8:00 AM
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Oronasal Fistulas Over Time: A Systematic Review and Meta-Analysis of Reported Oronasal Fistula Rates in the Literature
Background
Oronasal fistula (ONF) is a well-known complication of primary cleft palate repair which can lead to worsened speech, oral hygiene, and maxillary growth. Secondary closure of ONFs can be quite challenging as well, making the prevention of this complication of great interest. As surgical techniques have become more refined and there has been increasing access to educational resources and care delivery throughout the world, we anticipate there to be a temporal change in ONF rates. Therefore, we sought to evaluate how reported ONF rates in the literature have changed over time and whether site of care or other treatment factors influenced outcomes.
Methods
A systematic review and meta-analysis following PRISMA guidelines was performed of publications between January 1960 to November 2024 that reported fistula rate following cleft palate repair as a primary or secondary outcome. Case studies, case series with less than twenty patients, letters, surveys, and systematic reviews were excluded. Studies that reported a fistula repair rate rather than a fistula occurrence rate were also excluded. Studies reporting on secondary palatoplasty, secondary fistulas, submucous clefts, or syndromic patients were excluded. Study type, year of publication, treatment center type, continent, country, World Bank classification, cohort start and finish date, and fistula rates were all collected.
Results
223 publications with a total of 71,238 patients met inclusion criteria. The average fistula rate across all studies was 11.3% (95% CI: 9.8-12.8%). Most studies in our cohort were published in high-income countries (133, 59.6%), with fewer studies coming from upper-middle-income (44, 19.7%), lower-middle-income (36, 16.1%), and low-income countries (7, 3.1%). Fistula rates were found to vary significantly within our cohort when separated by publication decade (ANOVA, p=0.005), with an initial downward trend in fistula rates over time followed by a trend toward increasing reported fistula rates over the past 20 years. Average reported fistula rate was lowest in studies published in the 2000s (7.25%, [95% CI: 4.54%, 9.97%]), increased in the 2010s (9.98%, [95% CI: 8.07%, 11.9%]), and further increased in the 2020s (14.3%, [95% CI: 11.0%, 17.5%]). The reported fistula rate in studies published in the 2020s was significantly higher than those published in the 2000s (p=0.002) and 2010s (p=0.006). Prior to 2000, all studies in our cohort originated from high- and upper-middle- income countries. Post-2000, there were increasing numbers of studies from low- and middle-income countries, though they remained a minority of studies included.
Conclusions
Rates of fistula following cleft palate repair declined from early reports in the 1960s through the 2000s as expected but have interestingly shown a recent increase over the last two decades, as suggested by study publication year. This may suggest that in the early 21st century, surgical techniques and care significantly improved, or that publication bias may have been present in terms of greater reporting positive outcomes, though this warrants further investigation. Additionally, the rise of global health efforts and publications may account for this increase in reported fistula rates, leading to a more accurate depiction of cleft care outcomes globally.
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8:05 AM
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Public Insurance as a Predictor of Postoperative Emergency Department Utilization in Craniosynostosis Repair
Background: Postoperative emergency department (ED) visits are common across various procedures, often due to surgical complications, pain, and patient uncertainty about normal surgical recovery.(1-3) While major postoperative complications such as infections or dehiscence require acute medical management in the ED setting, many post-operative visit reasons are driven by minor postoperative symptoms such as fever or swelling.(1-3) In this study, we aim to identify the most common reasons for acute visits to the ED within 30 days following craniosynostosis surgery, with the intent of guiding improvements in postoperative care instructions, reducing postoperative ED visits and improving healthcare utilization.
Methods: A retrospective chart review of patients who underwent surgical repair of craniosynostosis at a tertiary children's hospital between 2003 and 2023 was performed. Patients with incomplete documentation and those lost to follow-up were excluded. The primary outcome was incidence of ED visits within a 30-day period after surgery. Secondary outcomes included duration until ED visit, visit indication, and insurance status.
Results: Among 794 patients who underwent surgical craniosynostosis repair, 38 (4.8%) required hospital-based acute care in the ED within 30 days postoperatively. The mean patient age was 9.0±3.4 months, with the majority being male (n=25, 65.8%). All patients underwent calvarial vault remodeling, with 13 (34.2%) who additionally had fronto-orbital advancement. The median time from surgery to ED visit was 9.5±7.7 days. The most common reasons included fever (8 visits, 21%), vomiting (7 visits, 18%), swelling (6 visits, 16%), and surgical site issues such as drainage, redness, or infection (6 visits, 16%). Other indications included surgical site bleeding (2 visits, 5%), trauma (2 visits, 5%), suture check (1 visit, 2.6%), ear pain (1 visit, 2.6%), seizure (1 visit, 2.6%), and upper respiratory tract viral illness (1 visit, 2.6%). There was a higher proportion of patients with public insurance compared to private insurance in the ED cohort versus those with no ED visits (p=0.011).
Conclusion: Fever, vomiting, and surgical site concerns are the primary drivers of ED visits after discharge from craniosynostosis surgery. Specific measures, such as providing caregivers with a clear postoperative fever management algorithm, educating families on recognizing normal versus abnormal symptoms, and scheduling early follow-up visits could potentially reduce avoidable ED visits.
References:
1. Kocher KE, Nallamothu BK, Birkmeyer JD, Dimick JB. Emergency department visits after surgery are common for Medicare patients, suggesting opportunities to improve care. Health Aff (Millwood). 2013;32(9):1600-1607. doi:10.1377/hlthaff.2013.0067
Toth M, Holmes M, Van Houtven C, Toles M, Weinberger M, Silberman P. Rural Medicare Beneficiaries Have Fewer Follow-up Visits and Greater Emergency Department Use Postdischarge [published correction appears in Med Care. 2015 Oct;53(10):908]. Med Care. 2015;53(9):800-808. doi:10.1097/MLR.0000000000000401
Vashi AA, Fox JP, Carr BG, et al. Use of hospital-based acute care among patients recently discharged from the hospital. JAMA. 2013;309(4):364-371. doi:10.1001/jama.2012.216219
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8:10 AM
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Predicting Nutritional Needs and Growth Patterns in Patients with Robin Sequence Following Mandibular Distraction Osteogenesis
Background:
Mandibular distraction osteogenesis (MDO) effectively treats airway obstruction in patients with Robin Sequence (RS), while promoting structural changes that facilitate more efficient feeding mechanisms and promote growth in infancy. Many children still require supplementation via gastrostomy tubes (G-tubes) and/or parenteral nutrition. Additionally, the temporal pattern of weight gain following MDO is poorly understood, particularly in the context of syndromic RS. The purpose of this study was to evaluate predictors of nutritional supplementation and weight trajectories of patients with syndromic and non-syndromic RS post-MDO.
Methods:
A retrospective review was conducted of RS patients who underwent MDO at a major children's hospital from May 2004 to November 2023. Those with less than one year of follow-up were excluded. Demographics, syndromic diagnoses, and feeding routes were collected. Weight-for-length (WFL) percentiles were recorded at birth, at time of MDO, and at one, three, six, and twelve months post-operatively. Observed and expected average daily weight gain (ADWG) were calculated using each patient's nearest standardized weight-for-length growth curve from their birth percentile. The percent difference between observed and expected ADWG was determined, with a positive value indicating greater weight gain than expected.
Results:
A total of 94 patients met inclusion criteria (20 in syndromic RS cohort, 74 in isolated RS cohort). Median corrected gestational age was 1.0 month at time of MDO surgery. Overall, 41.5% received a G-tube within the study period. Full oral intake was achieved at a median of 39 days post-MDO. Patients with syndromic RS required more time to achieve full oral intake than those with isolated RS (55 days vs. 37 days, p=0.039). Multivariable logistic regression revealed greater odds of G-tube placement in patients with sRS (p=0.011) and those born preterm (p=0.002). Initial mean WFL percentiles decreased from birth to time of MDO, but increased to above 50th percentile by 12 months post-MDO. At 12 months post-MDO, the syndromic RS cohort had significantly lower mean WFL percentiles (p=0.001). Despite this, the ADWG percent differences did not vary between syndromic and isolated RS cohorts from MDO to 12 months postoperatively (p=0.49).
Conclusion:
This study revealed significant improvements in oral intake and weight gain in patients with RS who underwent MDO, with both isolated and syndromic RS patients demonstrating positive growth trajectories. However, patients with syndromic diagnoses and preterm birth had greater G-tube requirements and required more time to achieve full oral intake. While those with syndromic RS exhibited lower WFL percentiles postoperatively, their growth aligned with expected ADWG, suggesting that with appropriate support, favorable outcomes are attainable in this population. Ultimately, tailored postoperative nutrition plans and close monitoring are critical for high-risk patients, particularly those with syndromic RS. These findings provide valuable insights and will aid physicians in counseling families on anticipated feeding and growth trajectories for patients with RS following MDO.
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8:15 AM
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Comparison of Clinical and Patient Reported Outcomes for Velopharyngeal Insufficiency after Cleft Palate Repair
Purpose:
After cleft palate repair, children are followed for clinical surveillance of velopharyngeal insufficiency (VPI), occurring in up to 30% of patients. While speech and language pathologists (SLP) clinically diagnose VPI, it may not always impact a patient's quality of life (QoL) enough to pursue additional management. In this study, we sought to determine if lower QoL, as measured by the validated Velopharyngeal Life Outcomes (VELO) instrument, is associated with the decision for further treatment of VPI.
Methods:
An IRB-approved cross-sectional study was performed with English- and Spanish-speaking patients presenting for routine follow up at a single institution's multidisciplinary cleft clinic (8/12/2024-12/16/2024). Caregivers completed the VELO questionnaire in clinic. Clinical evidence of VPI was defined as documentation of VPI or hypernasality in the most recent SLP note. Demographic and clinical data were extracted from electronic medical records. Patients were grouped into the following three categories based on SLP recommendations: scope/medical management for VPI recommended but family was uninterested, scope/medical management planned for VPI, and no scope/medical management recommended. Bivariate tests were used to measure associations between VELO scores and clinically evident VPI, treatment recommendation, insurance type, sex, race, cleft severity, syndromic cleft, age at primary repair (< or > 12 months), and type of primary repair.
Results:
In the final cohort (N=54), 51.9% were female and 88.9% White. Nearly half were insured by Medicaid (48.1%). Mean age at the time of the survey was 7.9 years (SD=3.0). More than half (55.8%) underwent palatoplasty before 12 months of age, and most (74.5%) underwent Furlow primary palatoplasty. Medicaid insurance (p=0.02) and clinical evidence of VPI (p<0.01) were associated with lower VELO scores. Among patients with clinical evidence of VPI, median VELO score of patients with scope/medical management planned was significantly lower (56.250) compared with those who were recommended scope/medical management but were uninterested (78.846) and those who were not offered scope or medical management (70.192) (p = 0.049). Median VELO scores were similar in the latter two groups (p=0.80).
Conclusion:
The impact of VPI on QoL adds critical context for decisions surrounding secondary speech surgery. Patients who pursued additional treatment for VPI had lower QoL as measured by the VELO instrument. However, those whose QoL was less affected by their VPI did not pursue further treatment, despite recommendations by the SLP. This emphasizes the need for shared decision-making and reveals a potential role for standardized QoL measurement in routine cleft care. Additionally, payor status negatively impacts access to care and may create barriers to speech therapy and surgery, thus necessitating close monitoring to ensure optimal surgical care.
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8:20 AM
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A Multi-Institution Analysis of Insurance Status Effects on Alveolar Bone Graft Timing
Objective: Prior studies have shown Medicaid insurance status as a predictor for delays in alveolar bone grafting (ABG) at single institutions (1,2). This study aims to study a larger cohort by combining data across three institutions to determine how insurance status, institutional factors, and patient demographics effect a patient's likelihood of receiving timely alveolar bone grafting. These institutions are in three cities and states with vastly different political leanings, median incomes, racial profiles, and population densities.
Methods: A retrospective analysis of all patients that received alveolar bone grafting was performed across three institutions. A total of 587 patients were included in the analysis, 251 from Institution #1, 225 from Institution #2, and 111 from Institution #3. Demographic data, insurance status, driving distance, and median income was collected. Age at surgery as a linear variable and binary variable of standard timing (<12 years) versus late timing (>12 years) was analyzed using Chi-Square, Cochran-Mantel-Haenszel Test, Factor Analysis of Variance, and multiple logistic regression with a significance assessed at α = 0.05.
Results: Results showed 51% of the cohort was male, the average age at surgery was 10.5 years, 29% received ABG after age 12, and 44% had Medicaid insurance. There were higher proportions of male (67% vs. 53%, p<0.01), Hispanic (23% vs 5.5%, p<0.0001), and Black/African American (11% vs 2%, p<0.0001) patients who had Medicaid rather than private insurance. Those with Medicaid were older 11.02 [9.73, 13.48] at the time of surgery compared to those with private insurance 10.28 [9.32, 11.71], p<0.0001. Those with Medicaid had a higher proportion of late surgeries compared to those with private insurance (38% vs 22%, p<0.0001). Those with Medicaid had lower absolute median income and lower household median income as a ratio to state median income (HI:SI), p<0.0001. Results of the logistic regression with late ABG timing as the outcome showed institution, gender, race, median income, and distance to center were not significant predictors, while insurance status and HI:SI were independently statistically significantly associated with having a delay (p = 0.0015, p= 0.0097).
Conclusion: Our results showed that Medicaid holders were more likely to be male, Hispanic or Black/African American, with lower median household income and have a later age at ABG. Results of this study support that regardless of the institution, patients with Medicaid are more likely to receive their alveolar bone graft after age 12 and that wealth is a predictor of timely care across the country. Further studies are needed to explore how to overcome the delay that patients with Medicaid are facing to provide this patient population with equitable care.
- Bushong EE, Patmon D, Pfershy H, Huffman C, Carlson A, Girotto J. Timing of Alveolar Bone Graft and Barriers to Care. Cleft Palate Craniofac J. Published online April 4, 2024. doi:10.1177/10556656241242695
- Badiee RK, Yang SC, Alcon A, Weeks AC, Rosenbluth G, Pomerantz JH. Disparities in Timing of Alveolar Bone Grafting and Dental Reconstruction in Patients With Clefts. Cleft Palate Craniofac J. 2023;60(5):639-644. doi:10.1177/10556656211073049
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8:25 AM
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Geographic Challenges in Cleft Lip and Palate Care in Oregon: A Mixed-Methods Analysis of Travel Burden and Patient-Reported Challenges
Purpose:
Oregon, a state with a large geographic area and significant rural population, has only one academic medical center offering pediatric subspecialty care. As a result, equitable access to pediatric surgical services remains a public health challenge. The Lancet's Global Surgery 2030 (1) lists timely access to essential surgical services as a key indicator of a functioning health system. Access to some services, including pediatric craniofacial surgery in Oregon, is limited due to geographic and socioeconomic factors. To date, this burden has not been analyzed. This study analyzes geographic access to cleft lip and palate (CLP) care and patient-reported barriers to care in Oregon at an academic center with pediatric fellowship-trained surgeons.
Methods:
Patients ages 0 - 2 years who received comprehensive CLP care from 2005 - 2023 at the single academic center in Oregon were included. The geographic distribution of each patient was recorded based on home address. Average one-way travel distance and number of visits were recorded. Additionally, a survey was sent by email to 380 families of past CLP patients to assess travel burden, financial costs, work leave required, and perceived difficulty of obtaining care. Univariate analysis and t-tests were performed to analyze cohort and survey results.
Results:
357 patients were included in the study. The mean distance each patient traveled one-way to receive CLP care was 94 miles. The median number of in-person visits required over the first two years of life was 10. The average cumulative travel per patient in the first two years exceeded 2,400 miles.
35 families completed the survey. Families who traveled more than 50 miles reported significantly greater perceived difficulty in obtaining care compared to those traveling less than 50 miles (mean difficulty scores: 25.7 vs. 9.7, p<0.05). These families also reported higher travel and related costs. 89% (n=31) of survey respondents missed work to seek care for their child. While 68% (n=15) of those traveling more than 50 miles indicated they would utilize outreach clinics, only 32% (n=7) reported access to one at the time of care.
Conclusion:
Patients receiving CLP care in Oregon face a significant travel burden due to the state's large geographic area and rural makeup. Families travel an average of 2,400 miles in their child's first two years, incurring financial strain. This burden is likely intensified by economic hardship, as three of the top four counties of patient origin have poverty rates exceeding state and national averages (13.4% vs. 12.1% and 11.5%, respectively). Survey responses reinforce that families who must travel farther report greater financial strain and higher perceived difficulty in obtaining care, as well as necessitating valuable time off work. Though patients in Oregon are receiving timely CLP surgical care by pediatric fellowship-trained specialists, the logistical and financial burden to families may mirror similar burdens seen in low- and middle-income countries. This study underscores the need for healthcare policies in Oregon that comprehensively support access to essential surgical care for families of children born with CLP.
References:
1) Global Surgery 2030: evidence and solutions for achieving health, welfare, and economic development
Meara, John G et al. The Lancet, Volume 386, Issue 9993, 569 - 624
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8:30 AM
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Comparing Mechanisms of Craniomaxillofacial Gunshot Wound Injuries: Fracture Patterns in Self-Inflicted, Assault, and Accidental Craniomaxillofacial Gunshot Wounds
Introduction:
Craniomaxillofacial gunshot wounds (GSWs) require complex surgical management, yet little is known about how fracture patterns vary by injury mechanism. This study compares fracture patterns among self-inflicted gunshot wounds (SIGSWs), assault-related GSWs, and accidental firearm injuries to inform reconstructive decision-making.
Methods:
A retrospective review of 448 patients with craniomaxillofacial GSWs treated at a tertiary care center (2018–2024) was conducted. Patients were categorized by injury mechanism: SIGSWs (n=195), assault-related (n=214), and accidental (n=39). Fracture patterns were analyzed across 24 craniofacial sites using radiographic imaging. Statistical comparisons were performed using chi-square tests and a multivariate logistic regression.
Results:
Fracture distribution varied significantly by injury mechanism (p<0.05). SIGSWs were more frequently associated with calvarial (p=0.015), temporal (p=0.007), cribriform plate (p=0.025), sphenoid (p=0.03), and frontal bone fractures (p=0.010). Assault-related injuries demonstrated the highest prevalence of mandibular fractures (p=0.151), while maxillary fractures were most common in accidental injuries (p=0.236). Temporal (OR 5.42, p=0.001), cribriform plate (OR 5.34, p=0.025), and frontal sinus (OR 8.88, p=0.002) fractures were significantly associated with 30-day mortality.
Conclusion:
Fracture patterns in craniomaxillofacial GSWs vary by injury mechanism, with SIGSWs more likely to involve critical cranial structures. These findings highlight the need for tailored reconstructive strategies based on injury etiology. Improved understanding of fracture distribution can optimize surgical planning and patient outcomes.
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8:35 AM
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Scientific Abstract Presentations: Craniomaxillofacial Session 7 - Discussion 1
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8:45 AM
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Understanding Psychosocial Outcomes in Pediatric Patients with Cleft Lip and / or Palate
Intro: Children with cleft lip and/or palate often encounter challenges with speech, feeding, and appearance. While the physical sequelae of cleft conditions are well-documented, the psychosocial outcomes in this population remain underexplored. Given the potential for these challenges to impact emotional and social functioning, this study aims to evaluate psychosocial outcomes, including family relationships, peer relationships, anger, anxiety, depression, and stress, among children with cleft conditions.
Methods: Participants were recruited from a tertiary care academic children's hospital. Participants with cleft conditions (cleft lip, cleft palate, or cleft lip and palate) were compared to controls recruited from the general pediatric clinic well-child visits at the same institution. Psychosocial outcomes, including family and peer relationships, anger, anxiety, depression, and stress, were assessed using Patient-Reported Outcomes Measurement Information System (PROMIS) self-report forms. PROMIS is a validated tool that enables standardized assessment of emotional health across various populations. PROMIS measures range from 1 (never) to 5 (always). T-test analyses were conducted to compare differences in outcomes between groups.
Results: Of 704 total patients, 353 were included: 118 in the control group, 146 with cleft lip and palate, 66 with isolated cleft palate, and 23 with isolated cleft lip. Children with isolated cleft palate had significantly higher anxiety scores than control patients (M = 2.21, SD = .80 vs. M = 1.95, SD = .70, p = .021); they also had higher depression scores (M = 2.17, SD = .82 vs. M = 1.63, SD = .70, p < .001) and higher psychological stress scores (M = 2.20, SD = .84 vs. M = 1.91, SD = .76, p = .018).
Similarly, children with cleft lip and palate also showed higher anxiety scores (M = 2.18, SD = .89 vs. M = 1.95, SD = .70, p = .024) and depression scores (M = 1.87, SD = .88 vs. M = 1.63, SD = .70, p = .018) compared to control patients. In contrast, patients with isolated cleft lip demonstrated no significant psychosocial differences in the assessed domains. Additionally, a subgroup analysis comparing patients with unilateral versus bilateral cleft lip and palate (n = 142) also revealed no significant differences in the assessed domains.
Conclusions: This study highlights the significant psychosocial challenges faced by pediatric patients with cleft conditions, particularly those with isolated cleft palate and comorbid cleft lip and palate. Elevated levels of anxiety, depression, and psychological stress were observed in these groups, suggesting that cleft palate may have a profound impact on emotional well-being. However, no significant differences were found in psychosocial outcomes between unilateral and bilateral cleft lip and palate cases, indicating that severity of cleft does not necessarily predict psychosocial distress. These findings underscore the need for integrated psychosocial support as part of comprehensive care for children with cleft palate to improve their overall quality of life and emotional health.
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8:50 AM
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Impact of Multidisciplinary Treatment on the Quality of Life of Patients with Apert Syndrome: A Longitudinal Analysis Using the HUI-3
Purpose:
Apert syndrome is a rare genetic disorder characterized by craniosynostosis, midface hypoplasia, and complex syndactyly, leading to significant functional and psychosocial challenges (1). This study aimed to evaluate the impact of multidisciplinary treatment on the quality of life (QoL) of patients with Apert syndrome using the Health Utilities Index (HUI-3) questionnaire (2), identifying the most affected domains and areas for therapeutic improvement.
Methods:
A retrospective, cross-sectional study was conducted on 20 patients diagnosed with Apert syndrome and treated at the Hospital das Clínicas, University of São Paulo, between 2000 and 2022. Patients aged ≥5 years who completed the HUI-3 questionnaire were included. The HUI-3 assesses eight QoL attributes: vision, hearing, speech, mobility, dexterity, emotion, cognition, and pain. Data were collected from clinical evaluations, photographic documentation, and medical records.
Results:
The most affected domains were speech (mean score: 0.42), cognition (0.75), and emotion (0.64). Over 60% of patients exhibited severe speech impairments, limiting effective communication. Cognitive deficits significantly impacted daily functioning, with all adult participants being unemployed. Emotional well-being was also compromised, with 62.5% of patients reporting unhappiness. Conversely, mobility (0.94) and dexterity (0.77) demonstrated better outcomes, with surgical interventions, such as syndactyly correction, improving function. Pain was the least affected domain, with most patients reporting minimal discomfort.
Conclusion:
Treatment significantly improves functional and aesthetic outcomes in patients with Apert syndrome; however, persistent deficits in speech, cognition, and emotional well-being highlight the need for comprehensive, long-term care (3). The findings emphasize the necessity of integrating speech therapy, cognitive rehabilitation, psychological support, and educational interventions into the treatment paradigm to enhance patient quality of life beyond surgical correction (4)(5).
References
1: Conrady CD, Patel BC, Sharma S. Apert Syndrome. 2023 Jun 1. In: StatPearls [Internet]. Treasure Island (FL): StatPearls Publishing; 2025 Jan–. PMID: 30085535.
2: Feeny D, Furlong W, Boyle M, Torrance GW. Multi-attribute health status classification systems. Health Utilities Index. Pharmacoeconomics. 1995 Jun;7(6):490-502. doi: 10.2165/00019053-199507060-00004. PMID: 10155335.
3: Raposo-Amaral CE, Denadai R, Oliveira YM, Ghizoni E, Raposo-Amaral CA. Apert Syndrome Management: Changing Treatment Algorithm. J Craniofac Surg. 2020 May/Jun;31(3):648-652. doi: 10.1097/SCS.0000000000006105. PMID: 31895846.
4: Alonso N, Fisher DM, Bermudez L, da Silva Freitas R. Cleft lip and palate treatment. Plast Surg Int. 2013;2013:372751. doi: 10.1155/2013/372751. Epub 2013 May 25. PMID: 23766900; PMCID: PMC3677641.
5: Lu X, Forte AJ, Sawh-Martinez R, Wu R, Cabrejo R, Gabrick K, Steinbacher DM, Alperovich M, Alonso N, Persing JA. Temporal Evaluation of Craniofacial Relationships in Apert Syndrome. J Craniofac Surg. 2019 Mar/Apr;30(2):317-325. doi: 10.1097/SCS.0000000000004836. PMID: 30358751.
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8:55 AM
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Ophthalmic Pathologies in Craniosynostosis: Prevalence and Risk Factors in the United States from the National Inpatient Sample, 2016-2022
Purpose: Craniosynostosis may affect vision development in children. Known ophthalmic manifestations of craniosynostosis include strabismus, papilledema, refractive errors, and amblyopia. Although existing literature has thoroughly investigated such comorbid ophthalmic findings, these studies have been largely limited to small sample sizes. This study uses a national inpatient database to estimate the prevalence of these ocular manifestations of craniosynostosis in the last decade, as well as their associations with sociodemographic and clinical drivers.
Methods: Hospital discharges with diagnoses of craniosynostosis were identified in the 2016-2022 National Inpatient Sample. Concurrent ophthalmic diagnoses were identified by ICD-10 codes. Admissions were additionally characterized by sociodemographic variables, such as age, sex, race, and insurance status, as well as clinical variables, such as hydrocephalus, Chiari malformation, and syndromic vs. non-syndromic craniosynostosis. A multivariable logistic regression model was used to evaluate independent predictors of comorbid ocular pathology (p<0.05).
Results: After accounting for random sampling, the final cohort included 39,850 discharges with craniosynostosis diagnoses from 2016-2022. The median age was 0 [IQR 0-2] years. The overall rate of ocular pathologies was 4.5% (1,800 discharges), which was split to 3.7% in non-syndromic cases and 7.1% in syndromic cases (p<0.001). The most common pathology was strabismus (1.5%), followed by papilledema (1.2%), refractive errors (0.8%), optic atrophy (0.2%), amblyopia (0.2%), and multiple pathologies (0.6%). The distribution of ophthalmic pathologies was different depending on syndromic diagnoses. The rate changed from 4.8% in. 2016 to 5.0% in 2022 and did not significantly change over the study period (p=0.13). In a multivariable model, hydrocephalus (OR: 1.86, 95% CI: 1.35-2.57, p<0.001), Chiari malformation (OR: 1.78, 95% CI: 1.31-2.42, p<0.001), and syndromic craniosynostosis (OR: 1.82, 95% CI: 1.64-2.02, p<0.001) predicted higher odds of concurrent ocular pathologies. When controlled by these factors, Black (OR: 1.21, 95% CI: 1.04-1.41, p=0.016) and Hispanic (OR: 1.24, 95% CI: 1.10-1.40, p<0.001) race/ethnicity, older age (OR: 1.03, 95% CI: 1.02-1.03, p<0.001), and Medicaid insurance status (OR: 1.15, 95% CI: 1.04-1.28, p=0.0095) predicted higher odds of ocular pathologies. The average hospital charge was significantly higher for patients with ocular comorbidities than those without such comorbidities ($107,552 [$55,548-$182,076] vs. $70,067 [$30,046-$136,818], p<0.001).
Conclusion: This study provides a novel estimate of prevalence for ophthalmic pathologies in craniosynostosis in the United States. The overall prevalence was approximately 4.5% with the majority of these cases comprising strabismus and papilledema. Risk factors for increased intracranial pressure, skull malformation, and orbital deformity predicts higher odds of ocular manifestations. Additionally, historically underserved populations, such as Black and Hispanic patients and publicly insured patients, may experience a disproportionate epidemiological, clinical, and financial burden from these comorbid conditions. Such findings emphasize the need for multidisciplinary partnership and early ophthalmology referral for both syndromic and non-syndromic craniosynostosis cases.
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9:00 AM
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The Impact of BMI on Surgical Complications Following Head and Neck Free-flap Reconstruction
BACKGROUND: Research has shown that in patients undergoing soft tissue microvascular reconstruction, obese BMI predisposes patients to increased complications. Prior head and neck research is scarce but has demonstrated increased risk of complications, such as post-operative bleeding and flap necrosis with a low pre-operative BMI. However, there is limited research assessing the relationship between BMI and post-operative complications for patients undergoing microvascular reconstruction for head and neck cancer. It is important to understand the impact of BMI on surgical outcomes to properly optimize and counsel patients pre-operatively. This study aims to assess if BMI extremes are a risk factor for complications following oncologic head and neck microvascular reconstruction.
METHODS: A prospectively maintained database was utilized to identify patients who underwent head and neck microvascular reconstruction following oncologic ablation between 2019 and 2024. Patients were categorized into six groups, according to CDC classifications of BMI based on their pre-operative BMI. Variables, including flap type, intraoperative complications, and post-operative medical and surgical complications were collected. Data were analyzed using SPSS software.
RESULTS: After controlling for reconstructions involving a skin paddle, 553 patients were included in the final cohort. The mean BMI was 26.5. The study included 55 underweight, 189 normal weight, 168 overweight, 82 obese, 41 severely obese, and 18 morbidly obese patients. Underweight patients had a significantly higher rate of salivary leak compared to other BMI categories (23.6% vs. 10%, p = 0.02). There were otherwise no significant differences in intraoperative complications, including anastomotic revision, thrombosis, neck hematoma, or airway issues. There were also no differences in post-operative flap compromise, hematoma, infection/cellulitis, dehiscence, or necrosis formation across BMI categories. Patients were further assessed according to flap donor site. The study included 311 thigh flap patients, 24 back flap patients, 119 fibula flap patients, and 96 forearm flap patients. Thigh flap patients with a severely and morbidly obese BMI had higher rates of overall recipient site complications (14.3% and 33.3% vs <10%, p = 0.044). Thigh flap patients with a severely obese BMI also had increased rates of flap loss (14.3% vs. <2.2%, p < 0.001) and dehiscence (12.5% vs. <10%, p = 0.029) compared to other BMI categories. Obese patients with a fibula flap had higher rates of salivary leak (15.4% vs. 6.1% or less), though this did not reach statistical significance (p = 0.051). There were no significant differences in complication rates across BMI groups for forearm flap patients.
CONCLUSIONS: Surgical complication rates in head and neck microvascular reconstruction vary depending on flap type and BMI. Patients at BMI extremes may face additional comorbidities that predispose to perioperative complications. It is unclear whether BMI alone is a risk factor for complications, or a proxy for additional comorbidities. Nonetheless, BMI extremes, both underweight and obese, correlate to increased perioperative complications, emphasizing the importance of preoperative optimization and pertinent patient counseling on surgical risks.
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9:05 AM
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Examining Sociodemographic Disparities in Diagnostic Delays and Surgical Management of Craniosynostosis: A 10-Year Review
Background
Socioeconomic status (SES) and non-White race have been associated with delayed surgery and more invasive procedures in patients with craniosynostosis.[1-3] While attention to sociodemographic (SD) factors in craniosynostosis care has increased, literature on their impact on diagnostic delays, complications, and hospital stays remains limited and inconclusive. This study examines the influence of SD variables on surgical management and outcomes in a diverse population at a tertiary care center.
Methods
We conducted a retrospective review of patients with craniosynostosis undergoing surgical intervention from 2019 to 2023 at a pediatric tertiary hospital. Patients with craniofacial syndromes or prior craniosynostosis surgery were excluded. Data on patient demographics, dates of diagnosis, surgical management, and postoperative outcomes were collected. SES was measured using the Area Deprivation Index (ADI). Primary outcomes included time intervals from first plastic surgery or neurosurgery appointment, diagnosis, and surgery, along with hospital stay and postoperative complications. Statistical analyses were performed using the Mann-Whitney U test and Chi-squared/Fisher's Exact tests.
Results
Of the 170 patients, 49.7% were White, 85.3% primarily spoke English, 71.6% had private insurance, 73.5% had a low ADI score, and 65.9% underwent minimally invasive procedures. Patients who underwent open procedures were older at the time of first specialty appointment (9.3 vs. 1.9 months, p<0.001), diagnosis (9.6 vs. 2.2 months, p<0.001), and surgery (14.0 vs. 3.0 months, p<0.001). Hispanic/Latino and Black/African American patients were more likely to undergo open procedures (52.6% and 52.4%, respectively, vs. 21.4% White, p=0.004). Publicly insured patients were also more likely to undergo open procedures (47.9% vs 28.1%, p = 0.014). There was no significant difference in patients undergoing open vs minimally invasive procedures across ADI categories, primary language, or interpreter requirements. Length of hospital stay was longer for patients undergoing open procedures (3 vs. 1 day p<0.001), publicly insured patients (1.5 vs. 1 day, p=0.027), Hispanic/Latino patients (2 vs. 1 day, p=0.006), and Black/African American patients (2 vs. 1 day, p=0.006). No significant differences were observed in complication rates across procedure types, interpreter requirements, race/ethnicity, or insurance types.
Conclusion:
Our study highlights that patients from lower SES, particularly Hispanic/Latinos, Black/African Americans and those publicly insured, experience longer referral delays to specialists, leading to later age at surgery and a higher likelihood of undergoing more invasive open procedures. These delays likely cause patients to miss the window for minimally invasive treatments, and they may be attributed to SD barriers, such as limited access to care, language barriers, or disparities in healthcare navigation.
References
1. Washington DM, Curtis LM, Waite K, Wolf MS, Paasche-Orlow MK. Sociodemographic Factors Mediate Race and Ethnicity-associated Childhood Asthma Health Disparities: a Longitudinal Analysis. J Racial Ethn Health Disparities. 2018;5(5):928-938. doi:10.1007/s40615-017-0441-2
2. Liu X, Rosa‐Lugo LI, Cosby JL, Pritchett CV. Racial and Insurance Inequalities in Access to Early Pediatric Cochlear Implantation. Otolaryngol Neck Surg. 2021;164(3):667-674. doi:10.1177/0194599820953381
3. Lynn JV, Ranganathan K, Bageris MH, Hart-Johnson T, Buchman SR, Blackwood RA. Sociodemographic Predictors of Missed Appointments Among Patients With Cleft Lip and Palate. Cleft Palate Craniofacial J. 2018;55(10):1440-1446. doi:10.1177/1055665618764739
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9:10 AM
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Reducing Cartilage Warp in Nasal Reconstruction: The Role of Collagen and Proteoglycans
Background: The use of costal cartilage in nasal reconstruction is often limited by its tendency to warp when cut. This study investigates the role of collagen fiber orientation and proteoglycan (PG) distribution in cartilage warp. Additionally, it evaluates the effectiveness of pre-treatment with hyperosmotic saline and collagen crosslinking in reducing warp.
Methods: Human costal cartilage specimens from the synchondrosis of rib 6 were scanned using a 7 Tesla MRI with optimized Diffusion Tensor Imaging parameters to map collagen fiber orientation. Histological analysis was used to map PG distribution. Resultant warp was quantified in transverse cartilage slices from the synchondrosis of 11 patients following immersion in isotonic saline (control), hyperosmotic saline, or methylglyoxal (MGO) for 24 hours.
Results: Distinct patterns of collagen fiber orientation were found in the synchondrosis, with anterior-posterior fibers in the mid/deep zones and a circumferentially oriented ring of fibers in the superficial layers. Histological analysis showed higher PG concentrations in the mid/deep zones. Pre-treatment with hyperosmotic saline significantly reduced warping immediately after slicing, and specimens maintained reduced warping up to 120 hours. MGO crosslinking at 4M concentration also significantly reduced warping immediately after sectioning, with a trend toward greater linearity at 120 hours.
Conclusions: This study describes the role of collagen fiber orientation and PG distribution in cartilage warping. Asymmetric disruption of the outer circumferential collagen ring combined with differential expansion of cartilage with higher PG concentration in deeper zones may result in differential expansion and therefore warp. Pre-treatment with hyperosmotic saline or collagen crosslinking reduces warping by balancing internal stresses and reinforcing the collagen network. These findings provide insights into minimizing cartilage warp post-sectioning and improving surgical outcomes.
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9:10 AM
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Reducing Cartilage Warp in Nasal Reconstruction: The Role of Collagen and Proteoglycans
Background: The use of costal cartilage in nasal reconstruction is often limited by its tendency to warp when cut. This study investigates the role of collagen fiber orientation and proteoglycan (PG) distribution in cartilage warp. Additionally, it evaluates the effectiveness of pre-treatment with hyperosmotic saline and collagen crosslinking in reducing warp.
Methods: Human costal cartilage specimens from the synchondrosis of rib 6 were scanned using a 7 Tesla MRI with optimized Diffusion Tensor Imaging parameters to map collagen fiber orientation. Histological analysis was used to map PG distribution. Resultant warp was quantified in transverse cartilage slices from the synchondrosis of 11 patients following immersion in isotonic saline (control), hyperosmotic saline, or methylglyoxal (MGO) for 24 hours.
Results: Distinct patterns of collagen fiber orientation were found in the synchondrosis, with anterior-posterior fibers in the mid/deep zones and a circumferentially oriented ring of fibers in the superficial layers. Histological analysis showed higher PG concentrations in the mid/deep zones. Pre-treatment with hyperosmotic saline significantly reduced warping immediately after slicing, and specimens maintained reduced warping up to 120 hours. MGO crosslinking at 4M concentration also significantly reduced warping immediately after sectioning, with a trend toward greater linearity at 120 hours.
Conclusions: This study describes the role of collagen fiber orientation and PG distribution in cartilage warping. Asymmetric disruption of the outer circumferential collagen ring combined with differential expansion of cartilage with higher PG concentration in deeper zones may result in differential expansion and therefore warp. Pre-treatment with hyperosmotic saline or collagen crosslinking reduces warping by balancing internal stresses and reinforcing the collagen network. These findings provide insights into minimizing cartilage warp post-sectioning and improving surgical outcomes.
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9:15 AM
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Disparities in the Timing of Cleft Surgeries Based on Insurance Coverage
Introduction: The timing of cleft-related surgeries, including lip repair, palate closure, and alveolar bone grafting (ABG), is critical for speech development, dental eruption, and overall facial growth. Prior studies have established a relationship between public insurance and delayed care (1). This study aims to investigate the timeline of care for patients with private versus public insurance, to determine when patients are experiencing the greatest delay.
Methods: A retrospective analysis was conducted on patients who completed all cleft-related surgeries, up to and including ABG at a primary cleft center since 2012. Patients were categorized based on public or private insurance status. Data collected included age at first appointment, age at ABG, number of missed appointments, cancellations, completed appointments, and reasons for missed appointments. Timing intervals were analyzed between first appointment to first surgery, lip repair to palate closure, and palate closure to ABG. Statistical analysis included Wilcoxon Rank Sum tests, chi-square tests, and Fisher Exact tests to compare outcomes between insurance groups.
Results: A total of 127 patients were included, with 48 (37.8%) publicly insured and 79 (62.2%) privately insured. Compared to privately insured patients, patients with public insurance were older at the time of their first cleft appointment (27.74 vs. 21.54 days, p=0.044) and at the time of their ABG (11.48 vs. 10.29 years, p=0.0029). The interval between first appointment to first surgery and the interval between lip repair to palate closure were comparable between groups (p=0.518 and p=0.779, respectively). However, publicly insured patients experienced a delay from palate closure to ABG (10.52 vs. 9.35 years, p=0.004). They were also more likely to have at least one no-show appointment (77.08% vs. 36%, p=0.0005) and have a higher number of no-show appointments (2 vs. 0, p<0.001). Additionally, publicly insured patients were more likely to have a cancelled appointment due to transportation issues (25% vs. 6%, p=0.0064). Total number of completed appointments between insurance groups were comparable (18.5 vs. 20, p=0.217).
Conclusion: While the timing of early-stage surgeries such as lip repair and palate closure was comparable between insurance groups, publicly insured patients experienced delays in establishing cleft care and during the longer interval between palate closure and ABG when orthodontics are completed. These delays may be exacerbated by systemic barriers including higher rates of missed appointments and transportation issues, despite a comparable number of completed appointments across insurance groups. These findings highlight disparities in access to timely cleft care for publicly insured patients, emphasizing the need for improved continuity of care, particularly during extended surgical gaps.
References:
1. Bushong EE, Patmon D, Pfershy H, Huffman C, Carlson A, Girotto J. Timing of Alveolar Bone Graft and Barriers to Care. Cleft Palate Craniofac J. Published online April 4, 2024. doi:10.1177/10556656241242695
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9:20 AM
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Scientific Abstract Presentations: Craniomaxillofacial Session 7 - Discussion 2
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