Cleft Palate-Craniofacial Journal最新文献

Objective: To develop consensus-based algorithms for genetic testing in patients with common craniofacial conditions.

Design: An online collaborative consisting of online meetings, independent work, and feedback across groups. Setting/Participants: A collaborative of genetics and pediatrics providers from three regional craniofacial centers (four institutions).

Methods: Collaborative participants agreed upon a shared initial framework, developed algorithms independently, and presented/tested the algorithms with a national audience. Algorithms were modified based on consensus feedback.

Results: The collaborative group developed final algorithms for genetic testing in patients with orofacial cleft, branchial arch conditions, and craniosynostosis.

Conclusions: Timely and accurate diagnosis of genetic conditions can support medical management recommendations that result in safer surgical interventions. Algorithms can help guide best-practices for testing, particularly in institutions without easy access to genetics providers.

Objective: To assess the effectiveness of an interactive iPad-based educational module (cleft iBook) in enhancing parent/caregiver education related to cleft lip and/or cleft palate (CL/P) care.

Design: A prospective study involving pre- and post-intervention surveys.

Setting: The study was conducted at a craniofacial clinic in a dedicated children's hospital specifically during initial consultations for CL/P care.

Participants: Thirty-two participants (parents/legal guardians/caregivers) ≥18 years of age, English and/or Spanish-speaking, and attending with a child with CL/P.

Interventions: Participants were provided with iPads and engaged with the interactive iPad-based educational module. Pre- and post-intervention surveys were administered.

Main outcome measure(s): The survey assessed the understanding of a CL/P diagnosis and management and usability of the iBook. Survey responses were graded on a 5-point Likert scale. Total scores for pre- and post-intervention surveys were compared.

Results: Utilizing the cleft iBook module before consultation significantly enhanced comprehension in multiple domains: prenatal development of CL/P, dento-facial molding, surgical techniques, steps after the initial consultation, postoperative care, and the necessity of long-term care for affected children (P < .01). Cumulative survey scores increased by 10.2 points, reflecting significantly improved responses regarding overall comprehension (P < .001).

Conclusions: The interactive, iPad-based cleft educational module emerges as a viable, digital strategy for providing education and empowerment to parents and caregivers navigating the challenges of caring for a child with CL/P. The cleft iBook serves as a readily accessible resource, fostering connections among the child, caregivers, and care team.

Objective: Development of an AI tool to assess velopharyngeal competence (VPC) in children with cleft palate, with/without cleft lip.

Design: Innovation of an AI tool using retrospective audio recordings and assessments of VPC.

Setting: Two datasets were used. The first, named the SR dataset, included data from follow-up visits to Skåne University Hospital, Sweden. The second, named the SC + IC dataset, was a combined dataset (SC + IC dataset) with data from the Scandcleft randomized trials across five countries and an intercenter study performed at six Swedish CL/P centers.

Participants: SR dataset included 153 recordings from 162 children, and SC + IC dataset included 308 recordings from 399 children. All recordings were from ages 5 or 10, with corresponding VPC assessments.

Interventions: Development of two networks, a convolutional neural network (CNN) and a pre-trained CNN (VGGish). After initial testing using the SR dataset, the networks were re-tested using the SC + IC dataset and modified to improve performance.

Main outcome measures: Accuracy of the networks' VPC scores, with speech and language pathologistś scores seen as the true values. A three-point scale was used for VPC assessments.

Results: VGGish outperformed CNN, achieving 57.1% accuracy compared to 39.8%. Minor adjustments in data pre-processing and network characteristics improved accuracies.

Conclusions: Network accuracies were too low for the networks to be useful alternatives for VPC assessment in clinical practice. Suggestions for future research with regards to study design and dataset optimization were discussed.

Objective: The influence of different surgical techniques on maxillary growth remains unclear. This study investigates the long-term impact of Furlow double-opposing Z-plasty versus straight-line repair (SLR) techniques on midface growth and subsequent orthognathic surgery.

Design: Retrospective cohort study.

Setting: Tertiary children's hospital.

Patients/participants: This study evaluated patients who underwent primary palatoplasty with Furlow or SLR techniques from 1994-2023. Patients were >14 years old at their most recent follow-up.

Interventions: No interventions were performed.

Main outcome measure(s): Primary outcomes were orthognathic surgery and orthognathic surgery recommendation rates to correct midface hypoplasia (MFH). Cephalometrics at the time of orthognathic surgery recommendation were traced to validate MFH.

Results: In total, 1857 patients underwent palatoplasty, of which 335 met inclusion criteria (49 SLR, 286 Furlow). Average age at last follow-up was 18.5±2.6 years. Patients who underwent Furlow versus SLR showed no significant difference in orthognathic surgery rates (p=0.428) or recommendation for orthognathic surgery rates (p=0.900). Patients recommended to undergo orthognathic surgery had more negative ANB angles (p<0.001) and smaller SNA angles (p<0.001) than patients not recommended for orthognathic surgery, demonstrating maxillary hypoplasia. Upon multivariate regression, patients with Veau III and IV clefts had an increased need for orthognathic surgery, p=0.047 and p=0.008, respectively.

Conclusions: Our findings suggest that higher cleft severity contributes to future orthognathic surgery. However, palatoplasty technique did not influence orthognathic surgery rates. Our results provide valuable data when surgeons are considering the impact of palatoplasty technique on sagittal growth restriction.

Fourteen Speech and Language Therapists/Pathologists (SLT/Ps) from 13 countries across 5 continents made up the International Confederation of Cleft Lip and Palate and Related Craniofacial Anomalies (ICCPCA) CLEFT 2022 Speech Taskforce. Following a group consensus activity led by an external facilitator using Lightning Design Thinking principles, "task-shifting" was identified as the topic for this Taskforce. Absence and scarcity of SLT/Ps in many parts of the world have led to non-SLT/Ps delivering speech and language therapy services to individuals with cleft lip +/- palate. This narrative is the first known attempt to develop a framework, describing the different types of providers and their scope of practice.

Objective: While previous literature has investigated the psychosocial impact and aesthetic satisfaction associated with post-operative scarring for certain pediatric craniofacial conditions, the impact of the scar burden resulting from craniosynostosis surgery has not been adequately studied.

Participants: SCAR-Q was shared with patients ages 8 and older. Thirty-two complete patient responses were recorded.

Interventions: SCAR-Q is a PROM that consists of three independent scales - appearance, symptoms, and psychosocial impact - associated with a scar.

Main outcome measures: Mann-Whitney U, linear regression, and Pearson correlation tests were used to evaluate associations between the scales, in addition to patient characteristics such as sex and suture involvement.

Results: Mean ages at time of surgery and survey completion were 9.65 ± 10.10 months and 12.10 ± 3.92 years, respectively. Mean scale scores were 81.5 ± 17.9 for appearance, 86.8 ± 12.4 for symptoms, and 79.3 ± 25.7 for psychosocial impact. Higher patient dissatisfaction with scar appearance correlated with more scar-related symptoms (r = 0.389; p = 0.028) and a greater psychosocial impact (r = 0.725; p < 0.001). SCAR-Q scales did not significantly correlate with age at surgery, age at survey completion, type of synostosis, or type of surgery; however, female patients reported lower mean appearance (65.4 vs. 86.0; p = 0.012) and psychosocial impact (57.3 vs. 85.5; p = 0.010) scores when compared to their male counterparts.

Conclusions: It is vital that surgeons discuss patients' aesthetic satisfaction following craniosynostosis surgery in order to appropriately address and limit deleterious, long-term physical and psychosocial outcomes.

Introduction: The levator veli palatini (LVP) muscle has two segments with distinct roles in velopharyngeal function. Previous research suggests longer extravelar segments with shorter intravelar segments may lead to a more advantageous mechanism for velopharyngeal closure. The purpose of this study was to examine whether the distribution of the LVP intravelar and extravelar segments differs between children with cleft palate with and without VPI and controls.

Methods: The study included 97 children: 37 with cleft palate +/- lip with VPI, 37 controls, and 19 with cleft palate with normal resonance. Measures included mean LVP length, mean extravelar LVP length, and intravelar LVP length.

Results: Overall mean LVP length was similar (P = .267) between controls and children with cleft palate (with and without VPI). However, there was a significant difference (P < .001) between group for both intravelar and extravelar LVP lengths: the intravelar segment was significantly longer in those with VPI compared to controls and children with cleft palate and normal resonance; and the extravelar segment was significantly shorter in those with VPI compared to controls and children with cleft palate and normal resonance.

Conclusions: Results from this study demonstrate a significant difference between the distribution of the functional segments of the LVP among children with VPI, with a more disadvantageous distribution of the muscle segments among those with VPI.

Background: Orthognathic surgery, addressing skeletal and dental irregularities, is pivotal for patients suffering from malocclusion, facial asymmetry, and related disorders. With the internet becoming a primary source of health information, the accuracy, quality, and reliability of online Arabic-language patient-centered information on orthognathic surgery necessitate thorough evaluation. This study aimed to assess the quality, reliability, and readability of Arabic online resources about orthognathic surgery to ascertain their potential as reliable patient education tools.

Methods: An infodemiological approach was employed, analyzing the top 100 websites from Google, Yahoo, and Bing based on specific Arabic search terms related to orthognathic surgery. Websites were evaluated for affiliation, content specialization, and presentation and excluded based on predetermined criteria to ensure relevance and focus. Quality assessment was conducted using the DISCERN instrument, Journal of the American Medical Association (JAMA) benchmarks, and Health On the Net code evaluation. Readability was assessed using the Flesch-Kincaid Grade Level, Simplified Measure of Gobbledygook, and Flesch Reading Ease scales.

Results: Of 600 initial websites, 96 met the inclusion criteria. Most were affiliated with universities or medical centers (53.1%), followed by commercial (28.1%), and non-profit organizations (16.7%). The majority of websites (96.9%) were only partially related to orthognathic surgery. Content analysis revealed a moderate quality of information, with a median overall DISCERN quality rating of 3 out of 5. The JAMA benchmarks showed a lack of comprehensive adherence, with currency being the most achieved criterion. Readability assessments indicated the content was generally accessible yet highlighted the need for Arabic-specific readability evaluation tools.

Conclusion: The study reveals a critical need for enhancing Arabic online resources on orthognathic surgery. Despite the available content's moderate quality and acceptable readability, there is a substantial gap in providing comprehensive, patient-centered, and easily understandable information. Future efforts should focus on developing high-quality, reliable, and readable online resources to aid Arabic-speaking patients in making informed decisions about orthognathic surgery.

Objective: Prospectively validate the accuracy of smartphone-based digital cranial measurements for the diagnosis and treatment of deformational plagiocephaly and/or brachycephaly (DPB), compared with calipers used in the standard of care.

Design/methods: Bird's-eye-view head photos were captured via smartphone, and their heads were measured with hand calipers by an expert user. CI/CVAI/CVA were calculated from photos and caliper measurements, and from 3D photogrammetry of the head as ground truth. Digital and caliper measurements were compared against 3D-based ground truth using mean absolute error, Spearman correlation coefficient, and Bland-Altman method. Statistical significance between methods was assessed using Wilcoxon Rank-Sum test.

Participants: 71 infants aged 2-11 months (20 female, 51 male) with DPB.

Results: The mean absolute errors for CI, CVAI, CVA were 1.63 ± 1.44, 1.45 ± 1.29, 2.38 ± 1.86 mm for smartphone, and 2.60 ± 1.96, 1.43 ± 1.22, 2.04 ± 1.81 mm for calipers, respectively. The correlation coefficients for CI, CVAI, CVA between smartphone and ground truth were 0.90, 0.94, 0.80 (p < 0.001), and 0.87, 0.93, 0.84 (p < 0.001) between calipers and ground truth, respectively. Bland-Altman results were (0.08, [-4.18, 4.34]), (-0.05, [-3.85, 3.76]), (-0.82, [-6.52, 4.87]) for smartphone, and (1.41, [-4.34, 7.15]), (0.28, [-3.37, 3.94]), (0.16, [-5.18, 5.49]) for caliper measurements respectively. Digital and caliper measurements were similar (p = 0.12) except for CI, where digital measurements were more accurate (p = 0.04).

Conclusion: Smartphone-based cranial measurements have very high correlation with 3D-based ground truth, and they are comparable or superior to caliper measurements. Digital measurements can be performed in pediatric offices or from home to help with the early detection and treatment of DPB.