Cleft Care Delivery Research Articles

Global Prevalence and Burden of Orofacial Clefts: A Systematic Analysis for the Global Burden of Disease Study 2019.

Orofacial clefts, in particular cleft lip and cleft palate, are among the most common congenital anomalies. Despite guidelines recommending early surgical correction, a global backlog of untreated patients persists. This has made orofacial clefts an attractive target for global cleft care initiatives. The most recent global burden of orofacial clefts was estimated to be 529,758.92 disability-adjusted life years (95% uncertainty interval: 362,492.88-798,419.69 disability-adjusted life years), whereas the global prevalence of orofacial clefts was estimated to be 4.6 million (95% uncertainty interval: 3.8-5.7 million). An inverse relationship exists between the Sociodemographic Index and the burden of orofacial clefts. Sub-Saharan Africa, Middle East/North Africa, and South Asia are the regions carrying the most significant burden of orofacial clefts. This manuscript provides updated estimates of the global burden and prevalence of orofacial clefts, acting as a guide to direct future investments, resources, and initiatives from individuals and organizations engaged in global cleft care delivery with the goal of building sustainable cleft care capacity where it is needed the most.

The First Hybrid International Educational Comprehensive Cleft Care Workshop.

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. Cross-sectional survey-based evaluation. International comprehensive cleft care workshop. Total of 489 participants. Three-day simulation-based hybrid comprehensive cleft care workshop. 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. 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). 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.

Clinical Practice Guidelines for the Management of Patients With Cleft Lip and Palate: A Systematic Quality Appraisal Using the Appraisal of Guidelines for Research and Evaluation II Instrument.

Patients with cleft lip and/or palate require complex and longitudinal care by a multidisciplinary cleft team. Unfortunately, delivery of cleft care is often fragmented, and care practices can vary significantly. Multiple clinical practice guidelines (CPGs) have been proposed to provide a standardized framework for cleft care delivery. As CPGs have gained popularity, there has been increasing demand to maintain the quality of existing guidelines. A comprehensive search of EMBASE, MEDLINE via PubMed, Scopus, Cochrane and grey literature sources published from January 1, 1990 to December 31, 2020 was conducted to identify CPGs for the care of cleft patients. The Appraisal of Guidelines for Research and Evaluation, 2nd edition II tool was used to assess the quality of selected CPGs. Intraclass coefficients were calculated to assess agreement among appraisers. Eleven guidelines were identified for study inclusion. One guideline was classified as "high" quality by Appraisal of Guidelines for Research and Evaluation II criteria, and the remaining guidelines were classified as "average" or "low" quality. The "Clarity of Presentation" domain achieved the highest mean score (76.9% ± 11.7%) across CPGs, whereas the "Rigor of Development" domain scored the lowest (35.6% ± 21.2%). Intraclass coefficients analysis reflected very good inter-rater reliability across all domains (0.853-0.987). These findings highlight significant variability in the quality of existing CPGs for the global management of patients with cleft lip and/or palate. The "Rigor of Development" domain reflects the greatest opportunity for improvement. Given these findings, future guidelines may prioritize incorporating a systematic review of existing evidence into recommendations.

Comprehensive Cleft Care Delivery in Developing Countries: Impact of Geographic and Demographic Factors.

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. 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. 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). 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.

Perceived Barriers to Comprehensive Cleft Care Delivery: Results From A Capacity-Building Educational Initiative and Implications.

We analyzed the perceptions of participants and faculty members in simulation-based comprehensive cleft care workshops regarding comprehensive cleft care delivery in developing countries. 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. 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). 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.

Sustainable Cleft Care: A Comprehensive Model Based on the Global Smile Foundation Experience.

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. Propose a path toward international sustainable cleft care based on the Global Smile Foundation experience. 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. 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.

Facilitators and Barriers to Implementing Standardized Outcome Measurement for Children With Cleft Lip and Palate

Identify facilitators and barriers to implementing standardized outcome measurement in cleft care. Cross-sectional, qualitative study. Participants included 24 providers and staff from a large, multidisciplinary cleft team in the southwest United States, 5 caregivers of children with cleft palate (with or without cleft lip) treated by this team, and 3 experts involved in implementing a cleft-specific standardized outcome measurement in the United Kingdom. Semistructured, qualitative interviews were conducted exploring perceived facilitators and barriers to implementing standardized outcome measurement in cleft care. Interviews were audio-recorded, transcribed, and analyzed for content. The Consolidated Framework for Implementation Research was used to guide the interviews and analysis. The analysis focused on the characteristics of standardized outcome measurement that directly influence its adoption. Participants identified both facilitators and barriers to implementing standardized outcome measurement. Facilitators included the strength and quality of evidence supporting improvements in cleft care delivery following implementation of standardized outcome measurement and the relative advantage of standardized outcome measurement over continuing the status quo. Barriers included the difficulty adapting standardized outcome measurement to meet local context and patient-specific needs and the complexity of implementing standardized outcome measurement. Providers, staff, and caregivers involved in cleft care perceive multiple benefits from standardized outcome measurement, while also recognizing substantial barriers to its implementation. Results from this study can be used to guide development of an implementation strategy for standardized outcome measurement that builds upon perceived strengths of the intervention and reduces perceived barriers.

The History and Mission of Smile Train, a Global Cleft Charity.

Cleft lip and/or palate (CLP) is a common congenital anomaly with a global impact. One organization attempting to decrease global burden of CLPs is Smile Train. Since 1999, Smile Train has empowered local medical providers to provide comprehensive and sustainable cleft care. Partner surgeons have performed more than 1.5 million operations for patients with CLPs in more than 90 countries. This article outlines the history and mission of Smile Train and details the organization's efforts to increase hospital-wide safety, provide education and training opportunities for partners, and use technology to improve the delivery of cleft care on a global scale.

Improved Early Cleft Lip and Palate Complications at a Surgery Specialty Center in the Developing World.

To evaluate complication rates following cleft lip and cleft palate repairs during the transition from mission-based care to center-based care in a developing region. We performed a retrospective review of 3419 patients who underwent cleft lip repair and 1728 patients who underwent cleft palate repair in Guwahati, India between December 2010 and February 2014. Of those who underwent cleft lip repair, 654 were treated during a surgical mission and 2765 were treated at a permanent center. Of those who underwent cleft palate repair, 236 were treated during a surgical mission and 1491 were treated at a permanent center. Two large surgical missions to Guwahati, India, and the Guwahati Comprehensive Cleft Care Center (GCCCC) in Assam, India. Overall complication rates following cleft lip and cleft palate repair. Overall complication rates following cleft lip repair were 13.2% for the first mission, 6.7% for the second mission, and 4.0% at GCCCC. Overall complication rates following cleft palate repair were 28.0% for the first mission, 30.0% for the second mission, and 15.8% at GCCCC. Complication rates following cleft palate repair by the subset of surgeons permanently based at GCCCC (7.2%) were lower than visiting surgeons ( P < .05). Our findings support the notion that transitioning from a mission-based model to a permanent facility-based model of cleft care delivery in the developing world can lead to decreased complication rates.

National Estimates of and Risk Factors for Inpatient Revision Surgeries for Orofacial Clefts.

To provide national estimates of the number and cost of primary and revision cleft lip and palate surgeries in the U.S. and to determine patient and hospital characteristics associated with disproportionate use of revision surgery. Retrospective cross-sectional study using data obtained from the 2003, 2006, and 2009 Kids' Inpatient Database. Inpatient. Children with CL, CP, or CLP undergoing inpatient cleft lip and/or palate surgery. Inpatient cleft lip and/or palate surgery. Orofacial cleft surgery estimates, estimates of primary versus revision surgeries, and estimated inflation-adjusted hospitalization costs. In 2009, there were a total of 2824 and 5431 hospitalizations for cleft lip and palate surgeries, respectively. Revision surgery accounted for 24.2% of cleft lip surgeries and 36.8% of cleft palate surgeries. Children with CLP (OR 1.87, 95% CI: 1.48-2.38), a syndromic diagnosis (OR 1.47, 95% CI: 1.16-1.87), or private insurance (OR 1.71, 95% CI: 1.41-2.09) were more likely to undergo cleft lip revision surgery. Similar risk factors were found for children undergoing cleft palate revision. Mean cost per hospitalization ranged from $7564 to $8393 in 2009, depending on surgery type, and did not change significantly (in 2009 U.S. $) between 2003 and 2009. Interventions to reduce revision surgery by improving results of primary surgery should be targeted in the population of identified high-risk (e.g., syndromic) patients. In addition, the association of health insurance status with revision surgery highlights the need to understand and address the impact of economic disparities on cleft care delivery.

Development of Volunteer International Craniofacial Surgery Missions

Volunteer surgical missions to provide cleft care to patients in developing countries has been done successfully for a number of years. Similar missions that provide craniofacial surgery introduce a dramatic step up in complexity. While articles have addressed protocols for the safe delivery of cleft care around the world, little has been written on volunteer craniofacial surgical missions. Komedyplast was established in March 2001 as a 501c(3) nonprofit organization to provide craniofacial surgical care to underserved populations and educate local surgeons in craniofacial principles. During 9 annual missions, the organization has provided surgical care to more than 150 patients with various complex, congenital, craniofacial conditions. The article addresses important safeguards that have been implemented to maximize safety and minimize risk.

Improving Education and Standards for Cleft Care in the Developing World

INTRODUCTION The partner hospital model identifies hospitals in the developing world to educate and enable local surgeons to deliver effective cleft care. This study aimed to determine the outcomes of this model on safety, education, and quality of surgical care. MATERIALS AND METHODS Twelve partner hospitals, sponsored by Smile Train for 5 or more years and distributed over 4 continents, were selected. Activities at each institution were evaluated using cleft surgical data, and electronic surveys were completed by hospital leadership. RESULTS A mean of 82% of patients with cleft at partner hospitals underwent sponsored surgeries. After partnership, all 12 hospitals implemented preoperative checklists for cleft surgery, and 5 hospitals implemented checklists for other surgeries. All hospitals had personnel who received safety training as a result of partnership. There was no change in 30-day reoperations or readmissions. Follow-up rate increased by 18% (P = 0.03). Facilities recruited 1.8 additional cleft surgeons (P < 0.01) and increased the number of cleft surgical trainees by a mean of 13.3 annually (P = 0.012); 2.5 ± 1.7 additional ancillary services were added, resulting in 75% of partner hospitals having a basic multidisciplinary cleft team (Surgery, Speech, and Dental), compared with 25% prior to partnership (P < 0.01). Total cleft surgeries, alveolar bone grafts, and percentage of secondary surgeries increased significantly as the length of partnership progressed (P < 0.01). CONCLUSIONS A model to enhance global education and delivery of cleft care through development and support of local hospitals increases both the volume and the quality of cleft care delivered at these institutions. Safety initiatives for cleft care demonstrate effects extending to global surgical care delivered at partner hospitals.

Cleft Care in Nigeria: Past, Present, and Future

To review the progress of orofacial cleft care in Nigeria and propose a viewpoint for the future. Review of the available literature on cleft care in Nigeria and survey of the status of ongoing cleft care in Nigerian centers. We employed a pretested self-administered questionnaire sent and returned through electronic mail. Coordinators of cleft care centers in Nigeria. Findings of literature search and responses to mailed questionnaires. Available literature suggests that the fate of orofacial cleft patients during the precolonial era in Nigeria remains unclear. However, there is evidence of surgical care delivery just before the end of the colonial era. We identified and contacted 39 existing cleft care delivery centers, of which 30 (76.9%) responded. The majority (69.2%) of the responding centers began cleft care delivery between 2006 and 2010; 73.3% have designated cleft clinic locations and 66.7% offer interdisciplinary care. All responding centers offer cheiloplasty, while 86.7% offer palatoplasty. Other aspects of cleft care are provided sparingly in most centers due to paucity of manpower. Challenges with hospital administration, securing bed and theater spaces, drug availability, and performing laboratory investigations were the common limitations reported. We advocate for improved cleft care delivery through removing administrative bottlenecks, fortifying existing centers, and mentoring younger colleagues for entry into underserved specialties. Concerted effort and international collaborations aimed at transforming some of the existing cleft centers to standard pediatric craniofacial centers are desirable.

Demographics of Cleft Care Providers in Africa and Reported Experience in Training and Practice: Direct Analysis of Continent-Based Practitioners

To elucidate the impact of several geographic, cultural, and socioeconomic variables on cleft care delivery in Africa, and to investigate the current status of cleft care delivery in Africa. Survey of practitioners attending the second Pan-African Congress on Cleft Lip and Palate (PACCLIP). The annual PACCLIP conference in Ibadan, Nigeria, West Africa, February 2007. To provide an analysis of the demographics and training experience of cleft care providers in Africa by collating information directly from the continent-based practitioners. Plastic surgeons and oral and maxillofacial surgeons provide the majority of cleft care. Most of the participants reported availability of formal training programs in their respective countries. The predominant practice settings were university and government-based. During training, half of the providers had encountered up to 30 cleft cases, and a quarter had managed more than 100 cases. Representation of visiting surgeons were equally distributed between African and non-African countries. This study provides initial and detailed analysis crucial to understanding the underlying framework of cleft care composition teams, demographics of providers, and training and practice experience. This awareness will further enable North American and other non-African plastic surgeons to effectively partner with African cleft care providers to have a further reaching impact in the region.

The Outcome for Secondary Alveolar Bone Grafting in the South West UK Region Post-CSAG

In 1998 the delivery of cleft care in the United Kingdom was examined by the Clinical Standards Advisory Group, the outcomes of which led to the wide restructuring of cleft services in the United Kingdom. We present a retrospective study evaluating the radiographic outcome of 53 consecutively performed alveolar bone grafts following the regional centralization of secondary alveolar bone grafting to the South West Cleft Centre between 2004 and 2006. A retrospective audit of one surgeon's outcome of 53 consecutively performed alveolar bone grafts, assessed radiographically using the Kindelan method. That 94% of bone-grafted sites achieved a successful radiographic outcome compares favorably with the previously published Clinical Standards Advisory Group data of 58% with Bergland scores. The radiographic outcome for alveolar bone grafting has improved with centralization. A small number of patients are being grafted after the ideal chronological age, and this needs to be addressed throughout the region. The Kindelan assessment provides a reliable method of early assessment for alveolar bone grafting.