Faculty Bibliography

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.

INTRODUCTION/BACKGROUND:Digital simulators are potential solutions to challenges facing surgical education. The authors sought to evaluate the reach and utilization of a freely-accessible craniofacial surgery digital educational simulator. More importantly, we compare usage patterns between web-based and mobile-based platforms. METHODS:A 3-way collaboration between academic, non-profit (myFace, New York, NY), and biotechnology (Biodigital, New York, NY) stakeholders in 2015 produced the Craniofacial Interactive Virtual Assistant Pro (CIVA-Pro). CIVA-Pro is a freely-accessible craniofacial surgery digital educational simulator. In addition to the web-based platform, a mobile-based platform was launched in 2017. Usage analytics were collected and analyzed. RESULTS:Since its launch, 751 registered users from 117 countries had accessed CIVA-Pro. The total number of sessions was 9531, including 7500 web and 2031 mobile sessions. The total screen time was 403.9 hours, 290.3 for the web and 113.6 for the mobile platform. Comparison of the mean monthly screen time and number of monthly sessions between platforms since 2017 demonstrated a significantly higher mean monthly screen time (60.1 ± 33.2 versus 29.4 ± 16.5 hours; P = 0.002) and number of sessions (110.2 ± 36.1 versus 58.1 ± 31.9; P < 0.0001) for the mobile-based platform. The mean screen time per session was comparable (P = 0.86). CONCLUSION/CONCLUSIONS:A freely available digital craniofacial surgery educational simulator designed for surgical trainees can achieve significant global reach. Significantly higher utilization of the mobile-based platform of the simulator as compared to the web-based platform reinforces the need to invest in user-friendly, easily accessible, and widely available digital educational resources by key stakeholders to ensure optimal plastic surgery trainee education.

Pierre Robin sequence is the constellation of micrognathia, glossoptosis, and tongue-based airway obstruction. When airway obstruction is severe, feeding, growth, and respiratory demise are at risk. Neonatal mandibular distraction osteogenesis is a technique which improves tongue-based airway obstruction and avoids tracheostomy in patients with severe expressions of Pierre Robin sequence. Its efficacy in relieving airway obstruction is well documented, and it has become the surgical intervention of choice at many craniofacial centers. However, this is an uncommon procedure which can be performed within the first weeks of life, offering little space for a learning curve. The success of neonatal distraction and avoidance of complications is highly dependent on proper surgical technique. This report provides a brief overview of the disease, details the technique of the senior surgeon with captioned videos, describes the protocol used at our institution and reports long-term outcomes with a case description.

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.

Children with cleft and craniofacial conditions commonly present with concurrent airway anomalies, which often manifest as sleep disordered breathing. Craniofacial surgeons and members of the multidisciplinary team involved in the care of these patients should appreciate and understand the scope of airway pathology as well as the proper means of airway assessment. This review article details the prevalence and assessment of sleep disordered breathing in patients with craniofacial anomalies, with emphasis on indications, limitations, and interpretation of polysomnography.

Background/Purpose: b-Tricalcium phosphate (b-TCP), the most common synthetic bone replacement product, is frequently used in craniofacial reconstruction. Although solid b-TCP can be absorbed over time, the slow degradation rate (1%-3%/year) predisposes this product to exposure, infection, and fracture, limiting its use in the growing face where implants are required to grow and remodel with the patient. Our tissue engineering laboratory has successfully leveraged 3D printers to manufacture 3D-printed bioactive ceramic (3DPBC) scaffolds composed of b-TCP in an architecture which optimizes the needs of rigidity with efficient vascular ingrowth, osteogenesis, and degradation kinetics. The latter qualities are further optimized when the osteogenic agent dipyridamole (DIPY) is used. This long-term animal study reports on the new degradation kinetics profile achievable through this novel manufacturing and tissue engineering protocol. Methods/Description: Twenty-two 1-month-old (immature) New Zealand white rabbits underwent creation of unilateral 10 mm calvarial defects with ipsilateral 3.5 +/- 3.5 mm alveolar defects. Each defect was repaired with b-TCP 3DPBC scaffolds coated with 1000 mM DIPY. Rabbits were killed at 8 weeks (n = 6), 6 months (n = 8), and 18 months (n = 8). Bone regeneration and scaffold degradation were calculated using micro-CT images and analyzed in Amira software. Cranial and maxillary suture patency and bone growth were qualitatively analyzed using histologic analysis.
Result(s): Results are reported as a percentage of volumetric space occupied by either scaffold or bone. When comparing time points 8 weeks, 6 months, and 18 months, scaffolds showed significant decreased defect occupancy in calvaria (23.6% +/- 3.6%, 15.2% +/- 1.7%, 5.1% +/- 3.4%; P < .001) and in alveoli (21.5% +/- 3.9%, 6.7% +/- 2.7%, 0.1% +/- 0.2%; P < .001), with annual degradation rates 55.9% and 94.2%, respectively. Between 8 weeks and 18 months, significantly more bone regenerated in calvarial defects (25.8% +/- 6.3% vs 55.7% +/- 10.3%, P < .001) and no difference was found in alveolar defects (28.4% +/- 6.8% vs 32.4% +/- 8.0%, P = .33). Histology showed vascularized, organized bone without suture fusion.
Conclusion(s): The degradation kinetics of b-TCP can be altered through 3D printing and addition of an osteogenic agent. Our study demonstrates an acceleration of b-TCP degradation from 1% to 3% a year to 55% to 95% a year. Absorbed b-TCP is replaced by vascularized bone and there is no damage noted to the growing suture. This additive manufacturing and tissue engineering protocol has implication to future reconstruction of the craniofacial skeleton

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: The evolution of primary unilateral cleft lip repair represents a series of incremental modifications pioneered by a distinct group of master surgeons. It is through understanding the purpose of each evolutionary step, the limits and compromises of these steps, and the subsequent modifications which followed, can a greater understanding of the art of cleft lip repair be realized. This course will trace the conceptual development of unilateral cleft lip repair over the past 70 years using a novel, real-time computer-based cleft lip simulator. A first order accurate biophysics implementation within the simulator will be used to demonstrate the cleft lip repair techniques described to reveal the strengths and weaknesses of each stage of unilateral lip repair development. The course will begin with the Tennison Randall lower triangular lip repair, as it is still in common use today. This will be followed by Skoog, Wynn, and Mustarde adding an upper triangle to the lip repair. The various stages in the progression of the Millard repair will then be carefully traced along with the biophysics which are likely responsible for why Millard altered his original design. The modifications of the Millard design by other surgeons, and the reasons for them, will then be carefully traced. The modifications covered will be those of Noordhoff, Mohler, Cutting, Fisher, and others. The interaction between lip repair technique and primary correction of the cleft-lip nasal deformity will be discussed in detail. Simulator-based demonstrations will be augmented with patient examples from the senior author's clinical practice which illustrate the conceptual difficulties encountered at various stages in the historical development of primary unilateral cleft lip and nose repair. Methods/Description: The principal method used in this course will be real-time computer-based surgical simulation. A unilateral cleft lipnose model involving skin, mucosa, muscle, bone, cartilage, and teeth was derived from an MR scan of an adolescent with an unrepaired unilateral cleft. Alterations in the model are illustrated with first-order accurate biophysics using a new software base called projective dynamics. Surgical tools provided are scalpel, suture, hook, and undermine of both skin/mucosa and bone/cartilage. Surgical "'history" files are used to step through a succession of cleft lip repairs in the surgical eclectic. The presentation will be augmented with photographs from the senior author's long career further illustrating why successive alterations in technique were made

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: 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