Incomes to Outcomes: A Global Assessment of Disparities in Cleft and Craniofacial Treatment.

OBJECTIVE: Recent investigations focused on health equity have enumerated widespread disparities in cleft and craniofacial care. This review introduces a structured framework to aggregate findings and direct future research. DESIGN: Systematic review was performed to identify studies assessing health disparities based on race/ethnicity, payor type, income, geography, and education in cleft and craniofacial surgery in high-income countries (HICs) and low/middle-income countries (LMICs). Case reports and systematic reviews were excluded. Meta-analysis was conducted using fixed-effect models for disparities described in three or more studies. SETTING: N/A. PATIENTS: Patients with cleft lip/palate, craniosynostosis, craniofacial syndromes, and craniofacial trauma. INTERVENTIONS: N/A. RESULTS: One hundred forty-seven articles were included (80% cleft, 20% craniofacial; 48% HIC-based). Studies in HICs predominantly described disparities (77%,) and in LMICs focused on reducing disparities (42%). Level II-IV evidence replicated delays in cleft repair, alveolar bone grafting, and cranial vault remodeling for non-White and publicly insured patients in HICs (Grades A-B). Grade B-D evidence from LMICs suggested efficacy of community-based speech therapy and remote patient navigation programs. Meta-analysis demonstrated that Black patients underwent craniosynostosis surgery 2.8 months later than White patients (P < .001) and were less likely to undergo minimally-invasive surgery (OR 0.36, P = .002). CONCLUSIONS: Delays in cleft and craniofacial surgical treatment are consistently identified with high-level evidence among non-White and publicly-insured families in HICs. Multiple tactics to facilitate patient access and adapt multi-disciplinary case in austere settings are reported from LMICs. Future efforts including those sharing tactics among HICs and LMICs hold promise to help mitigate barriers to care.

Sociodemographic Factors Predict Long-Term Attrition from Multidisciplinary Cleft Clinic.

OBJECTIVE: Identify and describe factors associated with retention and attrition of patients during longitudinal follow-up at multidisciplinary cleft clinic. DESIGN: Retrospective cohort study. SETTING: Single, tertiary care center. PATIENTS, PARTICIPANTS: Patients born between 1995 and 2007 with a diagnosis of cleft palate with or without cleft lip attending multidisciplinary cleft clinic. INTERVENTIONS: None tested, observational study. MAIN OUTCOME MEASURE(S): Age at last clinical appointment with a multidisciplinary cleft team provider. Attrition was defined as absence of an outpatient appointment following 15 years of age. RESULTS: Six hundred seventy-eight patients were included. The average age at last appointment across the entire cohort was 13.1 years (IQR 6.6-17.2). Patients who were Black (HR 1.60, 95% CI 1.10-2.32, p = 0.014) and other races (HR 1.90, 95% CI 1.22-2.98, p = 0.004) were more likely to be lost to follow-up compared to white patients. Publicly insured patients were more likely to experience attrition than those who were privately insured (HR 1.30, 95% CI 1.03-1.65, p = 0.030). Estimated income was not significantly associated with length of follow-up (p = 0.259). Those whose residence was in the fourth quartile of driving distance from our center experienced loss to follow-up significantly more than those who lived the closest (HR 2.04, 95% CI 1.50-2.78, p < 0.001). CONCLUSIONS: There is a high degree of follow-up attrition among patients with cleft lip and palate. Race, insurance status, and driving distance to our center were associated with attrition in a large, retrospective cohort of patients who have reached the age of cleft clinic graduation.

Comprehensive Long-Term Outcomes Following Mandibular Distraction Osteogenesis.

OBJECTIVE: To describe long-term outcomes and complications following mandibular distraction osteogenesis (MDO) in a diverse patient cohort. DESIGN: Cross-sectional study. SETTING: Single tertiary-care pediatric center. PATIENTS: Forty-eight patients previously undergoing MDO with minimum 4-year follow-up. MAIN OUTCOME MEASURES: Respiratory outcomes, feeding patterns, dental development, motor/sensory nerve function, temporo-mandibular joint function, and postsurgical scarring. RESULTS: Forty-six patients with a median age of 7 years were evaluated. Of 20 nonsyndromic patients, none required additional airway procedures, none required continuous positive airway pressure (CPAP) during sleep, and 19 (95%) fed exclusively by mouth. Among 26 syndromic patients, 7 (27%) required CPAP and 8 (31%) were tube fed. Permanent first molar differences were seen in the majority of subjects; patterns of damage interfering with function were more common in syndromic (13/28, 46%) compared to nonsyndromic (5/24, 21%; P = .014) subjects. MDO prior to age two was associated with more frequent and worse dental damage (P = .001). Inferior alveolar nerve and marginal mandibular nerve function were fully intact in 37 (80%) and 39 (85%) of patients, respectively. Three patients (6%), all with associated genetic syndromes, demonstrated severe nerve impairment. By the Vancouver scar scale, ≥ 80% of surgical scars were rated in the most favorable category for each quality assessed. Temporomandibular joint dysfunction was rare. CONCLUSIONS: MDO shows highly favorable long-term respiratory, feeding, nerve, and scar outcomes in nonsyndromic patients, although permanent molar changes not precluding tooth viability are commonly seen. Patients with associated syndromes demonstrate respiratory and feeding benefits, but higher rates of dental and nerve abnormalities.

A Retrospective and Prospective Cohort Study Comparing Pediatric Patients With Cleft Lip and Palate From the United States and Guatemala.

Orofacial clefts (OFC) remain among the most prevalent congenital abnormalities worldwide. In the United States in 2010 to 2014, 16.2 of 10,000 live births are born with OFC compared with 23.6 of 10,000 in Alta Verapaz, Guatemala in 2012. Demographics and cleft severity scores were retrospectively gathered from 514 patients with isolated OFC at the Children's Hospital of Philadelphia scheduled for surgery from 2012 to 2019 and from 115 patients seen during surgical mission trips to Guatemala City from 2017 to 2020. Risk factors were also gathered prospectively from Guatemalan families. The Guatemalan cohort had a significantly lower prevalence of cleft palate only compared with the US cohort, which may be a result of greater cleft severity in the population or poor screening and subsequent increased mortality of untreated cleft palate. Of those with lip involvement, Guatemalan patients were significantly more likely to have complete cleft lip, associated cleft palate, and right-sided and bilateral clefts, demonstrating an increased severity of Guatemalan cleft phenotype. Primary palate and lip repair for the Guatemalan cohort occurred at a significantly older age than that of the US cohort, placing Guatemalan patients at increased risk for long-term complications such as communication difficulties. Potential OFC risk factors identified in the Guatemalan cohort included maternal cooking-fire and agricultural chemical exposure, poor prenatal vitamin intake, poverty, and risk factors related to primarily corn-based diets. OFC patients who primarily rely on surgical missions for cleft care would likely benefit from more comprehensive screening and investigation into risk factors for more severe OFC phenotypes.

Incorporation and Utilization of an Additional Needs Screener by Surgical Trainees for Comprehensive Care of Underserved and Underinsured Surgical Patients.

OBJECTIVE: Comprehensive, socially-minded healthcare has historically been delivered in the primary care setting. For underserved patient populations, however, a surgical care episode may serve as the health care access point. To maximize patient wellbeing during the perioperative period, our surgical center developed the Additional Needs Screener (ANS). Operationalized into practice by GME and UME trainees, this tool screens surgical patients across 3 domains (social, emotional, and immigration needs) and connects patients to partner organizations if appropriate. This study describes the pilot utilization of the ANS among underserved and underinsured surgical patients. DESIGN: Clinical quality improvement and retrospective cohort study of patients completing the ANS from implementation in September 2021 to September 2022. SETTING: The Hospital of the University of Pennsylvania, PA-a tertiary care center. PARTICIPANTS: One hundred and 10 underinsured and/or underserved patients completed at least 1 ANS domain. RESULTS: Patients were majority female (55F, 53M, 2 other) and Hispanic/Latinx (72%) with a median age of 38 (IQR = 34-48). Most patients spoke a primary language other than English (77%), and nearly all were either uninsured (82%) or received emergency medical assistance or Medicaid (14%) at referral. Patients demonstrated significant needs; 39% endorsed difficulty affording housing, 32% endorsed difficulty paying for food, 29% endorsed experiencing current life-interfering distress, and 75% had undocumented immigration status. Ultimately, 57% of screened patients accepted referrals to our needs response teams. CONCLUSIONS: Underserved and underinsured patients presenting for surgical care face significant challenges relating to social, emotional, and immigration needs. Through adoption of the ANS, trainees gained competency identifying and addressing these barriers in the perioperative period. Future works will focus on categorizing referral outcomes, developing interventions to increase patient trust, and improving screener dissemination.

Geospatial and Socioeconomic Factors Interact to Predict Management and Outcomes in Cleft Lip and Palate Surgery: A Single Institution Study of 740 Patients.

OBJECTIVE: Determine interactions between geospatial and socioeconomic factors influencing cleft lip and/or cleft palate (CL/P) management and outcomes. DESIGN: Retrospective review and outcomes analysis (n = 740). SETTING: Urban academic tertiary care center. PATIENTS: 740 patients undergoing primary (CL/P) surgery from 2009 to 2019. MAIN OUTCOMES MEASURES: Prenatal evaluation by plastic surgery, nasoalveolar molding, cleft lip adhesion, and age at CL/P surgery. RESULTS: Prenatal evaluation by plastic surgery was predicted by the interaction between higher patient median block group income and shorter patient distance from the care center (OR = 1.07, p = 0.022). Nasoalveolar molding was also predicted by the interaction between higher patient median block group income and shorter distance from the care center (OR = 1.28, p = 0.016), whereas cleft lip adhesion was predicted by higher patient median block group income alone (OR = 0.41, p < 0.001). Lower patient median block group income predicted later age at cleft lip (ß = -67.25, p = 0.011) and cleft palate (ß = -46.35, p = 0.050) repair surgery. CONCLUSIONS: Distance from the care center and lower median income by block group interacted to significantly predict prenatal evaluation by plastic surgery and nasoalveolar molding for patients with CL/P at a large, urban, tertiary care center. Patients living farthest from the care center who received prenatal evaluation by plastic surgery or who underwent nasoalveolar molding had higher median block group income. Future work will determine mechanisms perpetuating these barriers to care.

Impact of Illustrated Postoperative Instructions on Knowledge and Retention During a Cleft Lip and Palate Surgical Mission.

OBJECTIVE: To determine the impact of illustrated postoperative instructions on patient-caregiver knowledge and retention. DESIGN: Prospective study with all participants receiving an educational intervention. SETTING: Pediatric plastic surgical missions in Guatemala City, Guatemala, between 2019 and 2020. PARTICIPANTS: A total of 63 majority-indigenous Guatemalan caregivers of patients receiving cleft lip and/or palate surgery. INTERVENTION: Illustrated culturally appropriate postoperative care instructions were iteratively developed and given to caregivers who were surveyed on illustration-based and text-based information at preoperative, postoperative, and four-week follow-up time points. MAIN OUTCOME MEASURE: Postoperative care knowledge of illustration-based versus text-based information as determined by the ability to answer 11 illustration- and 8 text-based all-or-nothing questions, as well as retention of knowledge as determined by the same survey given at four weeks follow-up. RESULTS: Scores for illustration-based and text-based information both significantly increased after caregivers received the postoperative instructions (+13.30 ± 3.78 % SE, + 11.26 ± 4.81 % SE; P < .05). At follow-up, scores were unchanged for illustration-based (-3.42 ± 4.49 % SE, P > .05), but significantly lower for text-based information (-28.46 ± 6.09 % SE, P < .01). Retention of text-based information at follow-up correlated positively with education level and Spanish literacy, but not for illustration-based. CONCLUSIONS: In the setting of language and cultural barriers on a surgical mission, understanding of illustration-based and text-based information both increased after verbal explanation of illustrated postoperative instructions. Illustration-based information was more likely to be retained by patient caregivers after four weeks than text-based information, the latter of which correlated with increased education and literacy.

Introducing Surgical Equity into Contemporary Medical Education: Results From Operation Equity, a Pilot Curriculum.

OBJECTIVE: To date, education about health equity for early-stage healthcare trainees is largely situated outside of surgical disciplines. This study aims to evaluate the effectiveness of a surgical equity curriculum offered to a voluntary group of medical and graduate students. DESIGN: Mixed-methods cohort study from January to June 2021. Pre- and post-course surveys measured domains of attitudes, self-reported confidence, and knowledge via 5-point Likert scale and multiple-choice questions. Paired t tests were used to analyze quantitative responses. Qualitative responses were studied via iterative thematic analysis. SETTING: At the University of Pennsylvania in Philadelphia, PA which provides tertiary level, institutional care, 10, interdisciplinary 1.5-hour sessions were held over 1 semester, teaching surgical equity topics that spanned the peri-operative continuum. PARTICIPANTS: Twenty-four medical and graduate students from across the University of Pennsylvania enrolled. Twenty completed both surveys. RESULTS: From pre- to post-course, students improved across all domains. Students improved in their self-rated ability to identify strategies to talk about sensitive health topics with patients (pre: 20%, post: 90%) and identify strategies to address healthcare disparities in surgery (pre: 10%, post: 90%). Qualitatively, from pre- to post-course, more students could articulate the role of bias and identify opportunities for surgeons to engage in surgical equity. The course strengthened any pre-existing interest in surgical equity, and for 1 student, created interest in a surgical career where it had not previously existed. Many also expressed greater resolve to provide patient-centric care. CONCLUSIONS: Formal curricula can improve students' ability to advocate for surgical equity. A similar framework may fill a need for medical students interested in health equity and surgical careers at other institutions.

Machine Learning in Metopic Craniosynostosis: Does Phenotypic Severity Predict Long-Term Esthetic Outcome?

BACKGROUND: There have been few longitudinal studies assessing the effect of preoperative phenotypic severity on long-term esthetic outcomes in metopic craniosynostosis. This study evaluates the relationship between metopic severity and long-term esthetic outcomes using interfrontal angle (IFA) and CranioRate, a novel metopic synostosis severity measure. METHODS: Patients with metopic craniosynostosis who underwent bifrontal orbital advancement and remodeling between 2012 and 2017 were reviewed. Preoperative computed tomography head scans were analyzed for IFA and CranioRate, a machine learning algorithm which generates quantitative severity ratings including metopic severity score (MSS) and cranial morphology deviation (CMD). Long-term esthetic outcomes were assessed by craniofacial surgeons using blinded 3-rater esthetic grading of clinical photos. Raters assessed Whitaker score and the presence of temporal hollowing, lateral orbital retrusion, frontal bone irregularities and/or "any visible irregularities." RESULTS: Preoperative scans were performed at a mean age of 7.7±3.4 months, with average MSS of 6/10, CMD of 200/300, and IFA of 116.8±13.8 degrees. Patients underwent bifrontal orbital advancement and remodeling at mean 9.9±3.1 months. The average time from operation to esthetic assessment was 5.4±1.0 years. Pearson correlation revealed a significant negative correlation between MSS and age at computed tomography ( r =-0.451, P =0.004) and IFA ( r =-0.371, P =0.034) and between IFA and age at surgery ( r =-0.383, P =0.018). In multinomial logistic regression, preoperative MSS was the only independent predictor of visible irregularities (odds ratio=2.18, B =0.780, P =0.024) and preoperative IFA alone significantly predicted Whitaker score, with more acute IFA predicting worse Whitaker score (odds ratio=0.928, B =-0.074, P =0.928). CONCLUSIONS: More severe preoperative phenotypes of metopic craniosynostosis were associated with worse esthetic dysmorphology. Objective measures of preoperative metopic severity predicted long-term esthetic outcomes.

Prenatal Diagnosis of Cleft and Craniofacial Conditions: An International Analysis of Practice Patterns.

ABSTRACT: Prenatal diagnosis of congenital anomalies is associated with increased parental stress. Surgical prenatal counseling for parents expecting a child with an orofacial cleft has been shown to reduce parental anxiety through education and expectation setting. Despite the prevalence and significance of these conditions throughout the world, little is known of international counseling practices as they pertain to clefts and other craniofacial conditions, specifically regarding topics of counseling and discussions of ethical issues including interruption of pregnancy (IOP).In this study, 50 members of the International Society of Craniofacial Surgery, representing 18 countries, were surveyed. The questionnaire included questions about the number of consultations performed, for which conditions, setting of practice, and regional regulations pertaining to IOP. The authors report that the most frequent topics of discussion included initial treatment of clefts (89%), future surgeries (89%), and speech/language difficulties (85%). North American surgeons more frequently discussed cleft appearance (100%) thansurgeons from Central/SouthAmerica (64%) or Europe (64%, P = 0.019). Overall, 46% of surgeons surveyed believe it is ethical to give parents their opinion on IOP when asked. European surgeons were more likely to report that parents want to receive information about IOP at prenatal consultations (82%), compared to 25% from North America and 8% from Central/South America ( P < 0.001). The authors conclude that despite a lack of standard guidelines on conducting prenatal counseling appointments for cleft and craniofacial conditions, discussion topics were similar across country, practice type, and surgeon experience.

A Quantification of Scalp Thickness before and after Posterior Vault Distraction Osteogenesis.

SUMMARY: Previous literature has documented craniometric changes of the bony calvaria, increases in intracranial volume, and resolution of Chiari malformations following posterior vault distraction osteogenesis. No studies have analyzed changes to the soft-tissue envelope after posterior vault distraction osteogenesis. In this study, the authors aimed to provide objective measurements of scalp thickness in patients undergoing posterior vault distraction osteogenesis, utilizing facial soft-tissue thickness as a proxy to control for growth. The authors hypothesized that the soft tissues of the scalp are not made thinner by the distraction process, either in the region of distraction or in neighboring areas. Subjects who underwent posterior vault distraction osteogenesis for a craniosynostosis diagnosis who had high-resolution predistraction and post-distractor removal computed tomographic scans within 100 days of each operation were included. The scans were analyzed on Materialise Mimics version 21 software (Materialise, Ghent, Belgium). Six key craniometric landmarks (glabella, pogonion, zygion, vertex, euryon, and opisthocranium) were identified on the three-dimensional bone masks in the Frankfort horizontal plane. The points were overlaid onto soft-tissue thickness masks and thicknesses were recorded. Percent change in postoperative facial soft-tissue thickness was used to control for growth of the craniofacial soft-tissue envelope during the study interval. The preoperative and postoperative posterior vault distraction osteogenesis cohorts did not differ significantly at the glabella, pogonion, opisthocranium, vertex, and zygion. The corrected median thickness at the euryon was significantly increased in the post-posterior vault distraction osteogenesis cohort [2.67 mm (IQR, 2.49 mm to 4.02 mm) versus 5.26 mm (IQR, 3.83 mm to 7.82 mm), p = 0.002]. This is the first study to quantify changes in soft-tissue thicknesses preoperatively and postoperatively in patients undergoing posterior vault distraction osteogenesis, demonstrating maintenance of scalp thickness. . CLINICAL QUESTION/LEVEL OF EVIDENCE: Therapeutic, IV.

Postnatal outcomes and risk factor analysis for patients with prenatally diagnosed oropharyngeal masses.

OBJECTIVES: To describe our experience treating prenatally diagnosed oropharyngeal masses in a novel, multidisciplinary collaboration. To identifying outcomes and risk factors associated with adverse postnatal outcomes. METHODS: This is a sixty-two patient case series at an academic referral center. Patients with prenatally diagnosed oropharyngeal masses were identified through a programmatic database and confirmed in the electronic health record. RESULTS: Sixty-two patient with prenatally diagnosed oropharyngeal mass were identified, with prenatal imaging at our institution confirming this diagnosis in fifty-seven patients, short term outcomes analysis conducted on forty-four patients, and long-term outcomes analysis conducted on seventeen patients. The most common pathology was lymphatic malformations (n = 27, 47.4%), followed by teratomas (n = 22, 38.6%). The median mass volume from all available patient imaging (n = 57) was 60.54 cm3 (range 1.73-742.5 cm3). Thirteen pregnancies were interrupted, six infants expired, and thirteen cases had an unknown fetal outcome. Confirmed mortality was 6/57 patients with imaging-confirmed oropharyngeal masses (10.5%). Fourteen (56%) of the surviving patients (n = 25) were delivered by Ex Utero Intrapartum Treatment (EXIT) procedure and the median NICU stay was thirty-six days (range: 3-215 days). There was no association between airway compression/deviation/displacement, stomach size, polyhydramnios, or mass size and mortality. Seventeen patients had more than one year of follow-up (mean 5.3 ± 2.4 years). These seventeen patients underwent general anesthesia a total of ninety-two times (mean 5.4 ± 4.3) and had a total of twenty-three mass-related surgeries. The great majority of patients required an artificial airway at birth, feeding support, and speech/swallow therapy. CONCLUSIONS: Oropharyngeal mass involvement of key anatomic structures-the neck, upper thorax, orbit, and ear, has a greater association with mortality than mass size. Regardless of the size and involved structures, oropharyngeal masses are associated with a high burden of intensive medical care and surgical care beginning at or before birth.

The Center for Surgical Health: A Compassion-Driven Urban Surgical Mission.

We are the Center for Surgical Health (CSH), an academic community partnership that supports, educates, and advocates for vulnerable Philadelphians with surgical diseases, founded in 2016 by Dr. Jon B. Morris, a leader in surgical education and a general surgeon at the University of Pennsylvania, and Dr. Alan Herbst, a current third-year Penn general surgery resident. At the time, Dr. Morris, raised in a Reform Jewish household, had been participating in an RCIA Program to convert to Catholicism. The mission of providing surgery to uninsured patients, primarily undocumented individuals, by helping them obtain insurance and see Penn providers was seen by Dr. Morris as a form of Catholic charity, which he has continued to remain dedicated to as his faith in Jesus Christ has deepened. Dr. Herbst, now Associate Director of Clinics for the CSH, recalls working with Dr. Morris as a sub-intern during his conversion, beginning with passion and a neon poster board inviting people to "See the Surgeon." Since that time, the CSH has grown from an organization with 10 volunteers, called "personal patient navigators," who provide insurance support and advocacy at every step of the perioperative continuum, to one with over 50, who have now seen 156 patients and assisted in providing 49 needed procedures. Much of this growth has been brought about through the dedication and vision of Dr. Matthew Goldshore, the Deputy Director of the CSH and a fifth-year Penn general surgery resident, as well as Dr. Carrie Z. Morales, Associate Deputy Director of the CSH and a recent Perelman School of Medicine graduate. Through their leadership, and the talent and commitment of other members of the CSH board, overseen by Director Dr. Morris, the CSH now has policy and research divisions, a surgical equity curriculum, and continues to develop new ways of providing better care.

Risk factor analysis and outcomes of airway management in antenatally diagnosed cervical masses.

PURPOSE: To investigate antenatally-determined imaging characteristics associated with invasive airway management at birth in patients with cervical masses, as well as to describe postnatal management and outcomes. STUDY DESIGN: A retrospective analysis of 52 patients with antenatally diagnosed neck masses was performed using single-center data from January 2008 to January 2019. Antenatal imaging, method of delivery, management, and outcomes data were abstracted from the medical record and analyzed. RESULTS: Antenatal diagnosis of neck masses in this cohort consisted of 41 lymphatic malformations (78.8%), 6 teratomas (11.5%), 3 hemangiomas (5.8%), 1 hemangioendothelioma (1.9%), and 1 giant foregut duplication cyst (1.9%). Mean gestational age at time of diagnostic imaging was 29 weeks 3 days (range: 19w4d - 37w). Overall, 22 patients (42.3%) required invasive airway management at birth, specifically 18 patients (34.6%) required endotracheal intubation and 4 (7.7%) required tracheostomy. 15 patients (28.8%) underwent ex-utero intrapartum treatment (EXIT) for the purposes of securing an airway. Polyhydramnios, tracheal deviation and compression, and anterior mass location on antenatal imaging were significantly associated with incidence of invasive airway intervention at birth, EXIT procedure, and tracheostomy during the neonatal hospitalization (p < 0.025; Fisher's exact test). Logistic regression analysis demonstrated statistically significant association between increasing antenatally-estimated mass volume and incidence of invasive airway management at birth (p = 0.02). Post-natal cervical mass management involved surgical excision (32.7%), sclerotherapy (50%), and adjuvant therapy with rapamycin (17.3%). Demise in the neonatal period occurred in 4 (7.7%) patients. CONCLUSION: This series documents the largest single-center experience of airway management in antenatally diagnosed cervical masses. Fetal imaging characteristics may help inform the appropriate method of delivery, airway management strategy at birth, and prenatal counseling.