underreporting, Prevalence, and Epidemiological Trends of Orofacial Clefts in the Brazilian Amazon Region.

Cleft lip and palate are the most common congenital anomalies of the cranial segment worldwide. Particularly in low-income and middle-income countries, these conditions are associated with increased morbidity and mortality, socioeconomic challenges, and considerable psychological and social integration difficulties for affected individuals. This study aims to evaluate the epidemiological profile of patients with these conditions treated at a newly established specialized center in the Brazilian Amazonian Region. Data were extracted from medical records at a Reference Service for Clefts and Craniofacial Anomalies in the north of Brazil, covering the period from 2016 to 2020. These were compared with data from the official epidemiological health portal of the Brazilian Ministry of Health. The study recorded a total of 852 patients, with males comprising 54.4%. The predominant type of cleft was the transforaminal cleft, which accounted for 69.4% of cases, followed by postforamen clefts at 17.3%. The left side was more frequently affected in 63% of the cases. The primary surgical intervention performed was cheiloplasty, representing 39.5% of all procedures. Notably, 52% of the patients were from the interior regions of the state. The 2019 DataSUS data indicated a prevalence of cleft lip and palate in the State of Pará of 4.26 per 10,000 live births. However, data from this single specialized hospital showed a higher prevalence of 7.58 per 10,000 live births. These results may reflect underreporting of the number of cases reported in national official data sets.

Patient-Reported Esthetic Outcomes Following Secondary Rhinoplasty in Adult Patients with a Cleft lip and Palate.

Secondary rhinoplasty is a frequently requested revision procedure in patients with a history of cleft lip and palate (CLP). The satisfaction of patients with the esthetic outcome of the procedure has been infrequently studied. Adult patients with CLP who had undergone a secondary rhinoplasty between 2009 and 2019 were identified in a cross-sectional cohort that had filled the FACE-Q rhinoplasty module. The effect of the cleft type and the number and timing of the nasal revision surgeries were evaluated. Forty participants were included, median age of 26 years (range 18-59 years). Nine (23%) participants had a history of bilateral CLP. The median time form the last nasal operation to the study was 4.5 years (range 16 days to 10 years). The participants reported being satisfied with the appearance of the nose (median score 52, interquartile range [IQR] 44-60) but the satisfaction with the appearance of the nostrils (median score 44, IQR 30-64) was below the normative value for the scale. Patients with a history of bilateral CLP reported worse satisfaction across all the aspects of health-related quality of life assessed (P < .05 for all). No association was observed between the age at the first open rhinoplasty and satisfaction with the appearance of the nose or the nostrils. Following secondary rhinoplasty, adults with CLP are, in general, satisfied with their nose but less so with nostrils. Compared to unilateral CLP, bilateral CLP is associated with a worse health-related quality of life.

Prenatal growth patterns of the upper jaw complex with implications for laryngeal echolocation in bats.

Craniofacial morphology is extremely diversified within bat phylogeny, however growth and development of the palate in bats remains unstudied. The formation of both midline and bilateral orofacial clefts in laryngeally echolocating bats, morphologically similar to the syndromic and non-syndromic cleft palate in humans, are not well understood. Developmental series of prenatal samples (n = 128) and adults (n = 10) of eight bat species (two pteropodids, four rhinolophoids, and two yangochiropterans), and two non-bat mammals (Mus musculus and Erinaceus amurensis), were CT-scanned and cranial bones forming the upper jaw complex were three-dimensionally visualised to assess whether differences in palate development can be observed across bat phylogeny. Volumetric data of bones composing the upper jaw complex were measured to quantify palate growth. The premaxilla is relatively reduced in bats compared to other mammals and its shape is heterogeneous depending on the presence and type of orofacial cleft across bat phylogeny. The palatine process of premaxillary bones is lacking in pteropodids and yangochiropterans, whereas the premaxilla is a mobile structure which is only in contact caudally with the maxilla by a fibrous membrane or suture in rhinolophoids. In all bats, maxillary bones progressively extend caudally and palatine bones, in some cases split into three branches, extend caudally so that they are completely fused to another one medially prior to the birth. Ossification of the vomer and fusion of the maxillary and palatine bones occur earlier in rhinolophoids than in pteropodids and yangochiropterans. The vomer ossifies bilaterally from two different ossification centres in yangochiropterans, which is uncommon in other bats and non-bat mammals. Analysis of ontogenetic allometric trajectories of the upper jaw complex revealed faster development of maxillary, vomer, and palatine bones in yangochiropterans compared to other bats, especially rhinolophoids. Ancestral state reconstruction revealed that yangochiropterans have a higher magnitude of change in ossification rate compared to other bats and E. amurensis a lower magnitude compared to M. musculus and bats. This study provides new evidence of heterochronic shifts in craniofacial development and growth across bat phylogeny that can improve understanding of the developmental differences characterising nasal and oral emission strategies.

Cleft Lip and Palate Classification Through Vision Transformers and Siamese Neural Networks

This study introduces a novel approach for the diagnosis of Cleft Lip and/or Palate (CL/P) by integrating Vision Transformers (ViTs) and Siamese Neural Networks. Our study is the first to employ this integration specifically for CL/P classification, leveraging the strengths of both models to handle complex, multimodal data and few-shot learning scenarios. Unlike previous studies that rely on single-modality data or traditional machine learning models, we uniquely fuse anatomical data from ultrasound images with functional data from speech spectrograms. This multimodal approach captures both structural and acoustic features critical for accurate CL/P classification. Employing Siamese Neural Networks enables effective learning from a small number of labeled examples, enhancing the model’s generalization capabilities in medical imaging contexts where data scarcity is a significant challenge. The models were tested on the UltraSuite CLEFT dataset, which includes ultrasound video sequences and synchronized speech data, across three cleft types: Bilateral, Unilateral, and Palate-only clefts. The two-stage model demonstrated superior performance in classification accuracy (82.76%), F1-score (80.00–86.00%), precision, and recall, particularly distinguishing Bilateral and Unilateral Cleft Lip and Palate with high efficacy. This research underscores the significant potential of advanced AI techniques in medical diagnostics, offering valuable insights into their application for improving clinical outcomes in patients with CL/P.

Perception, attitude, and opinion of parents of cleft lip and palate patients treated with nasoalveolar molding (NAM): A cross-sectional questionnaire study.

The main goal of the nasoalveolar molding (NAM) is to mitigate the initial severity of the cleft, facilitating the subsequent surgical procedures. Nevertheless, the use of the appliance entails high stress levels for families. The aim of this study was to assess the perceptions, attitudes, and opinions of parents whose cleft-affected children underwent treatment using the NAM technique. A cross-sectional questionnaire study was conducted on a cohort of relatives of infants born with cleft lip and palate who were treated with the NAM appliance. The parents completed a 59-item questionnaire that had been previously validated. The initial cohort consisted of 104 families. The level of satisfaction was high, and there was a direct correlation with early diagnosis. Satisfaction levels varied depending on the cleft type, with a decrease in cases of bilateral presentation. Satisfaction was influenced by the newborns' adaptation and the absence of complications. Parents who exhibited lower levels of satisfaction contemplated terminating the treatment. Parents expressed high satisfaction with NAM treatment, due to effective management and understanding. Bilateral clefts and delayed diagnosis can significantly impact satisfaction. These results emphasize the importance of personalized approaches to address challenges in NAM treatment, particularly in instances of bilateral clefts and delayed diagnoses.

Speech Outcome Audit for Cleft Lip and Palate Patient Population Correlated With Veau Cleft Type, Surgical Repair Technique, and Patient Age

Objective: The objective of this study is to determine speech outcome differences for specific palate repair techniques and correlate these data with patient age at the time of operation. Methods: A retrospective study was conducted on a cohort of consecutive nonsyndromic patients who underwent cleft lip and/or palate repair at the authors' hospital between 2010 and 2020. Only those patients who participated in at least 4 years of follow-up accompanied by audio-video recording were included. The patient cohort was divided into 2 age groups, between 1 year and 2 years of age versus older than 2 years of age. Standardized perceptual speech evaluations and nasopharyngoscopy were performed by the authors' team to detect the presence of velopharyngeal insufficiency. Results: A total of 481 patients were initially evaluated, and 323 of these patients met all inclusion criteria and were subsequently selected for this study. One hundred seventy-two patients (n = 172) underwent palatal surgery using the Bardach 2-flap palatoplasty technique, 47 patients (n = 47) underwent repair through the von Langenbeck palatoplasty technique, 78 patients (n = 78) underwent repair through Veau-Wardill-Killner palatoplasty technique, and 26 patients (n = 26) underwent repair through double-opposing Z-plasty palatoplasty. The rate of residual velopharyngeal insufficiency was statistically similar among all palatoplasty techniques. A comparison between these two age groups revealed that patients younger than 2 years of age showed higher speech outcome success. Conclusion: High rates of speech success are achieved for patients younger than 2 years of age. Speech outcomes success is maximized by proper technique selection in relation to cleft type.

Palatal relaxing incisions versus nasopharyngeal relaxing incisions in Palatoplasty.

Bone denudation after conventional relaxing incisions could be a critical factor in inhibiting maxillofacial growth. To address this, alternative relaxing incisions were designed. Thus, this study aimed to compare the effectiveness of palatal relaxing incisions versus nasopharyngeal relaxing incisions in enhancing postoperative outcomes. A retrospective cohort study was conducted, involving a total of 120 patients divided into three groups: 40 patients have received modified Furlow palatoplasty with nasopharyngeal relaxing incisions (M.F + N.P.I palatoplasty), and 40 patients who received modified Furlow palatoplasty with palatal relaxing incisions (M.F + P.R.I palatoplasty). The other 40 patients received original Furlow palatoplasty without relaxing incisions (F palatoplasty). Data collected included gender, cleft type, cleft width, age at repair, velopharyngeal function, presence of palatal fistula, and follow-up. The chi-square test compared frequencies of sex, cleft type, postoperative fistula, and velopharyngeal outcomes across groups. The Mann-Whitney and independent t-tests compared mean values, with statistical significance set at p < 0.05. The mean age at repair was similar across groups, with follow-up periods ranging from 5 to 11 years. No significant differences were found among the M.F + N.P.I and M.F + P.L.I groups regarding gender, cleft type, cleft width, and age at repair. However, the F group had a significantly narrowest cleft width compared to the other groups. Postoperative outcomes showed no significant differences in velopharyngeal function among the three groups, but the F group had a significantly higher rate of palatal fistula (32.5%) compared to the M.F + P.L.I (10%) and M.F + N.P.I (7.5%) groups. A comparison of the two modified Furlow techniques revealed no significant differences in velopharyngeal closure rates or the incidence of velopharyngeal insufficiency and persistent palatal fistula across different Veau classifications. While both incisions showed similar impacts on palatoplasty outcomes, palatal relaxing incisions may expose more bone and pose a higher risk of secondary healing issues. Therefore, nasopharyngeal relaxing incisions are recommended as an effective and potentially preferable technique in palatoplasty whenever feasible. The current study suggests that, whenever feasible, nasopharyngeal relaxing incisions are advised as an effective and potentially superior technique in palatoplasty.

Clinical Outcomes after Cleft Palate Repair at a Major Referral Hospital in Addis Ababa, Ethiopia

Background: The two most common complications after primary palatoplasty are oronasal fistula and velopharyngeal insufficiency (VPI). To the best of our knowledge, there have been no studies examining postpalatoplasty speech outcomes in Ethiopia. Methods: A hospital-based retrospective cross sectional study design was used. A total of 149 patient records were reviewed, and 103 patients fulfilled all the inclusion criteria. These 103 cleft lip and palate patients who had undergone cleft palate repair at Yekatit 12 Hospital Medical College and who had undergone long-term speech assessment with the speech therapy department from January 2019 to December 2021 were included in this study. The bivariate/multivariate logistic regression was used to compare between different groups of patients, with the level of significance set at a P value of less than 0.05. Results: Moderate and severe hypernasality was seen in 45.7% of patients. Age at time of palatal surgery has significant association with incidence of hypernasality (P = 0.01). The type of cleft, sex of the patient, and type of surgery had no significant association with incidence of hypernasality. Oronasal fistula was seen in 25% of patients, with Pittsburgh type VI being the most common (40%). Conclusions: There is a high rate of VPI and oronasal fistula in this cohort of patients who underwent palatoplasty at Yekatit 12 Hospital. One of the factors for higher complication rate was older age at time of surgery. Therefore, patient education and strengthening the cleft care with training are paramount to ensure better results.

Dynamic and static nasolabial muscle anatomy of unilateral cleft lip adult patients based on magnetic resonance imaging data.

This study aims to obtain a three-dimensional reconstruction model based on magnetic resonance imaging (MRI) data of patients with different degrees of unilateral cleft lip and analyze the anatomy and changes in multiple groups of nasolabial muscles under dynamic and static conditions. One normal person and four adult patients with unilateral cleft lip were included, and MRI was performed under static (upper and lower lips closed naturally) and dynamic (pout and grin) conditions. 3D Slicer software was used to reconstruct the model and draw the anatomic morphology of nasolabial muscles. The distance between the junction (where the muscle merges into the orbicularis oris) of the levator muscle, zygomaticminor muscle, and zygomatic major muscle to the median sagittal plane, the starting point to the junction point, the dynamic and static junction points, and the angle between the connection of dynamic and static junctions and the horizontal plane were measured under three kinds of movements, and the ratio was calculated. In all patients, under dynamic and static conditions, the distance from the muscle junction to the median sagittal plane, their ratios of the cleft side to the non-cleft side were all greater than 1. While the ratio of the distance from the starting point of the muscle to the junction point is less than 1. At static conditions, the two ratios of the same muscle increased gradiently with the severity of the cleft, and the ratio of the zygomatic minor muscle was prominent in the same patient. The ratio of the cleft side to the non-cleft side was greater than 1, and the value for comparison was the angle of the line from the static to the dynamic junction and the horizontal plane. The symmetry of the insertion site of the orbicularis oris and the linear distance of both sides of the muscle are related to muscle and cleft types. The angle of muscle contraction on the cleft side is greater than that on the non-cleft side.

The Kumagai Method: Feeding Techniques Using the Pigeon Baby Cleft Palate Bottle.

This study aimed to identify the P-bottle feeding techniques systematically organized by Ms. Kumagai, an expert in nursing care for children with a cleft lip and/or palate (CLP), which were developed as she gained expertise in feeding affected children. We recruited three nurses who had mastered the Kumagai method for feeding with a P-bottle. Through analysis of participants' voices and videos during interviews, we focused on aspects such as dealing with a closed mouth, inserting the nipple in cases of unilateral and bilateral CLP, dealing with the child's movements after insertion, and key considerations when squeezing the bottle. The interview analyses revealed numerous techniques used by nurses to manage the difficulties encountered while feeding children, ensuring successful provision of nourishment. Specifically, the nurses employed techniques such as placing the nipple along the midline of the child's tongue and varying the application of force on the nipple depending on the cleft type. The nurses reported that the objectives of these techniques were to prevent ulcer formation and encourage the use of the tongue, simulating original feeding movements. We explored feeding techniques and the management of associated challenges. Our results suggest that the "Kumagai Method" could be valuable in improving feeding practices.

Alterations of senescence-associated markers in patients with non-syndromic cleft lip and palate

Non-syndromic cleft lip with or without cleft palate (NSCL/P) is one of the most common craniofacial anomalies. Abnormal Alu methylation in DNA of the pregnant mother may influence the abnormal development of the child. This study aimed to examine Alu methylation and cellular senescence in NSCL/P patients and their mothers as well as the correlation with the severity of NSCL/P. A total of 39 patients with NSCL/P and 33 mothers were enrolled. Of these patients, 6 were cleft lip only (CLO), 9 were cleft palate only (CPO), and 24 were cleft lip and palate (CLP). Alu methylation and senescence markers were determined in the white blood cells of NSCL/P patients, their mothers, and in the lip and palatal tissues of patients at the time of cheiloplasty and palatoplasty. Total Alu methylation was not significantly different between groups. However, a decrease in Alu hypermethylation, increased partial Alu methylation, RAGE, and p16 expression were shown in CLP, the most severe cleft type. Alu methylation in tissues did not differ between groups. In mothers, an increase in Alu methylation was observed only in the CLP. Therefore, the pathogenesis of NSCL/P may be related to Alu methylation of the mother promoting loss of Alu methylation and subsequently senescence in the children.

Oral Staphylococcus Species and MRSA Strains in Patients with Orofacial Clefts Undergoing Surgical Rehabilitation Diagnosed by MALDI-TOF MS.

This study investigated the occurrence and dynamics of oral Staphylococcus species in patients with orofacial clefts undergoing surgical rehabilitation treatment. Patients (n = 59) were statistically stratified and analyzed (age, gender, types of orofacial clefts, surgical history, and types of previous surgical rehabilitation). Salivary samples were obtained between hospitalization and the return to the specialized medical center. Microbiological diagnosis was performed by classical methods, and MALDI-TOF MS. MRSA strains (SCCmec type II, III, and IV) were characterized by the Decision Tree method. A total of 33 (55.9%) patients showed oral staphylococcal colonization in one, two, or three sampling steps. A high prevalence has been reported for S. aureus (including HA-, MRSA and CA-MRSA), followed by S. saprophyticus, S. epidermidis, S. sciuri, S. haemolyticus, S. lentus, S. arlettae, and S. warneri. The dynamics of oral colonization throughout surgical treatment and medical follow-up may be influenced by (i) imbalances in staphylococcal maintenance, (ii) efficiency of surgical asepsis or break of the aseptic chain, (iii) staphylococcal neocolonization in newly rehabilitated anatomical oral sites, and (iv) total or partial maintenance of staphylococcal species. The highly frequent clinical periodicity in specialized medical and dental centers may contribute to the acquisition of MRSA in these patients.

Cleft lip and palate surgery in the Otolaryngology and Head-neck surgery department of a Medical College Hospital in Bangladesh

Background: Cleft lip and palate (CLP) are one of the most common developmental anomalies in orofacial region. CLP surgery lies in the anatomical domain of Ear Nose Throat and Head - Neck Surgery. In Bangladesh specialty of plastic surgery is still in developing stage and plastic surgeons are not easily available everywhere. ENT and Head-Neck surgeon had to take up the responsibility of some of cleft patients. This article is a retrospective analysis of a series of 200 cases of cheiloplasty and palatoplasty done in ENT and Head-Neck Surgery department of Khulna City Medical College Hospital, Bangladesh. Results were satisfactory in our, patient and parent's consideration. Objective: Our aim is to discuss the demographic profile, presentations, degree &amp; type of cleft and outcome of CLP surgery in Otolaryngology and Head-Neck Surgery department of a Medical College Hospital of Bangladesh. We will also discuss the embryology of face &amp; historical background of the cleft surgery. Materials and Methods: This is a retrospective analysis of CLP surgery for 200 cleft patients. We based on inclusion and exclusion criteria for selection of patients. Majority of cleft lip patients were operated following the Millard modified rotation advancement technique and isolated cleft palate patients were operated following Von Langenbeck bipedicle flap and Veau-Wardil-Kilner V-Y pushback technique in the ENT and Head-Neck Surgery Department of a Medical College Hospital. Patients were advised for periodic follow up. Results: Total of 200 patients of CLP were operated in our department out of which male was 92 (46%) and female was108 (54%)), male and female ratio being 1:1.08. The age range was 3 months to 40 years with a mean of 8.38 years. Pre alveolar group both male and female comprised of 134 (67%) and alveolar group 66 (33%) patients and that of isolated cleft palate 7 (10.60%) patients. Pre alveolar unilateral CLP was high. The results of surgery were satisfactory in our postoperative evaluation. Patients and parents were also satisfied with the outcome. Conclusion: Majority of CLP patients don’t suffer functional problem apart from facial disfigurement. Restoration of normal anatomy, facial appearance and function can be achieved by surgery in appropriate time and age. Few cases may be associated with feeding difficulties, dental and hearing problems. Some cases are associated with other congenital anomalies and syndromes. Those cases demand team approach of different specialty.

Periodontal health and oral hygiene of children with orofacial clefts in Eastern China.

To comparatively assess the periodontal condition and oral hygiene of children and adolescents at different ages presenting with different types of orofacial clefts (OFCs). A total of 1608 patients aged 6-18 years who had not previously undergone periodontal treatment were enrolled in this study. Participants were categorized into two age groups: 6-12 years (Group I) and 13-18 years (Group II). Participants in both age groups were further classified into one of the three OFC-type subgroups: cleft lip only (without or with alveolar cleft), cleft lip and cleft palate, and cleft palate only. Periodontal health was determined by evaluating plaque formation and gingival status with reference to the Silness and Loe plaque index (PI), Loe gingival index (GI), and community periodontal index (CPI). Periodontal health and oral hygiene were not significantly different between Groups I and II for cleft type (p > 0.05). A significant difference was not observed in PI for cleft type among the groups (p > 0.05). In Group II, GI and CPI were significantly higher than in Group I (p < 0.05). According to our results, cleft type does not influence periodontal health of children and adolescents with OFCs. Age, however, influences periodontal diseases' prevalence and severity.

DNA Methylation Effects on Van der Woude Syndrome Phenotypic Variability.

Van der Woude Syndrome (VWS) presents with combinations of lip pits (LP) and cleft lip and/or cleft palate (CL/P, CPO). VWS phenotypic heterogeneity even amongst relatives, suggests that epigenetic factors may act as modifiers. IRF6, causal for 70% of VWS cases, and TP63 interact in a regulatory loop coordinating epithelial proliferation and differentiation in palatogenesis. We hypothesize that differential DNA methylation within IRF6 and TP63 regulatory regions underlie VWS phenotypic discordance. DNA methylation of CpG sites in IRF6 and TP63 promoters and in an IRF6 enhancer element was compared amongst blood or saliva DNA samples of 78 unrelated cases. Analyses were done separately for blood and saliva, within each sex and in combination, and to address cleft type (CL/P ± LP vs. CPO ± LP) and phenotypic severity (any cleft + LP vs. any cleft only). For cleft type, blood samples showed higher IRF6 and TP63 promoter methylation on males with CPO ± LP compared to CL/P ± LP and on individuals with CPO ± LP compared to those with CL/P ± LP, respectively. Saliva samples showed higher IRF6 enhancer methylation on individuals with CPO ± LP compared to CL/P ± LP and contrary to above, lower TP63 promoter methylation on CPO ± LP compared to CL/P ± LP. For phenotypic severity, blood samples showed no differences; however, saliva samples showed higher IRF6 promoter methylation in individuals with any cleft + LP compared to those without lip pits. We observed differential methylation in IRF6 and TP63 regulatory regions associated with cleft type and phenotypic severity, indicating that epigenetic changes in IRF6 and TP63 can contribute to phenotypic heterogeneity in VWS.

"A Retrospective Evaluation of the Impact of Prenatal Diagnosis of Facial Clefts. Does Prenatal Diagnosis Matter?"

With the advent of improved prenatal detection, some patients with facial clefting are diagnosed prenatally while others are diagnosed postnatally. There is limited data regarding the utility of prenatal diagnosis and how this affects care of patients with facial clefts. A retrospective chart review was performed. Children with incomplete demographic data and those with syndromic conditions were excluded. The data were analyzed via Fisher's exact tests and Kruskal-Wallis tests (p < 0.05). 106 patients met inclusion criteria. Facial clefting was diagnosed prenatally at different frequencies depending on type of facial cleft- patients with cleft palate alone were less likely to be identified prenatally (p < 0.0001). Patients diagnosed prenatally were seen by craniofacial specialists at an earlier age compared to those diagnosed after birth (0.27 months vs 0.7 months, p < 0.001). Similarly, those with prenatal diagnosis underwent surgery at a younger age compared to those who were diagnosed postnatally (median: 3.6 months vs 10.67 months, p < 0.001) and experienced shorter lag time (median: 3.4 months vs 8.4 months, p = 0.027) from consultation to surgery. Importantly, prenatal diagnosis resulted in pre-surgical therapy more often than in children diagnosed postnatally (86% vs 22.2%, p < 0.001). Our data suggests that patients with prenatal diagnosis of facial clefts were more likely to undergo pre-surgical therapy, presented to a craniofacial specialist at an earlier age, underwent surgery at an earlier age, and experienced less lag time between initial visit and surgery. More study is warranted to improve protocols for prenatal diagnoses to improve surgical outcomes.

Foreign-born 5-year-old children with cleft palate had poorer speech outcomes than their native-born peers.

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.

“Long term speech outcomes after using the Sommerlad technique for primary palatoplasty: a retrospective study in the Wilhelmina Children’s Hospital, Utrecht.”

ObjectivesAfter cleft lip and/or palate (CL/P) repair, children may develop velopharyngeal insufficiency (VPI) leading to speech imperfections, necessitating additional speech correcting surgery. This study examines the incidence of VPI and speech correcting surgery after Sommerlad’s palatoplasty for CL/P, and its association with various clinical features.Materials and methodsA retrospective cohort study was performed in the Wilhelmina Children’s Hospital in Utrecht and child records from 380 individuals with CL/P registered from 2008 to 2017 were retrospectively reviewed. Inclusion criteria comprised the diagnosis of CL/P, primary palatoplasty according to Sommerlad’s technique, and speech assessment at five years or older. Association between cleft type and width, presence of additional genetic disorders and postoperative complications (palatal dehiscence, fistula) were assessed using odds ratios and chi squared tests.ResultsA total of 239 patients were included. The VPI rate was 52.7% (n = 126) and in 119 patients (49.8%) a speech correcting surgery was performed. Severe cleft type, as indicated by a higher Veau classification, was associated with a significant higher rate of speech correcting surgeries (p = 0.033). Significantly more speech correcting surgeries were performed in patients with a cleft width >10 mm, compared to patients with a cleft width ≤10 mm (p < 0.001). Patients with oronasal fistula underwent significantly more speech correcting surgeries than those without fistula (p = 0.004). No statistically significant difference was found in the incidence of speech correcting surgery between patients with and without genetic disorders (p = 0.890).Conclusions/clinical relevanceVariations in cleft morphology, cleft width and complications like oronasal fistula are associated with different speech outcomes. Future research should focus on creating a multivariable prediction model for speech correcting surgery in CL/P patients.

Does surgical sequencing influence the accuracy of maxillary positioning in bimaxillary cleft orthognathic surgery?

To evaluate whether the sequence of osteotomies influences the accuracy of maxillary positioning in patients with cleft palate ± cleft lip undergoing bimaxillary orthognathic surgery (OGS). This was a prospective study of patients with Veau 2-4 clefts who underwent bimaxillary OGS at tertiary-care children's hospital over a 3-year period. The primary predictor variable was the sequence of osteotomies (maxilla-first versus mandible-first). The primary outcome of interest was the concordance between the planned and achieved maxillary position, as assessed using linear and angular measurements. Secondary study predictors were demographic and surgical variables. Differences between groups were compared using non-parametric independent samples tests for continuous measures (data reported as median and interquartile range, IQR) and chi-squared tests for categorical measures. For all analyses, p≤ 0.05 was considered significant. Subjects who underwent maxilla-first (n=15) and mandible-first (n=16) operations were comparable with regard to age, gender, cleft type, skeletal classification, segmental maxillary osteotomy, and magnitude of maxillary movement (p ≥ 0.09). The planned sagittal and vertical positions of the maxilla were similarly accurate between the two groups (p ≥ 0.68). Angular accuracy was also comparable (p ≥ 0.56) between the study groups. In patients with CP ± CL undergoing bimaxillary orthognathic surgery, use of mandible-first sequencing, when compared to maxilla-first sequencing, does not impact accuracy of maxillary positioning in the immediate post-operative period in well-selected patients.

Estimated Time of Arrival: Impact of Healthcare Disparities on Access to and Outcomes of Multidisciplinary Cleft Lip and Palate Care.

To determine the impact of sociodemographic and clinical factors on patient presentation into the cleft care pathway and determine how delayed interventions may affect post-surgical outcomes. Retrospective study. Multidisciplinary craniofacial clinics of two university hospitals. 135 patients with cleft lip and/or palate. Primary cheiloplasty, primary palatoplasty. Age at initial presentation, age at first surgery, lag time, delayed surgery, rate of return to the emergency department (ED), readmission rate, reoperations, and oronasal fistula development. Patients referred by OBGYN who underwent cheiloplasty had an earlier age at initial presentation (p < 0.01), earlier age at first surgery (p = 0.01), and a shorter lag time (p < 0.01) compared to children from other referral pathways. African American children had an older age at first surgery (p = 0.01) and a longer lag time (p = 0.02) when compared to non-African American children. Children with syndromes had an older age at first surgery (p < 0.01) and a longer lag time (p < 0.01) than children without syndromes. Patient race, cleft type, and syndromic status increased the odds of receiving delayed surgery. Patients who received delayed palatoplasty returned to the ED at a higher rate than patients who received non-delayed palatoplasty (p = 0.02). Our data suggest that referral source, race, and syndromic status influence the timeliness of cleft care. Surgeons should develop strong referral networks with local OBGYNs and hospitals to allow for an early entry into the cleft care pathway.