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.

BACKGROUND:Comprehensive cleft care is a multidisciplinary team endeavor. While untreated craniofacial conditions have multiple undue repercussions, cleft care in outreach settings can be fraught with significant perioperative morbidity risks.AIM:Propose updated quality assurance standards addressing logistic and operational considerations essential for the delivery of safe and effective cleft lip and /or palate (CL/P) care in low and middle-income countries (LMICs) settings.METHODS:Based on American Cleft Palate-Craniofacial Association (ACPA) quality standards, published literature, published protocols by Global Smile Foundation (GSF), and the senior author's three-decade experience, updated standards for outreach cleft care were synthesized.RESULTS:Ten axes for safe, effective, and sustainable cleft lip and palate care delivery in underserved settings were generated: 1) site assessment, 2) establishment of community partnerships, 3) team composition and credentialing, 4) team training and mission preparation, 5) implementation of quality assurance guidelines, operative safety checklists, and emergency response protocols, 6) immediate and long-term postoperative care, 7) medical record keeping, 8) outcomes evaluation, 9) education, and 10) capacity building and sustainability. Subsequent analysis further characterized essential components of each of those ten axes to delineate experience derived and evidence-based recommendations.DISCUSSION:Quality assurance guidelines are essential for the safe delivery of comprehensive cleft care to patients with CL/P in any setting. Properly designed surgical outreach programs relying on honest community partnerships can be effectively used as vehicles for local capacity building and the establishment of sustainable cleft care ecosystems.

Introduction: The authors analyzed the insights of participants and faculty members of Global Smile Foundation's Comprehensive Cleft Care Workshops concerning the barriers and interventions to multidisciplinary cleft care delivery, after stratification based on demographic and geographic factors. Methods: During 2 simulation-based Comprehensive Cleft Care Workshops organized by Global Smile Foundation, participants and faculty members filled a survey. Surveys included demographic and geographic data and investigated the most relevant barrier to multidisciplinary cleft care and the most significant intervention to deliver comprehensive cleft care in outreach settings, as perceived by participants. Results: The total response rate was 57.8%. Respondents reported that the greatest barrier to comprehensive cleft care was financial, and the most relevant intervention to deliver multidisciplinary cleft care was building multidisciplinary teams. Stratification by age, gender, and geographical area showed no statistical difference in reporting that the greatest barrier to cleft care was financial. However, lack of multidisciplinary teams was the most important barrier according to respondents with less than 5 years of experience ( P = 0.03). Stratification by gender, years in practice, specialty, and geographical area showed no statistical difference, with building multidisciplinary teams reported as the most significant intervention. However, increased training was reported as the main intervention to cleft care for those aged less than 30 years old ( P = 0.04). Conclusions: Our study delivers an assessment for barriers facing multidisciplinary cleft care delivery and interventions required to improve cleft care delivery. The authors are hoping that stratification by demographic and geographic factors will help them delineate community-specific road maps to refine cleft care delivery.

Introduction: Clefts of the lip and palate are leading congenital facial anomalies. Underserved patients with these facial differences lack access to medical care, surgical expertise, prenatal care, or psychological support. Moreover, the disease results in significant economic strains on patients and their families. While surgical outreach programs have attempted to fill this void, significant challenges facing international comprehensive cleft care persist. Objective: Propose a path toward international sustainable cleft care based on the Global Smile Foundation experience. Results: International sustainable comprehensive cleft care can be achieved by regulating surgical outreach programs. Regulation of these missions would ensure standardized care and encourage stakeholders to cooperate and adequately allocate funding and resources. Capacity building can be achieved through "diagonal" cleft care delivery models, multidisciplinary workshops, fellowship programs, research and quality assurance, as well as leveraging emerging technologies such as Augmented Reality. Conclusion: International comprehensive cleft care requires continuous collaborative efforts between visiting and local teams as well as international and national organizations. Standardizing and regulating current practices as well as promoting capacity building initiatives can contribute to sustainable cleft care.

Objective: To present preliminary efforts to establish an internationally agreed set of minimum core practice and best practice guidelines, along with overarching principles to promote safe and comprehensive cleft care globally. Design: Representatives from 6 national and international organizations collaborated to form a World Cleft Coalition. Representatives met monthly/bimonthly to compile standards for safe, comprehensive, and sustainable cleft care. Outcomes were circulated within each organization and to a small subset of external constituents for feedback. Results: A series of overarching principles were established for those involved in International Cleft Treatment Programs, based on the experience of participating organizations. The overarching principles are followed by a structured and detailed Recommended Practice for Ensuring Safe, Comprehensive and Sustainable Cleft Care, which includes minimum core and best practice for the following areas: surgical safety, quality control, patient education, patient selection, patient follow-up, comprehensive care, partnership with the host nations and professionals, training and exchanges for sustainability, and local capacity building. Conclusions: Outcomes aimed to provide a working document to define core principles for safe comprehensive cleft care, while balancing various levels of resources, geographic locations, appropriately trained health care professional specialists, and training limitations. The study highlights the process and benefits to a collaborative international working group not only to establish best practice but also to solicit and engage others in discussion of their experiences with building and supporting safe, high-quality, comprehensive, sustainable, worldwide cleft care.

Purpose: Global Smile Foundation (GSF) is a non profit foundation that provides comprehensive cleft care to underserved patients. GSF focuses on long-term follow up and sustainability of local healthcare teams, having engaged in 32 years of follow up in Ecuador. In 2012, GSF added presurgical NasoAlveolar Molding (NAM) therapy for their patients in Guayaquil, Ecuador, as part of its sustainability and empowerment initiative. We present longitudinal data on 189 patients treated with NAM and discuss the challenges/barriers to its completion. Methods: Data was collected from GSF surgical and dental records including patient diagnosis, completion/incompletion and length of NAM therapy. Surgeon, patient age, peri, intra, and post-operative procedural data, were collected for primary cleft lip/nose and palate repair, and any additional surgeries. Follow up clinical and photographic data was retrieved to document long-term outcomes. Results: A total of 207 patients were treated with pre-surgical therapy: 189 patients received NAM therapy, while 18 patients were treated with lip tape and/or nasal elevator. Of the 189 NAM patients who received NAM, long-term follow up was available for 96 patients (50.8 %), while 84 (44.4%) were lost to follow up or subsequently seen by another foundation, and 9 (4.8%) are currently undergoing NAM or awaiting primary surgery. Of the 96 patients with long term follow up, 70 (72.9%) had Unilateral Cleft Lip and Palate and 26 (27.1%) had Bilateral Cleft Lip and Palate; 64 (66.67%) were male and 32 (33.3%) were female. Of those 96 patients, 58 (60.4%) completed NAM therapy, 17 (17.7%) failed to complete it, and 21 (21.8%) had incomplete NAM documentation. The average age at NAM initiation was 36.36 ± 31.39 days (range: 0-157 days) and average length of NAM therapy was 118.98 ± 82.68 days (range: 1-222 days). Patients underwent an average of 2.13± 0.93 (range: 1-5) surgeries after NAM Initiation, with an average of 0.17 ± 0.43 (0-2) Cleft Lip/Nose Revisions, 0.06 ± 0.28 (0-2) GingivoPerioPlasty, 0.06 ± 0.28 (0-1) pre-maxillary setbacks, 0.07 ± 0.30 (0-2) fistula repairs, and 0.03 ± 0.17 (0-1) Velopharyngeal Insufficiency corrections. Over an 8 year period, 12 NAM providers were trained in Ecuador, 7 provided treatment in Ecuador, and 5 provided treatment internationally, making Ecuador a site for information exchange. Follow-up for NAM patients was an average of 2.00 ± 1.77 (0.22 - 6.67) years after NAM initiation and 1.45 ± 1.77 (0 - 6.52) years after their primary Cleft Lip/Nose Repair. This includes continued long term follow up and comprehensive cleft care in addition to NAM therapy. Conclusion: With yearly patient follow-up and year-round partnership with local professionals, our model shows successful long-term delivery of NAM therapy as part of a sustainable comprehensive cleft care strategy in outreach settings.

Many orthodontists working on patients with cleft lip and palate (CLP) have shown great enthusiasm for presurgical infant orthopedics (PSIO) to improve surgical outcomes with minimal intervention. Even though every clinician aims to use the best treatment modality for their patients, PSIO effects can be confounded by surgical type and timing of the primary repair, as is discussed in many studies. In such cases, one should be cautious when evaluating the particular outcomes for patients with CLP since it is difficult to differentiate the sole effect of an individual surgical or orthodontic intervention. As with any treatment methodology, nasoalveolar molding (NAM) has both benefits and limitations. Commonly cited concerns with NAM, and PSIO in general, include increased cost, increased burden of care, and a negative impact on maxillary growth. However, NAM cannot be deemed as having apparent long-term negative or positive effects on skeletal or soft tissue facial growth, based on previous studies. A review of the literature suggests that NAM does not alter skeletal facial growth when compared with the samples that did not receive PSIO. Nevertheless, the published studies on NAM show evidence of benefits to the patient, caregivers, the surgeon, and society. These benefits include documented reduction in severity of the cleft deformity prior to surgery and as a consequence improved surgical outcomes, reduced burden of care on the care givers, reduction in the need for revision surgery, and consequent reduced overall cost of care to the patient and society.

Purpose: Global Smile Foundation (GSF) is a not for profit foundation whose founders and volunteers have been providing cleft care to underserved communities around the world for 32 years. In 2012, GSF incorporated nasoalveolar molding (NAM) into its treatment model in Guayaquil, Ecuador. We present an evaluation of nasolabial aesthetic outcomes and scarring in patients treated with NAM prior to primary cleft lip repair versus patients who were not in similar outreach settings. Methods: The Cleft Aesthetic Rating Scale (CARS) used frontal photographs taken at least 1-year post primary cleft lip repair to assess the nose: tip, nostrils (symmetry, size, flaring) and upper lip (vermillion symmetry and continuity and length of the philtrum), ranging from 1 (very good) to 5 (very poor). Photos were standardized to reveal only the nasolabial area and excluded any time points after any additional surgical revision to the nasolabial area. Each NAM time point was matched to a control time point based on their age at primary cleft lip repair (maximum of 3 months) and their time post-op from primary cleft lift repair (maximum of 6 months). All included NAM and control patients had Unilateral Cleft Lip and Palate, and were from Ecuador. Patients with congenital syndromes other than cleft lip and palate (CLCP) affecting facial appearance were excluded. Twelve independent raters including three surgeons, three orthodontist, three pediatric dentists and three medical students rated the photographs. As a modification to CARS, raters were also asked to assess scar quality utilizing a previously developed scar subtype scoring system. Results: Of the 189 patients treated with NAM in Guayaquil since 2012, 96 patients had long term follow up, and 27 patients with 34 photographic time points qualified for inclusion, 15 (55.56%) male, 12 (44.44%) female. The average time post primary cleft lip repair was 2.19 ± 1.65 months (range: 0.98-7.37 months). Matched to the patients who had received NAM were 31 control patients with 34 photographic time points, 26 (83.87%) male, 5 (16.13%) female with an average time post primary cleft lip repair of 2.19 ± 1.63 months (range: 0.93-6.98 months) The average rating of Nasal aesthetics was significantly lower in patients who had received NAM compared to nasal controls (2.60 ± 1.05 vs 2.82 ± 1.12 (p <0.01).The average rating for lip aesthetics showed similar differences between patients who had received NAM and matched controls (2.23 ± 0.96 vs 2.56 ± 1.07, (p < 0.01). Similarly, the average rating of scar quality was significantly lower among patients who had received NAM (1.82 ± 0.93 vs 2.03 ± 0.89), (p < 0.01). Conclusion: Using the CARS and a modified scar rating scale, patients who had received NAM were found to have superior nose and lip aesthetic outcomes in comparison to non-NAM controls. This suggests that NAM is not only feasible in an outreach setting, but it is also associated with improved lip, nose and scar outcomes.

INTRODUCTION:We analyzed the perceptions of participants and faculty members in simulation-based comprehensive cleft care workshops regarding comprehensive cleft care delivery in developing countries.METHODS:Data were collected from participants and faculty members in 2 simulation-based comprehensive cleft care workshops organized by Global Smile Foundation. We collected demographic data and surveyed what they believed was the most significant barrier to comprehensive cleft care delivery and the most important intervention to deliver comprehensive cleft care in developing countries. We also compared participant and faculty responses.RESULTS:The total number of participants and faculty members was 313 from 44 countries. The response rate was 57.8%. The majority reported that the most significant barrier facing the delivery of comprehensive cleft care in developing countries was financial (35.0%), followed by the absence of multidisciplinary cleft teams (30.8%). The majority reported that the most important intervention to deliver comprehensive cleft care was creating multidisciplinary cleft teams (32.2%), followed by providing cleft training (22.6%). We found no significant differences in what participants and faculty perceived as the greatest barrier to comprehensive cleft care delivery (P = 0.46), or most important intervention to deliver comprehensive cleft care in developing countries (P = 0.38).CONCLUSIONS:Our study provides an appraisal of barriers facing comprehensive cleft care delivery and interventions required to overcome these barriers in developing countries. Future studies will be critical to validate or refute our findings, as well as determine country-specific roadmaps for delivering comprehensive cleft care to those who need it the most.

OBJECTIVE:Evaluate simulation-based comprehensive cleft care workshops as a reproducible model for education with sustained impact.DESIGN:Cross-sectional survey-based evaluation.SETTING:Simulation-based comprehensive cleft care workshop.PARTICIPANTS:Total of 180 participants.INTERVENTIONS:Three-day simulation-based comprehensive cleft care workshop.MAIN OUTCOME MEASURES:Number of workshop participants stratified by specialty, satisfaction with the workshop, satisfaction with simulation-based workshops as educational tools, impact on cleft surgery procedural confidence, short-term impact on clinical practice, medium-term impact on clinical practice.RESULTS:The workshop included 180 participants from 5 continents. The response rate was 54.5%, with participants reporting high satisfaction with all aspects of the workshop and with simulation-based workshops as educational tools. Participants reported a significant improvement in cleft lip (33.3 ± 5.7 vs 25.7 ± 7.6; P < .001) and palate (32.4 ± 7.1 vs 23.7 ± 6.6; P < .001) surgery procedural confidence following the simulation sessions. Participants also reported a positive short-term and medium-term impact on their clinical practices.CONCLUSION:Simulation-based comprehensive cleft care workshops are well received by participants, lead to improved cleft surgery procedural confidence, and have a sustained positive impact on participants' clinical practices. Future efforts should focus on evaluating and quantifying this perceived positive impact, as well reproducing these efforts in other areas of need.

Store-operated Ca2+ entry (SOCE) channels are highly selective Ca2+ channels activated by the endoplasmic reticulum (ER) sensors STIM1 and STIM2. Their direct interaction with the pore-forming plasma membrane ORAI proteins (ORAI1, ORAI2, and ORAI3) leads to sustained Ca2+ fluxes that are critical for many cellular functions. Mutations in the human ORAI1 gene result in immunodeficiency, anhidrotic ectodermal dysplasia, and enamel defects. In our investigation of the role of ORAI proteins in enamel, we identified enamel defects in a patient with an ORAI1 null mutation. Targeted deletion of the Orai1 gene in mice showed enamel defects and reduced SOCE in isolated enamel cells. However, Orai2(-/-) mice showed normal enamel despite having increased SOCE in the enamel cells. Knockdown experiments in the enamel cell line LS8 suggested that ORAI2 and ORAI3 modulated ORAI1 function, with ORAI1 and ORAI2 being the main contributors to SOCE. ORAI1-deficient LS8 cells showed altered mitochondrial respiration with increased oxygen consumption rate and ATP, which was associated with altered redox status and enhanced ER Ca2+ uptake, likely due to S-glutathionylation of SERCA pumps. Our findings demonstrate an important role of ORAI1 in Ca2+ influx in enamel cells and establish a link between SOCE, mitochondrial function, and redox homeostasis.