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Background and Aim: The burden of orofacial clefts (OFCs) has declined globally except in sub-Saharan Africa, with a great disease burden in North Africa. Children with OFCs have a high risk of malnutrition, and African countries have some of the highest malnutrition rates. This scoping review assessed the status of research on OFCs and nutrition and feeding among children living in African countries., Methods: We followed the Joanna Briggs Institute guidelines for conducting scoping reviews. We searched eleven databases for articles on malnutrition and feeding among children with OFCs living in African countries. No restriction was done by type of study or publication date. Books, book chapters, and reviews were excluded. Only publications in English language were included. We extracted information about the publication year, study design, setting, location, participants' age, data collection methods, international collaboration, and funding. We classified articles into studies assessing (1) the impact of nutritional deficiencies during pregnancy on OFCs, (2) the impact of OFCs on malnutrition, (3) feeding problems in children with OFCs, and (4) the impact of nutritional status on OFCs repair outcomes. We calculated frequencies and used bar charts and a map., Results: Out of 208 search results, 36 were duplicates, and 25 eventually fit the inclusion criteria, with 52% retrieved from Google Scholar. About 80% of the studies were from four countries: Nigeria, South Africa, Ghana, and Uganda; 72% were hospital-based and 52% were cross-sectional. The most frequent data collection method was clinical examination and questionnaires. Most studies focused on feeding problems in children with OFCs (44%) and the impact of OFCs on malnutrition (32%). International collaboration was observed in six studies, with one study showing South-South collaboration. Only two studies were funded., Conclusions: There is a predominance of under-funded descriptive research not indexed by international databases. Minimal research has been directed to population-level OFC preventive programs in primary healthcare settings and assessing interventions supporting children with OFCs. A research agenda is needed to prioritize research needs and secure funds to support South-South collaboration to address the nutrition and feeding-related problems associated with OFCs., (© 2024. The Author(s).)

Objective: This retrospective study investigated the influence of palatal fistula (PF) formation after double opposing Z-plasty (DOZ) on speech outcomes in patients with cleft palate (CP), focusing on cleft width and palatal length as predictors of velopharyngeal insufficiency (VPI)., Methods: This study included 1117 patients with CP (579 males, 538 females) who underwent DOZ, performed by a single surgeon, between 1988 and 2017. Demographic characteristics, cleft dimensions, history of PF formation, and speech outcomes were investigated. Speech evaluations were performed at a minimum age of five to assess nasal emission, hypernasality, compensatory articulation, intelligibility, necessity for VPI surgery, and speech therapy. Logistic regression analysis was performed., Results: Speech assessments were conducted at the median age of five (interquartile range [IQR], 5-6 years). Overall, 96.5% of patients achieved 'socially acceptable speech' after DOZ. Patients with PF history showed greater cleft width and experienced higher rates of hypernasality, nasal emission, and VPI on videofluoroscopy (VFS) compared to those without PF history (mean, 11.4 mm vs. 7.1 mm; 28.4% vs. 23.6%; 34.8% vs. 14.9%, 38.5% vs. 14.0%, 40.6% vs. 28.3%, respectively; all p < 0.0001). Cleft width was significantly associated with VPI-related speech outcomes in the multivariate logistic regression analysis, affecting both perceptual and VFS-measured outcomes., Conclusions: A wider CP gap significantly increased the risk of VPI-related speech difficulties after DOZ. Cleft width is a more critical predictor of adverse speech outcomes than the presence of small-to-medium-sized PFs. Patients with a history of PF and wider cleft gaps require targeted interventions and intensified follow-up to effectively manage and improve speech outcomes., (Copyright © 2024 British Association of Plastic, Reconstructive and Aesthetic Surgeons. Published by Elsevier Ltd. All rights reserved.)

Study Objectives: Severe respiratory distress of neonates with Robin sequence is traditionally managed by surgery. Stanford orthodontic airway plate treatment (SOAP) is a nonsurgical option. The study aimed to determine whether SOAP can improve polysomnography parameters of neonates with Robin sequence., Methods: Polysomnography of neonates with Robin sequence treated with SOAP at a single hospital were retrospectively analyzed. Patients without polysomnography at all 4 time points (pre, start of, mid, and posttreatment) were excluded. Data were analyzed using a linear mixed effects model., Results: Sixteen patients were included. All patients had cleft palate. The median age (minimum, maximum) at the start of treatment was 1.1 months (0.3, 5.1) with the treatment duration of 4.5 months (3.5, 6.0). The mean obstructive apnea-hypopnea index (95% confidence interval) decreased from 39.3 events/h (32.9, 45.7) to 12.2 events/h (6.7, 17.7) ( P < .001), obstructive apnea index decreased from 14.1 (11.2, 17.0) events/h to 1.0 (-1.5, 3.5) events/h ( P < .001), and oxygen nadir increased from 79.9% (77.4, 82.5) to 88.2% (85.5, 90.8) ( P < .001) between pre and start of treatment. Respiratory improvements were sustained during and after the treatment. All patients avoided mandibular distraction osteogenesis or tracheostomy following SOAP., Conclusions: As being a rare diagnosis, the number of participants was, as expected, low. However, the current study shows that SOAP can improve polysomnography parameters, demonstrating its potential utility before surgical interventions for neonates with Robin sequence and cleft palate experiencing severe respiratory distress., Citation: Choo H, Sidell DR, Kim J-W, Ahn H-W, Day HS, Sullivan SS. Nonsurgical improvement of severe upper airway obstruction in infants with Robin sequence and cleft palate using Stanford orthodontic airway plate treatment. J Clin Sleep Med . 2024;20(11):1807-1817., (© 2024 American Academy of Sleep Medicine.)

Background: Speech in individuals with cleft lip and/or palate (CLP) is a complex myriad of presenting symptoms. It is uniquely associated with the structural difference of velopharyngeal insufficiency (VPI), together with a wide and heterogeneous range of other aetiologies which often co-occur. The nature of the speech sound disorder (SSD) including VPI may also change over the course of an individual's care pathway. Differences in terminology and approaches to analysis are currently used, resulting in confusion internationally. Additionally, current diagnostic labels and classification systems in SSD do not capture the complexity and full nature of speech characteristics in CLP., Aims: This paper aims to explore the different aetiologies of cleft palate/VPI speech and to relate aetiology with speech characteristic(s). In so doing, it attempts to unravel the different terminology used in the field, describing commonalities and differences, and identifying overlaps with the speech summary patterns used in the United Kingdom and elsewhere. The paper also aims to explore the applicability of current diagnostic labels and classification systems in the non-cleft SSD literature and illustrate certain implications for speech intervention in CLP., Methods and Procedures: The different aetiologies were identified from the literature and mapped onto cleft palate/VPI speech characteristics. Different terminology and approaches to analysis are defined and overlaps described. The applicability of current classification systems in SSD is discussed including additional diagnostic labels proposed in the field., Outcomes and Results: Aetiologies of cleft palate/VPI speech identified include developmental (cognitive-linguistic), middle ear disease and fluctuating hearing loss, altered oral structure, abnormal facial growth, VPI-structural (abnormal palate muscle) and VPI-iatrogenic (maxillary advancement surgery). There are four main terminologies used to describe cleft palate/VPI speech: active/passive and compensatory/obligatory, which overlap with the four categories used in the UK speech summary patterns: anterior oral cleft speech characteristics (CSCs), posterior oral CSCs, non-oral CSCs and passive CSCs, although not directly comparable. Current classification systems in non-cleft SSD do not sufficiently capture the full nature and complexity of cleft palate/VPI speech., Conclusions and Implications: Our attempt at identifying the heterogeneous range of aetiologies provides clinicians with a better understanding of cleft palate/VPI speech to inform the management pathway and the nature and type of speech intervention required. We hope that the unravelling of the different terminology in relation to the UK speech summary patterns, and those used elsewhere, reduces confusion and provides more clarity for clinicians in the field. Diagnostic labels and classification require international agreement., What This Paper Adds: What is already known on the subject Speech associated with cleft palate/velopharyngeal insufficiency (VPI) is a complex myriad of speech characteristics with a wide and heterogeneous range of aetiologies. Different terminology and speech summary patterns are used to describe the speech characteristics. The traditional classification of cleft palate/VPI speech is Articulation Disorder, although evidence is building for Phonological Disorder and contrastive approaches in cleft speech intervention. What this paper adds to existing knowledge This paper explores the range of aetiologies of cleft palate/VPI speech (e.g., altered oral structure, abnormal facial growth, abnormal palate muscle and iatrogenic aetiologies) and attempts to relate aetiology with speech characteristic(s). An attempt is made at unravelling the different terminology used in relation to a well-known and validated approach to analysis, used in the United Kingdom and elsewhere. Complexities of current diagnostic labels and classifications in Speech Sound Disorder to describe cleft palate/VPI speech are discussed. What are the potential or actual clinical implications of this work? There needs to be a common language for describing and summarising cleft palate/VPI speech. Speech summary patterns based on narrow phonetic transcription and correct identification of aetiology are essential for the accurate classification of the speech disorder and identification of speech intervention approaches. There is an urgent need for research to identify the most appropriate type of contrastive (phonological) approach in cleft lip and/or palate., (© 2023 The Authors. International Journal of Language & Communication Disorders published by John Wiley & Sons Ltd on behalf of Royal College of Speech and Language Therapists.)

Objective: To characterize the onset and prevalence of conductive hearing loss (CHL) in pediatric patients with cleft palate (CP) prior to palatoplasty with an enhanced audiologic protocol., Design: Retrospective cohort study., Setting: Multidisciplinary cleft and craniofacial clinic at a tertiary care center., Patients: Patients with CP who received audiologic workup pre-operatively. Patients with bilateral permanent hearing loss, expiration prior to palatoplasty, or no pre-operative data were excluded., Interventions: Patients with CP born February 2019 to November 2019 who passed newborn hearing screening (NBHS) received audiologic testing at 9 months of age (standard protocol). Patients born December 2019 to September 2020 underwent testing prior to 9 months of age (enhanced protocol)., Main Outcome Measures: Age of identification of CHL in patients after implementation of the enhanced audiologic protocol., Results: The number of patients who passed their NBHS in the standard protocol (n = 14, 54%) and the enhanced protocol (n = 25, 66%) did not differ. Infants who passed their NBHS, but demonstrated hearing loss on subsequent audiologic testing did not differ between enhanced (n = 25, 66%) and standard cohort (n = 14, 54%). Of patients who passed NBHS in the enhanced protocol, 48% (n = 12) had CHL identified by 3 months, and 20% (n = 5) by 6 months of age. With the enhanced protocol, patients who did not undergo additional testing post NBHS significantly dropped from 44.9% (n = 22) to 4.2% (n = 2) ( P  < .0001)., Conclusion: Even with passed NBHS, CHL is still present for infants with CP pre-operatively. Earlier and more frequent testing for this population is recommended., Competing Interests: Declaration of Conflicting InterestsThe author(s) declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article. This retrospective cohort study was approved by the Vanderbilt University institutional Review Board (#190840).

Objective: To determine whether performing tonsillectomy at the time of Furlow palatoplasty for the treatment of cleft palate related velopharyngeal insufficiency (VPI) incurs increased surgical complications or compromises speech outcomes., Design: A retrospective review of patients who had Furlow palatoplasty and the outcomes of surgery in the treatment of cleft palate related VPI., Setting: A single academic center between January 2015 and January 2022., Participants: Patients with submucous cleft (SMC) palate or patients with prior straight line primary palatoplasty presenting with VPI., Interventions: Simultaneous conversion Furlow palatoplasty and tonsillectomy., Main Outcome Measure(s): Primary outcome measures include preoperative and postoperative Modified Pittsburgh Weighted Speech Scale (mPWSS), and postoperative surgical complications., Results: Eight patients (25%) underwent Furlow palatoplasty and concomitant tonsillectomy, while 24 patients (75%) underwent Furlow palatoplasty alone. A significantly lower median postoperative mPWSS score, corresponding to better velopharyngeal function, was reported for patients in the Furlow-tonsillectomy group (0, IQR 0-0) compared to the Furlow only group (1, IQR 0-9, p  =  0.046). No surgical complications were encountered in either group. Five patients (20.8%) in the Furlow only group required subsequent surgery for persistent VPI. No patients in the Furlow-tonsillectomy group required additional surgical treatment for VPI (0%, p  =  0.16)., Conclusions: Tonsillectomy at time of Furlow palatoplasty is utilized in patients with both VPI and baseline tonsillar hypertrophy to lessen the risk of postoperative obstructive breathing. Tonsillectomy performed concurrently with Furlow palatoplasty is safe, without increased risk of surgical complications, and does not compromise post-Furlow palatoplasty speech outcomes., Competing Interests: Declaration of Conflicting InterestsThe author(s) declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.

Purpose: Treatment fatigue is a complex, multidimensional, multicausal, and subjective phenomenon that is not yet well explored and understood in the area of speech therapy. The purpose of this study was to investigate speech-related treatment fatigue and speech treatment dropout in parents and children with a cleft (lip and) palate receiving or having received speech treatment for cleft-related articulation disorders., Method: Thirteen participants were included in this study ( n = seven parents with a median age of 40 years and n = six children with a median age of 10 years). Qualitative semistructured interviews were conducted separately with parents and children to investigate their experiences with cleft speech treatment. An inductive thematic approach was used to analyze the data and construct different themes. Rigor of the data was verified by conducting an investigator triangulation and by performing member checks., Results and Conclusions: The analyses of the interviews yielded three major themes of importance to the children and their parents: (a) physical symptoms of treatment fatigue, (b) psychological symptoms of treatment fatigue, and (c) from treatment fatigue to treatment dropout. Physical symptoms of treatment fatigue were mainly related to transportation burden. On a psychological level, speech treatment may potentially lead to a cognitive-emotional overload. These feelings are primarily related to the practical issue of scheduling required treatment sessions in the family agenda. The decision to discontinue speech treatment was reported to be multifactorial. In this decision-making process, data suggested that the child's perspective must be heard more.

Purpose: The purpose of this study was to examine the influence of socioeconomic deprivation on language and developmental outcomes in toddlers with cleft palate with or without cleft lip (CP ± L). Other factors known to influence language outcomes were also considered, including home language history, history of hearing problems, syndromic diagnoses, and sex., Method: A multicenter, cross-sectional study design was conducted. Data for 566 16-month-old toddlers with CP ± L were collected from 17 outpatient cleft palate clinics located throughout the United States. Outcome measures included the MacArthur-Bates Communicative Development Inventories, LENA Developmental Snapshot, age at first word as reported by the caregiver, and the Ages and Stages Questionnaires-Third Edition (ASQ-3). Multivariable linear or logistic regression was used to determine the influence of socioeconomic deprivation, as measured by the Area Deprivation Index, on language and developmental outcomes., Results: Greater socioeconomic deprivation significantly predicted poorer language outcomes in toddlers with CP ± L, including receptive vocabulary words ( p = .02), expressive vocabulary words ( p = .02), and late-developing gestures ( p = .02). Additionally, toddlers from less affluent neighborhoods produced their first words significantly later than their counterparts living in more affluent areas ( p < .01). Lower maternal education levels predicted significantly increased risk for problem solving delays ( p < .01), and patients with subsidized insurance were at significantly increased risk for personal-social delays on the ASQ-3 ( p < .01)., Conclusions: Children with CP ± L are susceptible to developmental delays associated with socioeconomic deprivation. These findings have implications for identifying a child's individual risk factors for developmental language disorders when conducting speech-language assessments. Future study should examine how inequities in care can be mitigated and addressed.

Importance: Cleft lip or palate is a prevalent birth defect, occurring in approximately 1 to 2 per 1000 newborns and often necessitating numerous hospitalizations. Specific rates of hospitalization and complication are underexplored., Objective: To assess the rates of airway infection-associated hospitalization, overall hospital admissions, in-hospital complications, and mortality among children with a cleft lip or palate., Design, Setting, and Participants: This nationwide, population-based cohort study used in-hospital claims data from the Federal Statistical Office in Switzerland between 2012 and 2021. Participants included newborns with complete birth records born in a Swiss hospital. Data were analyzed from March to November 2023., Exposure: Prevalent diagnosis of a cleft lip or palate at birth., Main Outcomes and Measures: Outcomes of interest were monthly hospitalization rates for airway infections and any cause during the first 2 years of life in newborns with cleft lip or palate. In-hospital outcomes and mortality outcomes were also assessed, stratified by age and modality of surgical intervention., Results: Of 857 806 newborns included, 1197 (0.1%) had a cleft lip and/or palate, including 170 (14.2%) with a cleft lip only, 493 (41.2%) with a cleft palate only, and 534 (44.6%) with cleft lip and palate. Newborns with cleft lip or palate were more likely to be male (55.8% vs 51.4%), with lower birth weight (mean [SD] weight, 3135.6 [650.8] g vs 3284.7 [560.7] g) and height (mean [SD] height, 48.6 [3.8] cm vs 49.3 [3.2] cm). During the 2-year follow-up, children with a cleft lip or palate showed higher incidence rate ratios (IRRs) for hospitalizations due to airway infections (IRR, 2.33 [95% CI, 1.98-2.73]) and for any reason (IRR, 3.72 [95% CI, 3.49-3.97]) compared with controls. Additionally, children with cleft lip or palate had a substantial increase in odds of mortality (odds ratio [OR], 17.97 [95% CI, 11.84-27.29]) and various complications, including the need for intubation (OR, 2.37 [95% CI, 1.95-2.87]), extracorporeal membrane oxygenation (OR, 2.89 [95% CI, 1.81-4.63]), cardiopulmonary resuscitation (OR, 3.25 [95% CI, 2.21-4.78]), and respiratory support (OR, 1.94 [95% CI, 1.64-2.29])., Conclusions and Relevance: In this nationwide cohort study, the presence of cleft lip or palate was associated with increased hospitalization rates for respiratory infections and other causes, as well as poorer in-hospital outcomes and greater resource use.

Background: This study evaluates a three-dimensional (3D) visualisation design combined with customized surgical guides to assist anterior maxillary segmental distraction osteogenesis (AMSDO) in correcting maxillary hypoplasia in adolescents with cleft lip and palate (CLP), focusing on treatment outcomes, satisfaction and the validity of 3D planning., Methods: This retrospective cohort study was conducted at a single hospital in China. Between January 2020 and December 2023, 12 adolescents with CLP with maxillary hypoplasia were included. An advanced 3D simulation was used to convey the treatment strategy to the patients and their families. A customized surgical guide and distraction osteogenesis device were designed. Cephalometric analysis evaluated AMSDO changes and long-term stability. Patient satisfaction was assessed. The Chinese version of the Child Oral Health Impact Profile was used to evaluate the children's oral health-related quality of life before and after treatment. The postoperative outcomes were compared with the planned outcomes by superimposing the actual postoperative data onto the simulated soft tissue models and calculating the linear and angular differences between them., Results: One patient experienced postoperative gingivitis, yielding an 8.33% complication rate. Most patients (83.33%) were highly satisfied with the target position, with the rest content. Cephalometric analysis showed significant improvements in various indices post-traction. Quality-of-life scores significantly improved post-treatment. The discrepancies in facial soft tissue between the simulated and actual results were within clinically satisfactory ranges., Conclusions: Digitally designed surgical guides effectively treat maxillary hypoplasia in adolescents with CLP, ensuring stability, reducing complications, reducing dependency on operator experience, and enhancing satisfaction and health outcomes. Although the simulated results were clinically acceptable, it is important to inform patients of potential variations in the predicted soft tissue., (© 2024. The Author(s).)

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., Competing Interests: The authors declare no conflict of interest., (©2024 The Author(s). Published by MRE Press.)

This study evaluated the results of a reproductible protocol indicating the need for a pharyngeal flap in children with cleft palate and velopharyngeal insufficiency (VPI). A retrospective review of all patients operated for a pharyngeal flap between 2010 and 2019 in our center was conducted. After exclusion of patients with primary VPI or residual fistulas, 31 patients' data were analyzed. Our main outcome measure was the improvement of the Borel Maisonny Classification (BMC) by at least 1 rank. Further analysis was made to evaluate the impact of age, type of cleft, and BMC before surgery on the gain in the velopharyngeal function. Of the 31 patients, success was achieved in 29 (93.5%, p<0.005). There was no significant correlation between age and gain in the velopharyngeal function (p = 0.137). There was no significant correlation between type of cleft and gain in the velopharyngeal function (p = 0.148). There was a significant correlation observed between the starting classification and gain in velopharyngeal function. The gain observed was greater as the initial velopharyngeal function was worse (p = 0.035). The use of an algorithm combining clinical assessment with a standardized classification of the velopharyngeal function proved to be a reliable tool for the indication of surgery in patients with VPI. A close follow up is essential in a multidisciplinary team., Competing Interests: Declaration of Competing Interest The authors declare that there is no conflict of interest., (Copyright © 2023. Published by Elsevier Masson SAS.)

Evaluate periodontal bone support of maxillary central incisors (MCI) in patients with bilateral complete cleft lip and palate (BCCLP). Determine if syndromic diagnosis, age at time of alveolar bone graft (ABG), presence of maxillary lateral incisor (MLI), history of dentofacial orthopedics, maxillary expansion, and pre-maxillary osteotomy are associated with the periodontal bone support of MCI., Retrospective radiographic study., Tertiary care children's hospital., One hundred seventy-nine patients with BCCLP (22 syndromic) who had post-operative ABG cone beam computed tomography (CBCT) scans taken between 2002-2018., Crown to root ( C/R) ratio of MCI measured on CBCT scans., The C/R ratio in 65% of MCI indicated periodontally compromised teeth. Presence of a MLI improved bone support on adjacent MCI when compared to those missing a MLI (51.4% vs 28.4%, P  = .010). There was no significant difference in C/R ratios for syndromic diagnosis, age at ABG, history of dentofacial orthopedics, maxillary expansion, and pre-maxillary osteotomy., The majority of MCI in patients with BCCLP are periodontally compromised but bone support is improved when cleft adjacent lateral incisors are present., Competing Interests: Declaration of Conflicting InterestsThe author(s) declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.

The objective of this study is to utilize the Nasometer to objectively assess velopharyngeal competence, specifically through the quantification of nasalance. Initial calibration of the nasometer was conducted on American adults. The objective of this study was to validate the use of the nasometer for the objective diagnosis of velopharyngeal insufficiency (VPI) in French children born with a total cleft lip and palate and to select relevant verbal stimuli for clinical practice., Material and Methods: The nasalance scores of 42 children aged 8 to 10 years old, born with a cleft lip and palate, were collected and compared with 50 control children. The scores were then analyzed in relation to 31 verbal stimuli from the French corpus created for this study (sentences and syllables). The most relevant threshold values were determined by receiver operating characteristic curves, which exhibited the highest sensitivity and specificity., Results: The results demonstrated statistically significant differences (p < 0.05) in the mean nasalance scores of the control and cleft groups for all verbal stimuli containing oral phonemes. Threshold values with good diagnostic accuracy were defined, and 15 verbal stimuli were selected for use in clinical practice., Conclusion: The nasalance threshold values defined in this study can be utilized for the objective diagnosis of velopharyngeal insufficiency (VPI) and the subsequent monitoring of French children aged 8 to 10 years old, born with a cleft lip and palate., Competing Interests: Declaration of competing interest The authors declare that they have no known competing financial interests or personal relationships that could have appeared to influence the work reported in this paper., (Copyright © 2024 Elsevier Masson SAS. All rights reserved.)

This study aimed to document the prevalence, severity, and risk factors of velopharyngeal dysfunction (VPD) in craniofacial microsomia (CFM) and to analyse differences in VPD-related speech characteristics between CFM patients without cleft lip and/or palate (CL/P), CFM patients with CL/P, and CL/P patients without CFM (control). A total of 223 patients with CFM were included, of whom 59 had a CL/P. Thirty-four CFM patients had VPD, including 20 with a CL/P. VPD was significantly more prevalent in CFM with CL/P than in CFM without CL/P (odds ratio (OR) 4.1, 95% confidence interval (CI) 1.9-8.7; P < 0.001). Multivariate logistic regression showed a significant association between CL/P and VPD in CFM patients (OR 7.4, 95% CI 2.1-26.3; P = 0.002). The presence of VPD was not associated with sex, the laterality or severity of CFM. Speech problems related to VPD appeared to be similar among the different groups (CFM without CL/P, CFM with CL/P, CL/P without CFM). As 15.2% of all CFM patients and 8.5% of CFM patients without CL/P had VPD, it is proposed that all patients with CFM, with or without CL/P, should be assessed by a speech and language therapist for the potential risk of VPD., Competing Interests: Competing interests None., (Copyright © 2024 The Authors. Published by Elsevier Inc. All rights reserved.)

This report chronicles the case of an adolescent female with cleft palate associated severe hypoplastic maxillary Class III deformity. Treatment involved anterior maxillary segmental distraction osteogenesis (AMSDO) in conjunction with pre-distraction and post-distraction orthodontics. Following pre-distraction orthodontics, AMSDO was performed using a customized Hyrax distractor assembly. Post-distraction orthodontics helped stabilize distraction outcomes and finalize occlusion. Post-treatment, midface deficiency and prognathic profile improved dramatically with establishment of acceptable interincisal relationship and well-balanced functionally interdigitated occlusion. Three-year follow-up showed excellent morphologic and functional stability. AMSDO is a viable modality that contributes to effective stomatological rehabilitation of patients with cleft maxillary hypoplasia., (Copyright © 2024. Published by Elsevier Masson SAS.)

Aim: 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., Methods: 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., Results: 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., Conclusion: 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., (© 2024 The Author(s). Acta Paediatrica published by John Wiley & Sons Ltd on behalf of Foundation Acta Paediatrica.)

Objective: Children with cleft lip and/or palate (CL/P) are at increased risk for Sleep Disordered Breathing (SDB), particularly Obstructive Sleep Apnea (OSA). At our institution, routine screening for SDB is performed using the Chevrin Pediatric Sleep Questionnaire (PSQ). This analysis is a practice audit looking at the outcomes of screening children with CL/P., Design/setting/patients/participants: A single-center, retrospective analysis was done of all non-syndromic patients with CL/P over the age of 36 months over a 4-year period. Children with known OSA were eliminated from analysis., Main Outcome Measures: Univariate logistic regression was used to assess predictors for SDB (PSQ score > 8) amongst various patient, disease, and treatment characteristics. Outcomes of those screened were tracked., Results: Of the 239 patients in the study cohort, 43 (18%) had positive PSQs. These subjects were more likely to have class III dental occlusion with maxillary retrusion (OR = 2.65, 95% CI: 1.2-5.8, p = 0.02). There were no differences amongst age, type of cleft, Veau classification, BMI, or history of pharyngeal surgery. One third of the group did not complete recommended testing. Twenty-five subjects with positive sleep screening underwent subsequent polysomnography and 21 (84%) had OSA., Conclusion: Routine screening reveals a significant proportion of patients with CL/P with symptoms suggestive of OSA. While several patients did not complete confirmatory testing, those who completed a PSG had a high rate of identification of OSA. After excluding children with known OSA, patients with SDB are also likely to have class III dental occlusion and maxillary retrusion., Competing Interests: Declaration of Conflicting InterestsThe authors declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.

Objective: Identification of patient factors influencing velopharyngeal function for speech following initial cleft palate repair., Design: A literature search of relevant databases from inception until 2018 was performed using medical subject headings and keywords related to cleft palate, palatoplasty and speech assessment. Following three stage screening data extraction was performed., Setting: Systematic review and meta-analysis of relevant literature., Patients/participants: Three hundred and eighty-three studies met the inclusion criteria, comprising data on 47 658 participants., Interventions: Individuals undergoing initial palatoplasty., Main Outcome Measures: Studies including participants undergoing initial cleft palate repair where the frequency of secondary speech surgery and/or velopharyngeal function for speech was recorded., Results: Patient factors reported included cleft phenotype (95% studies), biological sex (64%), syndrome diagnosis (44%), hearing loss (28%), developmental delay (16%), Robin Sequence (16%) and 22q11.2 microdeletion syndrome (11%). Meta-analysis provided strong evidence that rates of secondary surgery and velopharyngeal dysfunction varied according to cleft phenotype (Veau I best outcomes, Veau IV worst outcomes), Robin Sequence and syndrome diagnosis. There was no evidence that biological sex was associated with worse outcomes. Many studies were poor quality with minimal follow-up., Conclusions: Meta-analysis demonstrated the association of certain patient factors with speech outcome, however the quality of the evidence was low. Uniform, prospective, multi-centre documentation of preoperative characteristics and speech outcomes is required to characterise risk factors for post-palatoplasty velopharyngeal insufficiency for speech., Systematic Review Registration: Registered with PROSPERO CRD42017051624., Competing Interests: Declaration of Conflicting InterestsThe author(s) declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.

Background: Ectrodactyly-ectodermal dysplasia-cleft lip/palate (EEC) syndrome mainly affects ectodermal and mesodermal tissues. It is usually manifested as split hands and feet, ectodermal dysplasia, and orofacial clefting, along with other signs and symptoms. A multidisciplinary approach to treatment is required, in which dentists play an important role in identifying and treating various oral conditions that may be genetically linked to or may be the result of EEC syndrome., Case Presentation: The present case describes the oral condition of a young child suffering from EEC syndrome and presenting with peripheral giant cell granuloma (PGCG) in the mandibular anterior region. After obtaining a thorough medical and family history and a clinical examination, the lesion was surgically excised under local anesthesia. The patient was followed up at periodic intervals for the next twenty four months, during which no recurrence of the lesion was observed., Conclusion: This report highlights the role of a dentist in the management of the oral conditions of patients suffering from EEC syndrome., (© 2024. The Author(s).)

Oral causes of dysphagia in infancy may involve the lips, the tongue, or the palate. Whereas ankyloglossia is commonly diagnosed in infants with dysphagia, assessment of the need for surgical intervention may be less straightforward. Tongue size (macroglossia) may be associated with dysphagia as it may cause limitation of movement of the food or milk bolus by the lips or cheeks. Congenital conditions such as cleft lip and palate, micrognathia, or craniofacial microsomia may also be associated with dysphagia. Diagnosis and treatment of these conditions can be improved with the engagement of lactation and feeding experts as well as multidisciplinary craniofacial teams., Competing Interests: Disclosure The authors have no financial disclosures., (Copyright © 2024 Elsevier Inc. All rights reserved.)

Background: Submucous cleft palate (SMCP) is a congenital anomaly characterized by the presence of Calnan's triad. However, in clinical practice, it is common for individuals to exhibit one or two anatomical abnormalities within the triad. Furthermore, the definition of SMCP has been diverse and ambiguous in literature. Therefore, this study aimed to analyze the correlation between anatomical abnormalities and development of velopharyngeal insufficiency (VPI)., Methods: We conducted a retrospective analysis of 99 patients referred to our clinic for speech issues or anatomical abnormalities identified during routine oral examinations from January 2012 to June 2023. A single surgeon performed all physical examinations. We evaluated the presence of bony notch, zona pellucida, and bifid uvula, assigned a score to each abnormality, and analyzed their correlation with VPI. The correlation of each of the abnormalities with VPI development was examined, along with the relationship between the number of abnormalities and VPI., Results: Among the 99 patients, 27 were diagnosed with VPI. Only the bony notch had a significant correlation with VPI development. The incidence of VPI tended to increase with the presence of more anatomical abnormalities. VPI occurred in approximately 40% of patients exhibiting all three anatomical abnormalities., Conclusion: The study findings highlight the importance of meticulous intraoral examinations in patients with SMCP and careful monitoring of patients with a bony notch or two or more anatomical abnormalities., (Copyright © 2024 British Association of Plastic, Reconstructive and Aesthetic Surgeons. Published by Elsevier Ltd. All rights reserved.)

Purpose: To investigate whether a novel electropalatography (EPG) therapy, underpinned by usage-based phonology theory, can improve the accuracy of target speech sounds for school-aged children and adults with persistent speech sound disorder (SSD) secondary to cleft palate +/- lip., Method: Six consecutively treated participants (7-27 years) with long-standing speech disorders associated with cleft palate enrolled in a multiple baseline (ABA) within-participant case series. The usage-based EPG therapy technique involved high-volume production of words. Speech was assessed on three baselines prior to therapy, during weekly therapy, at completion of therapy, and 3 months post-therapy. Percent correct of target phonemes in untreated words and continuously connected speech were assessed through acoustic phonetic transcription. Intra- and inter-transcriber agreement was determined., Result: Large to medium treatment effect sizes were shown for all participants following therapy (15-33 sessions). Percentage of targets correct for untreated words improved from near 0% pre-therapy, to near 100% for most target sounds post-therapy. Generalisation of target sounds to spontaneous connected speech occurred for all participants and ranged from 78.95-100% ( M  = 90.66; SD = 10.14) 3 months post-therapy., Conclusion: Clinically significant speech change occurred for all participants following therapy. Response to the novel therapeutic technique is encouraging and further research is indicated.

Objective: To investigate the relationship between auditory-perceptual ratings of resonance and nasometry scores in children with cleft palate. Factors which may impact this relationship were examined including articulation, intelligibility, dysphonia, sex, and cleft-related diagnosis., Design: Retrospective, observational cohort study., Setting: Outpatient pediatric cranio-facial anomalies clinic., Patients: Four hundred patients <18 years of age identified with CP ± L, seen for auditory-perceptual and nasometry evaluations of hypernasality as well as assessments of articulation and voice., Main Outcome Measure: Relationship between auditory-perceptual ratings of resonance and nasometry scores., Results: Pearson's correlations indicated that auditory-perceptual resonance ratings and nasometry scores were significantly correlated across oral-sound stimuli on the picture-cued portion of the MacKay-Kummer SNAP-R Test (r values .69 to.72) and the zoo reading passage (r = .72). Linear regression indicated that intelligibility ( p  ≤ .001) and dysphonia ( p  = .009) significantly impacted the relationship between perceptual and objective assessments of resonance on the Zoo passage. Moderation analyses indicated that the relationship between auditory-perceptual and nasometry values weakened as severity of speech intelligibility increased ( P  < .001) and when children presented with moderate dysphonia ( p  ≤ .001). No significant impact of articulation testing or sex were observed., Conclusions: Speech intelligibility and dysphonia alter the relationship between auditory-perceptual and nasometry assessments of hypernasality in children with cleft palate. SLPs should be aware of potential sources of auditory-perceptual bias and shortcomings of the Nasometer when following patients with limited intelligibility or moderate dysphonia. Future study may identify the mechanisms by which intelligibility and dysphonia affect auditory-perceptual and nasometry evaluations., Competing Interests: Declaration of Conflicting InterestsThe author(s) declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.

Learning Objectives: After studying this article, the participant should be able to: (1) Describe the pathology of velopharyngeal dysfunction (VPD) as it relates to patients with a cleft palate. (2) Use the perceptual speech assessment and objective diagnostic tools to determine the presence or absence of VPD. (3) Describe the surgical options available for the treatment of patients with VPD. (4) Develop an evidence-based, customized treatment plan for VPD founded on objective considerations., Summary: To treat patients with cleft palate effectively, the surgeon must understand the diagnosis and surgical management of cleft-associated velopharyngeal dysfunction. The authors review diagnostic modalities including perceptual speech assessment, video nasendoscopy, fluoroscopy, magnetic resonance imaging, and nasometry. Surgical treatments including palatal lengthening with buccal myomucosal flaps, conversion Furlow palatoplasty, sphincter pharyngoplasty, and pharyngeal flap are discussed. Selection of an optimal surgical treatment is addressed., (Copyright © 2024 by the American Society of Plastic Surgeons.)

Objectives: Previous studies have also associated cleft palate with increased cholesteatoma risk. Despite this close relation, the incidence of cholesteatoma and associated otologic issues in patients with ectodermal dysplasia types highly associated with cleft palate (EDT-ACPs) has not been formally analyzed. This study provides insight to guide clinicians caring for patients with ED types associated with cleft palate., Methods: Individuals with TP63 disorders and Goltz syndrome/Focal Dermal Hypoplasia in the National Foundation for Ectodermal Dysplasia database were contacted for participation in an online REDCap survey from Sept-Dec '22. Descriptive statistics were generated using SAS JMP Pro 17 statistical software., Results: 65 individuals participated in the survey (response rate approx. 18 %). The median [IQR] age was 22 [14, 43], 41 (63 %) were female, and Ectrodactyly-Ectodermal Dysplasia-Cleft Lip/Palate Syndrome (EEC) was most common (n = 26, 40 %). We found that, among our respondents with a history of cleft palate, the incidence of cholesteatoma was 39 %. Among respondents without a history of cleft palate, the incidence of cholesteatoma was 13 % CONCLUSIONS: Otologic issues, such as cholesteatoma, can have permanent implications including hearing loss that can be minimized by early identification and treatment. The estimated incidence of cholesteatoma among our participants is far above the estimated incidence of cholesteatoma in the general population with and without a history of cleft palate, suggesting an independent contribution of EDT-ACPs to the risk of cholesteatoma., Competing Interests: Declaration of competing interest The authors declare that they have no known competing financial interests or personal relationships that could have appeared to influence the work reported in this paper., (Copyright © 2024 The Authors. Published by Elsevier B.V. All rights reserved.)

Objective: The present investigation examined how factors such as cleft type, age of primary palatal surgery, diagnosed syndromes, hearing problems, and malocclusions could predict persistent speech difficulties and the need for speech services in school-aged children with cleft palate., Methods: Participants included 100 school-aged children with cleft palate. Americleft speech protocol was used to assess the perceptual aspects of speech production. The logistic regression was performed to evaluate the impact of independent variables (IV) on the dependent variables (DV): intelligibility, posterior oral CSCs, audible nasal emission, hypernasality, anterior oral CSCs, and speech therapy required., Results: Sixty-five percent of the children were enrolled in (or had received) speech therapy. The logistic regression model shows a good fit to the data for the need for speech therapy (Hosmer and Lemeshow's χ 2 (8)=9.647,p=.291). No IVs were found to have a significant impact on the need for speech therapy. A diagnosed syndrome was associated with poorer intelligibility (Pulkstenis-Robinson's χ 2 (11)=7.120,p=.789). Children with diagnosed syndromes have about six times the odds of a higher hypernasality rating (Odds Ratio = 5.703) than others. The cleft type was significantly associated with audible nasal emission (Fisher'sexactp=.006). At the same time, malocclusion had a significant association with anterior oral CSCs (Fisher'sexactp=.005)., Conclusions: According to the latest data in the Cleft Registry and Audit Network Annual Report for the UK, the majority of children with cleft palate attain typical speech by age five. However, it is crucial to delve into the factors that may influence the continuation of speech disorders beyond this age. This understanding is vital for formulating intervention strategies aimed at mitigating the long-term effects of speech disorders as individuals grow older., Competing Interests: Declaration of competing interest There's no financial/personal interest or belief that could affect the objectivity of this paper., (Copyright © 2024 Elsevier B.V. All rights reserved.)

Objectives: Children with cleft palates often have comorbid eustachian tube dysfunction requiring pressure equalization tubes (PETs). PETs can relieve middle ear effusions that impede hearing. Ideal PET placement timing in this population is controversial. In this study, the audiologic exam passing rates of patients with cleft palate prior to and following PET insertion were assessed. Rates for patients receiving PETs at different ages were compared. It was hypothesized that earlier PET placement may benefit patients with additional months of improved hearing., Methods: A retrospective chart review was performed of patients with cleft palate between November 22, 2016 and November 22, 2021 at a tertiary center. Statistical analysis compared passing/normal audiologic exams in patients receiving PETs at different ages., Results: A total of 348 patients had cleft palate diagnoses, received PETs, and had adequate hearing data for inclusion. Those with PETs inserted at 3 months of age or less had an increase in percent of patients passing audiologic exams following versus prior to PET insertion of 13% (1.3 times improvement). Those receiving PETs between 7- and 12-months had the largest rate of improvement (42%) (2.4 times improvement); other groups had changes in passing rates between 31% and 40%. The rate of passing audiologic exams following PET insertion was high across all groups, ranging from 66% to 81%., Conclusion: This is one of the first studies exploring the timing of PET placement in this population and showed that patients receiving PETs at 3 months of age or younger passed subsequent audiologic exams at similar rates relative to those receiving PETs later in life., Level of Evidence: 3 Laryngoscope, 134:3391-3394, 2024., (© 2024 The American Laryngological, Rhinological and Otological Society, Inc.)

Background: Management of velopharyngeal insufficiency (VPI) in 22q11.2 deletion syndrome is challenging. The authors compared pharyngeal flap outcomes in children with 22q11.2 deletion syndrome to those with nonsyndromic cleft lip and palate (CLP) to assess risk of poor speech outcomes and negative sequelae., Methods: Children with 22q11.2 deletion syndrome or CLP treated with pharyngeal flap through a multidisciplinary VPI clinic between 2009 and 2020 were retrospectively reviewed. Preoperative and postoperative speech assessments, perioperative characteristics, and complications were identified., Results: Thirty-six children with 22q11.2 deletion syndrome and 40 with CLP were included. Age at surgery ( P = 0.121), preoperative velopharyngeal competence score ( P = 0.702), and preoperative resonance ( P = 0.999) were similar between groups. Pharyngeal flaps were wider ( P = 0.038) and length of stay longer in the 22q11.2 deletion syndrome group ( P = 0.031). On short-term follow-up 4 months after surgery, similar speech outcomes were seen between groups. At long-term follow-up greater than 12 months after surgery, 86.7% of 22q11.2 deletion syndrome versus 100% of CLP ( P = 0.122) children had improvement in velopharyngeal function; however, fewer children with 22q11.2 deletion syndrome (60.0%) achieved a completely "competent" velopharyngeal competence score compared with those with CLP (92.6%) ( P = 0.016). Nasal regurgitation improved for both groups, with a greater improvement in those with 22q11.2 deletion syndrome ( P = 0.026). Revision rate ( P = 0.609) and new-onset obstructive sleep apnea ( P = 0.999) were similar between groups., Conclusions: Children with 22q11.2 deletion syndrome have improved speech after pharyngeal flap, but they may be less likely to reach normal velopharyngeal function over the long term than those with CLP; however, negative sequelae do not differ. Improvement in nasal regurgitation is a uniquely positive outcome in this population., Clinical Question/level of Evidence: Risk, II., (Copyright © 2023 by the American Society of Plastic Surgeons.)

Purpose: Recent studies have suggested that children with an isolated cleft lip (CL) are more likely to develop middle ear disease and eustachian tube dysfunction (ETD) compared to the general population. This may be related to abnormal palatal musculature or an undiagnosed submucosal cleft palate (SMCP). We aim to determine the prevalence of SMCP in patients with CL who exhibit ETD., Materials and Methods: A retrospective chart review was performed for children with an isolated CL requiring tympanostomy tubes over a 20-year period at an academic tertiary care medical center. Demographic, clinical, and surgical data were collected., Results: Three hundred twelve patients had an isolated CL, and 29 (9.3 %) children required tympanostomy tubes. Of those, nine (31 %) were found to have a SMCP (7 males, 6 Caucasian). The average age at CL repair was 3.94 ± 1.03 months, and the average age at tympanostomy tube placement was 13.68 ± 13.8 months. All nine patients had chronic otitis media, with four having mild conductive hearing loss and three having moderate conductive hearing loss. The SMCP was diagnosed at the time of CL diagnosis (4), after CL diagnosis with the diagnosis of chronic otitis media/ETD (2) and after a diagnosis of chronic otitis media/ETD., Conclusion: Middle ear disease or eustachian tube dysfunction in a patient with an isolated cleft lip should raise suspicion for an accompanying undiagnosed SMCP., Competing Interests: Declaration of competing interest The authors report no conflicts of interest., (Copyright © 2024. Published by Elsevier Inc.)

Purpose: To evaluate how prosthetic management affects the otological and audiological state of infants with cleft lip and palate by preventing or treating otitis media (OM)., Materials and Methods: Thirty infants with cleft lip and palate (L/P) were assigned to three equal groups according to the age of prosthetic intervention; Group I: immediately after birth, Group II: 2 months old, Group III: 5 months old. Assessment of middle ear function by tympanometry and hearing quality by auditory brainstem response (ABR) under natural sleep was conducted before and after prosthetic treatment every month till 10 months of age. Data from the study groups were compared., Results: No statistically significant differences were found between Gp I and Gp II in the 2nd, 3rd, and 4th months for right and left ears (p > 0.05). In the 5th month, statistically significant differences between the three groups were found in tympanometry for right (p = 0.011) and left (p = 0.024) ears also, in ABR for right (p = 0.007) and left (p = 0.011) ears. Tympanometric readings starting from the 6th till the 10th month showed no statistically significant differences between the three groups (p >0.05). The final ABR outcomes of the 10th month indicated statistically significant differences between the three groups for both ears (p = 0.027)., Conclusions: Early prosthetic care could delay the development of OM, so it could potentially improve the otological and audiological state in infants with cleft L/P. However, prosthetic treatment may not be able to completely prevent or eliminate middle ear disorders., (© 2024 by the American College of Prosthodontists.)

Regardless of the underlying cause for speech impairment in speakers with cleft palate, a universal consequence of cleft palate is reduced speech intelligibility. Still, there is no standardised approach for measuring intelligibility for speakers with cleft speech. The current study aimed to determine the relationship between orthographic transcription (OT)-based measures, interval-scale ratings, and visual analog scale (VAS) ratings for perceptual judgements of intelligibility in speakers with cleft palate as judged by speech-language pathologists (SLPs). The speaker participants were six speakers with velopharyngeal insufficiency secondary to cleft palate. Four sets of sentences from the Hearing in Noise Test were recorded from each speaker. A total of 14 SLPs provided their intelligibility judgement on these speaker's recordings by word-by-word orthographic transcriptions, a visual analog scale (0-100), and a 5-point interval rating scale. A Spearman rank correlation test indicated a negative, strong correlation between OT-based measurements and VAS scores ( r  = -.94; p  = 0.01) and between OT-based measurements and interval rating scores ( r = -.77, p  = 0.01). A strong, positive correlation was found between scores obtained from VAS and interval rating scales ( r  = .83, p  = 0.05). The strong relationship between the objective measure of intelligibility (i.e. OT-based measure) and a subjective measure of intelligibility (i.e. VAS and interval scale) supports using a less time-consuming VAS as a substitute for orthographic transcription in measuring intelligibility in cleft palate speech.

Despite a satisfactory primary repair, velopharyngeal insufficiency (VPI) may be a sequel of soft palate clefts, resulting in hypernasality and phonation disorders. In order to increase the function of the pharyngeal flap during a secondary pharyngoplasty, we have developed a strip pharyngoplasty technique involving the middle constrictor muscle of the pharynx. This article describes the successive steps of the intervention and discuss its indications, advantages and limitations. Strip pharyngoplasty as a secondary functional surgery for persistent VPI after primary cleft palate repair is an attractive surgical procedure, particularly adapted to cases with defective muscle contraction by providing a muscle contingent in addition to the mucosal flap., Competing Interests: Declaration of Competing Interest The authors declare that they have no known competing financial interests or personal relationships that could have appeared to influence the work reported in this paper., (Copyright © 2023 Elsevier Masson SAS. All rights reserved.)

Background: Magnetic resonance imaging (MRI) is the only imaging modality capable of directly visualizing the levator veli palatini (LVP) muscles: the primary muscles responsible for velopharyngeal closure during speech. MRI has been used to describe normal anatomy and physiology of the velopharynx in research studies, but there is limited experience with use of MRI in the clinical evaluation of patients with velopharyngeal insufficiency (VPI)., Methods: MRI was used to evaluate the velopharyngeal mechanism in patients presenting for VPI management. The MRI followed a fully awake, nonsedated protocol with phonation sequences. Quantitative and qualitative measures of the velopharynx were obtained and compared with age- and sex-matched individuals with normal speech resonance., Results: MRI was completed successfully in 113 of 118 patients (96%). Compared with controls, patients with VPI after cleft palate repair had a shorter velum (P < 0.001), higher incidence of LVP discontinuity (P < 0.001), and shorter effective velar length (P < 0.001). Among patients with persistent VPI after pharyngeal flap placement, findings included a pharyngeal flap base located inferior to the palatal plane [11 of 15 (73%)], shorter velum (P < 0.001), and higher incidence of LVP discontinuity (P = 0.014). Patients presenting with noncleft VPI had a shorter (P = 0.004) and thinner velum (P < 0.001) and higher incidence of LVP discontinuity (P = 0.014)., Conclusions: MRI provides direct evidence of LVP muscle anomalies and quantitative evaluation of both velar length and velopharyngeal gap. This information is unavailable with traditional VPI imaging tools, suggesting that MRI may be a useful tool for selecting surgical procedures to address patient-specific anatomic differences., (Copyright © 2023 by the American Society of Plastic Surgeons.)

Competing Interests: Declaration of competing interest The authors declare that they have no known competing financial interests or personal relationships that could have appeared to influence the work reported in this paper.

Objective: To compare the effectiveness of short-term ventilation tubes compared to surveillance on conductive hearing loss in children with non-syndromic orofacial clefting involving the muscular palate., Introduction: Chronic otitis media with effusion is a common finding in children with cleft palate. The accepted convention is insertion of short-term ventilation tubes at the time of palate repair, but some centres are choosing conservative management. Each approach has its advantages but there is currently no consensus on the most appropriate management in children with non-syndromic cleft palate., Inclusion Criteria: Children <18 years with cleft lip and palate, or isolated cleft palate, not associated with a genetic syndrome, who have been diagnosed with chronic otitis media with effusion., Methods: A systematic search of MEDLINE, CINAHL, Embase and Scopus databases was conducted. Grey literature searches were conducted through Central Register of Controlled Trials, Clinicaltrials.gov and ProQuest. Two reviewers screened the studies, conducted critical appraisal, assessed the methodological quality, and extracted the data. Where possible, studies were pooled in statistical meta-analysis with heterogeneity being assessed using the standard Chi-squared and I 2 tests., Results: Four studies met the inclusion criteria but were of low quality with a moderate risk of bias. Only data on hearing thresholds could be pooled for analysis which found no statistically significant difference. Other outcomes were presented in narrative form. Certainty of evidence for all outcomes was deemed low to very low using GRADE criteria., Conclusions: No definitive conclusions can be drawn regarding most effective management at improving conductive hearing loss. Missing data and inconsistent reporting of outcomes limited capacity for pooled analysis., Competing Interests: Declaration of Conflicting InterestsThe author(s) declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.

Surgical intervention can contribute to the development of velopharyngeal insufficiency (VPI) leading to hypernasality and regurgitation. In this case, a patient with a history of bilateral buccal flaps used for her primary CP repair presented to clinic with hypernasality and VPI as assessed by speech exam and imaging. She underwent repeat bilateral buccal flap palatal lengthening with division of the pedicles 3 months later. Three months after her division, her hypernasality score improved from moderate to mild and her posterior gap decreased. This study concluded buccal flaps can be used a second time for patients needing palatal revisions for VPI., Competing Interests: Declaration of Conflicting InterestsThe author(s) declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.

Objective: This study analyzed the systemic and oral abnormalities in individuals with Kabuki syndrome (KS) that might be investigated to enhance the early diagnosis and treatment by a multidisciplinary team, minimizing the consequences to the individual's health., Study Design: Clinical examination was conducted on 15 individuals to investigate orodental alterations such as tooth abnormalities and cleft lip and/or palate, and the patient records were also reviewed to investigate systemic diseases such as cardiopathies, infectious and immunologic diseases, nephropathies, and delayed neuropsychomotor development., Results: All individuals with KS presented cleft lip and/or palate, 11 (73.34%) tooth abnormalities, 5 (33.34%) congenital cardiopathies, 12 (80%) infectious or immunologic diseases, 1 (6.67%) nephropathy, and 14 (93.34%) had an intellectual disability., Conclusion: Individuals with KS often have dental anomalies such as hypodontia, cleft or palate, and systemic disorders such as congenital heart disease and infectious diseases. Intellectual disability is present in most cases. These alterations should be investigated as early as possible to prevent the increase in morbidity in these individuals., Competing Interests: Declaration of interest None., (Copyright © 2023 Elsevier Inc. All rights reserved.)

The aim was to establish a specific and definite connection between non-syndromic orofacial cleft patients and associated congenital heart disease (CHD). Following PRISMA guidelines, selective databases were searched for data collection. Studies showing a definite association of CHD with orofacial cleft were included, and studies non-specific of the association of orofacial cleft with CHD were excluded. Data extraction criteria were study design, frequency of CHD in overall non-syndromic orofacial cleft and in specific cleft type, and most prevalent congenital cardiac anomaly. DerSimonian Laird random effects model was used to estimate the pooled proportion of CHD, along with corresponding 95% confidence intervals (CIs) for each measure. Publication bias was assessed using Fail-Safe N analysis and the Rosenthel approach. Of a total of 182 articles searched, only 30 studies were assessed. The overall pooled estimate of the proportion of CHD in total cleft lips/palates was 16% (95% CI: 13-19). The odds of developing CHD in cleft palates was 4.08 times more as compared to cleft lips with 95% CIs of 3.86-4.33, and 1.65 more as compared to cleft lips and palates both with 95% CI of 1.52-1.68. We affirm the upsurging prevalence of CHD in non-syndromic cleft children and vehemently propose that it is of utmost importance to inculcate it in practice and policy-making to screen all non-syndromic orofacial cleft children for congenital cardiac anomaly. This study was registered on PROSPERO (ID no. CRD42023391597) on February 24, 2023., (© 2024 Japanese Teratology Society.)

Brainstem evoked response audiometry (BERA) is the most established and recommended objective audiometric method for the clinical diagnosis of hearing impairment in high-risk infants. It is unclear whether infants with orofacial clefts meet the criteria for the high-risk group. This retrospective cohort study evaluated the need for diagnostic BERA in infants with cleft palate with or without cleft lip by assessing the predisposition to and diagnosis of congenital hearing impairment. Data from 122 patients treated at a single cleft centre were evaluated. BERA was conducted at the time of palate repair at 4-6 months of age. Clinical follow-up was analysed up to 4 years. The presence of a syndrome was examined as a risk factor for congenital hearing impairment. Among the 122 patients, four had congenital sensorineural or mixed hearing loss requiring hearing aids. All affected patients had syndromes in addition to the cleft. Most patients with elevated hearing thresholds had transient conductive hearing loss. Most suspected sensorineural hearing loss initially diagnosed was refuted. However, a higher incidence of sensorineural hearing loss was found in patients with syndromic clefts, supporting the diagnostic use of BERA with initial surgery only in patients with syndromic clefts., (Copyright © 2023 The Authors. Published by Elsevier Inc. All rights reserved.)

Objective: To present the design and methodology for an actively enrolling comparative effectiveness study of revision palatoplasty versus pharyngoplasty for the treatment of velopharyngeal insufficiency (VPI)., Design: Prospective observational multicenter study., Setting: Twelve hospitals across the United States and Canada., Participants: Individuals who are 3-23 years of age with a history of repaired cleft palate and a diagnosis of VPI, with a total enrollment target of 528 participants., Interventions: Revision palatoplasty and pharyngoplasty (either pharyngeal flap or sphincter pharyngoplasty), as selected for each participant by their treatment team., Main Outcome Measure(s): The primary outcome is resolution of hypernasality, defined as the absence of consistent hypernasality as determined by blinded perceptual assessment of a standard speech sample recorded twelve months after surgery. The secondary outcome is incidence of new onset obstructive sleep apnea. Statistical analyses will use propensity score matching to control for demographics, medical history, preoperative severity of hypernasality, and preoperative imaging findings., Results: Study recruitment began February 2021. As of September 2022, 148 participants are enrolled, and 78 have undergone VPI surgery. Enrollment is projected to continue into 2025. Collection of postoperative evaluations should be completed by the end of 2026, with dissemination of results soon thereafter., Conclusions: Patients with VPI following cleft palate repair are being actively enrolled at sites across the US and Canada into a prospective observational study evaluating surgical outcomes. This study will be the largest and most comprehensive study of VPI surgery outcomes to date., Competing Interests: Declaration of Conflicting InterestsThe author(s) declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.

Objectives: to audit the surgical management of infants born with non-syndromic cleft lip and palate (CLP) at an Australian cleft unit in a large tertiary paediatric hospital., Design: Retrospective cohort study., Setting: A tertiary Cleft centre., Patients: 193 infants born with non-syndromic CLP were referred to the centre and underwent primary repair of their CLP between 2009 and 2020.Main Outcome Measures: (1) The timing and surgical repairs performed; (2) the frequency of postoperative complications; (3) the frequency of secondary Cleft surgery; and (4) the total Cleft-related operations performed for infants born with CLP., Results: Four different surgical repair techniques were performed by six surgeons, and postoperative complications were uncommon (n = 14). Rates of oronasal fistula surgery (10.5% at five years of age; 14.3% at eight years of age) and velopharyngeal insufficiency surgery (8.7% at five years of age; 14.3% at eight years of age) were not significantly different across the surgical repair groups (p-value >0.05) and were comparable to international Cleft centres. Children underwent an average of four operative procedures in this audit period, including primary Cleft repair, ear, nose and throat surgery, and dental care. Surgery for managing Eustachian tube dysfunction was the most common surgical intervention following primary Cleft repair., Conclusions: Children born with non-syndromic CLP have a high early operative burden, with outcomes similar across the spectrum of techniques and surgeons., Competing Interests: Declaration of Conflicting InterestsThe author(s) declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.

Introduction: Cleft lip-palate is the most common craniofacial congenital anomaly. Patients with Cleft lip palate require treatment with a multidisciplinary approach from birth to enable independent feeding and physiological growth. In the past, the fabrication of therapeutic devices for a child with a cleft lip palate was executed through conventional dental impression materials , with the risk of suffocation. The use of a digital workflow minimizes impression-related risks and streamlines procedures., Methods: This study aims to propose a hybrid workflow that can combine the advantages of digital workflow with the advantages of analog workflow that can be applied daily by clinicians treating cleft lip-palate-affected patients., Results: The device created was immediately accepted by the patient allowing autonomous nutrition. Evaluation of the effectiveness of the device was done by body weight assessment every 15 days., Conclusion: The patient had growth comparable to that of a child born healthy., (© 2023 Special Care Dentistry Association and Wiley Periodicals LLC.)

Aarskog-Scott syndrome (AAS), also known as facio-digito-genital syndrome, is a rare heterogenous syndrome characterized by facial dysmorphism, brachydactyly, and genetic abnormalities. Although severe craniofacial abnormalities have been reported in AAS, little is known about speech and resonance issues in AAS. Specifically, published data to date have only indicated reports of hypernasality associated with a cleft palate in AAS. This case report provides clinical and anatomic information surrounding hypernasal speech in the absence of an overt cleft palate in a patient with AAS., Competing Interests: Declaration of Conflicting InterestsThe author(s) declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.

Objective: The objective of this study was to use data from Smile Train's global partner hospital network to identify patient characteristics that increase odds of fistula and postoperative speech outcomes., Design: Multi-institution, retrospective review of Smile Train Express database., Setting: 1110 Smile Train partner hospitals., Patients/participants: 2560 patients., Interventions: N/A., Main Outcome Measure(s): Fistula occurrence, nasal emission, audible nasal emission with amplification (through a straw or tube) only, nasal rustle/turbulence, consistent nasal emission, consistent nasal emission due to velopharyngeal dysfunction, rating of resonance, rating of intelligibility, recommendation for further velopharyngeal dysfunction assessment, and follow-up velopharyngeal dysfunction surgery., Results: The patients were 46.6% female and 27.5% underweight by WHO standards. Average age at palatoplasty was 24.7 ± 0.5 months and at speech assessment was 6.8 ± 0.1 years. Underweight patients had higher incidence of hypernasality and decreased speech intelligibility. Palatoplasty when under 6 months or over 18 months of age had higher rates of affected nasality, intelligibility, and fistula formation. The same findings were seen in Central/South American and African patients, in addition to increased velopharyngeal dysfunction and fistula surgery compared to Asian patients. Palatoplasty technique primarily involved one-stage midline repair., Conclusions: Age and nutrition status were significant predictors of speech outcomes and fistula occurrence following palatoplasty. Outcomes were also significantly impacted by location, demonstrating the need to cultivate longitudinal initiatives to reduce regional disparities. These results underscore the importance of Smile Train's continual expansion of accessible surgical intervention, nutritional support, and speech-language care., Competing Interests: Declaration of Conflicting InterestsThe author(s) declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.

Previous research on children with cleft lip and palate (CLP) reported unequivocal findings with regard to language skills, with the majority suggesting persistent difficulties in early childhood. While expressive language deficits improved with age, receptive language skills were consistently lower than peers. Further study investigating the long term and persistent impact of language deficits amongst school-aged children with CLP is warranted. This was a cross-sectional study, aimed to determine the language abilities and explore the associated risk factors in Malay speaking children with CLP in Malaysia. Fifty-two children with CLP aged 7- to 12-year-old participated in this study. Language skills were assessed using the Malay Preschool Language Assessment Tool and the adapted Subway-School-age Language & Assessment Measures. Findings revealed that 14 (26.92%) school-aged children with CLP demonstrated language deficits. Children with CLP performed significantly poorer in reading comprehension (p = 0.031) and narrative (p = 0.026) skills. It was found that the age significantly influenced total receptive language score (β = 0.421, p = 0.003) and total expressive language score (β = 0.477, p = 0.000). Findings suggested that children with CLP may continue to have persistent language deficits into their school-age years. Recommendations for regular monitoring of language performance especially for those from younger age groups is warranted to help maximize school attainment., Competing Interests: No, authors have no competing interests., (Copyright: © 2024 Hui et al. This is an open access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited.)

Objective: Patients with a cleft require structured procedures to achieve feasible treatment results. Since many treatment protocols coexist without being superior to one another, this study investigated the Saarland University Hospital treatment concept for patients with unilateral and bilateral clefts to evaluate its effects upon dental arch dimensions until the early mixed dentition., Material and Methods: Digitized plaster models were used for data collection. Records of 83 patients (Cleft n = 41 [UCLP n = 28, BCLP n = 13], Non-Cleft Control n = 42) comprised 249 casts. The evaluation included established procedures for measurements of edentulous and dentate jaws. Statistics included Shapiro-Wilk, Friedmann, Wilcoxon and Mann-Whitney-U-Tests for the casts. The level of significance was set at p < 0.05., Results: The cast analysis showed an approximation of arch dimensions towards those of age-matched patients without a cleft until early mixed dentition. The mean values of patients with and without cleft lip and palate were almost indistinguishable when compared in primary and/or early mixed dentition., Conclusions: The evaluated treatment concept leads to feasible outcomes regarding dental arches in patients with unilateral and bilateral clefts compared to an age-matched non-cleft control., Clinical Relevance: The evaluated treatment concept leads to favorable outcomes until early mixed dentition., (© 2024. The Author(s).)

Background: Palatal lengthening is becoming a first-line treatment choice for cleft patients with velopharyngeal insufficiency (VPI). As cleft palate-related surgical outcomes are age dependent, speech outcomes may be similarly affected by patient age at the time of treatment. The primary goal of this study is to determine whether there are age-related speech outcome differences when double opposing buccinator myomucosal flaps are used as part of a palatal lengthening protocol and whether these outcome differences preclude utilization of this technique for specific patient age groups., Methods: A retrospective study was performed on consecutive nonsyndromic patients with VPI who underwent treatment using double opposing buccinator myomucosal flaps at our hospital between 2014 and 2021. Patients who completed the 15-month follow-up were stratified by age. Group A aged between 2 and 7 years (n = 14), group B aged 8 and 18 years (n = 23), and group C aged older than 18 years (n = 25) were included. Standardized perceptual speech evaluations and nasopharyngoscopy were performed. Hypernasality, soft palate mobility, and lateral palatal wall mobility were assessed both preoperatively and at a 15-month postoperative interval. Complications were also recorded. The χ2 test was used for statistical comparison., Results: All of the age-stratified patient groups in this study showed significant improvement in hypernasality, soft palate mobility, and lateral wall mobility (P < 0.01), with no statistically significant differences between the different patient age groups. Overall speech success was achieved in 69.4% of patients. Patients in group A achieved 78.6% speech success, patients in group B achieved 78.3% speech success, and patients in group C achieved 56% speech success, with no statistically significant differences being shown regarding speech success between the different patient age groups (P > 0.05)., Conclusions: Regardless of age, palatal lengthening via double opposing buccinator myomucosal flaps similarly improves speech outcomes., Competing Interests: Conflicts of interest and sources of funding: none declared., (Copyright © 2024 Wolters Kluwer Health, Inc. All rights reserved.)