Scientific Abstract Presentations: Craniomaxillofacial Session 1 Thursday, September 26 Thu, Sep 26 9:45-11:45 a.m. PDT

PURPOSE: Box osteotomy (BO) or facial bipartition (FB) are used for the surgical correction of hypertelorism. Both procedures have been shown to successfully reduce interdacryon distance and the overlying soft tissue. There is a paucity in the literature assessing the degree to which bony resection translates into soft tissue movement. This study analyzes the bony and soft tissue changes achieved with BO and FB.

METHODS: A retrospective review was conducted evaluating patients who underwent BO or FB at our institution from 2005-2023. Demographics, medical history, and perioperative data were recorded. Correction of the interdacryon distance and intercanthal distance were measured on pre- and post-operative computed tomography scans. Three-dimensional craniometric data was derived using Mimics software. Paired and Independent t-tests were utilized. The ratio of bone-to-soft tissue change was computed by calculating the change in the intercanthal distance for every 1mm change in the interdacryon distance.

RESULTS: Twenty-nine patients were included, of which 20 underwent FB and 9 BO. There was a significant improvement in the interdacryon distance (28.8mm to 22.1mm; p<0.001) and intercanthal distance (43.5mm to 38.2mm; p<0.001) for FB patients. Similarly, there was a significant improvement in the interdacryon distance (37.1mm to 25.8mm; p<0.001) and intercanthal distance (47.7mm to 39.9mm; p<0.001) for BO patients. The percent change in the interdacryon distance was 22.4% (FB) and 30.4% (BO; p=0.017), whereas the percent change in the intercanthal distance was 11.0% (FB) and 14.7% (BO, p=0.193). The ratio of bone-to-soft tissue change was 1-to-0.8 across both cohorts (BO: 1-to-0.9, FB: 1-to-0.7; p=0.502).

CONCLUSIONS: Box osteotomy and facial bipartition effectively correct hypertelorism, demonstrating significant bone and soft tissue changes. Our findings revealed that 1mm of interorbital bone resection correlated with 0.8mm change in the overlying soft tissue for both hypertelorism-correcting procedures.

Background
Geographic Information Systems (GIS) have evolved as a powerful tool for characterizing epidemiological trends, patterns of care utilization, and limitations in care access. Patients with cleft lip and/or palate (CL/P) require long-term care, with numerous surgeries and therapies to treat the aesthetic and functional consequences of their diagnosis (1). Access to appropriate and timely cleft care depends on a variety of factors. In addition to physical distance, cleft epidemiology, race, financial disparities, and social history can negatively affect a patient's ability to receive care. In this study we utilize population level analysis and GIS to generate a demand index for approved cleft care within the United States (US).

Methods All births between 2018-2022 were queried from the National Vital Statistics System (NVSS) Restricted-Use Vital Statistics Data. For each US county, CL/P birth prevalence per 10,000 births and population density were calculated. The Social Vulnerability Index (SVI) was utilized to account for the degree of social inequality for each county (2). Approved cleft centers were identified from the ACPA website and geocoded to calculate driving distances between county centroids and the nearest center. Birth prevalence, population density, SVI, distance to the nearest center, and centers within a 50km radius were used to generate a composite score of cleft demand.

Results Nine hundred and seventy counties had a score less than 25, 1930 counties had scores between 25 and 49.99, 172 counties had scores between 50 and 74.99, and 59 counties had scores between 75 and 100. The highest scoring county, Bibb County, GA had a CL/P birth prevalence of 16.2 per 10,000 births, population density of 241.46 persons per km2, distance to the nearest cleft center of 207.69 km, and an SVI of 0.96. Nevada (83.6), Montana (76.0), and Georgia (74.1) were the states with the highest average demand scores.

Conclusions Understanding the interplay between the numerous spatial and non-spatial barriers to care is crucial to optimize care delivery. Our composite score quantifies cleft burden, socioeconomic disadvantage, and geographic barriers to provide a measure of the of demand for approved cleft care in each US county. By describing the geospatial relation of these barriers, our study demonstrates the power of GIS for identifying areas with limited access to approved cleft care teams. Demand indices, like this, have demonstrated a variety of applications in health policy, research, and administration to expand access to patients not covered by local supply (3). Utilizing these score components can help tailor future ACPA interventions, outreach efforts, and planning of cleft care centers.

References:

1. Wells-Durand E, Buchel A, Tuen YJ, et al. What Does Cleft Lip and Palate Care Cost? The Time and Economic-Associated Burden of Care From Birth to Maturity. Plast Surg. Published online October 9, 2023:22925503231203216. doi:10.1177/22925503231203216
2. CDC/ATSDR Social Vulnerability Index (SVI). Published January 18, 2024. Accessed February 6, 2024. https://www.atsdr.cdc.gov/placeandhealth/svi/index.html
3. Calovi M, Seghieri C. Using a GIS to support the spatial reorganization of outpatient care services delivery in Italy. BMC Health Serv Res. 2018;18:883. doi:10.1186/s12913-018-3642-4

Background: Post-operative fever is a known complication following open surgical repair of craniosynostosis. However, its etiology, risk factors, and clinical significance is poorly understood. This often results in additional work-up and patient cost. The aim of this study is to address the incidence, potential risk factors, and subsequent complications associated with fever following open surgical repair of craniosynostosis.

Methods: A retrospective review of all patients who underwent open surgical correction of craniosynostosis at Hershey Medical Center between January 2017 and December 2022 was performed. Post-operative hyperthermia was defined as a temperature above 38°C. Logistic regression and Wilcoxon rank-sum tests were performed for analyses.

Results: Of the 75 patients included, 23 (30%) developed a post-operative fever. Average time to onset was 25 hours after surgery (range: 0.5-54hr) with an average time to maximum temperature of 30.96 hours (range: 1.5-54.5hr). Maximum temperatures ranged from 38.2-39.6°C with an average of 38.6°C. Only one child underwent an infectious work-up including CBC, urinalysis, respiratory viral panel, COVID19, chest X-ray and head imaging with no infectious source identified. Only one child was given prophylactic post-operative antibiotics due to multiple concurrent procedures. The other 22 children in the fever group had resolution of the temperature with Tylenol alone within 14.75 hours from fever onset, on average. Of the patients who developed a fever, 5 (27.1%) sustained iatrogenic dural violation during surgery. There was no association between intraoperative durotomy (p=0.52), estimated blood loss (p=0.42), intra-operative blood administration (p=0.68), weight (p=0.39), or age (p=0.19) and development of post-operative fever. Fever development was not associated with development of post-operative complications within one year, return to operating room, 30-day readmission (p=0.38) or increased hospital length of stay (p=0.20).

Conclusions: Approximately 1/3 of children developed post-operative fever following open surgical repair of craniosynostosis. These fevers resolved spontaneously or with the aid of Tylenol. Intraoperative durotomy, EBL, and blood administration were not associated with fever. Post-operative fever was not associated with post-operative complications or 30-day readmission. These results support hyperthermia as part of the natural post-operative course of craniosynostosis repair. They also provide support for withholding routine fever work-up to decrease unnecessary testing and financial burden, unless otherwise clinically warranted.

Background: Nasoalveolar molding (NAM) has been used to optimize outcomes in patients with cleft lip and palate (CLP) deformities, particularly for those with wider clefts. While this presurgical intervention has been associated with improved surgical outcomes, its effect on long-term postoperative outcomes, such as midface hypoplasia (MFH), is poorly understood. This study analyzes aggregate cephalometric data to assess long-term MFH among patients with complete unilateral CLP (CUCLP) who undergo NAM.

Patients and Methods: A systematic review was conducted following PRISMA guidelines. Studies reporting cephalometric measurements from lateral radiographs of patients with repaired CUCLP who underwent presurgical NAM versus no-PSIO (control) were included. Studies of bilateral cleft cases or unoperated clefts were excluded. Cephalometrics were assessed by pooled analysis and meta-analysis.

Results: A total of 2,063 articles were identified; five met the inclusion criteria. Post-operative cephalometrics were reported for 381 patients (176 NAM, 205 no-PSIO) at an average age of 9.7±2.9 years. Pooled analysis of the NAM and no-PSIO cohorts revealed that the NAM cohort had significantly smaller SNA angles (76.5°±6.7° vs. 78.2°±4.0°; p<0.001) and smaller ANB angles (1.4°±5.1° vs. 3.3°±2.8°; p=0.002). A subanalysis of patients with cephalometrics at eight years or older was performed (n=249 patients, 11.7±4.0 years). Compared to controls, the NAM cohort had significantly smaller SNA angles (74.6°±8.0° vs. 77.6°±4.4°; p<0.001) and smaller ANB angles (-0.8°±6.0° vs. 5.2°±3.7°; p<0.001). Meta-analysis further demonstrated that NAM was associated with a smaller SNA angle (Mean Difference [MD] -2.32, [-3.8 to -0.84]; p<0.01) and ANB angle (MD -2.60 [-3.72 to -1.48]; p<0.01).

Conclusion: This study reveals that patients with CUCLP treated with presurgical NAM had greater MFH than those without PSIO treatment. Before opting for NAM, clinicians should consider the potential sagittal growth restrictions experienced by patients with CLP, evaluate severity, and balance the need for NAM with the possible need for future orthognathic surgeries. Further evaluation of other interventions along the cleft care timeline for their effect on MFH is warranted, as this study only assesses one presurgical treatment modality.

Background Free flap reconstruction for pediatric head and neck defects poses unique operative and postoperative challenges. Successful reconstruction requires multidisciplinary care with consideration given to the patient's future growth and the effects of oncologic care on wound healing and repeat operations (1). This study aims to describe the reconstructive techniques and outcomes for pediatric patients undergoing head and neck reconstruction.

Methods A retrospective chart review was conducted for all pediatric patients undergoing head and neck reconstruction from January 2017 to September 2023 at a tertiary pediatric hospital. Information on patient demographics, oncologic history, reconstructive approach including flap choice and recipient vessel selection, perioperative data, and complications were collected. Descriptive statistical analysis was performed.

Results Of the 51 patients included in this study, 58.8% were male, and the average age was 11.7 years old (std 5.8). Oncologic defects comprised 66.7% of the cohort, most commonly in the maxilla (50.0%) and mandible (32.8%). 47.1% of oncologic defects were benign lesions, while 52.9% were malignant. Traumatic (15.7%) and congenital (13.7%) defects were also reconstructed. Free flap reconstruction was necessary in 94.1% of patients, with the most common flaps including the anterolateral thigh flap (35.4%), fibula flap (25.0%), and radial forearm flap (12.5%). The facial artery and vein were used as the recipient vessels in 72.9% of cases. Hardware, including plates, screws, and titanium mesh was used in 56.9% of cases. Operative time averaged 8.3 hours, with 10.1 hours of total anesthesia time.

Postoperatively, 92.2% were admitted to the Pediatric Intensive Care Unit and the remainder were transferred to the floor. Return to the OR for flap compromise was required for 20.8% of patients, with flap salvage successful in 45.5% of attempts. Recipient-site dehiscence was the most common complication (22.9%), followed by recipient-site infection (4.2%), donor-site dehiscence (4.2%), hematoma (4.2%), and seroma (4.2%). Partial flap loss was seen in 14.6% of patients and total flap loss in 6.3% of patients. 11 patients (22.9%) returned to the operating room to address a complication or harvest new flap, on average 23.9 days after their initial surgery. The average ICU stay was 8.4 days, with an average length of stay of 13.9 days. The average length of follow-up was 1.6 years. Survival within this cohort is 94.1%.

Conclusion Free flap reconstruction is the standard of care for pediatric head and neck oncologic, traumatic, and congenital defects. Maxillectomy and mandibulectomy are the two most common procedures requiring free flap reconstruction, with the ALT and fibula flap being the most common choice. With multidisciplinary care, both successful surgical outcomes

References

1. Roasa FV, et al. Pediatric free flap reconstruction for head and neck defects. Curr Opin Otolaryngol Head Neck Surg. 2018;26(5):334-339.

Background: Beckwith-Wiedemann Syndrome (BWS) is a congenital overgrowth and cancer predisposition disorder that presents with macroglossia in 85% of affected patients. While roughly 25% of patients with BWS require tongue reduction surgery, the natural history of mild macroglossia is not well characterized.

Methods: Medical records of patients with BWS and macroglossia seen between 2004-2024 were reviewed. BWS index of macroglossia (BIG) scores were prospectively assigned. Molecular diagnoses, polysomnography (PSG) data, surgical incidence, percentage mosaicism, and clinical scores were assessed using two-tailed t-tests and multivariate linear models.

Results: A total of 335 patients with BWS and macroglossia were included, 176 (52.5%) males and 159 (47.5%) females. Within this cohort, 242 patients who did not require surgery in the first three years of life were followed in our clinic, 152 (45.4%) had sleep studies, and 161 (48.1%) were assigned BIG scores. Among non-surgically managed patients with two sleep studies, their obstructive apnea-hypoxia index (OAHI, 7.2 (1.8-12.7) vs. 3.9 (1.9-6.6), p=0.022) decreased over time. Similarly, BIG score (2 (1-2) vs 1, p<0.001) decreased over time. Both OAHI (r=0.323, p<0.001) and oxygen saturation percentage nadir (r=0.310, p<0.001) improved as patients aged. For the 116 (34.6%) patients who had surgery (median age 1.1 (0.6-2.5) years), those with upper airway obstruction were significantly younger than those with a craniodental developmental concerns (0.7 (0.3-1.4) vs 2.9 (2.0-3.8), p<0.001.)

Conclusion: Macroglossia can improve over time in some patients with Beckwith-Wiedemann syndrome. Patients with upper airway obstruction have surgery at a younger age than those with craniodental developmental concerns.

PURPOSE: Recombinant human bone morphogenetic protein (rhBMP-2) with demineralized bone matrix (DBM) has served as a viable synthetic alternative to iliac crest bone graft (ICBG) in alveolar bone grafting (ABG). Although revision rates are low with both graft materials, future dental rehabilitation requires adequate bone within the repaired alveolar cleft. This study compares graft success rates among rhBMP-2/DBM versus ICBG versus ICBG+rhBMP-2 for ABG.

METHODS: A retrospective cohort study was conducted for patients undergoing ABG from 2017-2023. Clinical graft success was defined as no indication for revision ABG. Successful bony bridge formation and their respective bridge thickness were assessed based on CBCT imaging taken at least six months postoperatively (average 17.8±14.2 months). Bridge thickness was calculated based on the height of the graft site from the cementoenamel junction to the cleft-adjacent tooth root apex while ensuring the Hounsfield units measured in grafted areas were consistent with mineralized bone.

RESULTS: Among 432 patients included, 284 rhBMP-2/DBM, 196 ICBG, and 47 ICBG+rhBMP-2 index procedures were reported. The mean age at bone grafting was 10.7±3.6 years for ICBG+rhBMP-2 patients, 10.2±3.2 years for ICBG patients, and 9.3±2.9 years for rhBMP-2/DBM patients, The clinical graft success rate across the 527 procedures was 95.9%, with comparable success rates across all three cohorts (rhBMP-2/DBM: 95.1% vs. ICBG: 95.4% vs. ICBG+rhBMP-2: 97.9%; p=0.694). Based on CBCT imaging, 88.9% of the ICBG+rhBMP-2 cohort had demonstrated successful bony formation, which was significantly higher compared to ICBG (89.4% vs. 65.3%; p=0.031) and rhBMP-2/DBM (88.9% vs. 61.5%; p=0.023). The average bridge thickness was higher in the ICBG+rhBMP-2 cohort compared to ICBG (7.4±2.6mm vs.4.7±1.9mm; p<0.001) and rhBMP-2/DBM (7.4±2.6mm vs. 4.4±1.2mm; p<0.001).

CONCLUSION: Our preliminary findings demonstrate that the use of ICBG with rhBMP-2 appears to result in more bony bridge formation and greater bridge thickness. With a higher rate of bony bridge formation and bridge thickness, dental rehabilitation of the repaired alveolar cleft space with implants or orthodontic movement of existing teeth may be more feasible.

Background: Up to 75% of patients with cleft palate require surgical correction of midface hypoplasia following palate repair. The influence of different surgical techniques on maxillary growth remains unclear. Our study investigates the long-term impact of palatoplasty techniques on midface growth and subsequent orthognathic surgery. Methods: This institutional, retrospective cohort study evaluated patients without a craniofacial syndrome who underwent primary palatoplasty with Furlow double-opposing Z-plasty or straight-line repair techniques from 1994-2023. Patients were at least 14 years of age at their most recent follow-up visit. Our primary outcome was the need for orthognathic surgery to correct midface hypoplasia. Results: In total, 1,857 patients underwent palatoplasty, of which 332 met inclusion criteria (48 straight-line, 284 Furlow). Average age at last follow-up was 18.5±2.6 years. The orthognathic surgery rates based on Veau class were 46.7% (Veau I), 11.1% (Veau II), 60.9% (Veau III), and 70.2% (Veau IV). When comparing Furlow vs. straight-line repair, there was no significant difference in orthognathic surgery rates, even when stratified based on Veau class (p>0.05). Upon multivariate regression, patients with earlier repair (Odds Ratio [OR]: 0.82; p=0.031), Veau III (OR: 3.77; p=0.002), Veau IV (OR: 5.19; p<0.001), and more palatal fistula repairs (OR: 1.93; p=0.037) had increased orthognathic surgery rates. Conclusion: Our findings suggest that higher cleft severity and increased surgical intervention contribute to future orthognathic surgery. However, palatoplasty technique did not influence orthognathic surgery rates. Consequently, surgeons should perform their preferred palate repair technique when considering sagittal growth restriction.

Background & Purpose A relatively newer technique to treat velopharyngeal insufficiency (VPI) is autologous fat grafting to the soft palate and posterior pharyngeal wall, with the ultimate goal of reducing hypernasal resonance. This study aims to assess the efficacy of this treatment in mitigating hypernasality in patients with VPI.

Methods This retrospective case review focused on two community surgeons who performed fat injections between 2015 and 2023 on patients with cleft and non-cleft VPI. We identified 31 patients who had both preoperative and postoperative speech scores, containing a hypernasality rating by a cleft team speech-language pathologist. Hypernasality ratings were converted to an adapted scale of 0-3, ranging from balanced resonance to severe hypernasality. Ratings were based on the Cleft Audit Protocol for Speech (CAPS) and the Cleft Audit Protocol for Speech- Augmented (CAPS-A). Speech measures such as nasal air emission and intelligibility were not consistently documented and, as a result, were not reported in this study.

Results The mean length of follow-up time was 25 months. Complete resolution of VPI was achieved in 14 patients (45%); reduction in hypernasality without full resolution of symptoms was achieved in 7 patients (23%); no improvement with resonance post-injection was achieved in 10 patients (32%); and one patient demonstrated mixed resonance post-injection without a reduction in hypernasality. When comparing preop and postop speech scores in the same patient, a one-sample t-test showed a mean reduction of 0.73 (95% confidence interval –0.48 to –0.98, p<0.0001).

Conclusion Preliminary results indicate that at our site, fat injection was effective in reducing hypernasality for patients with VPI. Future research should include additional speech measures (e.g. intelligibility ratings, speech-related quality of life, nasal air emission, etc.), rates of obstructive sleep apnea, and comparison to other surgical speech interventions (e.g. synthetic injection, Furlow palatoplasty, sphincter pharyngoplasty, and posterior pharyngeal flap).

Background: An easy-to-use tool to objectively measure intraoral anatomy with meaningful clinical correlations may improve care for patients with Beckwith-Wiedemann syndrome (BWS), who commonly have symptomatic macroglossia.

Methods: Children aged 2-17 years with BWS were prospectively enrolled between 12/2021-01/2024. Digital intraoral photographs were taken, and morphometric measurements were made using ImageJ software. Relationships between morphometric measures and outcomes including BWS clinical score, percentage mosaicism, and incidence of tongue reduction surgery were examined using t-tests and multivariate linear models.

Results: Fifty-six morphometric measurements were obtained in 49 patients with BWS. Mouth area, width, and height differed across the BWS molecular subtypes (p<0.05). Right-to-left tongue width (p=0.022) and mouth width (p=0.049) were larger in those with loss of methylation at IC2 (IC2 LOM) than in other BWS variants. Patients with paternal uniparental isodisomy of chromosome 11p15 (pUPD11) had narrower mouths than others (p=0.040). Those with tongue reduction surgery had more tongue ridging than those without surgery (p<0.01). There were correlations between mouth area and BWS clinical score (p=0.046), tongue width and BWS clinical score (p=0.040), and tongue length and percentage mosaicism (p=0.012).

Conclusion: Straightforward intraoral morphometric measurements are associated with phenotypic burden in BWS. Tongue morphology varies across the BWS spectrum, with IC2 LOM presenting with wider tongues and mouths, and pUPD11 having narrower mouths. Tongue ridging is more common in those selected for surgery. Obtaining these measurements is safe and may be adopted at low costs across centers caring for children with BWS or others at risk of upper airway obstruction.

Background Distraction osteogenesis emerged as an alternative to cranial vault remodeling, permitting gradual increase of the cranial vault volume with less invasive undermining and reconstruction. Although previous studies have compared cranial vault remodeling with distraction, there has been little investigation between the use of spring or traditional rigid distractors. The aim of this study is to systematically review the existing literature to compare spring and traditional distraction for the treatment of craniosynostosis. Methods A systematic review was performed in accordance with the PRISMA guidelines. Studies investigating cranial vault distraction osteogenesis with springs or traditional distractors were included. The data collected included patient demographics, history of previous craniofacial surgery, type of craniosynostosis, surgical procedure, distraction protocol, operative time, blood loss, length of stay, cephalic index, recurrence, secondary procedures, complications, and follow-up duration. Results Sixty-five articles, representing 2,406 patients (1,193 spring and 1,213 traditional) were included. The average follow-up duration was 5.02±3.18 years for springs and 3.39±1.39 years for traditional (p<0.001). Patients who underwent spring distraction were younger (4.8 [4.6, 5.8] versus 18.7 [17.7, 19.4] months (p<0.001). Patients underwent spring distraction for sagittal (n=915, 77%), multisuture (n=187, 16%), lambdoid (n=26, 2%), coronal (n=22, 2%), metopic (n=20, 2%), and mixed/unspecified (n=23, 2%) craniosynostosis. Patients underwent traditional distraction for multisuture (n=215, 18%), coronal (n=92, 8%), sagittal (n=86, 7%), lambdoid (n=27, 2%), metopic (n=9, 1%), and mixed/unspecified (n=784, 65%) craniosynostosis. The operative time was significantly shorter for spring distraction (104 [78, 131] versus 127 [115, 170] minutes, p<0.001) with significantly less operative blood loss (50 [48, 63] mL vs. 300 [149, 300] mL, p<0.001) and lower percentage of patients requiring blood transfusion (23% [0%, 66%] versus 46% [46%, 81%], p<0.001). Hospital stay was significantly shorter after spring distraction (2 [1.5, 4.8] versus 4.2 [4, 9.4] days, p<0.001). The median preoperative cephalic index was lower in the spring cohort (69.2 [67.7, 70.7] versus 83.5 [73.3, 94.6], p<0.001). The median cephalic index at last follow-up remained lower in the spring group (75.6 [75.2, 76] versus 84.7 [80.3, 88.7], p<0.001). Spring distraction had a larger median increase in unadjusted intracranial volume (34.7% [31.6%, 38.1%] versus 21.0% [16.9%, 25.0%], p<0.001). The median minor complication rate (2.2% [0.6%, 2.9%] versus (5.5% [0%, 16%], p<0.001) and major complication rate requiring reoperation (7.4% [0%, 10.5%] versus 8.4% [8.3%, 15.8%], p<0.001) were lower in the spring group. Infections requiring reoperation were lower in the spring group (0% [0, 2%] vs. 3.5% [0%, 4.5%], p<0.001), however device exposures requiring reoperation were not significantly different for patients who underwent spring (1% [0%, 5.1%]) and traditional (2.6% [2.1%, 3.3%]) distraction (p=0.13).
Conclusion Patients underwent spring distraction primarily for sagittal synostosis at a younger age, while patients underwent traditional distraction for multisuture synostosis at an older age. Compared to traditional distraction, spring distraction required less operative time, resulted in less blood loss, shorter hospital stays, and lower complication rates. Patients who underwent spring distraction had larger increases in intracranial volume, although this is unadjusted for normal growth and confounded by the younger age at time of operation.

Background Current practice for primary cleft palate repair involves performing the procedure on an inpatient basis, where patients are admitted for an overnight hospital stay. As a result of rising national health care cost and hospital burden, there has been an increasing shift towards the implementation of ambulatory surgery protocols. Same-day discharge for pediatric patients undergoing surgery has been associated with benefits including lower risk for nosocomial infection, lower rates of anxiety and psychological burden, faster return to a normal sleep pattern, and good parental satisfaction rates (1,2). However, current available literature on same-day discharge for primary cleft palate repair is limited as studies describe outpatient procedures, which may include patients kept overnight with discharge at less than 24 hours (3). Therefore, the objective of this study was to compare outcomes following primary cleft palate repair in patients discharged on the same calendar day to patients admitted for overnight stay.

Methods This was a single-surgeon, retrospective review of 40 patients undergoing primary cleft palate repair performed consecutively from September 2018 to June 2023. Demographic characteristics, medications administered intraoperatively and postoperatively, 30-day readmission, reoperation, wound complication, and postoperative complication rates, and 1-year fistula rates were collected from electronic medical records. Fischer's exact test and nonparametric Mann-Whitney U tests were performed for data analysis.

Results: 40 total patients were identified. Following primary cleft palate repair, 20 patients were discharged on the same calendar day and 20 patients were admitted for overnight stay. There were no significant differences in rates of 30-day readmission, 30-day reoperation, 30-day wound complications, or 30-day postoperative complications between groups (p>0.05). Two instances of wound dehiscence on the oral surface were noted at postoperative follow-up visits within the group admitted for overnight stay (5%). These wounds healed without fistulas at 1-year follow-up, and no fistulas were reported at 1-year follow-up in either group. Preoperatively, there was a significantly higher proportion of patients admitted for overnight stay that were Veau class III (55%), as compared to patients discharged on the same day (15%) (p= 0.019). When comparing number of doses of specific medications received postoperatively, patients admitted for overnight stay received significantly more oxycodone at median of 2 doses (IQR 1.00 - 3.75) and acetaminophen at a median of 4 doses (IQR 3.00 - 5.00) than patients with same-day discharge with a median of 1 dose (IQR 0 - 1.0, p < 0.001). Additionally, there was no significant difference in rates of caregiver contact with the clinic following patient discharge and prior to first follow-up between groups (p= 0.523).

Conclusion: Same-day discharge following primary cleft palate repair may be safely undertaken in a low-risk patient population, resulting in similar short-term outcomes and 1-year fistula incidence as patients admitted for overnight stay.

References:

1. Al-Thunyan AM, Aldekhayel SA, Al-Meshal O, Al-Qattan MM. Ambulatory cleft lip repair. Plast Reconstr Surg. Dec 2009;124(6):2048-2053. doi:10.1097/PRS.0b013e3181bcf305

2. de Luca U, Mangia G, Tesoro S, Martino A, Sammartino M, Calisti A. Guidelines on pediatric day surgery of the Italian Societies of Pediatric Surgery (SICP) and Pediatric Anesthesiology (SARNePI). Ital J Pediatr. Mar 12 2018;44(1):35. doi:10.1186/s13052-018-0473-1

3. Kantar RS, Cammarata MJ, Rifkin WJ, Plana NM, Diaz-Siso JR, Flores RL. Outpatient versus Inpatient Primary Cleft Lip and Palate Surgery: Analysis of Early Complications. Plast Reconstr Surg. May 2018;141(5):697e-706e. doi:10.1097/prs.0000000000004293

Background: Patients with cleft lip and palate often require surgical correction of velopharyngeal insufficiency (VPI), a condition associated with difficulty with speech and swallowing. Associated comorbidities such as a craniofacial syndrome may place patients at an increased risk of poor speech outcomes. This study evaluates our long term experience with syndromic patients undergoing palatoplasty and aims to assess their need for VPI surgery.

Methods: A retrospective chart review was conducted of patients with syndromic cleft lip and palate who received care at a tertiary children's hospital from 2004-2024. Patient characteristics, syndromic diagnosis, cleft phenotype and Veau class were abstracted. The need for VPI surgery after cleft palate repair and the timing to VPI surgery were recorded. Descriptive statistics, chi-squared, and independent t-tests were conducted to analyze differences in rates of VPI surgery between various craniofacial syndromes. A Kaplan-Meier analysis was performed to compare time from cleft repair to VPI surgery between syndromes.

Results: A total of 207 syndromic patients were identified with 190 meeting inclusion criteria. The mean follow-up time was 8.01±5.48 years. The most commonly presented syndromes included 22q11.2 deletion syndrome (22q11.2DS) (n=35, 18.4%), Stickler syndrome (n=26, 13.7%), and CHARGE syndrome (n=15, 7.9%). A total of 20 (10.5%) patients had Robin Sequence (RS) associated with a craniofacial syndrome. Cleft types included 27 (14.2%) submucous cleft, 59 (31.1%) Veau I, 80 (42.1%) Veau II, 7 (3.7%) Veau III and 17 (9.0%) Veau IV. Overall, 31(16.3%) patients underwent VPI surgery at 4.33±3.16 years after their initial cleft palate repair, with the highest VPI rate amongst Veau I patients. Patients with 22q11.2DS underwent VPI surgery at higher rates (45.7%) compared to Stickler syndrome (15.3%, p=0.013), CHARGE syndrome (6.67%, p=0.008), and other patients with isolated cleft palate (19.1%, p=0.044). Following initial VPI surgery, only 6.2% of 22q11.2DS patients required additional VPI surgery; however, 62.5% of 22q11.2DS continued to have moderate/severe hypernasality. Kaplan-Meier analysis revealed a significantly shorter time period between cleft palate repair and VPI surgery in syndromic patients (p=0.044), when adjusting for age at cleft palate repair, cleft phenotype (submucosal, cleft lip and/or palate), and Veau class.

Conclusion: This retrospective study highlights the variability in rates and timing of VPI surgery in patients with syndromic cleft lip and palate. Patients with 22q11.2DS had significantly higher rates of VPI surgery. Additionally, our data suggest an accelerated timeline between cleft palate repair and VPI surgery for syndromic patients. These findings highlight the nuanced clinical course of VPI among patients with different craniofacial syndromes, and variability in the success of speech outcomes following surgical treatment of VPI. Therefore, meticulous preoperative planning with an individualized approach to management is imperative in this population.

Purpose: The timing of primary repair in non-syndromic cleft palate remains controversial. Recent evidence suggests earlier repair is associated with a lower incidence of velopharyngeal insufficiency (VPI). We aim to evaluate these findings in a large cohort study using causal inference

Methods: All non-syndromic cleft palate repairs in California were extracted between 2000-2021 from the California Health Care Access and Information (HCAI) database. These cases were linked with VPI surgery following cleft palate repair based on unique identifiers. The incidence of VPI surgery was evaluated with propensity score matching. Early cleft palate repair was defined as < 7 months of age versus traditional cleft palate repair at over 11 months of age. Standardized mean differences (SMD) were measured before and after matching for potential confounders including sex, race, payer, and distance from patient home to hospital.

Results: 52,007 cleft palate repairs were included of which 12,169 (23.3%) were repaired early and 39,838 (76.7%) were repaired late. Early cleft palate repairs were found to have a VPI incidence of 1.2% (13/1,000), compared with 6.1% (61/1000) in the traditional repair cohort. Post-matching, the average treatment effect of early repair was a 6.3% reduction in VPI (p<.001, 95% CI -6.3- -5.4%). All covariate SMDs were less than |0.1| after matching.

Conclusion: Our cohort study demonstrates a significantly reduced incidence of VPI in children with primary cleft palate repair prior to 7 months of age. These findings in light of recent trial data suggest that craniofacial centers should consider early cleft palate repair in appropriate patients.

Purpose:

This study aimed to investigate frontal sinus volume as a potential indicator of cranial compensatory growth in unoperated normocephalic nonsyndromic sagittal craniosynostosis (NNSC) patients compared to age- and sex-matched controls. Previous studies have indicated that frontal sinus volume is suppressed in unoperated craniosynostosis and may be an intracranial space conservation phenomenon.

Methods:

Head CTs from 21 unoperated NNSC patients at our institution were utilized in this study and matched with age- and sex-matched control subjects. Frontal sinus volumes were measured using Syngo Via, and statistical analysis was performed. Inclusion criteria for patients included presence of a frontal sinus with unoperated sagittal craniosynostosis, whereas inclusion criteria for controls included presence of a frontal sinus and no craniofacial trauma. Pearson correlation coefficient and significance values were calculated to evaluate the relationship between frontal sinus volume and craniosynostosis.

Results:

Six of 21 patients with NNSC were found to have presence of frontal sinus volumes, compared to controls where all patients had presence of a frontal sinus. When compared to age-sex matched controls, our analysis revealed a Pearson correlation coefficient of 0.993 and a high significance of p < 0.001, indicating a strong association between decreased-to-absent frontal sinus volume in unoperated normocephalic nonsyndromic sagittal craniosynostosis patients.

Conclusion:

The findings of this study demonstrate a correlation between decreased-to-absent frontal sinus volume in patients with NNSC in comparison to normal controls. These findings highlight the potential utility of frontal sinus volume as a diagnostic tool in identifying the degree of compensation that the skull has undergone in nonsyndromic sagittal craniosynostosis

Introduction Craniosynostosis can lead to anatomic deformation of the cranial vault, skull base, and orbits. Select phenotypes of craniosynostosis are associated with ophthalmologic comorbidities including strabismus, refractive changes, and anisometropia. Prior studies have shown refractive values to decrease post-operatively(1). The objective of this study is to determine the incidence of ophthalmologic comorbidities and compare the severity of refractory errors before and after cranial vault remodeling (CVR) for craniosynostosis.

Methods Retrospective review of patients who underwent surgical correction for craniosynostosis with perioperative ophthalmology exams was conducted. Data points gathered included gender, race/ethnicity, craniosynostosis phenotype, surgical approach, surgical repair technique, syndromic, second craniosynostosis surgery required, strabismus type, refractory errors, development of amblyopia, and development of anisometropia. Cycloplegic refraction data was collected and subdivided into pre-operative, early post-operative (1 month to 6 months), and post-operative (1 year to 3 years) periods. A between subjects repeated measure ANOVA was performed comparing the pre-operative to early post-operative refraction, pre-operative to 2-year post-operative refraction, and early post-operative to 2-year post-operative refraction for the variables noted above. Paired T-tests were conducted to assess postoperative changes in refraction.

Results Our cohort included 168 patients and was 68% male and 80% white. The average age at repair was 11 months and 11% had a diagnosis of syndromic craniosynostosis. The most common craniosynostosis phenotype was sagittal (46%), followed by metopic (23%), unicoronal (11%), multisuture (9%), bicoronal (7%), and lambdoid (3%). Of all patients, 27% had strabismus, 60% of strabismus was a form of exotropia/exophoria, and 95% of the cohort had refractory errors- 40% had hyperopia alone and 41% had hyperopic astigmatism. Overall, 14% of the cohort developed amblyopia and 4% developed anisometropia. Results of the ANOVA showed no statistically significant difference in refraction changes between any of the variables for either the pre-operative to 6-month post-operative, pre-operative to 2-year post-operative, or the early post-operative to 2-year post-operative comparison. Paired t-tests showed that when all children were combined, refraction decreased by -0.44 (p<0.01) from the pre-operative to 1-year post-operative period and by -0.83 (p<0.01) from the pre-operative to 3-year post-operative period. There was no difference in refraction from the early post-operative period to the 1-, 2-, or 3-year post-operative period.

Conclusion Post-operative changes in refraction occur in children with craniosynostosis up to at least 3 years post-operatively. This study supports that there are no patient-specific variables available for predicting postoperative refractory changes in this patient population. The collected data re-emphasizes the need for long-term ophthalmology follow-up and integration of ophthalmology onto multidisciplinary craniosynostosis care teams for all children with craniosynostosis to identify development or alteration in ophthalmologic comorbidities.

1. Ntoula E, Nowinski D, Holmstrom G, Larsson E. Ophthalmological findings in children with non-syndromic craniosynostosis: preoperatively and postoperatively up to 12 months after surgery. BMJ Open Ophthalmol. 2021;6(1):e000677. Published 2021 Apr 26. doi:10.1136/bmjophth-2020-000677

PURPOSE: Early cleft lip repair (ECLR) is a safe and effective strategy for improving oronasal symmetry without nasoalveolar molding (NAM). Leveraging the anatomic plasticity of the neonatal skeleton, ECLR is hypothesized to reduce the burden of secondary procedures compared with traditional lip repair (TLR). The burden of these additional procedures places emotional, physical, and financial strain on patients, families, and the healthcare system. This study compares the long-term financial impact of ECLR and TLR±NAM.

METHODS: A retrospective cohort study was conducted on all patients with non-syndromic unilateral cleft lip who underwent primary lip repair by three craniofacial surgeons between 2004–2023. Exclusion criteria were bilateral cases, CL repair at older than 6 months of age, or less than 2 years of follow-up. Data on patient demographics, cleft characteristics, and revision surgeries were collected. Patients undergoing ECLR were compared to TLR±NAM with coarsened exact matching for cleft characteristics and surgeon. Hospital and physician costs were determined based on total charges for each intervention using institutional data. A budget-impact model was constructed to simulate the long-term costs associated with each cohort. One-way and probabilistic sensitivity analyses were used for cost analysis. The associated costs for ECLR and TLR were calculated and adjusted to 2023 United States dollars.

RESULTS: A total of 1,184 patients underwent primary cleft lip repair, of which 358 patients (145 ECLR, 47 TLR+NAM, 166 TLR-NAM) were eligible for the matching process. After matching, 130 patients (65 ECLR, 20 TLR+NAM, 45 TLR-NAM) were included. The mean follow-up time across all patients was 6.1±3.4 years. The median gestationally-corrected ages of the ECLR and TLR±NAM cohorts were 0.9 months and 3.3 months, respectively (p<0.001). The mean number of preoperative appointments for the ECLR and TLR±NAM cohorts were 1.1±0.3 (range 1–3) and 2.6±4.0 (range 1–17), respectively (p=0.002). The ECLR cohort had a significantly lower overall revision rate (10.7% vs. 26.9%, p=0.001). Stratifying based on NAM utilization, the ECLR cohort demonstrated persistently lower revision rates (12.3% ECLR vs. 40.0% TLR+NAM vs. 51.1% TLR-NAM, p<0.001). The total cost of ECLR was estimated to be $45,776 vs. $51,389 for TLR±NAM, resulting in $5,557 cost savings per patient. Probabilistic sensitivity analysis found ECLR to be cost-effective over TLR 94.1% of the time.

CONCLUSIONS: Early cleft lip repair was found to be cost-effective over traditional methods 94.1% of the time, saving approximately $5,557 in costs per patient. Our findings suggest that early cleft lip repair has the potential to decrease the burden of healthcare costs and secondary procedures for patients and families.

Purpose: This study evaluates the effect of postoperative helmet therapy (PHT) on long-term fronto-orbital symmetry and relapse outcomes in patients with unicoronal synostosis. Methods: Patients with unicoronal synostosis who underwent cranial vault remodeling and fronto-orbital advancement (CVR+FOA) between 2004-2023 were reviewed retrospectively. Patients with computed tomography (CT) imaging >3 months from CVR+FOA were included. Patients with and without PHT were compared to assess symmetry and surgical relapse of the fronto-orbital region. Mimics software (Materialise, Leuven, Belgium) was used to measure cranial anthropometrics from CT images. To measure anterior fronto-orbital position, a point was placed along the frontal bandeau at the mid-orbit of each hemisphere and the distance in relation to the coronal plane was measured. Fronto-orbital symmetry was calculated as the difference in anterior fronto-orbital position of the left and right hemispheres. Paired t-tests compared this symmetry measurement in the PHT vs non-PHT groups. Results: Twenty-four patients met inclusion criteria (12 PHT, 12 non-PHT). Patients were followed for 6.80±4.92 years. Average time from CVR+FOA to relapse evaluation (CT measurements) was 43.95±45.14 months. Patients with PHT had increased fronto-orbital symmetry compared to patients without PHT, trending towards significance (5.03±4.15mm vs 2.74±1.71mm, p=0.092). One patient without PHT required surgery for relapse. No patients with PHT required surgery for relapse. Conclusions: Radiographic data suggests that postoperative helmet therapy in patients with unicoronal synostosis may increase long-term fronto-orbital symmetry. Future studies will seek to increase cohort size to determine definitive significance.

BACKGROUND: Following bimaxillary orthognathic surgery, postoperative orthodontics are essential to facilitating tooth movement and refining final occlusion. Guiding elastics are an important component of this, helping the resting length of the musculature, connective tissue, and bones adapt to their new position.1 Accordingly, most centers opt for a 2-6 week course of elastics postoperatively, typically initiated with placement at the end of surgery.2 However, some patients, such as those with behavioral challenges, airway compromise, or medical conditions such as seizure disorders, may not be able to have elastics placed during surgery and have delayed initiation of elastic therapy. The purpose of this work was to evaluate whether delay in initiation of elastic therapy after surgery prolonged the time to achieving the desired occlusion in patients with cleft palate undergoing bimaxillary orthognathic surgery.

METHODS: This prospective cohort study included patients who underwent bimaxillary orthognathic surgery with a single surgeon at a tertiary academic center over a three-year period. The primary study variable was the use of guiding elastics (6 oz. heavy in a class II or class III pattern based upon the initial diagnosis) post-operatively, with initiation either immediately post-operatively (placed in the OR and maintained throughout early healing) or delayed post-operatively (no elastics placed in the OR and elastic therapy initiated at 2 weeks post-operatively and then maintained thereafter). Patients in the latter group had delayed elastics initiation due to behavioral issues, concern for airway obstruction, or other complicating medical conditions (e.g., active seizure disorder). Patients were followed weekly for the first six weeks after surgery. Demographics, perioperative clinical data, and cephalometric measurements were abstracted from the medical record. The primary outcome was the whether the desired occlusion was able to be achieved within 6 weeks of surgery and the time to achieving the desired occlusion.

RESULTS: Thirty-four patients were identified and met the study criteria. The mean age was 18.5 + 2.0 years, and 19 (55.6%) subjects were female. Twenty-two subjects had class III skeletal malocclusions; 12 subjects had class II skeletal malocclusions. Segmental maxillary osteotomies were performed in 9 subjects. Cleft severity was distributed as follows: Veau 2 (10), Veau 3 (17), Veau 4 (7). Twenty subjects had elastic therapy initiated immediately after surgery and fourteen subjects had elastic therapy initiated at 2-weeks post-operatively. All patients achieved the planned occlusion within 6 weeks post-operatively. There were no significant differences between the groups with regard to age, gender, type of malocclusion, segmental osteotomy, or cleft type (p > 0.10) Patients with immediate elastics achieved the planned occlusion at 2.1 + 0.8 weeks post-operatively, compared to 4.6 + 1.3 weeks post-operatively for those with delayed initiation of elastic therapy (p < 0.01).

CONCLUSIONS: In patients with a history of cleft palate undergoing bimaxillary orthognathic surgery, the desired occlusion can be achieved within 6 weeks of surgery whether guiding elastics are placed immediately after surgery or in a delayed fashion. Delayed initiation of post-operative guiding elastics increases the time to spontaneously achieving the planned occlusion by an average of 2.5 weeks.

REFERENCES: 1. Nocher AF, McMullan RE, Pierse D. Leaflet to aid postoperative placement of elastics after orthognathic surgery. British Journal of Oral and Maxillofacial Surgery. 2012;50(3):275-276. 2. Benato L, Miotto AV, Molinari RL, et al. Body mass index and weight loss in patients submitted to orthognathic surgery: a prospective study. Dental Press J Orthod. 2023;28(5):e2323107.