Faculty Bibliography

BACKGROUND:Gingivoperiosteoplasty can avoid secondary alveolar bone grafting in up to 60 percent of patients with a cleft. However, preoperative predictors of success have not been characterized. This study reports on the preoperative alveolar segment position most favorable for successful gingivoperiosteoplasty. METHODS:The authors performed a single-institution, retrospective review of patients with a unilateral cleft who underwent nasoalveolar molding. Alveolar segment morphology was directly measured from maxillary dental models created before and after nasoalveolar molding. Statistical analysis was performed to identify parameters associated with the decision to perform gingivoperiosteoplasty and its success, defined as the absence of an eventual need for alveolar bone grafting. RESULTS:Fifty patients with a unilateral cleft who received nasoalveolar molding therapy were included in this study (40 underwent gingivoperiosteoplasty and 10 did not). Eighteen alveolar morphology and position characteristics were tested, including cleft gap width, horizontal and vertical positions of the alveolar segments, alveolar stepoff, and degree of alveolar segment apposition. Post-nasoalveolar molding vertical rotation of the greater segment and the percentage of segment alignment in the correct anatomical zone were statistically significant predictors of the decision to perform gingivoperiosteoplasty (86 percent predictive power). Cleft gap, greater/lesser segment overlap, alveolar segment alignment, greater segment horizontal rotation, and alveolar segment width following nasoalveolar molding were significant predictors of gingivoperiosteoplasty success (86.5 percent predictive power). CONCLUSIONS:Greater segment vertical rotation and proper alveolar segment anatomical alignment are positive predictors of the decision to perform gingivoperiosteoplasty. Post-nasoalveolar molding evidence of proper alignment and direct contact between the alveolar segments were significant predictors of successful gingivoperiosteoplasty. CLINICAL QUESTION/LEVEL OF EVIDENCE/METHODS:Risk, III.

Background/Purpose: In October 2012, a freely available, Internetbased cleft simulator was created in partnership among academic, nonprofit, and industry sectors. The purpose of this educational resource was to address disparities in surgical education resulting from resident work-hour restraints in developed countries and limited resources in developing nations. This report assesses demographics, utilization, and global impact of our simulator, in its fifth year since inception. Methods/Description: Simulator modules demonstrate surgical anatomy, markings, detailed procedures, and intraoperative footage to supplement digital animation. Available data regarding number of users, sessions, countries reached, and content access was collected. Surveys evaluating the demographic characteristics of registered users and simulator utility were collected by direct e-mail.
Result(s): The total number of simulator new and active users reached 2865 and 4086 in June 2017, respectively. A steady increase in number of new users (217-327), active users (407-555), and sessions (1956-2304) was noted from January 2016 to June 2017. From March 2015 to June 2017, our simulator was accessed in an increasing number of countries (85-136). In the same time frame, the number of sessions was 11 176, with a monthly average of 399.0 +/- 190.0. Developing countries accounted for 35% of sessions. New users generated the majority of sessions (59.8% +/- 8.5%), and the average session duration was 9.0 +/- 7.3 minutes. This yields a total simulator screen time of 100 584 minutes (1676 hours) and an average of 3725 minutes (62 hours) per month. A total of 151 users responded to our survey, the majority of whom were surgeons or trainees (87%) specializing in plastic, maxillofacial or general surgery (89%). Most users found the simulator to be useful (88%), at least equivalent or more useful than other resources (83%), and used it for teaching (58%).
Conclusion(s): Internet-based distribution of a freely available cleft surgery simulator can deliver an interactive teaching platform that reaches the intended target audience, is not restricted by socioeconomic barriers to access, and is judged to be useful by surgeons. Our simulator has reached more than 4000 active users since inception. The great majority of users are surgeons or surgical trainees. The total screen time over approximately 2 years exceeded 1600 hours. This suggests that future educational simulators of this kind may be sustainable by stakeholders interested in reaching this target audience

Background/Purpose: 3D-printed bioactive ceramic (3DPBC) scaffolds composed of beta-tricalcium phosphate (b-TCP) and coated in the osteogenic agent dipyridamole have been previously shown to heal critically sized calvarial defects in an adult animal model. This bone tissue engineering construct has yet to be applied in a pediatric craniofacial model and there has been evidence that other osteogenic agents such as BMP-2 can prematurely fuse growing sutures. The purpose of this study is to apply the described bone tissue engineering construct in a pediatric growing animal model and 1) quantify osteogenic potential in a growing calvarium; 2) maximize the scaffold design and dipyridamole (DIPY) concentration for the growing calvarium; and 3) characterize the effects of this bone tissue engineering construct on the growing suture. Methods/Description: Bilateral calvarial defects (10 mm) were created in 5-week-old New Zealand White rabbits (n = 14) 2 mm posterior and lateral to the coronal suture and sagittal sutures, respectively. 3DPBC scaffolds were constructed in quadrant form composed by varying pore dimensions (220, 330, and 500 mum). Each scaffold was collagen coated and soaked in varying concentrations of DIPY (100, 1000, and 10 000 muM). Controls comprised empty defects and collagen-coated scaffolds. Scaffolds were then placed into the calvarial defects to fill the bone space. Animals were euthanized 8 weeks postoperatively. Calvaria were analyzed using micro-computed tomography and 3D reconstruction.Mixed model analyses were conducted considering pore size and dosage effects on bone growth (a = 0.05).
Result(s): Scaffold group healing presented bone formation throughout the scaffold structure (defect marginal and central regions) while bone healing in empty sites was restricted to the defect margins, confirming its critical size dimension at 8 weeks in vivo. No significant difference in bone formation was detected when experimental groups were collapsed over pore size (P > .40). When pore size was collapsed over DIPY concentration, higher mean values were observed for the DIPYimmersed groups, and significance was shown between the 1000-muM and collagen groups (P < .05). Pore size and DIPY interaction was more pronounced for the 330-mum pore size where both the 100-and 1000-mum dosages presented significantly higher bone formation compared to collagen (P < .05). Across all concentrations of DIPY, including 10 000 mM (10 times greater than the experimental concentration, yielding the highest bone formation), sutures remained patent.
Conclusion(s):We present an effective bone tissue engineering scaffold design and dipyridamole concentration that significantly improve bone growth in a pediatric growing calvarial model and preserves cranial suture patency

Background/Purpose: Surgical education is increasingly relying on digital and online resources. However, the educational benefit of learning through digital media has not been rigorously tested in the field of cleft care. We present a prospective, randomized, blinded trial comparing the educational efficacy of a textbook vs digital simulation in teaching cleft lip repair. Methods/Description: Medical student volunteers (N = 35) were asked to draw a cleft lip repair on a standardized patient photograph of a complete unilateral cleft lip deformity (Pretest). They were then randomized to 1 of 2 study groups that demonstrated unilateral cleft lip repair markings: textbook (n = 17) or digital simulation (n = 18). After 20 minutes of study, volunteers were asked to draw a cleft lip repair a second time on the same standardized photograph (Posttest). Volunteers were then shown the educational material provided to the other study group and given a validated survey to compare the educational value of both resources. Cleft lip marking was graded in a blinded manner on 2 separate occasions using a 0-to 10-point scale created by 2 senior cleft surgeons. Paired and independent t tests were used to compare differences between groups. Intrarater reliability was evaluated using intraclass correlation coefficients (ICCs).
Result(s): Intrarater reliability was excellent for both pretest (ICC = 0.94; P < .001) and posttest (ICC = 0.95; P < .001) grading. Pretest performances between the textbook and simulator groups were comparable (0.82 +/-1.17 vs 0.64+/-0.95; P = .31). There was significant improvement in posttest performance compared to pretest in both the textbook (3.50 +/- 1.62 vs 0.82 +/- 1.17; P < .001) and simulator (6.44 +/- 2.03 vs 0.64 +/- 0.95; P < .001) groups. However, significantly greater improvement was demonstrated by the simulator group when compared to the textbook group (5.81 +/- 2.01 vs 2.68 +/- 1.49; P < .001). Participants thought the simulator increased interest (3.91 +/- 1.01 vs 2.31+/-1.21; P < .001); allowed learning (3.83+/-0.95 vs 2.17 +/-1.20; P < .001); was stimulating (3.74+/-0.98 vs 1.69+/-0.87; P < .001), clearer (3.66+/-1.08 vs 2.17+/-1.22; P < .001), and effective in teaching (4.14 +/- 0.94 vs 2.31 +/- 1.21; P < .001); and that they were likely to be recommended to others (4.00+/-1.11 vs 2.14+/-1.19; P < .001) more than the textbook.
Conclusion(s): A prospective, randomized, blinded study demonstrates superior cleft lip repair learning through digital simulation compared to textbook. Learners subjectively found digital simulation to be the superior educational medium. The cleft and craniofacial society should strongly consider investment into digital media platforms to teach future cleft surgeons

Background/Purpose: Outpatient primary cleft palate surgery (PCPS) has been implemented in many cleft centers; however, the prevalence of this procedure is unknown and its safety has been called into question. We queried the American College of Surgeons National Surgical Quality Improvement Program Pediatric database (ACS NSQIPPediatric) to evaluate perioperative complications associated with PCPS. Methods/Description: The ACS NSQIP-Pediatric database was reviewed from 2012 to 2015 using Current Procedure Terminology (CPT) codes for PCPS. Patients undergoing concurrent bone grafting or cleft lip surgery were excluded. Patients aged 5 years or younger were included. The goal of our study was to compare 30-day perioperative complications following outpatient vs inpatient PCPS. Statistical analyses were carried out using SPSS (Version 21.0. Armonk, NY: IBM Corp).
Result(s): We identified 4191 (2760 inpatient vs 1431 outpatient) eligible patients. The majority of patients were males (52.6%). Plastic surgeons performed these procedures most frequently (80.3%) followed by otolaryngologists (18.7%). Tympanostomy tube insertion was the most common concurrent procedure (17.1%). Mean age in days and weight in kilograms at surgery were 485.5 +/- 319.2 and 9.7 +/- 3.8, respectively. Mean age (509.3 +/- 346.9 vs 473.2 +/- 303.1; P < .001) and weight (9.9 +/- 4.0 vs 9.6 +/- 3.8; P = .01) were significantly higher in the outpatient group. The inpatient group included a significantly higher proportion of patients with congenital abnormalities (25.0% vs 21.2%; P = .01), history of stroke (1.0% vs 0.3%; P = .02), cardiac risk factors (14.4% vs 11.7%; P = .02) and oxygen dependence (1.8%vs 0.8%; P = .01). Univariate analysis showed that rates of superficial (3.5% vs 2.0%; P = .01) and deep (2.2% vs 1.0%; P = .003) wound dehiscence were significantly higher in the outpatient group. The rates of reoperation (1.2 vs 0.4; P = .02) and readmission (3.2 vs 1.5; P = .01) were significantly higher in the inpatient group. Mortality at 30 days was comparable between groups. After controlling for confounders, rates of superficial (OR = 1.99, P = .01, 95% CI [1.22, 3.24]) and deep (OR = 2.22, P = .01, 95% CI [1.25, 3.95]) wound dehiscence remained significantly higher in the outpatient group, whereas reoperation (OR = 2.8, P=.04, 95%CI [1.04, 7.14]) and readmission (OR=1.92, P= .02, 95% CI [1.14, 3.23]) rates were significantly higher in the inpatient group.
Conclusion(s): Outpatient PCPS is a common practice and appears to have an acceptable safety profile in appropriately selected patients. Outpatient surgery has a higher risk for wound complications. Inpatient surgery is associated with greater reoperation and readmission. Preoperative evaluation of patient risk factors and comorbidities is critical for optimal outcomes

Background/Purpose: Presurgical infant orthopedics has been employed since 1950 as an adjunctive neonatal therapy for the correction of cleft lip and palate. Most of these therapies did not address deformity of the nasal cartilage in unilateral and bilateral cleft lip and palate as well as the deficiency of the columella tissue in infants with a bilateral cleft. The nasoalveolar molding (NAM) technique is a unique approach to presurgical infant orthopedics to reduce the severity of the initial cleft of the alveolar and the nasal deformity, particularly in patients with bilateral cleft lip and palate. Methods/Description: In infants with bilateral cleft lip and palate, the premaxilla may be protrusive, mobile, and may show varying degree of asymmetrical displacement and rotation. In some instances, the premaxilla may be everted placed on top of the nasal tip with a very short columella length. Protruded premaxilla and the associated nasal deformity present a special challenge for the surgeon in achieving optimal repair during primary reconstructive surgery. This study session will demonstrate the NAM technique to treat patients with severe bilateral cleft lip and palate. The technique of correcting the protruded and asymmetrically displaced premaxilla, molding the alar cartilage and nonsurgical columella elongation will be discussed. Appliance design and weekly adjustment of the NAM appliance to accomplish the desired result will be presented. Special emphasis will be placed on leveling the premaxilla in asymmetric cases; retracting premaxilla in incomplete bilateral clefts and management of complications during the course of the NAM therapy will be discussed. For the successful outcome, the surgeon has to take the advantage of the NAM therapy during the primary repair. Surgical technique of 1-stage lip nose and alveolus surgery utilizing the presurgical preparation of infants with bilateral cleft lip and palate with NAM therapy will be discussed in detail. Long-term outcome of patients treated with NAM and primary reconstruction of nose lip and alveolus will be presented

Background/Purpose: Gingivoperiosteoplasty (GPP) performed concurrent with lip repair is an option for treating bony alveolar deficiency in patients with orofacial clefts. GPP has been demonstrated to produce bony continuity, eliminating the need for alveolar bone grafting (ABG) in two-thirds of treated cleft sites. The purpose of this study was to assess if early bone formation as produced by successful GPP influences maxillary canine eruption. Methods/Description: A retrospective chart review was conducted to identify patients born between January 1, 2000, and December 31, 2007, with unilateral complete cleft lip and alveolus, with or without cleft palate. Patients were included if they had successful GPP or ABG, and had panoramic or maxillary CBCT radiographs available at age 5 to 9 (T1) and 9 to 12 (T2) years, with a minimum of 6 months between radiographs. Panoramic images were excluded if a head positioning error produced an occlusal plane greater than 15degree from perpendicular to midline. Panoramic images were used to assess maxillary canine sector, angulation relative to midline and ipsilateral occlusal plane, and cusp tip height from ipsilateral occlusal plane. CBCT images were used to assess the horizontal distance between the canine cusp tip and the maxillary arch form. Clinical charts were reviewed to determine if canines erupted successfully or required intervention. Finally, canine mesial-distal and labio-lingual position after eruption was assessed using occlusal photographs. Outcomes in GPP and ABG groups were compared, and results were stratified by ipsilateral lateral incisor presence or absence.
Result(s): Seventy-nine patients met inclusion; 24 had successful bone fill after GPP, and 55 after ABG. In patients with cleft-side lateral incisors present, no significant differences were found between GPP and ABG groups in canine angulation, height, sector, eruptive outcome, or timing of eruption. When spontaneous canine eruption occurred, there was a statistically nonsignificant trend to more mesial eruptive position in patients who were treated with GPP. In patients with cleft-side lateral incisor agenesis, initial canine angulation did not differ. Patients who were treated with GPP demonstrated 10.8degree +/- 11.1degree spontaneous canine uprighting from T1 to T2, while canine angulation was maintained in the ABG group; this difference was statistically significant (P = .001). The GPP group demonstrated greater canine descent from T1 to T2, resulting in significantly less distance from the occlusal plane (5.8 +/- 4.8 mm) compared to the ABG group (9.4+/-4.2 mm). Horizontal distance to arch did not differ between the groups. In the GPP group, 75% of patients demonstrated successful spontaneous canine eruption, compared to 41% in the ABG group, though this did not reach statistical significance (P = .146).
Conclusion(s): Gingivoperiosteoplasty favorably influenced the angulation, height, and eruptive success of cleft-side canines in patients. These benefits were predominantly noted in patients with congenital absence of lateral incisors

Background/Purpose: LeFort III distraction osteogenesis is commonly recommended for children with syndromic craniosynostosis to reduce exorbitism, improve airway function, and decrease dysmorphism. This purpose of this study is to report on the long-term clinical outcomes of patients with syndromic craniosynostosis patients who have undergone early primary subcranial LeFort III distraction osteogenesis and who have been followed longitudinally through skeletal maturity. Methods/Description: Retrospective review of all patients who underwent LeFort III distraction osteogenesis between the ages of 3 and 11 years and were followed throughout development with longitudinal dental, medical, radiographic, and photographic evaluations conducted through skeletal maturity and beyond. Inclusion criteria entailed having preoperative medical photographs and cephalometric studies at 6 months and 1, 5, and 10 years postoperatively after the primary LeFort III distraction osteogenesis as well as cephalometric documentation 6 months and 1 year after the secondary midface advancement after skeletal maturity.
Result(s): Seventeen patients fulfilled inclusion criteria, with a mean age of 5.7 years at the time of initial LeFort III distraction. The mean advancement of point A was 14.9 mm anteriorly and 2.7 mm inferiorly along the x- and y-axis, respectively. Orbitale moved 10.5 mm anteriorly and 2.2 mm inferiorly along the x- and y-axis, respectively. At 10 years postoperatively, point A moved 3.4 mm anterior along the xaxis and 4.7 mm inferiorly along the y-axis, while orbitale moved 0.4 mm posteriorly and 3 mm inferiorly along the x- and y-axis, respectively. At the time of skeletal maturity, there was a return of occlusal disharmony from normal mandibular growth and a return of proptosis owing to remodeling of orbitale inferiorly, and the lateral orbital rim posteriorly, while the globe continued to grow in the anterior vector. All but 1 study patient underwent or is scheduled to undergo a secondary midface advancement at the LeFort III and LeFort I level after skeletal maturity was attained.
Conclusion(s): The data demonstrate that patients who undergo early LeFort III distraction osteogenesis before the age of mixed dentition will still most likely need a secondary midface advancement after skeletal maturity is reached given that there is a small degree of anterior growth at the level of the maxilla and no anterior growth at orbitale over time

Background/Purpose: Financial constraints are driving hospitals toward shortening patient stay and favoring outpatient surgery when appropriate. This study compares perioperative complications between the outpatient and inpatient settings in patients undergoing primary cleft lip surgery (PCLS) and identifies risk factors associated with complications and longer lengths of stay. Methods/Description: The American College of Surgeons National Surgical Quality Improvement Program Pediatric database (ACS NSQIP-Pediatric) was reviewed from 2012 to 2015 using Current Procedure Terminology (CPT) codes for PCLS. Patients older than 5 years or undergoing concurrent cleft palate surgery were excluded. The objective of our study was to compare perioperative complications following outpatient vs inpatient PCLS. Statistical analyses were performed using SPSS (version 21.0; IBM Corp, Armonk, NY).
Result(s): We identified 3142 (1721 inpatient vs 1421 outpatient) eligible patients. The majority of patients were males (63.0%) and underwent unilateral PCLS (78.5%). Plastic surgeons were the most frequent providers (85.1%) performing these procedures followed by otolaryngologists (14.0%). The most commonly performed concurrent procedures were cleft lip rhinoplasty (24.2%) and tympanostomy tube insertion (4.7%). Tissue grafting and gingivoperiosteoplasty were each performed in 1.2% of patients. Mean age in days and weight in kilograms at surgery were 200.8+/-223.3 and 7.0+/-3.2, respectively. Mean age (222.6+/-258.7 vs 182.9+/-187.7; P < .001) and weight (7.1 +/- 3.2 vs 6.9 +/- 3.5; P = .03) were significantly higher in the outpatient group. The inpatient group included a significantly higher proportion of patients with cardiac risk factors (12.9% vs 9.4%; P = .002) and oxygen dependence (1.1% vs 0.4%; P = .02). Rates of surgical site infections, wound dehiscence, reoperation, readmission, 30-day mortality, cardiac arrest, transfusion requirements, reintubation and operative time were comparable between groups on univariate analysis. Multivariate regression showed that an underlying structural pulmonary abnormality was significantly associated with a longer hospital length of stay (B = 4.94, P = .001, 95% CI [2.21,7.66]). No other significant associations were identified on multivariate analysis.
Conclusion(s): Surgical site infections, wound dehiscence, reoperation, readmission, 30-day mortality, and other perioperative complications are comparable in patients undergoing outpatient and inpatient PCLS. Patient selection remains the cornerstone for safe practice. Increasing health care fiscal constraints warrant considering outpatient PCLS for appropriate candidates

Background/Purpose: Skeletal Class III malocclusion with maxillary hypoplasia results in anterior-posterior discrepancy of the upper and lower incisors position and lip incompetence. This affects the ability to achieve appropriate placement of tongue and management of the intraoral air pressure for sound production, resulting in perceived nasal emission and abnormal articulatory gestures. The aim of this study is to investigate the relationship between occlusal disharmony, velopharyngeal competence, and speech outcomes in patients with skeletal Class III malocclusion undergoing orthognathic surgery. Methods/Description: Seventy-five consecutive patients between 2015 and 2017 who underwent orthognathic surgery secondary to maxillary hypoplasia were evaluated on type of orthognathic procedure (LeFort I only; LeFort I and III; LeFort I and BSSO); amount of anterior and vertical advancement achieved, severity of skeletal Class III malocclusion, and lip incompetence. Patients were divided into 3 groups: cleft lip and palate, syndromic, and those with noncleft/nonsyndromic skeletal deformity. Each group received speech assessments preoperatively and 3 months postoperatively (velopharyngeal competence, resonance and articulatory integrity, and the Pittsburgh Weighted Speech Score [PWSS]).
Result(s): Following exclusionary analysis, 58 patients were included in the study; cleft lip and palate (n = 28), syndromic (n = 15), and noncleft/nonsyndromic skeletal (n =15). Preoperatively, the cleft palate and syndromic groups with increased mean skeletal discrepancy and lip incompetence values presented with higher total mean PWSS scores, and mean nasal emission values than the noncleft/nonsyndromic group (P <= .001). Postoperatively, the cleft group that underwent LFI only presented with insignificantly improved total PWSS (P <= .99) and nasality (P <= .28) scores. The syndromic patients who had the most severe skeletal discrepancy preoperatively and who underwent an LFI and LFIII combined procedure continued to present with significantly higher mean nasal emission values postoperatively than cleft and noncleft/ nonsyndromic patients undergoing LFI only or LFI and BSSO (P < .001). There was no significant difference in the amount of anterior advancement achieved and degree of velopharyngeal competence.
Conclusion(s): Our results show that orthognathic surgery to correct skeletal disharmony does not provide increased compromise to the velopharynx. However, the severity of the skeletal Class III malocclusion and lip position, especially those with a syndromic diagnosis, increases presence of nasal emission pre- and postoperatively. This study suggests that severity of skeletal Class III malocclusion can contribute to the perception of nasality post orthognathic surgery. Severity of skeletal discrepancy should be considered as an additional iatrogenic factor related to perceived velopharyngeal competence in orthognathic surgery