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Objective: The objective of this paper is to conduct a systematic review that summarizes the cost-effectiveness of cleft lip and/or palate (CL/P) care in low- and middle-income countries (LMICs) based on existing literature., Design: We searched eleven electronic databases for articles from January 1, 2000 to December 29, 2020. This study is registered in PROSPERO (CRD42020148402). Two reviewers independently conducted primary and secondary screening, and data extraction., Setting: All CL/P cost-effectiveness analyses in LMIC settings., Patients, Participants: In total, 2883 citations were screened. Eleven articles encompassing 1,001,675 patients from 86 LMICs were included., Main Outcome Measures: We used cost-effectiveness thresholds of 1% to 51% of a country's gross domestic product per capita (GDP/capita), a conservative threshold recommended for LMICs. Quality appraisal was conducted using the Joanna Briggs Institute (JBI) checklist., Results: Primary CL/P repair was cost-effective at the threshold of 51% of a country's GDP/capita across all studies. However, only 1 study met at least 70% of the JBI criteria. There is a need for context-specific cost and health outcome data for primary CL/P repair, complications, and existing multidisciplinary management in LMICs., Conclusions: Existing economic evaluations suggest primary CL/P repair is cost-effective, however context-specific local data will make future cost-effectiveness analyses more relevant to local decision-makers and lead to better-informed resource allocation decisions in LMICs., 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: Describe the first hybrid global simulation-based comprehensive cleft care workshop, evaluate impact on participants, and compare experiences based on in-person versus virtual attendance., Design: Cross-sectional survey-based evaluation., Setting: International comprehensive cleft care workshop., Participants: Total of 489 participants., Interventions: Three-day simulation-based hybrid comprehensive cleft care workshop., Main Outcome Measures: Participant demographic data, perceived barriers and interventions needed for global comprehensive cleft care delivery, participant workshop satisfaction, and perceived short-term impact on practice stratified by in-person versus virtual attendance., Results: The workshop included 489 participants from 5 continents. The response rate was 39.9%. Participants perceived financial factors (30.3%) the most significant barrier and improvement in training (39.8%) as the most important intervention to overcome barriers facing cleft care delivery in low to middle-income countries. All participants reported a high level of satisfaction with the workshop and a strong positive perceived short-term impact on their practice. Importantly, while this was true for both in-person and virtual attendees, in-person attendees reported a significantly higher satisfaction with the workshop (28.63 ± 3.08 vs 27.63 ± 3.93; P  = .04) and perceived impact on their clinical practice (22.37 ± 3.42 vs 21.02 ± 3.45 P  = .01)., Conclusion: Hybrid simulation-based educational comprehensive cleft care workshops are overall well received by participants and have a positive perceived impact on their clinical practices. In-person attendance is associated with significantly higher satisfaction and perceived impact on practice. Considering that financial and health constraints may limit live meeting attendance, future efforts will focus on making in-person and virtual attendance more comparable.

Importance: In Ethiopia, more than 70% of infants with cleft lip and/or palate (CL/P) lack access to surgery. Infants who are untreated can experience severe malnutrition and extreme social stigma resulting in abandonment. Utilities are standardized measures of health-related quality of life (HRQOL) that inform health care resource allocation. However, CL/P utilities are missing from low- and middle-income countries (LMICs)., Objective: To elicit utilities for untreated and surgically treated children with CL/P with consideration for social determinants of health from patient-proxy and societal participants., Design, Setting, and Participants: This cross-sectional study used patient proxies and societal participants in Addis Ababa, Ethiopia, from July 1, 2019, to January 30, 2020. Eligible patient proxies were caregivers of children younger than 18 years with nonsyndromic CL/P who were untreated or received surgery. Proxies were necessary as most patients were 0 to 4 years old and cannot reliably self-report. Eligible societal participants were 18 years and older with no family history of CL/P., Exposures: Surgical treatment and social determinants of health., Main Outcomes and Measures: Participants measured utilities using the visual analog scale (VAS), time trade-off (TTO), and standard gamble (SG)., Results: In this study, 312 patient proxies and 135 societal participants were recruited. Mean (SD) utilities for untreated CL/P ranged from 0.57 (0.23) to 0.70 (0.22) from patient proxies and from 0.35 (0.21) to 0.8 (0.23) from societal participants, depending on utility instrument and cleft type. Surgical treatment was associated with a better HRQOL from the patient-proxy perspective (VAS, 0.17; 95% CI, 0.09 to 0.26; TTO, 0.15; 95% CI, 0.05 to 0.25) from the societal perspective (VAS, 0.21; 95% CI, 0.16 to 0.26; TTO, 0.17; 95% CI, 0.13 to 0.22; SG, 0.11; 95% CI, 0.06 to 0.15). Social determinants of health that were associated with patient-proxy utilities were income above the national mean (VAS, 0.10; 95% CI, 0.02 to 0.17; TTO, 0.11; 95% CI, 0.02 to 0.20), and religion (Christian vs other: TTO, -0.10; 95% CI, -0.17 to -0.03). From the societal perspective, the association between treatment and utilities was smaller in females compared with males (TTO, -0.05; 95% CI, -0.10 to -0.01)., Conclusions and Relevance: The findings of this study suggest that CL/P disease severity and surgical impact in Ethiopia were undervalued by previous estimates from high-income countries and were associated with social determinants of health. Utility studies from participants from LMICs are feasible and necessary for representing HRQOL in LMICs and addressing health inequalities.

Background: There is evidence that patient-specific plate fixation for Le Fort I osteotomies (LFI) is more stable than traditional plate fixation. The purpose of this study was to evaluate stability of LFI in patients with cleft lip and palate and determine stability differences between patient-specific and stock plates., Methods: Consecutive patients with cleft lip and palate who underwent isolated LFI by one surgeon (B.L.P.) between 2016 and 2021 were included. The predictor variable was type of plate used for fixation (patient-specific or stock). The outcome variable was magnitude of relapse in the vertical (nasion to A point) and horizontal planes (basion to A point) at 1 year after LFI using 3-dimensional cone beam computed tomography. Statistical analysis included independent samples t , Mann-Whitney U , Fisher exact, and chi-square tests. Values of P < 0.05 were significant., Results: The sample included 63 subjects; 23 in the patient-specific group (36.5%) and 40 in the stock group (63.5%). Groups were comparable by sex, race, age at surgery, cleft type, presence of pharyngeal flap, and magnitude of horizontal movement ( P > 0.136 for all). Subjects who underwent patient-specific plate fixation were less likely to have greater than or equal to 1-mm change at 1 year in the horizontal (4.3% versus 50.0%; P < 0.001) and vertical planes (4.3% versus 65.0%; P < 0.001) compared with stock plates. For patients who had greater than 10-mm horizontal advancement, the patient-specific plates had significantly less relapse (patient-specific plates, 0.105 ± 0.317 mm; stock plates, 1.888 ± 1.125 mm; P = 0.003)., Conclusion: Patient-specific plate fixation of LFI is more stable and demonstrates less relapse after 1 year than stock plates., Clinical Question/level of Evidence: Therapeutic, III., (Copyright © 2024 by the American Society of Plastic Surgeons.)

Background: The CLEFT-Q, a questionnaire developed and validated specifically for cleft patients, contains seven appearance scales. The International Consortium of Health Outcomes Measurement (ICHOM) has incorporated only some CLEFT-Q appearance scales in the Standard Set to minimize burden. This study evaluates which appearance scales provide the most meaningful information in the different cleft types at specific ages, for the most efficient cleft appearance outcome assessment., Methods: Within this international multicenter study, outcomes of the seven appearance scales were collected, either as part of the ICHOM Standard Set, or as part of the field test study performed to validate the CLEFT-Q. Analyses were performed in separate age groups and cleft types, and involved univariate regression analyses, trend analyses, t tests, correlations, and floor and ceiling effects., Results: A total of 3116 patients were included. Scores for most appearance scales showed a downward trend by age group, with the exception of the Teeth and Jaw scales. In all cleft types, several scales correlated strongly with each other. No floor effects were observed, but ceiling effects were found in several scales in different age groups, most often in the CLEFT-Q Jaw scale., Conclusions: A proposition for the most meaningful and efficient appearance outcome assessment in cleft patients is made. It was composed so that recommendations are of value for different cleft protocols and initiatives. Suggestions for the use of scales in the ICHOM Standard Set at different ages are given, and also from a clinical perspective. Use of the CLEFT-Q Scar, Lips, and Nose scales will provide additional relevant information., (Copyright © 2023 by the American Society of Plastic Surgeons.)

Background: Measuring the patient perspective is important in evaluating outcomes of cleft care. Understanding how treatment outcomes vary depending on cleft type may allow for better planning of treatments, setting of expectations, and more accurate benchmarking efforts. The CLEFT-Q is a patient-reported outcome measure for patients with cleft lip and/or palate., Methods: The 12 CLEFT-Q scales measuring appearance (i.e., face, nose, nostrils, lips, cleft lip scar, teeth, and jaws), function (i.e., speech), and health-related quality of life (i.e., psychological, school, social, and speech-related distress) were field tested in a cross-sectional study in 30 centers in 12 countries. Patients with cleft lip and/or cleft palate aged 8 to 29 years were recruited from clinical settings. Differences in CLEFT-Q scores by cleft subtypes were evaluated using one-way analysis of variance or Kruskal-Wallis H tests, with Tukey or Dunn procedure with Bonferroni corrections post hoc analyses, respectively. Scores are presented using radar charts to visualize all outcomes simultaneously., Results: The field test included 2434 patients. Scores on all CLEFT-Q scales varied significantly with cleft subtype. Patients with unilateral or bilateral cleft lip and/or palate scored lower on all appearance scales compared with patients with cleft palate or unilateral incomplete cleft lip. Scores on the speech function and speech-related distress scales decreased with each progressive group in the Veau classification. Patients with complete bilateral cleft lip and palate scored lowest on the social, school, and psychological scales., Conclusions: Patient-reported outcomes measured with the CLEFT-Q vary significantly with cleft type. Visualizing multiple outcomes simultaneously with radar charts allows for an understanding of a patient's overall status in a single graph.

Objective: To test the feasibility of implementing a high-fidelity cleft palate simulator during a workshop in Santiago, Chile, using a novel video endoscope to assess technical performance., Design: Sixteen cleft surgeons from South America participated in a 2-day cleft training workshop. All 16 participants performed a simulated repair, and 13 of them performed a second simulated repair. The repairs were recorded using a low-cost video camera and a newly designed camera mouth retractor attachment. Twenty-nine videos were assessed by 3 cleft surgeons using a previously developed cleft palate objective structured assessment of technical skill (CLOSATS with embedded overall score assessment) and global rating scale. The reliability of the ratings and technical performance in relation to minimum acceptable scores and previous experience was assessed., Results: The video setup provided acceptable recording quality for the purpose of assessment. Average intraclass correlation coefficient for the CLOSATS, global, and overall performance score was 0.69, 0.75, and 0.82, respectively. None of the novice surgeons passed the CLOSATS and global score for both sessions. One participant in the intermediate group, and 2 participants in the advanced group passed the CLOSATS and global score for both sessions. There were highly experienced participants who failed to pass the CLOSATS and global score for both sessions., Conclusions: The cleft palate simulator can be practically implemented with video-recording capability to assess performance in cleft palate repair. This technology may be of assistance in assessing surgical competence in cleft palate repair.

Background: The CLEFT-Q is a validated instrument designed to elicit patient-reported outcomes among people affected by cleft lip and/or palate. However, it has not been reported how use of CLEFT-Q data alters routine cleft care. This study analyzed the impact of CLEFT-Q data integration on patient care and clinical decision-making., Methods: Patients were sequentially, prospectively evaluated during scheduled cleft team visits. The CLEFT-Q was completed before the clinic encounter, but results were initially masked from the surgeon and family. In the encounter, a study observer characterized patients' verbalized attitudes across 7 specific domains of appearance and function, and the provisional assessment and plan was noted. CLEFT-Q data were then introduced into the clinical encounter and discussed. Discordance between patients' initially verbalized attitudes and their self-reported scores on the CLEFT-Q was documented along with any resultant modifications to their care plan., Results: Seventy patient visits were observed; the mean patient age was 12.7 years (range, 8 to 19 years). Forty-one patients (59%) had cleft lip and palate/alveolus and 29 (41%) had isolated cleft palate. Discordance was observed in 36% of visits and in 9.2% of specific domains assessed. Highest discordance rates were observed in domains of psychosocial function (12.5%), speech function/distress (11.6%), and lips/lip scar appearance (11.6%). No age group or sex was associated with increased discordance. Integration of CLEFT-Q results altered the assessment and plan in 11 visits (16%)., Conclusion: The CLEFT-Q provides clinically relevant insight into patient perspectives that are not captured by routine interview and examination alone, and regularly leads to a change in the management plan., (Copyright © 2023 by the American Society of Plastic Surgeons.)

Purpose: This study aimed to evaluate the efficiency of the porcine tongue for palatoplasty simulation compared to 3D-printed simulators and their surgical education role., Materials and Methods: A total of 18 senior cleft surgeons participated in a palatoplasty simulation-based workshop conducted using porcine tongue simulators and 3D-printed simulators. This workshop consisted of a didactic session followed by a hands-on simulation session. Each participant independently used both simulators to perform Furlow double-opposing Z-plasty, which was assessed and scored by senior cleft surgeons using a scoring system including organizational flexibility and ductility, anatomical design simulation, proper incision, proper suturing, and convenience of operation. A paired t test was used for data statistical analysis and a P value < 0.05 was regarded as a statistically significant difference., Results: All senior cleft surgeons strongly agreed that the simulation-based workshop was a valuable learning experience, and both simulators were useful and easy to manipulate (P = 1.00). The results of this comparative study showed that a porcine tongue palatoplasty simulator had an effectively significant difference in terms of organizational flexibility and ductility (P = 0.04), and suturing was better than the 3D-printed palatoplasty simulator (P < 0.01). There were no significant differences between the simulators regarding anatomical design simulation (P = 0.76) and incision simulation (P = 0.65)., Conclusion: Both porcine tongue simulator and 3D-printed simulator have their unique strengths in surgical education for palatoplasty. Thus, the combined use of a porcine tongue and a 3D-printed cleft palate simulators are efficient as an educational model to practice Furlow double-opposing Z- palatoplasty. The porcine tongue simulators are superior in terms of organizational flexibility, ductility, and suturing simulators, while with the 3D-printed simulator, various palatoplasty techniques can be repeatedly practiced with better-simulated face and oral cavity., (© 2023. BioMed Central Ltd., part of Springer Nature.)

Background: Cleft palate repair is a challenging procedure for cleft surgeons to teach. A novel high-fidelity cleft palate simulator has been described for surgeon training. This study evaluates the simulator's effect on surgeon procedural confidence and palatoplasty knowledge among learners., Methods: Plastic surgery trainees attended a palatoplasty workshop consisting of a didactic session on cleft palate anatomy and repair followed by a simulation session. Participants completed a procedural confidence questionnaire and palatoplasty knowledge test immediately before and after the workshop., Results: All participants reported significantly higher procedural confidence following the workshop (p < 0.05). Those with cleft palate surgery experience had higher procedural confidence before (p < 0.001) and after (p < 0.001) the session. Palatoplasty knowledge test scores increased in 90 percent of participants. The mean baseline test score was 28 ± 10.89 percent and 43 ± 18.86 percent following the workshop. Those with prior cleft palate experience did not have higher mean baseline test scores than those with no experience (30 percent versus 28 percent; p > 0.05), but did have significantly higher scores after the workshop (61 percent versus 35 percent; p < 0.05). All trainees strongly agreed or agreed that the simulator should be integrated into training and they would use it again., Conclusions: This study demonstrates the effective use of a novel cleft palate simulator as a training tool to teach palatoplasty. Improved procedural confidence and knowledge were observed after a single session, with benefits seen among trainees both with and without previous cleft experience.

Objective: The objectives of this study were to assess economy of hand motion of residents, fellows, and staff surgeons using a high-fidelity cleft palate simulator to (1) stratify performance for the purpose of simulator validation and (2) to estimate the learning curve., Design: Two residents, 2 fellows, and 2 staff surgeons performed cleft palate surgery on a high-fidelity cleft palate simulator while their hand motion was tracked using an electromagnetic hand sensor. The time, number of hand movements, and path length of their hands were determined for 10 steps of the procedure. The magnitude of these metrics was compared among the 3 groups of participants and utilized to estimate the learning curve using curve-fitting analysis., Results: The residents required the most time, number of hand movements, and path length to complete the procedure. Although the number of hand movements was closely matched between the fellows and staff, the overall total path length was shorter for the staff. Inverse curves were fit to the data to represent the learning curve and 25 and 113 simulation sessions are required to reach within 5% and 1% of the expert level, respectively., Conclusion: The simulator successfully stratified performance using economy of hand motion. Path length is better matched to previous level of experience compared to time or number of hand movements.

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.)

Purpose: We evaluated hearing loss and general intelligence among persons with auricle anomalies and cleft lip and/or alveolus and/or cleft palate (CLAP)., Methods: A nationwide cross-sectional study of data recorded during 1966-2019, as mandatory pre-military recruitment of individuals., Results: Of 3 182 892 adolescents, 548 were diagnosed with auricle anomalies and 2072 with CLAP. For the latter, the adjusted odds ratios for the low, low to medium and medium general intelligence categories compared to the highest category were 1.4 [95% CI 1.5-1.2], 1.2 [95% CI 1.4-1.1] and 1.1 [95% CI 1.2-0.9] respectively. The corresponding values for the auricle anomalies were not significant., Conclusions: General intelligence was impaired among individuals with CLAP, but no significant correlation was found among individuals suffering from auricle anomalies., (© 2023 John Wiley & Sons A/S. Published by John Wiley & Sons Ltd.)

Purpose: Simulation course serves as an efficient supplement to the experiential learning inside the operation room in surgical residency programs. Furlow double-opposing Z-plasty (DOZ) remains an important and challenging surgical technique in cleft palate repair, but the simulation is currently limited. This study aims to develop a blended learning module with low-cost and easy-to-promote DOZ simulation., Methods: Comparison among the common rodent experimental animals and the readily available porcine tongue was made to select the ideal animal for surgical simulation, with which a DOZ learning module was developed. Oral surgery residents were enrolled to attend the blended learning module. An after-session questionnaire and a quiz were distributed to the participants to collect their feedback of the course perceptions and to assess their learning outcomes., Results: The porcine tongue had more advantages in simulating the human soft palate over the common experimental animals. The surgical procedure of DOZ could be well designed and simulated on the porcine tongue. The vast majority of the participant liked the learning module regarded it as beneficial for the comprehension if DOZ principles and procedures. With the help of the porcine tongue simulator, the blended learning module helped the participants grasp the designing principles of DOZ procedure and promote their associative learning., Conclusions: The blended learning module incorporating the porcine tongue simulator can help oral surgery residents master the principles and procedures of DOZ palatoplasty., (© 2022 American Dental Education Association.)

Background: This study assessed technical performance in cleft palate repair using a newly developed assessment tool and high-fidelity cleft palate simulator through a longitudinal simulation training exercise., Methods: Three residents performed five and one resident performed nine consecutive endoscopically recorded cleft palate repairs using a cleft palate simulator. Two fellows in pediatric plastic surgery and two expert cleft surgeons also performed recorded simulated repairs. The Cleft Palate Objective Structured Assessment of Technical Skill (CLOSATS) and end-product scales were developed to assess performance. Two blinded cleft surgeons assessed the recordings and the final repairs using the CLOSATS, end-product scale, and a previously developed global rating scale., Results: The average procedure-specific (CLOSATS), global rating, and end-product scores increased logarithmically after each successive simulation session for the residents. Reliability of the CLOSATS (average item intraclass correlation coefficient (ICC), 0.85 ± 0.093) and global ratings (average item ICC, 0.91 ± 0.02) among the raters was high. Reliability of the end-product assessments was lower (average item ICC, 0.66 ± 0.15). Standard setting linear regression using an overall cutoff score of 7 of 10 corresponded to a pass score for the CLOSATS and the global score of 44 (maximum, 60) and 23 (maximum, 30), respectively. Using logarithmic best-fit curves, 6.3 simulation sessions are required to reach the minimum standard., Conclusions: A high-fidelity cleft palate simulator has been developed that improves technical performance in cleft palate repair. The simulator and technical assessment scores can be used to determine performance before operating on patients.

Background: Patients with cleft lip and/or palate can undergo numerous procedures to improve appearance, speech, dentition and hearing. We developed a cleft-specific patient-reported outcome instrument to facilitate rigorous international measurement and benchmarking., Methods: Data were collected from patients aged 8-29 years with cleft lip and/or palate at 30 hospitals in 12 countries between October 2014 and November 2016. Rasch measurement theory analysis was used to refine the scales and to examine reliability and validity. Normative CLEFT-Q values were computed for age, sex and cleft type., Results: Analysis led to the refinement of an eating and drinking checklist and 12 scales measuring appearance (of the face, nose, nostrils, teeth, lips, jaws and cleft lip scar), health-related quality of life (psychological, social, school, speech distress) and speech function. All scales met the requirements of the Rasch model. Analysis to explore differential item functioning by age, sex and country provided evidence to support the use of a common scoring algorithm for each scale for international use. Lower (worse) scores on CLEFT-Q scales were associated with having a speech problem, being unhappy with facial appearance, and needing future cleft-related treatments, providing evidence of construct validity. Normative values for age, sex and cleft type showed poorer outcomes associated with older age, female sex and having a visible cleft., Interpretation: The CLEFT-Q represents a rigorously developed instrument that can be used internationally to collect and compare evidence-based outcomes data from patients aged 8-29 years of age with cleft lip and/or palate., Competing Interests: Competing interests: Anne Klassen and Karen Wong Riff are co-developers of the CLEFT-Q and, as such, could potentially receive a share of any licence revenues as royalties based on their institutions’ inventor sharing policy if the CLEFT-Q is used in a for profit study., (© 2018 Joule Inc. or its licensors.)

Objective: The goal of treatment for individuals with cleft lip and/or palate (CL/P) is to improve physical, psychological, and social health. Outcomes of treatment are rarely measured from the patient's perspective. The aim of the study was to develop a conceptual framework for a patient-reported outcome (PRO) instrument for individuals with clefts (CLEFT-Q) by developing an in-depth understanding of issues that individuals consider to be important., Design: The qualitative methodology of interpretive description was used. Setting, Participants, and Intervention: We performed 136 individual in-depth interviews with participants with clefts of any age, presenting for cleft care, across 6 countries. Parents were involved if the child was more comfortable. Interviews were audio-recorded, transcribed verbatim, and coded using constant comparison. The data were used to develop a refined conceptual framework., Results: Participants described concepts of interest in 3 top-level domains, each of which included subdomains: appearance (face, nose, nostrils, teeth, lips, jaw, cleft lip scar), health-related quality of life (psychological, social, school, speech-related distress), and facial function (speech, eating/drinking). Participants were able to describe changes over time with regard to the 3 domains., Conclusions: A conceptual framework of concepts of interest to individuals with CL/P formed the basis of the scales in the CLEFT-Q. Each subdomain represents an independently functioning scale. Understanding what matters to patients is essential in guiding PRO measurement.

Background: A surgical robot offers enhanced precision, visualization, and access and the potential to improve outcomes in cleft palate surgery. The goal of this study was to investigate the feasibility of using the da Vinci robot for cleft palate repair in infants using a cleft palate simulator test bed., Methods: A high-fidelity cleft palate simulator was developed that allows performance of a robotic cleft palate repair procedure. A complete cleft palate repair was performed with the da Vinci Si with 5-mm instruments and the da Vinci Xi with 8-mm instruments. The advantages of the robotic approach were assessed in comparison with using standard instruments. For each system, arm repositioning, collisions, instrument and endoscope excursion, wrist orientation, and vision were compared for 12 steps of the repair., Results: The cleft palate simulator provided a reproducible platform for testing robotic cleft palate surgery. The advantages of the robotic approach were the ability to articulate a miniature wrist intraorally with superior visualization, increased ambidexterity, and improved ergonomics compared with using standard instruments. Cleft palate repair with the Xi was superior to the Si with respect to arm repositioning, instrument collisions and excursion, and wrist orientation. However, Xi performance remained suboptimal because of the larger instruments., Conclusions: Robotic cleft palate repair using the da Vinci system offers advantages compared with the traditional approach. Cleft palate repair is more feasible with the Xi and 8-mm instruments. However, performance is limited by the instrumentation, which requires modification to ensure safety and efficacy., Clinical Question/level of Evidence: Therapeutic, V.

Objective: Segmental Le Fort I osteotomy through the cleft is a common strategy to narrow the alveolar cleft in adults. This study compared skeletal stability between single and segmental Le Fort I osteotomies in patients with unilateral cleft lip and palate (UCLP)., Materials and Methods: This retrospective analysis examined 45 adults with complete UCLP-associated class III deformities who underwent bimaxillary surgery with either single (n = 30) or segmental (n = 15) Le Fort I advancement. Cone beam computed tomography (CBCT) scans of the facial skeleton were acquired before surgery, 1-week postsurgery, and at follow-up. Measures of landmarks from the CBCT images for the two treatment groups were compared for translation (left/right, posterior/anterior, superior/inferior) and rotation (yaw, roll, pitch)., Results: Postsurgery, the downward movement of the maxilla was larger in the segmental group than the single group. At follow-up, the maxilla moved backward in both groups, and upward in the segmental group. The mandible moved forward and upward and rotated upward in both groups. The amount of upward movement and rotation was larger in the segmental group than the single group., Conclusions: Two years after bimaxillary surgery in patients with UCLP-associated class III deformity, greater relapse was found after segmental Le Fort I osteotomies in vertical translation of the maxilla and mandible, and pitch rotation of the mandible compared with single Le Fort I osteotomies., Clinical Relevance: The vertical relapse of the maxilla was larger after segmental Le Fort I advancement compared with single Le Fort I advancement in clefts., (© 2024. The Author(s), under exclusive licence to Springer-Verlag GmbH Germany, part of Springer Nature.)

Introduction: Patient-reported outcome (PRO) instruments should be developed according to rigorous guidelines in order to provide clinically meaningful, scientifically sound measurement. Understanding the methodology behind instrument development informs the selection of the most appropriate tool. This mixed methods protocol describes the development of an internationally applicable PRO instrument, the CLEFT-Q, for evaluating outcomes of treatment for cleft lip and/or palate (CL/P)., Methods and Analysis: The study includes three main phases that occur iteratively and interactively. In phase I, we determine what concepts are important to patients regarding their outcome. A conceptual framework for the CLEFT-Q is formed through a systematic review and an extensive international qualitative study. The systematic review ascertains what concepts have previously been measured in patients with CL/P. The qualitative study employs interpretive description and involves in-depth interviews with patients in high-income and lower-middle income countries. Preliminary items are generated from the qualitative data. Preliminary scales are then created for each theme in the framework. Cognitive debriefing interviews and expert clinician input are used to refine the scales in an iterative process. In phase II, the preliminary scales are administered to a large international group of patients with CL/P. The modern psychometric method of Rasch Measurement Theory analysis is employed to define the measurement characteristics. The preliminary scales are shortened based on these results. In phase III, further tests assess reliability, validity and responsiveness of the instrument., Ethics and Dissemination: The study is approved by Research Ethics Boards for each participating site. Findings from this study will be published in open access peer-reviewed journals and presented at national and international conferences. Integrated knowledge translation is employed to engage stakeholders from the outset of the study. Successful execution of the CLEFT-Q will result in an internationally applicable PRO instrument for children and young adults with CL/P., Competing Interests: KWYWR, ET, TG, CRF, ALP and AFK declare that they have no competing interests. SJC started Modus Outcomes, a company that designs PRO instruments, after the inception of this study., (Published by the BMJ Publishing Group Limited. For permission to use (where not already granted under a licence) please go to http://www.bmj.com/company/products-services/rights-and-licensing/.)

Background: Cleft palate repair is a challenging procedure to learn because of the delicate tissue handling required and the small confines of the infant oral cavity. As a result, cleft palate simulators have previously been described to augment cleft palate repair training. Although valuable, they lack the fidelity for this complex procedure., Methods: A high-fidelity cleft palate simulator was evaluated by staff and fellows in pediatric plastic surgery who provided feedback on its realism, anatomical accuracy, and effectiveness as a training tool. The simulator was implemented within a training workshop following a didactic session on cleft palate repair and anatomy. A test was administered to each participant before and immediately after the workshop to assess knowledge transfer. Perceived confidence of performing a repair following the workshop was also assessed, as was the workshop's effectiveness., Results: Overall, participants agreed that the simulator is anatomically accurate and realistic and strongly agreed that the simulator is a valuable training tool. The average test score increased from 25 percent before the workshop to 77.27 percent after the workshop. Overall, participants of the workshop felt more confident performing a repair and strongly agreed that the workshop was valuable and effective., Conclusions: A high-fidelity cleft palate simulator has been evaluated as realistic, anatomically accurate, and valuable as a training tool. The simulator was successfully integrated into a training workshop, which resulted in significant knowledge increase on anatomy and the procedure and perceived confidence and comfort in performing a cleft palate repair.

Objective: To investigate the stability of major versus minor Le Fort I maxillary advancements in unilateral cleft lip and palate (UCLP) patients., Design: A retrospective longitudinal study was undertaken on 30 nonsyndromic UCLP patients treated with the same protocol at The Hospital for Sick Children, Toronto, Canada. Patients were grouped into major and minor movement groups based on planned surgical advancement. Standard lateral cephalometric radiographs were taken preoperatively (T1), immediately postoperatively (T2), and at least 1 year postoperatively (T3). Skeletal and dental variables were measured using cephalometric analysis. Stability was compared between groups using repeated-measures analysis of variance. Linear regression analysis was used to assess the relationship between advancement and relapse for the entire study population., Results: A mean maxillary advancement of 9.8 mm and 4.9 mm was seen for the major (n = 10) and minor (n = 20) movement groups, respectively. The mean skeletal horizontal relapse was 1.8 mm (18%) for the major advancement group and 1.5 mm (31%) for the minor advancement group. There was no significant difference in skeletal horizontal relapse between the groups (P > .05). The correlation coefficient (r) between linear horizontal advancement and relapse was calculated to be .31 (P > .05). Dental horizontal relapse was not significant for either the major or minor groups, and no significant difference was found between the groups (P > .05)., Conclusion: Skeletal and dental relapse was found to be unrelated to the amount of maxillary linear advancement using conventional Le Fort I osteotomies in UCLP.

Purpose: To investigate the stability of single-piece versus segmental (2-piece) maxillary advancement in patients with unilateral cleft lip and palate (UCLP) treated using conventional Le Fort I orthognathic surgery., Patients and Methods: A retrospective study was undertaken in 30 patients with nonsyndromic UCLP treated with the same surgical and orthodontic protocol from 2002 through 2011. Standard lateral cephalometric radiographs were taken preoperatively, immediately postoperatively, and at least 1 year postoperatively. Patients were divided into single-piece and segmental Le Fort I groups based on planned surgical movement. Postoperative movements were compared between groups using repeated measures analysis of variance., Results: The mean skeletal horizontal advancement was 7.3 and 7.5 mm in the single-piece and segmental groups, respectively. The skeletal horizontal relapse was 1.3 mm (18%) for the single-piece group and 1.9 mm (25%) for the segmental group. The skeletal surgical extrusion was 2.7 mm for the 2 groups. The skeletal vertical relapse was 0.6 mm (22%) and 1.5 mm (56%) for the single-piece and segmental groups, respectively. The mean dental horizontal postoperative movement was an advancement of 0.4 mm for the single-piece group and a relapse of 0.2 mm (3%) for the segmental group. The mean dental vertical relapse was 0.1 mm (4%) for the single-piece group and 0.3 mm (11%) for the segmental group. There was no statistically significant difference in relapse between the single-piece and segmental groups for all movements (P>.05)., Conclusion: Skeletal and dental relapse was similar between single-piece and segmental maxillary advancements using conventional Le Fort I orthognathic surgery in patients with UCLP., (Copyright © 2014 American Association of Oral and Maxillofacial Surgeons. Published by Elsevier Inc. All rights reserved.)

Objective: Individuals born with cleft lip and palate may face difficulties in speech function, nutrition, facial aesthetics, and long-term care. These difficulties may increase the risk of psychological and psychiatric diseases. This work aimed to test if the variant allele of COMT was carried more frequently among individuals that have psychological and psychiatric outcomes within a cohort of patients born with cleft lip and palate., Method: DNA extraction from saliva of two hundred and fifteen individuals born with cleft lip with and/or palate and genotyping was performed, and the frequency of COMT rs4818 alleles was determined. The domain 'Psychological Function' of Cleft-Q™ was used to generate scores for analysis. The scores were computed, and differences in genotype or allele frequencies between individuals with psychological function scores 60 or above and 59 or below were compared. The history of psychiatric illness (family history of psychiatric disease or self-reported psychiatric illness) was registered., Results: Genotype and allele frequencies were compared between individuals with and without a family history of psychiatric illness. Individuals with lower Psychological Function (Cleft-Q™) scores were more likely to be GG (P = .04) or carriers of allele G (P < .001). The reported psychiatric illness and positive family history of psychiatric illness were compared to COMT rs4818 allele and genotype frequencies of individuals without these indicators, and individuals with psychiatric illness and positive family history of psychiatric illness were more likely to carry allele G (P = .03 and P = .008, respectively)., Conclusion: The study confirms previously suggested role of COMT rs4818 in psychiatric and psychological outcomes in a distinct cohort of patients born with cleft lip and palate., (© 2022 John Wiley & Sons A/S. Published by John Wiley & Sons Ltd.)

Objective : Orofacial clefts such as cleft lip, cleft palate, and cleft lip and palate are the most frequent congenital anomalies of the head and neck. The purpose of this study was to determine the current demographics for orofacial clefts in Canada. Methods : A request for data from all Canadian provinces (excluding Quebec due to incompatibilities with provincial coding systems) for the fiscal years 2002-2003 to 2007-2008 was submitted to the Canadian Institute for Health Information. Variables evaluated included gender, cleft type, gestational age, birth weight, income quintile, and institution health region. Results : Over the period studied, the prevalence of orofacial clefts ranged from 11.0 to 15.3 per 10,000 live births (1 in 654 to 1 in 909 live births). The distribution of cleft types for live births with orofacial clefts was 17% for cleft lip, 41% for cleft palate, and 42% for cleft lip and palate, of which cleft lip and cleft lip and palate were male dominant (62% and 66% male, respectively) and cleft palate was female dominant (56% female). Saskatchewan and Manitoba had significantly higher cleft birthrates (P < .05) compared with the other provinces. Birth weight and gestational age (but not income quintile) were significantly (P < .0001) lower for newborns with orofacial clefting compared with those with no cleft. Conclusions : Canada has one of the highest orofacial cleft birthrates in the world (prevalence of 12.7 per 10,000 live births, approximately 1 in 790 live births). This study presents an updated demographic of orofacial clefts in Canadian newborns and may be useful in predicting the burden of anticipated health care.

Objective: This study explored the experiences of adolescent girls with cleft lip and palate who were preparing for orthognathic surgery., Design: A qualitative study, based on a phenomenological approach, explored adolescents' experiences. Two 1-hour semistructured interviews were conducted before surgery, and member checking was employed to verify findings with each participant., Setting: A tertiary care pediatric hospital in central Canada., Participants: A criterion sampling technique was used to recruit a sample of seven participants with cleft lip and palate ranging in age from 15 to 20 years who were scheduled for cleft orthognathic surgery., Results: PARTICIPANTS described experiencing teasing, bullying, and stares. In many cases, participants sought to resist and dismiss negative social attitudes about their visible difference while working to combat this with a positive view of who they really are. To varying degrees, participants felt constricted in engaging in the community as they were worried what others thought of their visible difference. They worked at finding ways to reduce the impact of social stigma through coping strategies, social supports, and reconstructive surgery., Conclusions: Adolescent girls can experience strain associated with living with a facial difference; however, they find strategies to cope with the perception of difference. Reconstructive surgery is viewed as a means to increase confidence. Further intervention is needed in understanding and addressing stigma and fostering resiliency related to female adolescents living with a facial difference.

Objective: Cleft lip and/or palate (CLP) is the most common congenital craniofacial anomaly. As a first step toward developing a quality of life (QOL) questionnaire for CLP patients, our team conducted a systematic literature review to identify studies that measured child- or proxy-reported outcomes of CLP., Design: PUBMED, CINAHL, EMBASE and PsycINFO were searched from their inception to July 2010 to identify studies that measured health-related concepts in CLP patients. Abstract and title screening was performed by two screeners. Full texts of all potentially relevant papers were obtained and examined by two reviewers. We identified publications that measured health concepts and categorized them to form a preliminary conceptual framework of CLP QOL issues., Results: A total of 4594 publications were identified. Twenty-six studies met our inclusion criteria. Research involved CLP patients living in nine countries with sample sizes ranging from 23 to 661. Health concepts were measured using 29 different questionnaires. No patient-reported outcome (PRO) instrument measuring QOL concerns of CLP patients currently exists. CLP-specific health concepts measured to date were categorized into a preliminary conceptual QOL framework with the following categories: physical, psychological and social health., Conclusions: Our review has helped to identify areas of health that have been well researched using either a patient or proxy patient-reported outcome instrument (e.g., self-concept; behavior) and areas where more research is required., (Copyright © 2011 British Association of Plastic, Reconstructive and Aesthetic Surgeons. Published by Elsevier Ltd. All rights reserved.)

Objective: To investigate the use of intravenous ketorolac and iliac crest bupivacaine infusion in the management of iliac crest donor-site pain in the pediatric cleft population. The null hypothesis was there is no difference with respect to pain scores between ketorolac and iliac crest bupivacaine infusion as analgesic adjuncts to intravenous opioids., Methods: A total of 54 children and adolescents (27 boys, 27 girls) undergoing alveolar cleft repair or Le Fort I osteotomy were assigned randomly in a prospective, single-blinded fashion to one of three groups: intravenous ketorolac plus iliac crest normal saline infusion, intravenous ketorolac plus iliac crest bupivacaine infusion, or iliac crest bupivacaine infusion alone. Iliac crest infusions and ketorolac were administered for 48 hours or until discharge, whichever occurred first. All patients received morphine via a patient-controlled analgesia device., Main Outcome Measure(s): Primary outcome was pain score, and secondary outcomes were morphine consumption and satisfaction scores., Results: Pain scores, morphine consumption, and satisfaction scores were not significantly different among groups. Estimated costs were significantly higher for bupivacaine infusion than intravenous ketorolac., Conclusions: Iliac crest donor-site pain is well managed in this patient population. Intravenous ketorolac and iliac crest bupivacaine infusion provide comparable analgesia for iliac crest bone graft donor-site pain in children and adolescents.

Morphological characteristics of the "cleft face" were analyzed by calculating proportion indices in young adults who had undergone surgery in childhood for cleft lip and/or palate: 592 in 37 subjects with unilateral clefts and 432 in 27 with bilateral clefts. Three areas of the face with 16 indices based on 12 projective linear measurements were analyzed in each subject: 5 indices in the general face, 6 in the upper face, and 5 in the lower. Although a balanced relationship was found in two thirds of the indices assessed, severe disproportions, which greatly influence appearance, were noted in about one quarter. In the general face, the most frequent abnormal anthropometric findings contributing to severe disproportions were small upper-face height and a severely high or moderately narrow mandible. In the soft tissues, disproportions included wide nose, small nasal tip protrusion, and short or long columella. Deviations from normality were rarely noted in the nose and upper-lip height, mouth width, total facial height, and width of the upper face. In the upper face, severe disproportions occurred much more frequently in bilateral cleft subjects (67.6%) than in unilateral clefts (30.3%). In the lower face, however, severe disproportions were twice as frequent among unilateral clefts (39.4%) than bilateral (14.7%). The results, although interesting, require complementary preoperative data for reliable analysis of the adult "cleft face."

Background: Patients with alveolar cleft unrepaired suffer from nasal deformities of different magnitude. Bone and cartilage grafts are harvested through several incisions. In this study, we present a method to simultaneously correct nasal deformities and repair alveolar cleft using grafts from the nasal septum., Patients and Methods: All 6 patients with unilateral cleft lip and palate have alveolar cleft unrepaired combined with nasal deformity. Computed tomography scans and 3-dimensional-printed models of vomer and ethmoid bone were used for the purpose of preoperative design and for assessing the magnitude of deformity. Grafts of bone and cartilage from deviated septum were harvested by septoplasty through which dorsum deviation was corrected. Bone grafts from vomer and ethmoid were then fixed to the prepared alveolar cleft to repair the defect and elevate the alar base. Septal cartilage was adjusted into different shapes of grafts and deformities of nasal tip, nostrils, and columella were then corrected by rhinoplasty to restore the symmetry of the nose., Results: Symmetry of nostrils was improved. The height of alar base on the cleft side was elevated to the level close to the noncleft side. Deviation of the septum, nasal dorsum, and columella was corrected. Projection of the nasal tip was adjusted to facial midline. Midface aesthetics was generally improved., Conclusion: Application of septal grafts reduce the number of incisions. One-stage repair of alveolar cleft and nasal deformities, with the aid of digital design, improves the postoperative experience and the general outcome of the surgery., Competing Interests: The authors report no conflicts of interest., (Copyright © 2022 by Mutaz B. Habal, MD.)

Abstract: Many patients with cleft palate in developing countries never receive postoperative speech assessment or therapy. The use of audiovisual recordings could improve access to post-repair speech care. The present study evaluated whether English-speaking speech-language pathologists (SLPs) could assess cleft palate patients speaking an unfamiliar language (Tamil) using recorded media. Recordings obtained from Tamil-speaking participants were rated by 1 Tamil-speaking SLP and 3 English-speaking SLPs. Ratings were analyzed for inter-rater reliability and scored for percent correct. Accuracy of the English SLPs was compared with independent t tests and Analysis of Variance. Sixteen participants (mean age 14.5 years, standard deviation [SD] 7.4 years; mean age of surgery of 2.7 years, SD 3.7 years; time since surgery: 10.8 years, SD 5.7 years) were evaluated. Across the 4 SLPs, 5 speech elements were found to have moderate agreement, and the mean kappa was 0.145 (slight agreement). Amongst the English-speaking SLPs, 10 speech elements were found to have substantial or moderate agreement, and the mean kappa was 0.333 (fair agreement). Speech measures with the highest inter-rater reliability were hypernasality and consonant production errors. The average percent correct of the English SLPs was 60.7% (SD 20.2%). English SLPs were more accurate if the participant was female, under eighteen, bilingual, or had speech therapy. The results demonstrate that English SLPs without training in a specific language (Tamil) have limited potential to assess speech elements accurately. This research could guide training interventions to augment the ability of SLPs to conduct cross-linguistic evaluations and improve international cleft care by global health teams., Competing Interests: The authors report no conflicts of interest., (Copyright © 2022 by Mutaz B. Habal, MD.)

Purpose: Patients born with a cleft palate often suffer from velopharyngeal insufficiency (VPI) due to the soft palate musculature's abnormal structure. Surgical correction of the palate at a young age can hinder maxillary growth, requiring surgical correction of the maxillomandibular discrepancy at skeletal maturity. Orthognathic surgery can then cause or further exacerbate VPi in these patients. The purpose of this paper is to assess cleft-orthog-nathic patients under the lens of persistent or newly-developed VPi, to understand outcomes and generate a targeted management algorithm., Methods: A retrospective study was performed inspecting cleft-orthognathic patients presenting to a single surgeon. Patients with sufficient follow-up were placed into predefined algorithmic cohorts by their VPi development pattern in relation to orthognathic surgery. They were further stratified into groups by level of adherence to our algorithm to evaluate VPi outcomes. Demographic factors, risk factors, and outcomes were compared between groups via Welch t test and Fisher exact test., Results: Fifty-one patients were examined, including 16 with VPI. Velopharyngeal insufficiency fully resolved amongst all algorithmically adherent patients and remained in nonadherent patients ( P  < 0.001)., Conclusions: Our targeted algorithm may improve symptoms and the management of VPI in cleft-orthognathic patients. Multi-centered studies with larger sample sizes and prospective studies are encouraged to validate our proposed treatment algorithm further., Competing Interests: The authors report no conflicts of interest., (Copyright © 2022 by Mutaz B. Habal, MD.)

Background: Hemifacial microsomia (HFM), which involves multiple sites with different levels of severity, is the second most common congenital craniofacial deformity after cleft lip and palate. However, three-dimensional (3D) measurements of mandibular deformities have not yet been studied in detail. The objective of this study is to investigate the method of 3D measurements of mandibular deformities in HFM patients., Methods: A total of 48 HFM patients were included in this study. All clinical treatment for patients was performed in the Plastic Surgery Hospital of the Chinese Academy of Medical Sciences at Peking Union Medical College from June 2006 to June 2020. The patients' 3D computerized tomography scan data were processed using medical imaging software, following four iterative steps: 3D reconstruction, mirroring, differential analysis, and partition., Results: The characteristics of the mandibular bone in HFM patients are mainly presented as follows: (1) compared to the normal side, the part of the bone body that extends from the ascending ramus to the pogonion (Po-NB) is analyzed using a dynamic process: less fullness-fullness-more fullness; (2) absences were frequently observed among the angular zones, that is, the height of the ascending ramus is deficient., Conclusions: HFM is a complicated condition with numerous variations in clinical presentation. We employed both 3D image reconstruction and computerization image processing techniques to investigate asymmetrical mandibular deformity in HFM patients in detail and with great accuracy. This will be of great use to clinicians for disease management., (Copyright © 2022 The Chinese Medical Association, produced by Wolters Kluwer, Inc. under the CC-BY-NC-ND license.)

Objectives: Some adults with cleft lip and palate (CLP) require orthognathic surgery due to skeletal deformity. This prospective study aimed to (1) compare skeletal stability following bimaxillary surgery for correction of class III deformity between patients with unilateral CLP (UCLP) and bilateral CLP (BCLP), and (2) identify risk factors of stability., Materials and Methods: Adults with CLP and skeletal class III deformities who underwent surgery-first bimaxillary surgery were divided into two groups according to cleft type: UCLP (n = 30) and BCLP (n = 30). Skeletal stability was assessed with measures from cone beam computed tomography images of the maxilla and mandible taken before treatment, 1-week and ≥ 1 year postsurgery for translation (left/right, posterior/anterior, superior/inferior) and rotation (yaw, roll, pitch); multiple regression analysis examined risk factors., Results: At follow-up, the maxilla moved upwards in both groups, and backwards in the UCLP group. The mandible moved forward and upward, shifted to the cleft (deviated) side, and rotated upward in both groups. The amount of surgical advancement was a risk factor for sagittal stability in the maxilla (ß = -0.14, p < 0.05). The mandible had three risk factors for sagittal stability: age (ß = -0.23, p < 0.05), surgical team (ß = -1.83, p < 0.05), and amount of surgical setback (ß = -0.32, p = 0.001)., Conclusions: Two years after bimaxillary surgery, patients with UCLP had a higher sagittal relapse of the maxilla compared with patients with BCLP, which was due to a greater surgical advancement in the patients with UCLP., Clinical Relevance: Surgery-first bimaxillary surgery results in favorable treatment outcomes for correction of cleft-related class III deformity. Severity of jaw discrepancy and surgeons should be considered in the surgical design of overcorrection., (© 2021. The Author(s), under exclusive licence to Springer-Verlag GmbH Germany, part of Springer Nature.)

Cleft lip and/or palate (CL/P), the most prevalent congenital anomaly, is understood to negatively affect a wide range of child development. Since the concept remains controversial, because most published work is from cross-sectional studies, we examined the neurodevelopmental trajectories in participants with CL/P through a longitudinal comparison with the general population during early childhood using data from a nationwide birth cohort study in Japan. The linear mixed models for each domain of the Ages and Stages Questionnaire, third edition (ASQ-3), were used to detect differences in standardised mean scores between groups. The ASQ-3 is a general neurodevelopmental screening tool comprising communication, gross motor, fine motor, problem-solving, and personal-social domains. Participants' neurodevelopment was determined semi-annually from 6 to 36 months of age. The trajectories of standardised mean scores in each domain showed several significant differences between the control and CL/P groups, with the maximum difference at 24 months of age in the communication domain. Indeed, CL/P was associated with significantly lower scores in the communication (coefficient: -3.31, 95% CI: -5.09 to -1.14), problem-solving (coefficient: -3.13, 95% CI: -5.07 to -1.18), and personal-social domains (coefficient: -1.99, 95% CI: -3.87 to -0.11). Trajectories of ASQ-3 scores suggest neurodevelopmental delays in children with CL/P., (© 2022 Scandinavian Division of the International Association for Dental Research. Published by John Wiley & Sons Ltd.)

Abstract: Orofacial clefts (OFCs) are the most common craniofacial congenital anomalies, and its prevalence is highest among Asian populations. The aim of this retrospective case-control study is to evaluate the effect of parental consanguinity on the frequency of OFCs at Jordan University Hospital over a 15-year-period. The study group consists of all patients with OFCs presented to the major tertiary referral center in Jordan during the last 15 years, along with age and gender-matched controls. The authors analyzed the risk of different predictors, including consanguinity, on the development of OFCs, both cleft lip with or without cleft palate (CL/P) and cleft palate only. A total of 332 participants were included in this study, with a mean age of 74.36 (±48.75) months. The authors included 129 (38.9%) OFCs, and 203 (61.1%) controls. The percentage of parental consanguinity among OFCs group was 41.1%, compared to only 24.1% for controls, a difference that was statistically significant (P = 0.001). On logistic regression analysis, the authors found that parental consanguinity is a significant predictor for the occurrence of OFCs (P = 0.007), where people with consanguineous marriage have 2 times higher risk (odds ratio of 0.504, with 95% confidence interval 0.306-0.830) to have offspring with OFCs. Moreover, lower birth weight babies are also significantly more associated with OFCs (P = 0.014), with an odds ratio of 1.819 (95% confidence interval 1.131 2.926). Among the Jordanian population, the authors found that consanguinity and lower birth weight were the only variables significantly associated with the development of OFCs., Competing Interests: The authors report no conflicts of interest., (Copyright © 2021 by Mutaz B. Habal, MD.)

Background: The Quality of Life Adolescent Cleft Questionnaire is a measure of quality of life in cleft lip/palate (CL/P) and includes items on pre-treatment and post-treatment status. Items, however, were originally organized in a factor structure that prevents a formal pre-treatment to post-treatment comparison. Additionally, the questionnaire was tested in older patients., Aim: We aimed to explore a factor structure that allows a comparison of pre- to post-treatment status in children and adolescents with CL/P., Design: The sample comprised 60 children and adolescents with CL/P. The scale was divided into two groups of items (24 comparing pre-treatment and post-treatment status and 26 measuring current quality of life). Two different exploratory and confirmatory analyses were conducted (one for each group of items). Sources of criterion validity were investigated with measures of self-esteem and self-efficacy., Results: The results supported a 6-factor structure for the pre-treatment and post-treatment items. In the second group of items, 9 items were removed due to inadequate functioning and a final 4-factor solution was obtained. The criterion validity of factors was good., Conclusion: The proposed factor solution might be more useful to detect the perceived satisfaction in different areas and can be used in younger patients., (© 2021 The Authors. International Journal of Paediatric Dentistry published by BSPD, IAPD and John Wiley & Sons Ltd.)

Background: Left clefts occur twice as frequently as right ones. The sidedness has been suggested to influence certain outcomes. Some surgeons consider a right cleft more challenging to repair. This is often attributed to their reduced prevalence. The authors question whether this may be caused by morphologic differences. The authors' hypothesis is that there are anthropometric differences between left and right complete cleft lips., Methods: Patients with complete unilateral cleft lip, with or without cleft palate, operated on at the age of 3 to 6 months, between 2000 and 2018, by a single surgeon, were included. Eight standardized anthropometric measurements of the cleft lip, collected just before cleft lip repair, compare lip and vermillion dimensions and ratios between left and right clefts., Results: One hundred thirty-nine left and 80 right unilateral cleft lips were compared. A significant difference was found between left and right clefts for cleft-side to non-cleft-side ratios comparing the lateral lip element vertical heights and vermillion heights., Conclusions: Patients with right cleft lips have a greater degree of lateral lip element hypoplasia, demonstrating greater deficiencies of lateral lip element vertical height and vermillion height when compared to patients with left clefts. This has clinical implications for preoperative assessment, choice of surgical technique, and postoperative and long-term outcomes., (Copyright © 2022 by the American Society of Plastic Surgeons.)

Aim: To assess treatment outcome and 1-year stability of LeFort I advancement in patients with complete cleft lip and palate., Methods: Thirty-five patients (age 20.65 ± 2.20 years) with unilateral (n = 25) or bilateral (n = 10) complete cleft lip and palate who underwent LeFort I advancement were included.Lateral cephalograms before surgery (T1), immediately postsurgery (T2), and at 1-year follow-up (T3) were superimposed, and the position of anterior nasal spine (ANS), A-point, and U1 Tip assessed using an x, y coordinate system. Differences between landmark positions at the 3-time points were analyzed using paired sample t-tests, with a significance defined as α ≤ 0.05., Results: The mean surgical advancement in the horizontal direction (T2-T1) was 6.50 ± 2.62 mm at ANS (P < 0.001) and 7.05 ± 2.51 mm at A-point (P < 0.001). At a 1-year follow-up (T3-T2), the mean horizontal relapse at ANS was -1.41 ± 1.89 mm (P < 0.001) and -0.79 ± 1.48 mm at A-point (P 0.003). Mean horizontal relapse was 21.7% and 11% of surgical advancement when assessed at ANS and A-point, respectively. The central incisor tip position remained stable during the postsurgical period (0.12 ± 2.11 mm, P 0.732). At A-point, the mean vertical surgical change (T2-T1) was -0.96 ± 2.57 mm (P < 0.001). No significant post-treatment (T3-T2) vertical changes were detected at ANS or A-point. Phenotypic stability was excellent, with all patients maintaining positive overjet at 1-year follow-up., Conclusions: LeFort I advancement in complete cleft lip and palate is stable, with less than a 2 mm relapse after 1-year. Surgical overcorrection by 10% to 20% is recommended to compensate for the expected skeletal relapse., Competing Interests: The authors report no conflicts of interest., (Copyright © 2021 by Mutaz B. Habal, MD.)

Le Fort I osteotomy is the most common procedure for maxillary advancement in cleft patients, and a significant relapse is expected in the postoperative period. This study purpose is to evaluate the maxillary relapse and associated factors in cleft lip and palate (CLP) patients submitted to standard Le Fort I advancement. This systematic review sought studies in PubMed, SCOPUS, and Cochrane and included papers that evaluated the maxillary relapse with at least 1-year postoperative period. The quality assessment was adapted following PRISMA statement. The initial electronic search found 345 papers, and the final selection included 10 studies. In total, 275 CLP patients were evaluated 198 had unilateral CLP and 41 had bilateral CLP. Isolated maxillary advancement was performed in 166 patients, while 109 had bimaxillary surgery. The mean relapse in horizontal and vertical plane was 1.2 mm (19.7%) and 1.0 mm (29.4%), respectively. In conclusion, a relapse after maxillary advancement in patients with CLP is expected, being higher in vertical plane. Patients with UCLP and those who underwent bimaxillary surgery had a higher relapse rate. Due to the miscellaneous data and methodologies, future prospective clinical trials should apply rigorous selection of CLP patients sample and methods for variable analysis to obtain more accurate results.

Background: Treatment for patients with diverse craniofacial conditions is complex and long-term. Craniofacial conditions profoundly influence health-related quality of life, and patient- and parent-reported outcomes provide a critical and complementary perspective on the multidisciplinary treatment of patients. However, little is known regarding the health-related quality of life among children with diverse craniofacial conditions. The purpose of this study was to systematically review the literature regarding patient- and parent-reported outcomes measures for patients with diverse craniofacial conditions., Methods: Articles from the PubMed, PsychINFO, CINAHL, Embase/MEDLINE, Scopus, and Web of Science databases that used patient- and/or parent-reported outcome instruments in patients with diverse craniofacial conditions were reviewed. Diagnoses included were cleft lip and/or palate, craniosynostosis, microtia, craniofacial microsomia, facial vascular malformations, and congenital nevi across pediatric populations (0 to 22 years of age)., Results: Six hundred ninety articles were identified, and 155 were selected for inclusion. One hundred twenty different health-related quality-of-life tools were used to analyze factors such as physical, psychological, or social function. Of these, the 10 most common psychometrically tested tools were identified in 59 studies. Five tools had both parent and patient versions. Two tools were developed and validated for patients with diverse craniofacial conditions, but neither was developed for nonadolescent children., Conclusions: Many parent- and patient-reported instruments are used to measure varying health-related quality of life factors in this population, but no tool exists that was developed and psychometrically tested in different facial deformities that measures comprehensive health-related quality of life issues across all pediatric ages. This study will guide the development of new tools to measure the parent and patient health-related quality-of-life perspective in patients with diverse craniofacial conditions.

Objectives: Timing of cleft palate repair is controversial. We aim to assess whether timing of cleft palate repair affects rates of inpatient complications, length of stay (LOS), and cost of stay., Methods: The Healthcare Cost and Utilization Project Kids' Inpatient Database 2009 was queried for all admissions with a primary diagnosis of cleft palate during which cleft palate repair was performed as a primary procedure. Age 6 months or less was termed "early" repair, while age >6 months was termed "standard" repair. Patients age >3 years old, inpatient stays >30 days, and those stays in which a cleft lip repair was performed were excluded. Logistic regressions were used to model the probability of complications. Generalized linear models and a natural log link function were used for LOS and hospital charges, using SAS 9.4., Results: We included 223 early and 1482 standard repair patients. Early repairs were exclusively performed in urban hospitals (P < 0.001). Eighty-nine patients experienced a total of 100 complications, including respiratory failure (N = 53), airway obstruction (N = 18), and oropharyngeal hemorrhage (N = 13). We found no significant difference in complication rate or total hospital charges in the 2 groups. The earlier repair group had a slightly longer LOS (P = 0.048)., Conclusion: Over 85% of United States cleft palate repairs are performed after 6 months of age. All early repairs were performed at urban hospitals, and had slightly longer LOS. There was a 5.1% overall complication rate. Available data revealed no significant difference in complication rates between early repair and standard repair groups., Level of Evidence: 3b., Competing Interests: The authors report no conflicts of interest., (Copyright © 2020 by Mutaz B. Habal, MD.)

Abstract: The purpose of this study was to analyze the morphology of the nasomaxillary complex of Iranian healthy infants of 2, 4, 6, and 12-month-old.In this cross-sectional study, 232 healthy Fars infants of 2, 4, 6, and 12-month-old referred to Mashhad health centers were evaluated. Photographic images were taken from the infants' frontal view at rest position. Anthropometric landmarks including width of the mouth, nose, columella, width and height of the philtrum, and height of the upper lip were measured by Photoshop software.The authors did not find any considerable differences in nasolabial morphology of Fars children between boys and girls, except for columellar width and the width of superior philtrum at all intervals. The mean columellar width decreased with increasing age in both sexes. The width of the lower philtrum decreased from 2 to 4 months in both sexes, but increased from 4 to 6 months. In girls, the mean height of the right and left philtrum and height of the upper lip increased by increasing age from 2 to 4 months. But it consistently reduced from 4 months to 6 and 12-month-old. In boys, the mean height of the right and left philtrum and height of the upper lip decreased from 2 to 4 months. It showed a mild increase from 4 to 6 months, and a relatively large reduction from 6 to 12 months.This study provides useful information for reconstructive surgeries of the nasolabial region in the population studied., Competing Interests: The authors report no conflicts of interest., (Copyright © 2020 by Mutaz B. Habal, MD.)

Objective: The objectives of this study were to evaluate health-related quality of life (HRQoL) in 4-7-year-old children treated for cleft lip and/or palate (CL/P) compared to healthy controls and to estimate a possible association with cleft type, gender, age, and surgical re-interventions., Materials and Methods: A total of 171 children with CL/P (mean age 5.7 years) and 186 healthy controls (mean age 5.5 years) were included in the study. Sixty-four (37.4%) children experienced both cleft lip and palate, 56 (32.8%) cleft lip only, and 51 (29.8%) cleft palate only. HRQoL in children was assessed by Kiddy-KINDL and COHIP-14 questionnaires and in parents by KINDL-p. Total score and dimensions of each questionnaire were compared between cleft children, their parents, and controls., Results: The total scores and dimension scores of Kiddy-KINDL showed similar values between CL/P and control groups, except for "self-esteem" dimension (p = 0.036). The comparison of Kiddy-KINDL and KINDL-p showed a statistically significant total score (82.11 vs. 80.44, p = 0.047). The CL/P group presented significantly worse values with respect to controls in total score of COHIP-14 (10.53 vs 5.01, p < 0.001) and in all its dimensions., Conclusions: Children treated for CL/P had a negative impact on HRQoL at early age compared to controls. Significant differences were found in the psychological and functional dimensions related to lip and palatal affectation, respectively. Parents reported significantly worse scores than their children. HRQoL measurement provided valuable complementary information to better inform to parents and make clinical decisions in children with CL/P at early age., Clinical Relevance: Our findings suggested that Spanish children between 4 and 7 years of age with a history of surgically treated CL/P experienced a poorer HRQoL when compared with their non-cleft peers.

Purpose: Postoperative hip pain is commonly reported after anterior iliac crest harvest for alveolar bone grafting. The goal of this study is to describe our institution's experience and examine the efficacy of our pain management protocols., Methods: A retrospective review was performed by abstracting demographic, operative, and pain management data from January 2011 to April 2013. Paired t-tests and Fisher exact tests were used to examine differences when comparing 2 groups, while ANOVA was used to examine difference between the 3 protocols for harvest and pain management: trapdoor technique and local anesthetic injection (TD+LAI), TD and pain catheter (TD+PC), and split crest and LAI., Results: Eighty-four patients, 52 males (61.9%), averaging 8.8 years old (±2.9) were included. Postoperatively, 17 (71%) patients in the PC group received IV narcotics compared to 27 (45%) in those without a PC (P = .03). When comparing all 3 protocols, no significant difference was found in IV morphine usage or duration of IV morphine treatment. In subgroup analysis, when patients in the groups TD+PC versus TD+LAI were examined, those in the TD+PC group had significantly shorter hospital stays and were more likely to go home postoperative day 1 (P = .03; P = .04)., Conclusions: Overall, patients tolerated alveolar bone grafting well regardless of harvest technique or pain management approach. While indwelling PCs did not significantly decrease IV morphine usage, these patients had significantly shorter lengths of stays.

Background: The aim of this retrospective case series study was to compare three-dimensional postsurgical outcomes of patients with cleft lip and palate following maxillary advancement., Methods: Fifty consecutive cleft lip and palate patients who underwent whole-pieced Le Fort I advancements were assigned to the major (advancement ≥ 5 mm) or minor (advancement < 5 mm) groups. Three-dimensional surgical simulation was used for presurgical evaluation and planning. Virtual triangles of the presurgical, simulated, and 6-month postoperative stages were used for comparison. Translational and angular changes of each endpoint (A-point, MxR, and MxL) on the virtual triangles and reference planes were recorded and analyzed. Relationships between possible related variables and outcome discrepancies from simulations among all subgroups were also investigated., Results: Analysis of covariance and the least significant difference test revealed that the outcome discrepancy measurements were affected by different combinations of independent variables. The reliability test showed high consistency of the authors' method for three-dimensional measurements., Conclusions: The actual surgical outcomes of cleft lip and palate patients differed from the virtual simulations. The outcome discrepancies are impacted by multiple factors. The outcome discrepancies of all rotational surgical corrections (roll, yaw, and pitch) were positively correlated to the degree of planned surgical movement. Meanwhile, bilateral cleft lip and palate patients are more likely to incur outcome discrepancies in yaw correction with major maxillary advancement. However, a maxillary advancement cutoff value of 5 mm would not necessarily lead to significant translational outcome discrepancies among cleft lip and palate patients., Clinical Question/level of Evidence: Therapeutic, IV.

Background: The intermediate cleft tip rhinoplasty is performed in childhood to address residual tip asymmetries during the most critical period of psychosocial development. The authors describe and evaluate long-term outcomes of that approach for the unilateral cleft lip and palate patient based on the concept of individual restoration of each abnormal anatomical component., Methods: Photomorphometric analyses of unilateral cleft lip and palate patients (n = 50) who underwent intermediate cleft tip rhinoplasty using the component restoration technique were evaluated preoperatively (time 0) and postoperatively at less than 1 year (time 1), 1 to 3 years (time 2), and more than 3 years (time 3) and compared to age-matched unilateral cleft lip and palate control patients. Nasal relationships (alar symmetry, nasal tip protrusion-to-alar base width ratio, and height-to-width dimensions for the cleft and noncleft nostrils) were compared over time using a linear mixed-effect model., Results: At time 0, both groups demonstrated similar nasal relationships, with the exception of a wider cleft-side nostril in relationship to height in the rhinoplasty group. The component restoration technique improved all four nasal relationships at all postoperative time points compared with time 0 in a statistically significant manner, whereas control patients did not demonstrate significant changes at the corresponding ages. Long-term differences at time 3 revealed a trend toward improved alar symmetry and cleft-side and non-cleft-side nostril dimensions, and a significant improvement in the nasal tip protrusion-to-alar base width ratio in intermediate cleft tip rhinoplasty-treated versus control patients (p = 0.002)., Conclusion: The component restoration technique for the unilateral intermediate cleft tip rhinoplasty improves nasal relationships toward normal immediately and in a sustained manner for at least 3 years., Clinical Question/level of Evidence: Therapeutic, III.