Cost-effectiveness of orthodontics: a systematic review.

BACKGROUND: Health economic evaluation is a methodology to maximize health benefits and minimize opportunity costs and is of increasing importance in informing resource allocation decisions in healthcare. OBJECTIVE: This systematic review aims to assess the availability and quality of economic evaluations of any orthodontic interventions and summarize the conclusions of these studies. SEARCH METHODS: A thorough search of the literature was carried out including terms related to orthodontic interventions and economic evaluation between January 2000 and February 2022. MEDLINE, EMBASE, SCOPUS, Web of Science, NHS Economic Evaluation Database, and Cochrane were searched. Grey literature was searched and further hand-searching was performed on the reference lists of relevant systematic reviews. SELECTION CRITERIA: Studies on cleft lip and palate surgery and sleep apnoea were excluded due to the multi-disciplinary nature of these conditions which might alter the applicability of the result to orthodontic interventions. Two independent reviewers selected studies for inclusion. DATA COLLECTION AND ANALYSIS: Data extraction was carried out through full text analysis of included studies by two authors. The quality of each study was assessed according to the Drummond 10-point Checklist and the National Institute for Health and Care Excellence (NICE) Quality Appraisal Checklist for Economic Evaluations. Characteristics and conclusions of included articles were descriptively summarized. RESULTS: Sixteen articles met inclusion criteria. Most of the included articles were trial-based studies, with only one model-based study. Orthodontic interventions assessed included crossbite correction, functional appliance treatment and orthognathic surgery. Most studies were carried out in Europe and specifically in Sweden. The quality of included studies was generally low with 69% of studies reported as having serious limitations according to the NICE Checklist. CONCLUSION: This review highlighted a lack of economic evaluations for orthodontic interventions, and limitations of existing economic evaluations. Recommendations on future research are provided. REGISTRATION: The protocol for the systematic review was registered on the NIHR Database (www.crd.york.ac.uk/prospero, CRD42021220419).

Poverty and Risk of Cleft Lip and Palate: An Analysis of United States Birth Data.

BACKGROUND: The relationship between poverty and incidence of cleft lip and cleft palate remains unclear. The authors investigated the association between socioeconomic status and cleft lip with or without cleft palate and cleft palate only in the United States after controlling for demographic and environmental risk factors. METHODS: The U.S. 2016 and 2017 natality data were utilized. Proxies for socioeconomic status included maternal education, use of the Special Supplemental Nutrition Program for Women, Infants, and Children, and payment source for delivery. Multiple logistic regression controlled for household demographics, prenatal care, maternal health, and infant characteristics. RESULTS: Of 6,251,308 live births included, 2984 (0.05 percent) had cleft lip with or without cleft palate and 1180 (0.02 percent) had cleft palate only. Maternal education of bachelor's degree or higher was protective against, and delayed prenatal care associated with, cleft lip with or without cleft palate (adjusted ORs = 0.73 and 1.14 to 1.23, respectively; p < 0.02). Receiving assistance under the Special Supplemental Nutrition Program for Women, Infants, and Children was associated with cleft palate only (adjusted OR = 1.25; p = 0.003). Male sex, first-trimester tobacco smoking, and maternal gestational diabetes were also associated with cleft lip with or without cleft palate (adjusted ORs = 1.60, 1.01, and 1.19, respectively; p < 0.05). Female sex, prepregnancy tobacco smoking, and maternal infections during pregnancy were associated with cleft palate only (adjusted ORs = 0.74, 1.02, and 1.60, respectively; p < 0.05). CONCLUSIONS: Increased incidence of orofacial clefts was associated with indicators of lower socioeconomic status, with different indicators associated with different cleft phenotypes. Notably, early prenatal care was protective against the development of cleft lip with or without cleft palate. CLIINCAL QUESTION/LEVEL OF EVIDENCE: Risk, III.

Considerations for Payment Bundling in Cleft Care.

SUMMARY: Since the introduction of the Bundled Payments for Care Improvement initiative, progress has been made in piloting bundled payment models to improve care coordination and curtail health care expenditures. In light of improvements in patient outcomes and the concomitant reduction in health care spending for certain high-volume and high-cost procedures, such as total joint arthroplasty and breast reconstruction, the authors discuss theoretical considerations for bundling payments for the care of patients with orofacial clefts. The reasons for and against adopting such a payment model to consolidate cleft care, as well as the challenges to implementation, are discussed. The authors purport that bundled payments can centralize components of cleft care and offer financial incentives to reduce costs and improve the value of care provided, but that risk adjustment based on the longitudinal nature of care, disease severity, etiologic heterogeneity, variations in outcomes reporting, and varying definitions of the episode of care remain significant barriers to implementation.

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.

Data analysis of recent cleft lip and palate surgery in Korea using the National Health Insurance Service (NHIS) database.

BACKGROUND: No national epidemiological investigations have been conducted on several aspects of cleft lip and palate surgery. METHODS: The study was performed using the data of 5727 people who underwent cleft lip and palate surgery during the 6-year period from 2013 to 2018 archived by the National Health Information Database (NHID) of the Health Insurance Review and Assessment Service. Disease and operation codes were utilized for the analysis. RESULTS: Most patients underwent surgery during the first 5 years of life. The number of operations per patient remained almost constant during the study period, but hospital costs per patient gradually increased. When hospital costs per patient were subdivided by institution type, they were found to have steadily increased on an annual basis at all types with the exception of hospital .Since 2016, average hospital stay per operation decreased slightly, but the number of outpatient visits tended to increase. CONCLUSIONS: Accessibility to tertiary hospitals has improved and patients increasingly choose to be treated at larger hospitals in metropolitan areas. Furthermore, these tendencies are expected to be maintained. Despite reductions in hospitalization stays, the cost of treatment, including operation costs, have increasing considerably. Changes in the insurance system are required to reduce financial burdens imposed on patients and on the National Health Insurance Service.

Cleft and Craniofacial Team Orthodontic Care in the United States: A Survey of the ACPA.

OBJECTIVE: To better understand the capacity for orthodontic care, service features, and finances among members of the American Cleft Palate-Craniofacial Association (ACPA). DESIGN: Cross-sectional survey. SETTING: ACPA-approved multidisciplinary cleft teams. PARTICIPANTS: Cleft team coordinators. INTERVENTIONS: Coordinators were asked to complete the survey working together with their orthodontists. MAIN OUTCOME MEASURE: Model for orthodontic care. RESULTS: Coordinators from 82 out of 167 teams certified by ACPA completed the survey (response rate = 49.1%). Most orthodontists were private practice volunteers (48%) followed by university/hospital employed (22.8%). Care was often delivered in community private practice facilities (44.2%) or combination of university and private practice facilities (39.0%). Half of teams reported offering presurgical infant orthopedics (PSIO), with nasoalveolar molding being the most common. Cleft/craniofacial patients typically comprise 25% or less of the orthodontists' practices. The presence of a university/hospital-based orthodontist was associated with higher rates of offering PSIO (P < .001) and an increased percentage dedication of their practice to cleft/craniofacial care (P < .001). CONCLUSION: Orthodontic models across ACPA-certified teams are highly varied. The employment of full-time craniofacial orthodontists is less common but is highly correlated with a practice with a high percentage of cleft care and the offering of advanced services such as PSIO. Future work should focus on how to effectively promote such roles for orthodontists to ensure high-level care for cleft/craniofacial patients requiring treatment from infancy through skeletal maturity.

What is the Cost of Free Cleft Surgery in the Middle East?

BACKGROUND: This project explores the costs of cleft lip and/or palate surgeries in Palestine and Sudan, two low- and middle-income countries (LMIC), in the Middle East. Our purpose is to examine the veracity of advertisements from international cleft organizations claiming that "250 US dollars (USD) covers the cost of a single cleft surgery." We hypothesize that the actual cost of surgery is greater than 250 USD. METHODS: Costs for each cleft surgery were organized broadly into 5 categories: hospital charges, personnel (time and money spent for health professionals to travel to LMIC, including lost wages), tests, consumables, and reusables. Each item was priced at market value during the time of data collection. Following itemization of actual costs, we compared the costs per cleft surgery among four surgical practice models: (1) visiting international surgical teams, (2) visiting international surgeon working with local teams, (3) local teams working at government hospitals, and (4) local teams working at private hospitals. RESULTS: Our results suggest that 250 USD is an underestimate of actual costs per cleft surgery in all models. The most expensive model in both Palestine and Sudan was the first model, visiting international teams performing all team functions; the cheapest surgical model in both countries was a local team working at government hospitals. The largest cost for any of these models is travel and lost wages for international team members. Eliminating this single cost (travel) decreases overall cost tremendously, but still does not approach the advertised cost of 250 USD. CONCLUSIONS: We conclude that 250 USD underestimates the actual costs to perform a single cleft surgery in Palestine and Sudan. If international cleft organizations are genuinely committed to creating sustainable international cleft programs, they should focus exclusively on training local professionals to perform surgery in hospitals of their own choosing.

Anaesthesia for cleft lip surgeries in a resource poor setting: techniques, outcome and safety.

INTRODUCTION: Cleft lip and palate is one of the more common congenital malformation and the most common craniofacial anomalies in children. The treatment is expensive and requires specialised care. Access to this care in middle and low income countries is compounded by socioeconomic status of patients and their relation and also the inadequacy of expertise in medical personnel and infrastructure. Objective: the study aimed to review the techniques of anaesthesia used in a low resource setting in terms of the techniques, outcome, and safety. METHODS: This is a retrospective review of 79 cases done in a resource poor setting. Information regarding the patients, surgeries and modes of anaesthesia were retrieved from the case notes. RESULTS: A total of 62 patients were operated with incomplete cleft accounting for 37 (59.7%), complete 23(37.1%), and 2 (3.2%) as bilateral. Forty-six (74.2%) of patients had their surgery done with ketamine anaesthesia without endotracheal intubation, 14 (22.6%) had regional anaesthesia and 2 patients (3.2%) had general anaesthesia with endotracheal intubation. CONCLUSION: This study demonstrates that with careful planning and expertise, cleft lip repair can be done safely in resource poor setting.

The impact and cost-effectiveness of the Amref Health Africa-Smile Train Cleft Lip and Palate Surgical Repair Programme in Eastern and Central Africa.

INTRODUCTION: Cleft lip with or without cleft palate (CLP) is a congenital malformation that causes significant morbidity in low and middle income countries. Amref Health Africa has partnered with Smile Train to provide CLP surgeries since 2006. METHODS: We analyzed anonymized data of 37,274 CLP patients from the Smile Train database operated on in eastern and central Africa between 2006 and 2014. Cases were analyzed by age, gender, country and surgery type. The impact of cleft surgery was determined by measuring averted Disability-Adjusted Life Years (DALYs) and delayed averted DALYs. We used mean Smile Train costs to calculate cost-effectiveness. We calculated economic benefit using the human capital approach and Value of Statistical Life (VSL) methods. RESULTS: The median age at time of primary surgery was 5.4 years. A total of 207,879 DALYs were averted at a total estimated cost of US$13 million. Mean averted DALYs per patient were 5.6, and mean cost per averted DALY was $62.8. Total delayed burden of disease from late age at surgery was 36,352 DALYs. Surgical correction resulted in $292 million in economic gain using the human capital approach and $2.4 billion using VSL methods. CONCLUSION: Cleft surgery is a cost-effective intervention to reduce disability and increase economic productivity in eastern and central Africa. Dedicated programs that provide essential CLP surgery can produce substantial clinical and economic benefits.

Orofacial clefts in Brazil and surgical rehabilitation under the Brazilian National Health System

The objective of this study was to investigate the prevalence of live births with orofacial clefts in Brazil from 2009 to 2013, according to Brazil's federative units and regions, and correlate it with the number of corrective surgery procedures for cleft lip and palate performed through the Brazilian National Health System in the same period. The data were collected from the National Live Birth System (SINASC in Portuguese) and the Hospital Information System (SIH-SUS in Portuguese). The results showed that the average prevalence of oral clefts in the period was 5.86 per 10,000 live births, with differences observed between the federative units and the regions. The correlation between the prevalence of orofacial clefts and the number of medical procedures associated with this anomaly was statistically significant (r=0.94; p <0.001). This result indicates that greater investments are being made in areas with a higher number of cases. The relationship between supply and demand for corrective surgeries suggests that only 18.91% of the live births with orofacial clefts in this period may have received this service under the Brazilian National Health System.

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

OBJECTIVE: 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. DESIGN: Retrospective cross-sectional study using data obtained from the 2003, 2006, and 2009 Kids' Inpatient Database. SETTING: Inpatient. PATIENTS: Children with CL, CP, or CLP undergoing inpatient cleft lip and/or palate surgery. INTERVENTIONS: Inpatient cleft lip and/or palate surgery. MAIN OUTCOME MEASURES: Orofacial cleft surgery estimates, estimates of primary versus revision surgeries, and estimated inflation-adjusted hospitalization costs. RESULTS: 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. CONCLUSIONS: 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.

Minority and Public Insurance Status: Is There a Delay to Alveolar Bone Grafting Surgery?

OBJECTIVE: This study sought to determine the timing of alveolar bone grafting (ABG) surgery among children with cleft lip with or without cleft palate (CL±P) with regard to race and insurance status. DESIGN: A retrospective chart review of consecutive patients receiving ABG surgery was conducted. A multivariate regression model was constructed using predetermined clinical and demographic variables. SETTING: A large, urban cleft referral center. PATIENTS, PARTICIPANTS: Nonsyndromic patients with CL±P were eligible for study inclusion. INTERVENTIONS: ABG surgery using autogenous bone harvested from the anterior iliac crest. MAIN OUTCOME MEASURE: The primary outcome of interest was age at ABG surgery. RESULTS: A total of 233 patients underwent ABG surgery at 8.1 ± 2.3 years of age. African American and Hispanic patients received delayed ABG surgery compared with Caucasian patients by approximately 1 year (P < .05). There was no difference in ABG surgery timing by insurance status (P > .05). CONCLUSIONS: The timing of ABG surgery varied by race but not by insurance status. Greater resources may be needed to ensure timely delivery of cleft care to African American and Hispanic children.

Ethics in Numbers: Auditing Cleft Treatment in Mexico and Beyond.

Plastic surgeons around the globe are implementing projects that mix audit with medical research to ensure and improve the level of care offered to patients with cleft lip and palate. Drawing on recent literature on "audit culture" and the global growth of "performance indicators" as a form of governance, I demonstrate the conjugation of ethics and the production of numerical indicators in cleft treatment. By standardizing documentation, cleft treatment audit programs facilitate evidence-based medicine and a form of reflexive self-governance. However, the abstraction that accompanies standardization is amplified as corollary data practices travel. In emerging as the answer to improving treatment, these projects lock out the politico-economic factors that mediate medical care in resource poor settings. This danger is compounded by the tendency of numerical governance to replace political conversation with technocratic expertise.

Orofacial clefts in Brazil and surgical rehabilitation under the Brazilian National Health System

Abstract The objective of this study was to investigate the prevalence of live births with orofacial clefts in Brazil from 2009 to 2013, according to Brazil’s federative units and regions, and correlate it with the number of corrective surgery procedures for cleft lip and palate performed through the Brazilian National Health System in the same period. The data were collected from the National Live Birth System (SINASC in Portuguese) and the Hospital Information System (SIH-SUS in Portuguese). The results showed that the average prevalence of oral clefts in the period was 5.86 per 10,000 live births, with differences observed between the federative units and the regions. The correlation between the prevalence of orofacial clefts and the number of medical procedures associated with this anomaly was statistically significant (r=0.94; p <0.001). This result indicates that greater investments are being made in areas with a higher number of cases. The relationship between supply and demand for corrective surgeries suggests that only 18.91% of the live births with orofacial clefts in this period may have received this service under the Brazilian National Health System.

Barriers to Reconstructive Surgery in Low- and Middle-Income Countries: A Cross-Sectional Study of 453 Cleft Lip and Cleft Palate Patients in Vietnam.

BACKGROUND: Despite health system advances, residents of low- and middle-income countries continue to experience substantial barriers in accessing health care, particularly for specialized care such as plastic and reconstructive surgery. METHODS: A cross-sectional household survey of patients seeking surgical care for cleft lip and/or cleft palate was completed at five Operation Smile International mission sites throughout Vietnam (Hanoi, Nghe An, Hue, Ho Chi Minh City, An Giang, and Bac Lieu) in November of 2014. RESULTS: Four hundred fifty-three households were surveyed. Cost, mistrust of medical providers, and lack of supplies and trained physicians were cited as the most significant barriers to obtaining surgery from local hospitals. There was no significant difference in household income or hospital access between those who had and had not obtained cleft surgery in the past. Fewer households that had obtained cleft surgery in the past were enrolled in health insurance (p < 0.001). Of those households/patients who had surgery previously, 83 percent had their surgery performed by a charity. Forty-three percent of participants did not have access to any other surgical cleft care and 41 percent did not have any other access to nonsurgical cleft care. CONCLUSIONS: The authors highlight barriers specific to surgery in low- and middle-income countries that have not been previously addressed. Patients rely on charitable care outside the centralized health care system; as a result, surgical treatment of cleft lip and palate is delayed beyond the standard optimal window compared with more developed countries. Using these data, the authors developed a more evidence-based framework designed to understand health behaviors and perceptions regarding reconstructive surgical care.

Not All Clefts Are Created Equal: Patterns of Hospital-Based Care Use among Children with Cleft Lip and Palate within 4 Years of Initial Surgery.

BACKGROUND: This study compares hospital-based care and associated charges among children with cleft lip, cleft palate, or both, and identifies subgroups generating the greatest cumulative hospital charges. METHODS: The authors conducted a retrospective cohort study of cleft lip, cleft palate, or cleft lip and palate who underwent initial surgery from 2006 to 2008 in four U.S. states. Primary outcome was hospital-based care-emergency, outpatient, inpatient-within 4 years of surgery. Regression models compared outcomes and classification tree analysis identified patients at risk for being in the highest quartile of cumulative hospital charges. RESULTS: The authors identified 4571 children with cleft lip (18.2 percent), cleft palate (39.2 percent), or cleft lip and palate (42.6 percent). Medical comorbidity was frequent across all groups, with feeding difficulty (cleft lip, 2.4 percent; cleft palate, 13.4 percent; cleft lip and palate, 6.0 percent; p < 0.001) and developmental delay (cleft lip, 1.8 percent; cleft palate, 9.4 percent; cleft lip and palate, 3.6 percent; p < 0.001) being most common. Within 30 days of surgery, those with cleft palate were most likely to return to the hospital (p < 0.001). Hospital-based care per 100 children within 4 years was lowest among the cleft lip group, yet comparable among those with cleft palate and cleft lip and palate (p < 0.001). Cumulative 4-year charges, however, were highest among the cleft palate cohort (cleft lip, $56,966; cleft palate, $106,090; cleft lip and palate, $91,263; p < 0.001). Comorbidity, diagnosis (cleft lip versus cleft palate with or without cleft lip), and age at initial surgery were the most important factors associated with the highest quartile of cumulative hospital charges. CONCLUSIONS: Cleft lip and palate children experience a high rate of hospital-based care early in life, with degree of medical comorbidity being a significant burden. Understanding this relationship and associated needs may help deliver more efficient, patient-centered care.

Economic Valuation of the Global Burden of Cleft Disease Averted by a Large Cleft Charity.

BACKGROUND: This study attempts to quantify the burden of disease averted through the global surgical work of a large cleft charity, and estimate the economic impact of this effort over a 10-year period. METHODS: Anonymized data of all primary cleft lip and cleft palate procedures in the Smile Train database were analyzed and disability-adjusted life years (DALYs) calculated using country-specific life expectancy tables, established disability weights, and estimated success of surgery and residual disability probabilities; multiple age weighting and discounting permutations were included. Averted DALYs were calculated and gross national income (GNI) per capita was then multiplied by averted DALYs to estimate economic gains. RESULTS: 548,147 primary cleft procedures were performed in 83 countries between 2001 and 2011. 547,769 records contained complete data available for the study; 58 % were cleft lip and 42 % cleft palate. Averted DALYs ranged between 1.46 and 4.95 M. The mean economic impact ranged between USD 5510 and 50,634 per person. This corresponded to a global economic impact of between USD 3.0B and 27.7B USD, depending on the DALY and GNI values used. The estimated cost of providing these procedures based on an average reimbursement rate was USD 197M (0.7-6.6 % of the estimated impact). CONCLUSIONS: The immense economic gain realized through procedures focused on a small proportion of the surgical burden of disease highlights the importance and cost-effectiveness of surgical treatment globally. This methodology can be applied to evaluate interventions for other conditions, and for evidence-based health care resource allocation.