Faculty Bibliography

INTRODUCTION/UNASSIGNED:Nasal duplication is a rare congenital deformity with many subtypes including supernumerary nostril. The challenge of surgical correction is to achieve nasal symmetry and restore nasal airflow. However, there is no defined protocol for treatment, especially with regard to presurgical therapy. METHODS/UNASSIGNED:We performed a review of literature of studies reporting on patients with supernumerary nostril to complete this review. We then report on a patient with supernumerary nostril who was treated with nostril expansion therapy prior to surgical intervention. RESULTS/UNASSIGNED:We identified 59 cases of nostril duplication. Because of the rarity of the condition, treatment protocols varied greatly. For our patient, preoperative nasal appliance therapy was implemented for 3 months prior to surgical intervention. Patient was followed-up regularly for 1 year. DISCUSSION/UNASSIGNED:Although literature on nasal duplication is scarce, there is a general agreement that early intervention has psychological, anatomic, and functional benefits to the patient. In our case report, nostril expansion therapy was easy to implement and facilitated surgical reconstruction, resulting in aesthetic outcome and expanded airway 1 year postoperatively.

BACKGROUND/UNASSIGNED:The aim of this study is to assess the effect of nasoalveolar molding (NAM) versus no-NAM on nasal morphology in patients with unilateral cleft lip and palate (UCLP) at the time of nasal maturity. METHODS/UNASSIGNED:A retrospective, single-institution review was conducted on all non-syndromic patients with UCLP. Inclusion criteria included age 14 years or above, unilateral cleft repair at the time of infancy, and adequate photography taken at nasal maturity and prior to rhinoplasty. Exclusion criteria included age less than 14 years, syndromic diagnosis, and rhinoplasty prior to nasal maturity. Ten parameters were measured twice from standardized clinical photographs using the Dolphin Imaging Software for establishment of intrarater reliability. Subjective analysis was achieved through completion of the Asher McDade grading scale by 3 expert cleft practitioners. RESULTS/UNASSIGNED:Nostril height, columellar angle, alar cant, vertical alar height, alar height angle, nasofacial angle, and nasolabial angle were found to be significantly less severe in patients who had undergone NAM in conjunction with surgical repair when compared with those who had undergone surgical repair alone. Asher McDade grading revealed significant improvement in nasal form, nasal symmetry/deviation, nasal profile, vermillion border, and overall score in patients who underwent NAM compared to no-NAM. CONCLUSION/UNASSIGNED:The use of presurgical NAM during infancy can improve nasal symmetry and nasal proportions at the time of nasal maturity.

OBJECTIVE/UNASSIGNED:To assess social and demographic influences on caregiver success and difficulty with nasoalveolar molding (NAM). DESIGN/UNASSIGNED:Retrospective review identified patients who began NAM between April 22, 2013, and April 18, 2017, at the New York University Langone Medical Center. Records were reviewed, and the following sociodemographic data retrieved: parental marital status, parental ages, number of siblings, distance traveled to clinic, insurance coverage, concurrent medical conditions, and need for an interpreter. PATIENTS/UNASSIGNED:Patients were included if complete charting was available; 106 patients met the inclusion criteria; 79 patients with unilateral and 27 with bilateral clefts. OUTCOME MEASURES/UNASSIGNED:Chart entries indicating incorrect appliance usage, emergency visits, phone calls, and noncompliance were recorded. Alveolar cleft gap closure was measured on pre- and posttreatment models in unilateral cases. Multiple regression analyses were performed to assess the influence of social variables on these outcomes. RESULTS/UNASSIGNED:= .019). CONCLUSIONS/UNASSIGNED:Alveolar cleft gap closure was more successful for older fathers, younger mothers, and married couples. Married couples were also less likely to experience treatment difficulties such as incorrect appliance usage or inadequate duration of wear, as were those with private insurance coverage.

OBJECTIVE/UNASSIGNED:Evaluate simulation-based comprehensive cleft care workshops as a reproducible model for education with sustained impact. DESIGN/UNASSIGNED:Cross-sectional survey-based evaluation. SETTING/UNASSIGNED:Simulation-based comprehensive cleft care workshop. PARTICIPANTS/UNASSIGNED:Total of 180 participants. INTERVENTIONS/UNASSIGNED:Three-day simulation-based comprehensive cleft care workshop. MAIN OUTCOME MEASURES/UNASSIGNED:Number of workshop participants stratified by specialty, satisfaction with the workshop, satisfaction with simulation-based workshops as educational tools, impact on cleft surgery procedural confidence, short-term impact on clinical practice, medium-term impact on clinical practice. RESULTS/UNASSIGNED:< .001) surgery procedural confidence following the simulation sessions. Participants also reported a positive short-term and medium-term impact on their clinical practices. CONCLUSION/UNASSIGNED:Simulation-based comprehensive cleft care workshops are well received by participants, lead to improved cleft surgery procedural confidence, and have a sustained positive impact on participants' clinical practices. Future efforts should focus on evaluating and quantifying this perceived positive impact, as well reproducing these efforts in other areas of need.

OBJECTIVE/UNASSIGNED:This systematic review aims to evaluate nasoalveolar molding (NAM) in the context of burden of care defined as physical, psychosocial, or financial burden on caregivers. SEARCH METHODS/UNASSIGNED:Following Preferred Reporting Items for Systematic Reviews and Meta-Analyses guidelines, 5 databases were searched from inception through December 24, 2019, for keywords and subject headings pertaining to cleft lip and/or palate and NAM. ELIGIBILITY CRITERIA/UNASSIGNED:Clinical studies on NAM with reference to physical (access to care, number of visits, distance traveled), psychosocial (caregiver perceptions, family interactions, breast milk feeding), and financial (direct and indirect costs) burden were included. DATA COLLECTION AND ANALYSIS/UNASSIGNED:Study selection was performed by 2 independent reviewers. RESULTS/UNASSIGNED:The search identified 1107 articles and 114 articles remained for qualitative synthesis. Burden of care domains were discussed but not measured in 43% of articles and only 25% assessed burden of care through a primary outcome. Of these, 20 articles reported on physical, 8 articles on psychosocial, and 12 articles on financial burden. Quality of evidence is limited by study design and risk of bias. CONCLUSION/UNASSIGNED:Nasoalveolar molding has been indiscriminately associated with burden of care in the literature. Although NAM may not be the ideal treatment option for all patients and families, the physical considerations are limited when accounting for the observed psychosocial advantages. Financial burden appears to be offset, but further research is required. Teams should directly assess the impact of this early intervention on the well-being of caregivers and advance strategies that improve access to care.

BACKGROUND:The authors present an institutional experience treating congenital and acquired temporomandibular joint (TMJ) ankylosis, detailing outcomes and potential risk factors of recurrence. METHODS:Retrospective chart review identified patients with TMJ ankylosis (1976-2019). Clinical records, operative reports, and imaging studies were reviewed for demographics, surgical operations, and ankylosis including maximal interincisal opening (MIO) and re-ankylosis. RESULTS:Forty-four TMJs with bony ankylosis were identified in 28 patients (mean age at any initial mandibular surgery: 3.7; range:0-14 years). Follow-up was 13.7 ± 5.9 years. Sixteen (57.1%) patients had bilateral ankylosis; 27(96.4%) had syndromes. Nine patients had congenital ankylosis, 16 had iatrogenic ankylosis (4.5 ± 3.7 years from initial distraction osteogenesis or autologous mandibular reconstruction) referred from outside institutions in 6 instances, and 3 had post-infectious ankylosis. Patients having their first mandibular operation at a younger age had more frequent reoperations for recurrent TMJ ankylosis, although this did not reach statistical significance. Mean improvement in MIO was 21.4 ± 7.3 mm. Ankylosis recurred in 21 (75%) patients. Five patients with congenital TMJ ankylosis required gastrostomy and remained at least partially dependent. Five patients had tracheostomy at the time of TMJ ankylosis surgery: 2 were eventually decannulated and 3 required repeat tracheostomy after ankylosis recurrence and remained tracheostomy-dependent. CONCLUSION/CONCLUSIONS:The clinical course of TMJ ankylosis in children affected by craniofacial differences is complex and typically involves a high rate of recurrence and multiple reoperations despite initial improvement in postoperative MIO. Younger age at initial mandibular surgery and number of operations require further investigation as potential predictors of recurrent TMJ ankylosis as well as tracheostomy and gastrostomy dependence.

Background/Purpose: Le Fort I advancement surgery is challenging in patients with clefts because of the palatal scar tissues. In this study, we investigated the outcome of Le Fort I advancement surgery (mild, moderate, and severe groups) and 1-year skeletal stability in patients with cleft lip and palate. Methods/Description: A retrospective chart review was performed to identify patients with nonsyndromic unilateral or bilateral cleft lip and palate who underwent maxillary Le Fort I advancement at skeletal maturity from 2013 to 2019. To satisfy the inclusion criteria, all patients had to have diagnostic quality cone beam computed tomography (CBCT) prior to surgery (T0), immediately postoperative (T1), and at 1-year follow-up (T2). A total of 59 patients (unilateral n = 34, bilateral n = 25) who underwent Le Fort I advancement was identified. Nineteen of these 59 patients were excluded due to insufficient radiographic records; thus, 40 patients with complete records were included in the study. The sample was comprised of 9 females and 31 males, with an average age of 19.1 +/- 3.21 years at the time of the surgery. Lateral cephalograms were extracted, traced, and superimposed using Dolphin Imaging software (V 11.95). Horizontal surgical movement (T0-T1) and postoperative relapse (T1-T2) at skeletal and dental level were quantified as linear changes at point A and upper incisor edge (U1-tip), respectively. Patients were divided into 3 groups according to the severity of surgical movement: mild (<5 mm, n = 9), moderate (5-10 mm, n = 20), and severe (>10 mm, n = 11). The statistical analysis was performed using 2-way repeated-measures ANOVA to test the difference of surgical movements and postoperative relapse between groups.
Result(s): The mean advancement (T0-T1) of all patients at point A was 8.1+/-2.8 mm and at U1-tip was 7.7+/-2.6 mm. In the mild, moderate, and severe groups, the mean advancement at point A were 4.6 +/- 1.3 mm, 7.7+/-1.1 mm, and 11.6+/-1.2 mm, and at U1-tip were 5.7+/-2.9 mm, 7.4 +/- 1.3 mm, and 10 +/- 2.6 mm, respectively. There were significant skeletal and dental advancements in all the 3 groups following Le Fort I surgery (P < .0001). At 1-year follow-up, the mean relapse (T1-T2) at point A was 1.2 +/- 1.1 mm and at U1-tip was 0.07 +/- 1.9 mm. When analyzed within the mild, moderate, and severe groups, the mean relapse at point A were 0.8 +/- 0.7 mm, 1.2 +/- 0.9 mm, and 1.9+/-1.5 mm and at U1-tip were -0.4+/-1.6 mm, 0.4+/-2.1 mm, and -0.2 +/- 1.5 mm, respectively. There was no significant difference in the relapse amount between the mild, moderate, and severe groups at skeletal and dental components (P > .05).
Conclusion(s): Le Fort I advancement surgery successfully corrected maxillary hypoplasia in patients with cleft lip and palate in all the 3 groups. This study also demonstrated that larger advancement in the severe group can result in equivalent skeletal stability when compared to the mild and moderate advancement. Though mild skeletal relapse was observed in all the 3 groups, none of the patients had to be reoperated

Background/Purpose: We present the first long-term outcomes analysis of the nasoalveolar molding (NAM) treatment protocol on patients with a cleft followed from birth to facial maturity. Methods/Description: Single-institution retrospective review of all patients with a cleft who underwent NAM between the years 1990 and 2000. All study patients completed cleft care treatment at the same institution and were followed by the same team members. Our institution's treatment protocol offers NAM to patients with a significant cleft nasal deformity and/or widely displaced alveolar segments. All patients underwent primary cleft lip and nasal repair prior to the age of 6 months. Gingivoperiosteoplasty (GPP) is performed, when possible, at the time of lip repair. Cleft palate repair is performed by 1 year of age. Collected data include surgical and orthodontic outcomes of cleft care such as cleft lip and palate repair, GPP, alveolar bone grafting (ABG), speech surgery for velopharyngeal insufficiency (VPI), palatal fistula repairs, orthognathic surgery, and revision surgery to the nose and/or lip.
Result(s): A total of 135 patients met the inclusion criteria. Mean length of follow-up was 18.8 years. Eighty-nine patients presented with a unilateral cleft (UNI) and 46 with a bilateral cleft (BI); 84% (113/135) of patients underwent GPP (UNI: 78% [69/89]; BI: 96% [44/46]), 43% (58/135) of patients underwent ABG (UNI: 40% [36/89]; BI: 48% [22/46]), 18% (24/135) of patients underwent speech surgery for VPI (UNI: 14% [12/89]; BI: 26% [12/46]), 3% (4/135) of patients underwent palatal fistula repair (UNI: 0% [0/89]; BI: 9% [4/46]), 31% (42/135) underwent orthognathic surgery (UNI: 22% [20/89]; BI: 48% [22/46]), and 11% (15/135) underwent revision surgery to lip, nose, or both prior to facial maturity (UNI: 9% [8/89]; BI: 15% [7/46]]. Of the patients who underwent GPP, 61% (69/113) did not require ABG (UNI: 65% [45/69]; BI: 55% [24/44]) and 42% (48/113) required neither ABG nor orthognathic surgery (UNI: 51% [35/69]; BI: 30% [13/44]).
Conclusion(s): Clinical outcomes of the NAM treatment protocol from birth to facial maturity demonstrate a low rate of revision surgery to the lip and nose, as well as a low fistula and VPI rate. The frequency of orthognathic surgery reported in this study is consistent with published data. In addition, 42% of patients who underwent NAM with GPP required neither ABG nor orthognathic surgery

Background/Purpose: Our study quantifies changes in skeletal, soft tissue profile, and globe position in patients with syndromic craniosynostosis after Le Fort I + III (LF I + III) surgery. Methods/Description: Patients with syndromic craniosynostosis who underwent LF I + III at the time of facial maturity were followed for at least 1 year. Each lateral cephalometric radiograph was traced using Dolphin Imaging software and superimposed at the sella. Changes in positions of the different landmarks at T0 (preoperatively), T1 (immediate postoperatively), and T2 (1 year postoperatively) were measured by the software. Sixty-seven soft tissue and skeletal landmarks were digitized and measured. LF III skeletal changes were measured by changes in lateral orbit and orbitale. LF I skeletal changes were measured at the A point and U1. Corresponding soft tissue profile and globe position were studied. All data were measured along the x-axis.
Result(s): Twelve patients included in our study have the following syndromes: Crouzon (n = 6), Pfeiffer (n = 2), Apert (n = 1), Antley-Bixler (n = 1), cleidocranial dysplasia (n = 1) and frontonaso dysplasia (n = 1). Nine patients had previous history of LF III distraction. Standard descriptive statistics was used. Data were analyzed using paired T test. Lateral orbit advanced 5.49 mm (T0-T1) on average, with a P value of 1.3-5, and 5.94 mm (T0-T2) on average; 0.45 mm (T1-T2) change with a P value of .96 suggests the lateral orbit is stable. Similar advancement at orbitale is observed at 5.68 mm (T0-T1) and 6.42 mm (T0-T2). The globe moved anteriorly by 1.98 mm (T0-T1) with a P value of .025 and anteriorly by 0.944mm(T0-T2). The change between T2 and T1 is 1.04 mm (P value: .26), which suggests the globe moved backward after postsurgical swelling subsided. The ratios of movement (globe to lateral orbit) between T0-T1 and T0-T2 are 31% and 16%, respectively. The decrease in ratio can be attributed to the reduction in soft tissue swelling at T2. Restoring position of the globe relative to the lateral orbit decreases the risk of exposure keratitis, keratoconjunctivitis sicca, and corneal ulceration. Anterior nasal spine and point A were advanced by 9.38 and 10.08 mm, respectively, between T0 and T1, and 9.01 mm and 8.51 mm, respectively, between T1 and T2. At the occlusal level, U1 advanced 10 mm and L1 moved back 1.45 mm between T0 and T1. Menton moved back 1.25 mm (T0-T1) but advanced by 2.48 mm (T0-T2). This change in direction is due to splint use at T1 as it rotates mandible clockwise.
Conclusion(s): In our cohort, LFI + III surgery improved both midface deficiency and proptosis in those with syndromic craniosynostosis.Combined Le Fort I + III surgery allows surgeons to perform differential corrections of the midface at the orbital and the dentition level. This is ideal for proptosis correction and establishing optimal jaw relationship

Background/Purpose: Temporomandibular joint (TMJ) ankylosis is an uncommon but debilitating condition which can affect feeding, speech, dental health, facial growth, and quality of life. We present an institutional experience treating congenital and acquired TMJ ankylosis, detailing outcomes and potential risk factors of recurrence. Methods/Description: Patients with ankylosis of the TMJ were identified through retrospective chart review (1976-2019). Clinical records, operative reports, and imaging studies were reviewed for demographics, surgical operations, and ankylosis including mean interincisal opening (MIO) and reankylosis.
Result(s): Forty-four TMJs with bony ankylosis were identified in 28 patients, 27(96.4%) of whom had syndromes. Mean age at any initial mandibular surgery was 3.7+/-3.6 (range: 0-14 years). Follow-up was 13.7 +/- 5.9 years. Sixteen (57.1%) patients had bilateral ankylosis. Nine cases of ankylosis were congenital, 16 were iatrogenic (4.5 +/- 3.7 years from initial distraction osteogenesis or autologous mandibular reconstruction) referred from outside institutions in 6 cases, and 3 were postinfectious. Patients having their first mandibular operation at a younger age had more frequent reoperations for recurrent TMJ ankylosis, although this did not reach statistical significance. Improvement in MIO was 21.4 +/- 7.3 mm. Ankylosis recurred in 21(75%) cases, 11 of which were iatrogenic, requiring an average of 2 reoperations (range: 1-8). Five patients with congenital TMJ ankylosis required gastrostomy and remained at least partially dependent. Five patients had tracheostomy at the time of TMJ ankylosis surgery: 2 were eventually decannulated and 3 required repeat tracheostomy after ankylosis recurrence and remained tracheostomy-dependent.
Conclusion(s): Craniofacial anomalies, younger age at mandibular surgery, and number of operations portend to increased risk of TMJ ankylosis as well as tracheostomy and gastrostomy dependence. Despite initial improvement in postoperative MIO, pediatric TMJ ankylosis is associated with high recurrence and multiple reoperations