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OBJECTIVE: To assess the barriers to obtaining care for patients with orofacial clefts through a survey of Florida-based orthodontists and families and an analysis of the Pediatric Health Information System (PHIS) database. DESIGN: A cross-sectional study utilizing multiple-choice questionnaires completed by Florida orthodontists and caregivers of patients who attended a Florida-based cleft and craniofacial clinic. Additionally, data from the PHIS database were analyzed to investigate national factors affecting the age of alveolar bone grafting (ABG). SETTING: Craniofacial team in Florida. PATIENTS/PARTICIPANTS: The survey included 39 orthodontists (7.1% response rate) and 48 caregivers (41% response rate) The PHIS study included 1182 patients. MAIN OUTCOME MEASURES: Barrier to orthodontic care and age of ABG. RESULTS: Orthodontic Survey: Among the surveyed orthodontists, 71% treated cleft/craniofacial patients, 37% accepted Medicaid, and 55% provided pro-bono care. Poor reimbursement was identified as the most common barrier (58%). Caregiver Survey: Most patients were insured by Medicaid (67%), with 55% incurring out-of-pocket expenses. PHIS Database: The average age of ABG was 10.3 years (SD = 3.2). Government funding was associated with a 6.0-month delay in ABG (p = 0.047) and residing in non-Medicaid expanded states was linked to a 6.0-month delay (p = 0.023). Post-Medicaid expansion status was also associated with a delay (p = 0.004). CONCLUSIONS: Access to oral care is difficult for patients with OFC. Despite both federal and state mandates, many financial and non-financial barriers still exist in accessing orthodontic care and a majority of patients experience significant out-of-pocket expenses despite statutorily mandated insurance coverage.

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OBJECTIVE: The inaugural Cleft Summit aimed to unite experts and foster interdisciplinary collaboration, seeking a collective understanding of velopharyngeal insufficiency (VPI) management. DESIGN: An interactive debate and conversation between a multidisciplinary cleft care team on VPI management. SETTING: A two-hour discussion within a four-day comprehensive cleft care workshop (CCCW). PARTICIPANTS: Thirty-two global leaders from various cleft disciplines. INTERVENTIONS: Cleft Summit that allows for meaningful interdisciplinary collaboration and knowledge exchange. MAIN OUTCOME MEASURES: Ability to reach consensus on a unified statement for VPI management. RESULTS: Participants agreed that a patient with significant VPI and a dynamic velum should first receive a surgery that lengthens the velum to optimize patient outcome. A global, multicenter prospective study should be done to test this hypothesis. CONCLUSION: The 1st Cleft Summit successfully distilled global expertise into actionable best-practice guidelines through iterative discussions, fostering interdisciplinary collaboration and paving the way for a transformative multi-center prospective study on VPI care.

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BACKGROUND: This study characterizes the potential loss of velar length in patients with a wide cleft and rescue of this loss of domain by local flap reconstruction, providing anatomic evidence in support of primary lengthening of the soft palate during palatoplasty. METHODS: A retrospective review was conducted of all patients with a cleft palate at least 10mm in width, who underwent primary palatoplasty with a buccal flap prior to 18 months of age over a 2-year period. All patients underwent primary palatoplasty with horizontal transection of the nasal mucosa, which was performed after nasal mucosa repair, but prior to muscular reconstruction. The resulting palatal lengthening was measured and the mucosal defect was reconstructed with a buccal flap. RESULTS: Of the 22 patients included, 3 (13.6%) had a history of Pierre Robin sequence, and 5 (22.7%) had an associated syndrome. No patients had a Veau I cleft, 7 (31.8%) had a Veau II, 12 (54.5%) had a Veau III, and 3 had (13.6%) a Veau IV cleft. All patients had a right buccal flap during primary palatoplasty. The mean cleft width at the posterior nasal spine was 10.6 ± 2.82mm, and mean lengthening of the velum after horizontal transection of the nasal mucosa closure was 10.5 ± 2.23mm. There were 2 (9.1%) fistulas, 1 (4.5%) wound dehiscence, 1 (4.5%) 30-day readmission, and no bleeding complications. CONCLUSIONS: Patients with a wide cleft palate have a potential loss of 1cm velar length. The buccal flap can rescue the loss of domain in palatal length, and potentially improve palatal excursion.

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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.

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OBJECTIVES: The study followed the modelling of postnatal growth of a healthy palate of the Central European (Czech) population sample based on transverse data on sex and age from 6 to 19 years. MATERIALS AND METHODS: Digitised 3D models of 212 healthy palatal surfaces were evaluated using 3D geometric morphometrics and superimpositions. The individuals were grouped based on age (preschool, younger and older school age, younger and older adolescents, young adults) and sex (♂ n = 101, ♀ n = 111). RESULTS: Female palatal development was non-linear and was interrupted between the 10-12 years and then proceeded intensively until the age of 15 when it ceased. In contrast, male-modelled growth was consistent throughout the follow-up and continued linearly until at least 19 years of age. The palate did not widen further with increasing age, and primarily palatal vaulting and heightening were found. The characteristics and distribution of areas with extensive modelled growth changes were comparable in females and males, as confirmed by the location of principal components (PC1 and PC2) within modal space and growth trajectories. The extent of sexual dimorphism increased from 15 years of age due to pubertal spurt combined with earlier completion of palatal development in females. CONCLUSIONS: The study showed modelled healthy palatal development from 6 years of age to early adulthood, which might be utilised as reference standards for the Central European population sample. CLINICAL RELEVANCE: The comparison of normal reference subjects with patients with cranio-maxillo-facial dysmorphologies represents the first step in diagnosing and establishing effective therapy.

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OBJECTIVE: The objective of this study is to evaluate online educational resources on cleft lip and palate teams. DESIGN: A Cross-Sectional Study. SETTING: An International, Multi-Institutional Study. PARTICIPANTS: All American Cleft Palate and Craniofacial Association-approved teams with websites. INTERVENTIONS: None. MAIN OUTCOME MEASURES: Websites were assessed for patient education and support content. Affiliation between presence of materials and U.S. Newsweek Top 100 Hospitals was also assessed. RESULTS: 187 teams were included. Presence of educational videos were available in 29.4% of websites, educational infographics in 18.2%, written materials in 66.8%, perioperative instructions in 19.3%, diagnosis information in 34.8% and treatment information in 63.1%. Information on team members were available on 77.0% of websites, resources in languages other than English in 38.5%, and support group resources in 25.7%. Cleft lip and palate diagnosis information was significantly associated with geographic region, with the largest impact from the West region (P = .03). There was a significant difference between teams affiliated with U.S. Newsweek Top 100 hospitals: Top 100 hospitals had a higher presence of educational infographics, perioperative instructions, definitions, diagnosis, and treatment (P < .01). There was no significant difference between presence of educational videos (P = .37). CONCLUSION: While many websites had basic educational materials, very few included detailed information on peri-operative planning, as well as additional forms of information including videos, infographics, and non-English languages. Providing comprehensive patient education materials online is an important supplement for patients with cleft lip and palate and should be prioritized by cleft teams.

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Compare the maxillary growth of patients with Unilateral Cleft treated with pre-surgical Functional Maxillary Orthopedic (FMO) and that of who underwent a surgical procedure with no previous use of appliances.Prospective study, the patients were divided into 2 groups. G1, was composed of 12 patients who received no pre-surgical intervention and cheilorhinoplasty at 6 months of age and G2, included 12 patients treated using Pre-Surgical Funtional Maxilary Orthopedic; they underwent no surgery during the study. Measurements were taken at three times: before the first month of life, at 6 months and between 9-12 months of age. Cleft distance (anterior, medium and posterior), maxillary width (anterior, medium and posterior) and minor and major segment width were analyzed.Alveolar Cleft decrease was 79.82% in G1 and 52% in G2. Posterior Cleft decrease was 24.1% in G1 and 41.77% in G2. Greater Segment Width increase was 24.53% for the patients in G1, and 37.47% for the patients in G2. As for Inter Canine Width, a decrease of 5.16% in G1 and an increase of 9.19% in G2 were found. Medium Arch Width only increased in a statistically significant manner in G2 9.02%.Surgery allowed for the closure of the alveolar cleft. FMO made it possible to close the anterior and the posterior clefts through the growth of the maxillary segments, increased the transverse growth of the maxilla and could prevent maxillary collapse. Each team must individually evaluate whether to indicate or not the use of preoperative orthopedics.

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Standard bone grafting between ages 6 and 12 has become the preferred treatment of choice for alveolar clefts. Given the importance of surgical timing in complete cleft palate repairs, it is important to identify any populations at-risk for delayed alveolar bone grafting. The purpose of this study is to identify whether a racial disparity is present nationally in the timing of alveolar bone grafting.Retrospective analysis Setting: Hospitals participating in the American College of Surgeons National Surgical Quality Improvement Program (ACS-NSQIP).Inclusion criteria involved patients who underwent alveolar bone grafting identified by current procedure terminology (CPT) code 42210 between years 2012 and 2019. Patients were stratified by age at time of operation based on the following parameters: early bone grafting (before 6 years of age), standard bone grafting (between 6 and 12 years of age), and late bone grafting (after 12 years of age).Racial and ethnic differences in the age of patients at the time of alveolar bone grafting.Overall, 20.28% of the cohort received alveolar bone graft after 12 years of age. African American (29.33%) and Hispanic (24.42%) patients received late alveolar bone grafting more frequently than other racial and ethnic groups (P < .001).Racial and ethnic disparities are present in the frequency at which patients receive late alveolar bone grafting for complete cleft palates. Given the suboptimal surgical results of late compared to standard alveolar bone grafting it is important to further investigate the driving factors of these disparities.

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The case series details 2 unusual cases of male newborns with cleft lip and palate (CLP) that later developed formula otorrhea. Both patients underwent bilateral myringotomies with the insertion of pressure equalizing (PE) tubes for chronic otitis media with effusion (OME). Chronic otorrhea associated with feeding occurred post-PE tube insertion and the otorrhea was later confirmed to be due to reflux of formula. Patients were treated with antibiotic ear drops, routine ear cleaning, anti-reflux medication, and reflux precautions. After definite cleft palate repair, formula otorrhea completely resolved. When patients with CLP develop chronic OME or otorrhea following PE tube placement, reflux of formula into the middle ear should be considered and treated accordingly.

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Objective: Using labial vestibular flap was performed to close the primary alveolar and hard palate cleft at the second stage of early 2-stage closure surgery for unilateral cleft lip and palate for minimizing the damage to the maxillary periosteum. We analyzed maxillary development to clarify the influence of cleft palate surgery. Design: Retrospective longitudinal study in 5 years after primary palatal closure. Setting: Institutional study Patients: Study subjects included 214 patients with nonsyndromic complete unilateral cleft lip and palate who were consecutively treated in our clinic. Main Outcome: We used a 3D dental model scanner to assess maxillary development in patients aged 3 months to 5 years after using either the conventional pushback method (PB) (51 cases) or 2-stage closure (Local palatal flap closure: LF [67 cases] and Labial vestibular flap closure: VF [96 cases]). Results: Comparing the measurement results, the major axis of maxilla, width, intercanine distance, and intermolar distance was significantly larger in the LF group compared to the PB group. After the age of 3, the cleft side of VF group had grown significantly to compare with LF group in width. It was also confirmed that the inserted labial mucosal flap itself grew. Enlargement of the labial mucosal flap was observed at all sites except the canine. Conclusion: Good maxillary growth occurred in the following order: VF groups > LF group > PB group. Poor growth was correlated with the extent of periosteal damage during surgery and the degree of postoperative bone surface exposure.

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Unoperated patients with bilateral complete cleft lip and palate (BCCLP), who do not receive orthopedic treatment in infancy and present with significant sagittal excess of the premaxilla, are among the most difficult patients to treat. Delay in primary reconstruction reduces the elastic characteristics of the tissues. The resulting rigidity of bony structures accompanied by the wide alveolar and palatal clefts associated with sagittal excess of the premaxilla pose a great challenge for both surgeon and orthodontist. There is no unique protocol for treatment of these conditions. This paper describes the use of orthopedic treatment in an unoperated 3-year-old male child with BCCLP and a protrusive premaxilla whose appearance and function were both severely affected. A modified Meazzini technique, as described in operated BCCLP patients with a downward-displaced premaxilla, was used. The device itself consists of the 2 independent components mutually connected by a 17 × 25 steel arch. The first is an active fan-type expander. The second consists of an acrylic cap covering the premaxilla and 3 brackets used for the steel arch application. They are connected by a rectangular steel 17 × 25 archwire. Reduction of the premaxillary sagittal excess of is carried out with an elastic chain. The use of this orthopedic appliance is a reliable and valuable presurgical treatment in the therapy of children with BCCLP and protrusive premaxilla whose orthopedic and surgical treatment have been delayed.

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Total aplasia of paranasal sinus (TAPS) is extremely rare, although the fact that partial aplasia is very common. TAPS seems to be limited to only 5 case reports in the literature until now. We present the case of a 29-year-old patient who has a syndromic face appearance but whose TAPS was detected incidentally. The maxillary, sphenoid, ethmoid, and frontal sinuses were totally aplastic. Furthermore, clinodactyly and high-arched palate were observed. The patient's appearance was consistent with a syndromic face because of some findings on inspection such as hypertelorism, shortening of the palpebral fissure, protruded and wide nasal base, high arched palate and zygomatic hypoplasia. The patient's profile was more suitable for Teacher-Collins syndrome than other syndromes, however, a certain diagnosis was not made genetically. To the best of our knowledge, this is the first reported association between TAPS and a syndromic condition.

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INTRODUCTION: Palate development involves a genetic regulation through a complex molecular mechanism that may be disrupted by environmental factors, resulting in impaired fusion and cleft palate formation. An encounter with a case of cleft palate due to dorsal tongue hamartoma prompted us to perform this systematic review. OBJECTIVE: To review the clinical profile and management approach for a case with cleft palate and tongue hamartoma. DESIGN: A systematic literature search was conducted using keywords related to cleft palate and tongue hamartoma in PubMed, Scopus, MEDLINE, and Scielo databases through December 2021, with no time or language restrictions. PATIENTS, PARTICIPANTS: Studies reporting patients with cleft palate and tongue hamartoma were included. MAIN OUTCOME MEASURE(S): Information related to clinical profile, diagnostic tests, histopathology, management, and outcomes were extracted.Fourteen relevant publications were identified with 16 cases reported so far. Among them, thirteen patients were females (81.25%), and 3 were males (18.75%). The age of presentation varied from birth to 19 years. Oral-facial-digital syndrome (type II) was the most commonly associated syndrome.Congenital tongue hamartoma with cleft palate is a rare presentation, which can present as an isolated entity or part of a syndrome. Genetic evaluation is warranted, particularly for multiple hamartomatous lesions. The preferred treatment is immediate excision of hamartoma while following a standard timeline for palatoplasty.

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OBJECTIVE: To examine levator veli palatini muscle composition in patients with nonsyndromic cleft palate and investigate the impact of Veau class. DESIGN: Prospective cohort study. SETTING: Tertiary care academic hospital. PATIENTS/PARTICIPANTS: Thirteen patients with nonsyndromic cleft palate were recruited. INTERVENTIONS: During primary palatoplasty, a sample of levator veli palatini muscle was excised and prepared for histological analysis. MAIN OUTCOME MEASURES: Fat and collagen content were determined utilizing Oil Red and Sirius red stains, respectively, while muscle fiber cross-sectional areas were calculated from H&E-stained samples, with analysis using histomorphometric methods. Immunofluorescent staining of myosin heavy chain isoforms was performed. RESULTS: Patients underwent repair at 10.8 months of age (interquartile range [IQR] 10.2-12.9). Fat content of the levator veli palatini muscle was low in both groups, ranging from 0% to 5.2%. Collagen content ranged from 8.5% to 39.8%; neither fat nor collagen content showed an association with Veau classes. Mean muscle fiber cross-sectional area decreased with increasing Veau class, from 808 µm2 (range 692-995 µm2) in Veau II to 651 µm2 (range 232-750 µm2) in Veau III (P = .02). There was also a nonsignificant decrease in proportion of type I muscle fibers with increasing Veau class (44.3% [range 31.4%-84.4%] in Veau II vs 35.3% [range 17.4%-61.3%] in Veau III). CONCLUSIONS: Muscle fiber area in levator veli palatini muscles decreases in Veau III clefts in comparison to Veau II. The impact of these differences in velopharyngeal dysfunction requires further analysis of a larger cohort.

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OBJECTIVE: To determine the prevalence and odds ratios for cleft lip and/or palate (CL/P) among infants prenatally exposed to opioids with or without neonatal opioid withdrawal syndrome (NOWS). DESIGN: This study represents an exploratory, retrospective cohort study design of newborn medical health records from 2011 to 2016. SETTING: Records were drawn from a regional health system located in South Central Appalachia. POPULATION AND STUDY SAMPLE: The original population yielded 3 cohorts of infants: (1) infants with opioid exposure (OE) but not requiring pharmacological intervention (OE; N = 168); (2) infants with NOWS requiring pharmacological intervention (N = 294); and (3) infants with no opioid exposure (NOE; N = 16 090), the primary comparison group. MAIN OUTCOME: Infants in the NOWS and OE groups showed significantly increased prevalence and odds ratios for CL/P when compared to those in the NOE group. RESULTS: Prevalence rates per 1000 live births for infants with OE (35.71) and infants with NOWS (6.80) were significantly higher than those for infants with NOE (1.37). Comparison of infants with OE to the NOE group revealed significantly increased odds for CL/P, isolated cleft palate (CP), cleft lip (CL), and cleft lip and palate (CLP) (27.05, 41.81, 19.26, 19.37, respectively; all Ps < .008). The odds ratios for infants with NOWS compared to the NOE group were significantly higher for CL/P and CP (5.00 and 10.98, respectively; Ps < .03) but not for CL and CLP. CONCLUSION: The results provide additional evidence that prenatal OE should be considered among the critical environmental risk factors that can contribute to CL/P.

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Cleft patients may develop an abnormal opening (fistula) between the oral and the nasal cavities. Surgical repair minimizes the adverse effect on speech and feeding. However, an obturator prosthesis is a nonsurgical approach to help close the communication. The purpose of the case report presented is to show the clinical use of an intraoral digital impression in the fabrication of obturator/speech aid appliances in children with cleft lip and palate deformity. Minimal adjustments were needed, and patients and caregivers responded positively. Prostheses demonstrated good stability and retention at delivery. The use of digital technology seems to have several benefits as an alternative method for capturing impressions, especially in young children with cleft lip and palate deformity.

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BACKGROUND: Common surgical techniques aim to turn the entire vomerine mucosa with vomer flaps either to the oral side or to the nasal side. The latter approach is widely performed due to the similarity in color to the nasal mucosa. However, we lack a histologic description of the curved vomerine mucosa in cleft lip and palate malformations. METHODS: We histologically examined an excess of curved vomerine mucosa in 8 patients using hematoxylin-eosin, periodic acid-Schiff, Elastin van Gieson, and Alcian blue stains. Tissue samples were obtained during surgery at 8 months of age. RESULTS: Our histological analysis of the mucoperiosteum overlying the curved vomer revealed characteristics consistent with those of an oral mucosa or a squamous metaplasia of the nasal mucosa, as exhibited by a stratified squamous epithelium containing numerous seromucous glands. Some areas showed a palisaded arrangement of the basal cells compatible with metaplasia of respiratory epithelium, but no goblet cells or respiratory cilia were identified. Abundant fibrosis and rich vascularity were present. CONCLUSION: The vomer mucosa showed no specific signs of nasal mucosa. These findings should be considered in presurgical cleft orthopedics and palatal surgery for further refinement. Shifting the vomer mucosa according to a fixed physiologic belief should not overrule other important aspects of cleft repair such as primary healing and establishing optimal form and function of palatal roof and nasal floor.

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Bilateral Tessier type 4 craniofacial clefts are extremely rare and disfiguring malformations with vision-threatening ramifications. To date, there is no consensus in the literature with respect to the ideal surgical technique and management of these patients. Emergent eyelid reconstruction and additional procedures may be required to protect the cornea and avoid further ophthalmic and surgical complications. We present our experience and challenges of managing a case of bilateral Tessier type 4 clefting with an emphasis on oculoplastic considerations.

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OBJECTIVE: Two main objectives were established. First objective was to determine the prevalence of the cleft lip and/or cleft palate (CL/P) in Thailand from 2012 to 2015 using the orofacial clefts (OFCs) registry and civil registration. Second objective was to conduct a quality control of this OFC registry especially for the Birth Defects Registration (BDR). DESIGN: Registry-based survey. SETTING: Analyzing data from the Thailand National Health Security Office. PARTICIPANTS: Registered patients with CL/P in Thailand from 2012 to 2015. INTERVENTION: None. MAIN OUTCOME MEASURE: Duplicated records were verified using National Identity Number (Thai ID#) and date of birth. The prevalence of CL/P and specific phenotypes was then calculated. From this prevalence estimate method, quality assurance of the OFCs registry was possible. RESULTS: For the main outcome, the population-weighted pool prevalence of CL/P was 2.14 per 1000 live births (95% confidence interval of 2.08-2.20). Thai ID# and expense reimbursement systems were the main factors driving this cases capturing. However, this OFCs registration still requires active case finding with clinical verification, improvement of staff training and databases networking. CONCLUSIONS: This study reported a very high CL/P prevalence of Thailand. Strengths and limitations of these OFCs registry and BDR were described.

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OBJECTIVE: To evaluate patency of circummaxillary sutures in children with Apert, Crouzon, and Pfeiffer Syndromes and to compare it to a nonsyndromic matched control group. DESIGN: Case-control study. SETTING: Tertiary care public hospital. MATERIALS AND METHODS: Thirty-eight computed tomography (CT) scans of patients affected by syndromic craniofacial synostosis (13 patients with Apert syndrome, 20 patients with Crouzon syndrome, and 5 patients with Pfeiffer syndrome), average age 5 ± 2.8 years, range 1.9 to 12 years, were compared to age- and sex-matched control CTs of 38 nonsyndromic children. Computed tomography scans of the study group had to be performed prior to any midfacial surgery. MAIN OUTCOME MEASURES: Midpalatal suture, zygomaticomaxillary sutures, and pterigomaxillary sutures were evaluated and scored. RESULTS: The syndromic group showed a significant earlier ossification of all sutures compared to the nonsyndromic group. Significant differences were already present in early childhood and continued through adolescence. CONCLUSIONS: Based on the differences in terms of maxillary sutural ossification identified, midfacial hypoplasia does not seem to be only secondary to premature cranial base ossification, but also to primary synostosis of facial sutures, thus providing new insights into the pathogenesis of midface deficiency in children with craniofacial-synostosis. Care should be taken when planning any maxillary orthopedics, such as expansion or maxillary protraction, given the high frequency of early fusion of circummaxillary sutures.

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