Age at Primary Cleft Lip Repair: A Potential Bellwether Indicator for Pediatric Surgery.

The bellwether procedures described by the Lancet Commission on Global Surgery represent the ability to deliver adult surgical services after there is a clear and easily made diagnosis. There is a need for pediatric surgery bellwether indicators. A pediatric bellwether indicator would ideally be a routinely performed procedure, for a relatively common condition that, in itself, is rarely lethal at birth, but that should ideally be treated with surgery by a standard age. Additionally, the condition should be easy to diagnose, to minimize the confounding effects of delays or failures in diagnosis. In this study, we propose the age at primary cleft lip (CL) repair as a bellwether indicator for pediatric surgery. METHOD: We reviewed the surgical records of 71,346 primary cleft surgery patients and ultimately studied age at CL repair in 40,179 patients from 73 countries, treated by Smile Train partners for 2019. Data from Smile Train's database were correlated with World Bank and WHO indicators. RESULTS: Countries with a higher average age at CL repair (delayed access to surgery) had higher maternal, infant, and child mortality rates as well as a greater risk of catastrophic health expenditure for surgery. There was also a negative correlation between delayed CL repair and specialist surgical workforce numbers, life expectancy, percentage of deliveries by C-section, total health expenditure per capita, and Lancet Commission on Global Surgery procedure rates. CONCLUSION: These findings suggest that age at CL repair has potential to serve as a bellwether indicator for pediatric surgical capacity in Lower- and Middle-income Countries.

The Smile Index: Part 2. A Simple, Prognostic Severity Scale for Unilateral Cleft Lip.

BACKGROUND: There is no universally accepted classification system for unilateral cleft lip that objectively quantifies the spectrum of disease, making it difficult to evaluate postoperative outcomes in the context of preoperative severity. METHODS: Anthropometric measurements and photographs were prospectively collected from unilateral cleft lip patients in Morocco, Bolivia, Vietnam, and Madagascar. Columellar angle, cleft width, nostril widths, vertical lip heights, and horizontal vermillion lengths were measured preoperatively and postoperatively. "Unacceptable" postoperative outcomes were defined as those with a cleft-side/non-cleft-side vertical lip height discrepancy greater than 3 mm, based on previous sociologic and cleft outcome studies. RESULTS: Of the 147 patients studied, 22 had unacceptable outcomes. Univariate logistic and multivariate logistic stepwise models showed that among preoperative characteristics, cleft width ratio (preoperative cleft width divided by commissure width) was the most significant predictor for unacceptable outcomes, controlling for surgeon experience. Cleft width ratio was normally distributed. Two severity categories were created based on iterative data and regression analysis: "severe" (cleft width ratio >0.5) and "not-severe" (cleft width ratio <0.5). Severe patients had a higher likelihood of unacceptable outcomes versus not-severe patients (OR, 2.9; 95 percent CI, 1.1 to 7.7; p = 0.029; 27 percent versus 11 percent). The probability of having unacceptable outcomes for severe individuals was higher versus not-severe individuals (positive predictive value, 73 percent versus 89 percent). CONCLUSIONS: Preoperative cleft width ratio greater than 0.5 is associated with having an unacceptable surgical outcome. The authors propose a simple, objective, and clinically reproducible scale to unify the language of unilateral cleft lip severity, as a step toward improving algorithms of care, directing surgical technique, guiding patient/family discussions, and optimizing patient outcomes. CLINICAL QUESTION/LEVEL OF EVIDENCE: Risk, III.

The Smile Index: Part 1. A Large-Scale Study of Phenotypic Norms for Preoperative and Postoperative Unilateral Cleft Lip.

BACKGROUND: Unilateral cleft lip has a spectrum of disease morphology, but severity classifications are difficult given the absence of accessible, objective assessment tools or reference data. The authors characterize the spectrum of cleft morphology before and after surgical repair for a large, multi-ethnic population using easily identifiable facial landmarks collected through a novel smart phone-based application. METHODS: Anthropometric measurements and standardized photographs were prospectively collected in Morocco, Bolivia, Vietnam, and Madagascar during medical missions in 2015 using an application designed specifically for the study. After data collection, two experienced cleft surgeons and two laypersons subjectively ranked photographs based on the degree of deformity/aesthetics. RESULTS: One hundred forty-seven patients were analyzed. Mean preoperative cleft width ratio was 0.4 ± 0.12. Nasolabial symmetry improved significantly from preoperatively to postoperatively for the following measurements: columellar angle (65 ± 17 degrees to 87 ± 8 degrees), nostril width ratio (1.7 ± 0.68 to 1.0 ± 0.22), philtral height ratio (0.8 ± 0.14 to 1.0 ± 0.14), and lip length ratio (0.9 ± 0.26 to 1.0 ± 0.11) (p < 0.001). Surgeon and layperson rankings showed high inter-rater reliability (r = 0.64, p < 0.001). Preoperatively, multivariate regression showed that cleft width ratio, nostril width ratio, and philtral height ratio were predictive of rank (p < 0.01). Postoperatively, philtral height ratio was most predictive of rank (p = 0.0097). Most cleft characteristics were not significantly different between countries. CONCLUSIONS: The authors present simpler, more straightforward measures to quantify preoperative and postoperative morphology/aesthetics and introduce a novel technology to streamline and standardize measurements to make data collection more accessible.

Patient Barriers to Accessing Surgical Cleft Care in Vietnam: A Multi-site, Cross-Sectional Outcomes Study.

BACKGROUND: Most people who lack adequate access to surgical care reside in low- and lower-middle-income countries. Few studies have analyzed the barriers that determine the ability to access surgical treatment. We seek to determine which barriers prevent access to cleft care in a resource-limited country to potentially enable barrier mitigation and improve surgical program design. METHODS: A cross-sectional, multi-site study of families accessing care for cleft lip and palate deformities was performed in Vietnam. A survey instrument containing validated demographic, healthcare service accessibility, and medical/surgical components was administered. The main patient outcome of interest was receipt of initial surgical treatment prior to 18 months of age. RESULTS: Among 453 subjects enrolled in the study, 216 (48%) accessed surgical care prior to 18 months of age. In adjusted regression models, education status of the patient's father (OR 1.64; 95% CI 1.1-2.5) and male sex (OR 1.61; 95% CI 1.1-2.4) were both associated with timely access to care. Distance and associated cost of travel, to either the nearest district hospital or to the cleft surgical mission site, were not associated with timing of access. In a sensitivity analysis considering care received prior to 24 months of age, cost to attend the surgical mission was additionally associated with timely access to care. CONCLUSIONS: Half of the Vietnamese children in our cohort were not able to access timely surgical cleft care. Barriers to accessing care appear to be socioeconomic as much as geographical or financial. This has implications for policies aimed at reaching vulnerable patients earlier.

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.

A Model of the Unmet Need for Cleft Lip and Palate Surgery in Low- and Middle-Income Countries.

BACKGROUND: There is a significant unmet need for the cleft lip and/or palate (CL/P) care in low- and middle-income countries (LMICs) ; however, country-level estimates that can be used to inform local and international cleft care program strategies are lacking. METHODS: Using data from Operation Smile surgical programs in twelve LMICs and country-level indicators from the World Health Organization and World Bank, we developed a model to estimate the proportion of individuals with CL/Ps older than respective surgery age targets for cleft lip and cleft palate surgery (1 and 2 years, respectively). After extrapolating this model to other LMICs with available indicator data, we combined these findings with estimates of CL/P prevalence among live births to estimate the total number of unrepaired CL/P cases in LMICs worldwide. RESULTS: The models were constructed from a total of 887 cases of cleft palate and 576 cases of cleft lip across the twelve countries. From these, we estimated that there are 616,655 cases of unrepaired CL/P (95 % CI 564,893-678,503) in the 113 countries with available data for extrapolation. The rate of unrepaired CL/Ps ranged from 2.5 per 100,000 population in Romania to 28.5 per 100,000 in Cambodia, respectively (median rate 10.7 per 100,000 population). CONCLUSIONS: Our model provides marked insight into the global surgical backlog due to cleft lip and palate. While the most populated LMICs have the largest number of unrepaired CL/Ps, low-income countries with relatively less healthcare infrastructure have exceptionally high rates (e.g., Cambodia, Afghanistan, and Nepal). These estimates can be used by local and international cleft care organizations to set program priorities, estimate resource requirements, and inform strategies to support cleft care.

The Global Surgery Partnership: An Innovative Partnership for Education, Research, and Service.

PROBLEM: An estimated two billion people worldwide lack access to adequate surgical care. Addressing surgical disparities requires both immediate relief efforts and long-term investments to improve access to care and surgical outcomes, train the next generation of surgical professionals, and expand the breadth of formative research in the field. While models exist for establishing short-term surgical missions in low- and middle-income countries, far less focus has been placed on models for multi-institutional partnerships that support the development of sustainable solutions. APPROACH: In 2011, the Global Surgery Partnership (GSP) was founded by an established children's hospital (Children's Hospital Los Angeles), an academic medical center (University of Southern California), and a nonprofit organization (Operation Smile) to build oral cleft surgical capacity in resource-poor settings through education, research, and service. OUTCOMES: Leveraging the strengths of each partner, the GSP supports three global health education programs for public health graduate students and surgical residents, including the Tsao Fellowship in Global Health; has initiated two international research projects on cleft lip and palate epidemiology; and has built upon Operation Smile's service provision. As of January 2015, Tsao fellows had operated on over 600 patients during 13 missions in countries including China, Vietnam, Mexico, and India. NEXT STEPS: The GSP plans to conduct a formal evaluation and then to expand its programs. The GSP encourages other global health organizations and academic and medical institutions to engage with each other. The partnership described here provides a basic model for structuring collaborations in the global health arena.

A Health Systems Perspective on the Mission Model for Cleft Lip and Palate Surgery: A Matter of Sustainability or Responsibility?

One in 700 children around the world are born with cleft lip and/or palate (CL/P). Although reconstructive surgery is widely available in high-income settings, over 2 billion people in low- and middle-income countries lack access to essential surgical care. The mission model has been demonstrated to be highly effective in responding to the global surgical workforce crisis, but has been questioned in regard to its sustainability, value, and overall impact. Through effective health systems integration, the mission model presents abundant opportunities for streamlined delivery and horizontal impact. Still, the primary goal of the mission model is direct care delivery; and although the value of sustainability is indisputably vital, we contend that the mission model, when executed responsibly, creates high-value, sustained impact on the individual lives of those presently in need. We furthermore advocate for the sustained commitment of implementing organizations, patient safety, local integration, and a new focus on patient centeredness as key elements of the responsible mission model.