A multi-centric, single-blinded, randomized, parallel-group study to evaluate the effectiveness of nasoalveolar moulding treatment in non-syndromic patients with complete unilateral cleft lip, alveolus and palate (NAMUC study): a study protocol for a randomized controlled trial.

BACKGROUND: Cleft lip and palate (CLP) are among the most common congenital anomaly that affects up to 33,000 newborns in India every year. Nasoalveolar moulding (NAM) is a non-surgical treatment performed between 0 and 6 months of age to reduce the cleft and improve nasal aesthetics prior to lip surgery. The NAM treatment has been a controversial treatment option with 51% of the cleft teams in Europe, 37% of teams in the USA and 25 of cleft teams in India adopting this methodology. This treatment adds to the already existing high burden of care for these patients. Furthermore, the supporting evidence for this technique is limited with no high-quality long-term clinical trials available on the effectiveness of this treatment. METHOD: The NAMUC study is an investigator-initiated, multi-centre, single-blinded randomized controlled trial with a parallel group design. The study will compare the effectiveness of NAM treatment provided prior to lip surgery against the no-treatment control group in 274 patients with non-syndromic unilateral complete cleft lip and palate. The primary endpoint of the trial is the nasolabial aesthetics measured using the Asher McDade index at 5 years of age. The secondary outcomes include dentofacial development, speech, hearing, cost-effectiveness, quality of life, patient perception, feeding and intangible benefits. Randomization will be carried out via central online system and stratified based on cleft width, birth weight and clinical trial site. DISCUSSION: We expect the results from this study on the effectiveness of treatment with NAM appliance in the long term along with the cost-effectiveness evaluation can eliminate the dilemma and differences in clinical care across the globe. TRIAL REGISTRATION: ClinicalTrials.gov CTRI/2022/11/047426 (Clinical Trials Registry India). Registered on 18 November 2022. The first patient was recruited on 11 December 2022. CTR India does not pick up on Google search with just the trial number. The following steps have to be carried out to pick up. How to search: ( https://ctri.nic.in/Clinicaltrials/advsearch.php -use the search boxes by entering the following details: Interventional trial > November 2022 > NAMUC).

Innovative Therapeutic Approaches for the Treatment of the Ocular Morbidities in Patients with EEC Syndrome.

Ectrodactyly-Ectodermal dysplasia-Clefting (EEC) syndrome is caused by heterozygous missense point mutations in the p63 gene, an important transcription factor during embryogenesis and for stem cell differentiation in stratified epithelia. Most of the cases are sporadic, related to de novo mutations arising during early-stage development. Familial cases show an autosomic dominant inheritance. The major cause of visual morbidity is limbal stem cell failure, which develops in the second to third decade of life. Patients often show ocular surface alterations, such as recurrent blepharitis and conjunctivitis, superficial microlesions of the cornea, and spontaneous corneal perforation and ulceration, leading to progressive corneal clouding and eventually visual loss. No definitive cures are currently available, and treatments to alleviate symptoms are only palliative. In this review, we will discuss the proposed therapeutic strategies that have been tested or are under development for the management of the ocular defects in patients affected by EEC syndrome: (i) gene therapy-based approaches by means of Allele-Specific (AS) siRNAs to correct the p63 mutations; (ii) cell therapy-based approaches to replenish the pool of limbal stem cells; and (iii) drug therapy to correct/bypass the genetic defect. However, as the number of patients with EEC syndrome is too limited, further studies are still necessary to prove the effectiveness (and safety) of these innovative therapeutic approaches to counteract the premature differentiation of limbal stem cells.

A Systematic Review of the Cost-Effectiveness of Cleft Care in Low- and Middle-Income Countries: What is Needed?

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.

The First Hybrid International Educational Comprehensive Cleft Care Workshop.

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.

Craniofacial Orthodontic Experience in CODA-Accredited Orthodontic Residency Programs.

To evaluate orthodontic care for patients with craniofacial anomalies (CFA) by identifying orthodontic residents' preparedness to treat certain conditions and willingness to receive more training in CFA.A 12-question survey was sent through the American Association of Orthodontics (AAO) organization to orthodontic residents. Questions were primarily designed to obtain information on the frequency with which they dealt with patients with CFA in their training, specific craniofacial conditions that orthodontic residents feel comfortable treating.A total of 150 participants out of 1066 responded. Of the 150 responses, 35% were first-year residents, 43% second year, and 22% were third-year residents. Thirty nine percent of residents saw 3 or more CFA patients during their residency followed by 24% that saw no patients with CFA. Forty five percent reported that 1 to 3 hours of lecture time was devoted to CFA per month. Sixty percent felt their training in CFA was not sufficient to feel comfortable treating these patients in practice. Specifically, 62% felt comfortable treating Down syndrome, 84% unilateral cleft lip and/or palate, and 64% bilateral cleft lip and/or palate, while the majority did not feel comfortable treating Pierre Robin sequence (68%), Cleidocranial dysplasia (65%), Crouzon syndrome (75%), Pfeiffer syndrome (80%), Treacher Collins syndrome (76%), Apert syndrome (76%), CHARGE syndrome (84%), and DiGeorge sequence (84%). Seventy eight percent of residents reported that they would like more training in treating craniofacial.Orthodontic residents did not feel comfortable treating patients with CFA. Majority of the residents felt that they would like to learn more about CFA.

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.

ABSTRACT: 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% ±â€Š21.2%). 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.

Occlusal Outcome of Orthodontic Treatment for Patients With Complete Cleft Lip and Palate.

AIM: To assess occlusal outcomes of orthodontic treatment for patients with complete cleft lip and palate. DESIGN: Retrospective assessment using the Peer Assessment Rating (PAR) index. SETTING: Consecutive patients treated by one consultant orthodontist at a tertiary care cleft center. PARTICIPANTS: One hundred twenty-seven patients with either complete unilateral cleft lip and palate (UCLP) or bilateral cleft lip and palate (BCLP) consecutively treated with fixed appliances. INTERVENTION: Fixed orthodontic appliance treatment and orthognathic surgery when required. OUTCOMES: The PAR index assessment was carried out by a calibrated-independent assessor. Treatment duration, the number of patient visits, and data on dental anomalies were drawn from patient records and radiographs. RESULTS: One hundred two patients' study models were assessed after exclusions. Mean start PAR score for UCLP (n = 71) was 43.9 (95% CI, 41.2-46.6, SD 11.5), with a mean score reduction of 84.3% (95% CI, 81.9-86.7, SD 10.1). The UCLP mean treatment time was 23.7 months with 20.1 appointments. Mean start PAR score for BCLP (n = 31) was 43.4 (95% CI, 39.2-47.6, SD 11.4), with a mean score reduction of 80.9% (95% CI, 76.3-85.5, SD 12.5). The BCLP mean treatment time was 27.8 months with 20.5 appointments. CONCLUSION: These results compare well with other outcome reports, including those for patients without a cleft, and reflect the standard of care provided by an experienced cleft orthodontist. As with high-volume surgeons, orthodontic treatment for this high need group is favorable when provided by a high-volume orthodontist. These findings may be used for comparative audit with similar units providing cleft care.

Long-term treatment outcomes from the perspective of a patient with unilateral cleft lip and palate.

The management of patients with orofacial cleft (OFC) often extends from diagnosis or birth well into adulthood and requires many different specialists within multidisciplinary teams (MDT). The aims of treatment are to restore form and function relating to hearing, speech, occlusion and facial aesthetics. People with OFCs that include the lip, alveolus and palate (cleft lip and palate (CLP)) require several different staged and coordinated surgical and non-surgical interventions, and the treatment pathway is associated with a heavy burden of care. Due to the extensive nature of the interaction with these patients, MDT members have opportunities to provide enhanced patient-centred care and support. This case report provides an overview of the current knowledge of the aetiology of OFC and the management of these patients. It provides a unique perspective from one of the coauthors who has a unilateral CLP (UCLP) and reports on his treatment experiences and long-term treatment outcomes. By having a better understanding of the impact of UCLP and treatment provided, MDT members can not only provide improved clinical treatment but also offer improved patient experiences for those with craniofacial anomalies, in particular, an increased awareness of the psychosocial challenges, they endure throughout their treatment pathway and beyond.

Gingivosupraperiosteoplasty following Presurgical Maxillary Orthopedics Is Associated with Normal Midface Growth in Complete Unilateral and Bilateral Cleft Patients at Mixed Dentition.

BACKGROUND: Passive orthodontic appliances and gingivosupraperiosteoplasty are adjuncts that can be used by surgeons at the time of primary cleft lip repair. These treatments, along with the surgical technique of cleft lip and palate repair, may impact midface growth. The objective of this study was to describe the authors' protocol for unilateral and bilateral cleft lip repair and to evaluate midfacial growth in a cohort of patients at mixed dentition who had undergone presurgical passive orthodontic appliance therapy and gingivosupraperiosteoplasty at the time of unilateral and bilateral cleft lip repair. METHODS: Fifteen complete unilateral and 15 complete bilateral cleft lip and palate patients underwent passive orthodontic appliance treatment and primary lip repair with gingivosupraperiosteoplasty. Lateral cephalograms were analyzed by three blinded reviewers. Mean cephalometric measurements at mixed dentition were compared to cephalometric values for noncleft patients, unilateral cleft lip and palate patients who did not undergo gingivoperiosteoplasty or presurgical treatment, and unilateral cleft lip and palate patients who underwent gingivoperiosteoplasty/nasoalveolar molding with independent samples t tests. RESULTS: Mean cephalometric values were within age-specific normal values for sella-nasion-A point, sella-nasion-B point, A point-nasion-B point, and facial axis. Eighty-seven (13/15) percent of unilateral cleft lip and palate patients and 93 percent (14/15) of bilateral cleft lip and palate patients did not exhibit skeletal class III malocclusion. There was no significant difference between cephalometric values for our patients and patients who did not receive gingivosupraperiosteoplasty or presurgical treatment or who underwent the gingivoperiosteoplasty/nasoalveolar molding protocol. CONCLUSIONS: Presurgical passive orthodontic appliances, combined with gingivosupraperiosteoplasty at the time of lip repair, leads to normal maxillary development in most patients at mixed dentition. Assessment of midface growth at skeletal maturity is required. CLINICAL QUESTION/LEVEL OF EVIDENCE: Therapeutic, IV.

A Comparative Assessment of Nasal Appearance following Nasoalveolar Molding and Primary Surgical Repair for Treatment of Unilateral Cleft Lip and Palate.

BACKGROUND: Although many cleft teams have adopted nasoalveolar molding to improve nasal form, few comparative studies have assessed the postoperative benefits of this treatment. Given that reported outcomes have been contradictory and that treatment involves considerable burden to families, the purpose of this study was to assess objective and subjective changes from nasoalveolar molding at approximately 5 years of age. METHODS: All patients with complete unilateral cleft lip and palate who underwent primary cheiloplasty performed by a single surgeon over a 7-year period were reviewed. Patient results were grouped into nasoalveolar molding or no-nasoalveolar molding. Cleft severity and aesthetic outcomes were assessed by panels of raters who independently ranked subject images at presentation, immediately preoperative (after molding), and at 5-year follow-up. Objective symmetry was measured using standard anthropometric analysis on three-dimensional images. RESULTS: Among 41 patients included, 16 successfully completed nasoalveolar molding. Both groups were similar at presentation; however, the nasoalveolar molding group had improved appearance following molding (p < 0.05). After surgery, at 5 years of age, the nasoalveolar molding group had better rank scores for overall appearance (p < 0.05), cleft nostril height, and cleft medial lip height (p < 0.05). Regression analysis revealed that nasoalveolar molding treatment was the most significant predictor of overall nasal appearance at 5 years, but that treatment team experience and initial severity were also significant predictors (p < 0.05). Qualitative audit following analysis identified favorable and unfavorable features of nasoalveolar molding. CONCLUSION: In children with complete unilateral cleft lip and palate, nasoalveolar molding was associated with better overall nasal aesthetics and improved cleft nostril height and cleft medial lip height at approximately 5 years of age. CLINICAL QUESTION/LEVEL OF EVIDENCE: Therapeutic, III.

Ortopedia prequirúrgica en el tratamiento de pacientes con fisura labio y paladar / Presurgical orthopedics in the treatment of patients with cleft lip and palate

RESUMEN: Introducción: Dentro del tratamiento estándar de los pacientes con fisura labio-máxilo-palatinas se encuentra la ortopedia prequirúrgica. Esta se realiza con el objetivo de modular la posición, alineación y estabilización de los segmentos maxilares previo a la primera cirugía. Pese a que es ampliamente utilizada, su efectividad es controversial. Métodos: Se realizó una búsqueda en Epistemonikos, la mayor base de datos de revisiones sistemáticas en salud, la cual es mantenida mediante el cribado de múltiples fuentes de información, incluyendo MEDLINE, EMBASE, Cochrane, entre otras. Se extrajeron los datos desde las revisiones identificadas, se analizaron los datos de los estudios primarios, posteriormente se realizó un metaanálisis y se preparó una tabla de resumen de los resultados utilizando el método GRADE. Resultados y Conclusiones: Se identificaron 14 revisiones sistemáticas que en conjunto incluyeron 57 estudios primarios, de los cuales, cinco corresponden a ensayos clínicos aleatorizados. Concluimos que no es posible establecer con claridad si la ortopedia prequirúrgica mejora o perjudica la morfología maxilar, debido a que la certeza de la evidencia ha sido evaluada como muy baja. Por su parte, la ortopedia prequirúrgica podría resultar en poca o nula diferencia en el crecimiento y desarrollo facial, las alteraciones oclusales, la satisfacción parental, la alimentación, el habla y la apariencia facial y nasolabial, pero la certeza de la evidencia es baja.

ABSTRACT: Introduction: Within the standard treatment of patients with labio-maxillo-palatal cleft is presurgical orthopedics. This procedure seeks to modulate the position, alignment and stabilization of the maxillary segments prior to the first surgery. Although it is widely used, its effectiveness is currently controversial. Methods: We searched in Epistemonikos, the largest database of systematic reviews in health, which is maintained by screening multiple information sources, including MEDLINE, EMBASE, Cochrane, among others. We extracted data from the systematic reviews, reanalyzed data of primary studies, conducted a meta-analysis and generated a summary of findings table using the GRADE approach. Results and conclusions: We identified 14 systematic reviews that included 57 primary studies, five of which correspond to randomized trials. We conclude that it is uncertain whether presurgical orthopedics improves or deteriorates maxillary morphology, because the certainty of the evidence has been very low. On the other hand, presurgical orthopedics may make little or no difference to facial growth and development, occlusal alterations, parental satisfaction, feeding, speech, and facial and nasolabial appearance, but the certainty of the evidence is low.

Engineered mucoperiosteal scaffold for cleft palate regeneration towards the non-immunogenic transplantation.

Cleft lip and palate (CL/P) is the most prevalent craniofacial birth defect in humans. None of the surgical procedures currently used for CL/P repair lead to definitive correction of hard palate bone interruption. Advances in tissue engineering and regenerative medicine aim to develop new strategies to restore palatal bone interruption by using tissue or organ-decellularized bioscaffolds seeded with host cells. Aim of this study was to set up a new natural scaffold deriving from a decellularized porcine mucoperiosteum, engineered by an innovative micro-perforation procedure based on Quantum Molecular Resonance (QMR) and then subjected to in vitro recellularization with human bone marrow-derived mesenchymal stem cells (hBM-MSCs). Our results demonstrated the efficiency of decellularization treatment gaining a natural, non-immunogenic scaffold with preserved collagen microenvironment that displays a favorable support to hMSC engraftment, spreading and differentiation. Ultrastructural analysis showed that the micro-perforation procedure preserved the collagen mesh, increasing the osteoinductive potential for mesenchymal precursor cells. In conclusion, we developed a novel tissue engineering protocol to obtain a non-immunogenic mucoperiosteal scaffold suitable for allogenic transplantation and CL/P repair. The innovative micro-perforation procedure improving hMSC osteogenic differentiation potentially impacts for enhanced palatal bone regeneration leading to future clinical applications in humans.

Barriers and Resources to Cleft Lip and Palate Speech Services Globally: A Descriptive Study.

BACKGROUND: Although a number of international cleft organizations and cleft professionals in low- and middle-income countries (LMICs) have built and supported comprehensive cleft care and speech therapy models to address the shortage of speech services in LMICs, the specific speech needs of individuals with cleft lip and palate (CLP) in such countries remain unknown. The objective of this study was to evaluate the barriers to accessing speech services for patients with CLP as well as the resources and models of speech services that are currently available for individuals with CLP in LMICs, with the goal of better understanding the needs of this population. METHODS: Qualitative and quantitative methods consisted of Smile Train partner surveys that were distributed June 25th to July 31st, 2018 worldwide. Surveys were distributed through Smile Train's online medical database, Smile Train Express, which every Smile Train partner uses to report their Smile Train sponsored treatment outcomes. A total of 658 Smile Train partners responded to the surveys. Respondents included surgeons, speech therapists, orthodontists, administrators and nurses who represented non-governmental organizations, hospitals (private or public), hospital groups, and private clinics. RESULTS: Results indicated that lack of resources, including access to local speech providers and language materials, as well as financial constraints such as patient travel and speech treatment costs, are the most commonly reported barriers to accessing speech services across all geographic regions surveyed. CONCLUSIONS: Improving access to CLP speech services in LMICs may require strategies that address lack of speech providers, language materials, and financial constraints.

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

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.

Treatment of Unilateral Cleft Lip and Palate Patient With Intraoral Tooth Tissue Borne Distractor and Facemask Therapy: A Case Report.

This case report demonstrates the successful use of intraoral distractor/hygenic rapid expander (HYRAX) for rapid maxillary expansion in anteroposterior direction with an adjunctive use of face mask therapy for anterior orthopedic traction of maxillary complex in a cleft patient with concave profile. The patient was a 13-year-old girl who reported with a chief complaint of backwardly positioned upper jaw and a severely forward positioned lower jaw. Therefore, a treatment was chosen in which acrylic bonded rapid maxillary expansion was done with tooth tissue borne intraoral distractor/HYRAX having a different activation schedule along with Dr Henri Petit facemask to treat maxillary retrognathism. As a result, crossbite got corrected and attained a positive jet with no bone loss in cleft area over a period of 5 months which was followed by fixed mechanotherapy achieving a well settled occlusion in 1 year. After completion of expansion and fixed mechanotherapy, ANB became +1 post-treatment which was -4 pretreatment. The prognathic profile was markedly improved by expansion and taking advantage of the remaining growth potential, thus minimizing the chances of surgery later in life. This provided a viable alternative to orthognathic surgery with good long-term stability.

Telemedicine in International Cleft Care: A Systematic Review.

BACKGROUND: Cleft lip and cleft palate (CLP) are among the world's most common congenital malformation and has a higher prevalence in developing nations due to environmental and genetic factors. Global efforts have been developed in order to prevent and treat the malformation. Telemedicine has been implemented in various humanitarian global missions with success and is currently the primary means of care due to COVID-19. OBJECTIVE: To assess the benefits and barriers of telehealth in the care of patients with CLP through a global approach. METHODS: Systematic review of the PubMed and Cochrane Review databases with relevant terms related to telemedicine in cleft lip and palate surgery. RESULTS: Eight articles fit the inclusion criteria and suggested benefits with the use of telemedicine in regard to education, preoperative, and postoperative care as well as increased access to underserved populations. Barriers included connectivity and accessibility concerns. CONCLUSION: Telehealth is a beneficial way to evaluate patients with CLP in developing countries with proper care and follow-up to reduce complications and to improve health outcomes.

Time-Driven, Activity-Based Costing of Presurgical Infant Orthopedics: A Critical Component of Establishing Value of Latham Appliance and Nasoalveolar Molding.

BACKGROUND: Value-based health-care reform requires assessment of outcomes and costs of medical interventions. In cleft care, presurgical infant orthopedics is still being evaluated for clinical benefits and risks; however, the cost of these procedures has been largely ignored. This study uses robust accounting methods to quantify the cost of providing two types of presurgical infant orthopedics: Latham appliance treatment and nasoalveolar molding. METHODS: This is a prospective study of patients with nonsyndromic cleft lip and/or palate who underwent treatment with presurgical infant orthopedics from 2017 to 2019 at two academic centers. Costs were measured using time-driven activity-based costing. Personnel costs, facility costs (operating room, clinic, and inpatient ward), and equipment costs were included. Travel expenses were incorporated as an estimate of direct costs borne by the family, but indirect costs (e.g., time off from work) were not considered. RESULTS: Twenty-three patients were treated with Latham appliance treatment and 14 were treated with nasoalveolar molding. For Latham appliance treatment, average total cost was $7553 per patient ($1041 for personnel, $637 for equipment, $4871 for facility, and $1004 for travel over 6.5 visits). Unilateral and bilateral costs were $6891 and $8860, respectively. For nasoalveolar molding, average cost totaled $2541 ($364 for personnel, $151 for equipment, $300 for facility, and $1726 for travel over 13 visits); $2120 for unilateral and $3048 for bilateral treatment. CONCLUSIONS: The major difference in cost is attributable to operative placement of the Latham device. Travel cost for nasoalveolar molding is often higher because of frequent clinical encounters required. Future investigation should focus on whether outcomes achieved by presurgical infant orthopedics justify the $2100 to $8900 expenditure for these adjunctive procedures.

Cleft Lip and Palate YouTube Videos: Content Usefulness and Sentiment Analysis.

OBJECTIVE: This study aimed to describe the content of YouTube videos about cleft lip and palate (CLP) and to describe the sentiment analysis of related comments. DESIGN: Retrospective, YouTube video, and sentiment analysis. METHODS: YouTube videos (N = 112) about CLP were examined, including demographics and ratings of quality and usefulness. Comments on CLP (N = 205) were classified by content, and measured from negative (-5) to positive (+5) with a sentiment analysis program. RESULTS: Videos were uploaded most often by universities and hospitals (25%) and health information sites (22%). Most videos were educational (49.1%) or described patient experiences (48.2%). The most frequent video topics were terminology, CLP treatment, and prenatal diagnosis information. Based on the comprehensiveness of CLP topics covered, videos were generally rated by authors as moderately useful (31.5%) or very useful (35.7%). Comments were mostly made by people with CLP (79%), and the median sentiment scores were +3, and -2 for positive and negative sentiments, respectively. General content categories were positive comments (24.8%), provided information about themselves (24.4%), negative comments (19.5%), comments on usefulness of video (17.6%), or advice (9.3%). While 36.1% of comments noted no difficulties, the remaining comments included difficulties with surgery pain (13.2%), psychological state (12.2%), embarrassment (11.7%), physical appearance (10.2%), long term therapy (9.8%), and speech problems (6.8%). CONCLUSIONS: Although useful and educational YouTube videos were available, the variability of content quality suggests that caution be used in relying on videos for information and that experts in CLP treatment should be consulted.

Secondary Cleft Lip Induced by Congenital Hemangioma: Report of a Rare Case.

Infant hemangioma is a relatively rare congenital disease. Ulcer and infection may occur in some cases. A few cases may develop into deformity and defect. Herein, we present a case of secondary cleft lip induced by congenital hemangioma and also our sequential treatment.

Treatment Experiences in Adults Born With Cleft Lip and/or Palate: A Whole of Life Survey in the United Kingdom.

OBJECTIVES: Adults with cleft lip and/or palate (CL/P) may report dissatisfaction with aesthetic and/or functional outcomes and express desire for further treatment. Although medical intervention can improve quality of life, surgical procedures require complex decision-making and can invoke psychological distress. The aim of this study was to gain a better understanding of ongoing treatment-related support needs by examining self-reported treatment experiences of a sample of adults born with CL/P. DESIGN: An online, mixed-methods survey was designed by the Cleft Lip and Palate Association in collaboration with the Centre for Appearance Research at the University of the West of England. A total of 207 eligible responses were received. Qualitative data were analyzed using inductive content analysis, while quantitative data were analyzed using descriptive statistics. RESULTS: Although participants were satisfied with surgical and dental/orthodontic outcomes overall, many reported ongoing difficulties. Some stated they would seek further treatment while others felt conflicted about risks and benefits. Some had refused treatment, citing treatment fatigue, anxiety, and/or concerns that treatment would be unsuccessful. More than 40% of participants were unaware of their entitlement to cleft-related treatment provided by the National Health Service, and many others had experienced difficulties accessing care. CONCLUSIONS: Although advances in health care may offer opportunities for adults to improve their quality of life, treatment decisions should be weighed with the support of a clinical psychologist where available. Guidance for young adults considering future treatment is also recommended. Finally, training and resources for local practitioners may improve general awareness of CL/P services and increase access to specialist care for adults with ongoing CL/P-related concerns.