Faculty Bibliography

This case presents a facially mature patient with Beckwith-Wiedemann Syndrome (BWS) who presented with severe class III malocclusion. Computed tomography imaging revealed an anterior crossbite of 19 mm and a narrow pharyngeal airway at the level of the tongue base precluding mandibular setback surgery. The patient was indicated for a LeFort III combined with a LeFort I advancement, each of 10 mm, for a 20 mm combined advancement. Stable, functional occlusion was achieved without airway compromise. This novel use of the combined LeFort III/I can restore stable class I occlusion in patients with BWS at risk for tongue base airway compromise.

OBJECTIVES/OBJECTIVE:Cleft lip repair has traditionally been performed as an inpatient procedure. There has been an interest toward outpatient cleft lip repair to reduce healthcare costs and avoid unnecessary hospital stay. We report surgical outcomes following implementation of an ambulatory cleft lip repair protocol and hypothesize that an ambulatory repair results in comparable safety outcomes to inpatient repair. DESIGN/SETTING/METHODS:This is a single-institution, retrospective study. PATIENTS/PARTICIPANTS/METHODS:Patients undergoing primary unilateral (UCL) and bilateral (BCL) cleft lip repair from 2012 to 2021 with a minimum 30-day follow-up. A total of 226 patients with UCL and 58 patients with BCL were included. INTERVENTION/METHODS:Ambulatory surgery protocol in 2016. OUTCOME MEASURES/METHODS:Variables include demographics and surgical data including 30-day readmission, 30-day reoperation, and postoperative complications. RESULTS:There were no differences in rates of 30-day readmission, reoperation, wound complications, or postoperative complications between the pre- and post-protocol groups. Following ambulatory protocol implementation, 80% of the UCL group and 56% of the BCL group received ambulatory surgery. Average length of stay dropped from 24 h pre-protocol to 8 h post-protocol. The 20% of the UCL group and 44% of the BCL group chosen for overnight stay had a significantly higher proportion of congenital abnormalities and higher American Society of Anesthesiology (ASA) class. Reasons for overnight stay included cardiac/airway monitoring, prematurity, and monitoring of comorbidities. There were no differences in surgical outcomes between the ambulatory and overnight stay groups. CONCLUSIONS:An ambulatory cleft lip repair protocol can significantly reduce average length of stay without adversely affecting surgical outcomes.

Background/Purpose: After LeFort I advancement surgery, soft tissue changes are unpredictable, especially in patients with orofacial clefts, as scar tissue from primary repair can alter soft tissue responses. Therefore, this study aimed to measure and evaluate soft tissue response following LeFort I advancement in skeletally matured patients with complete cleft lip and palate (CLP). Methods/Description: The cohort of 26 patients with non-syndromic CLP who underwent Le Fort I osteotomy between 2013 and 2019 and met the inclusion criteria. Patients were included if they had lateral cephalograms or CBCT at pre-operative (T1), immediately post-operative (T2), and one-year follow-up (T3). Patients who underwent nose/lip revision surgery before T3 were excluded. Four skeletal and dental hard-tissue (ANS, point A; A-point, upper incisor most labial; U1-most, upper incisor edge; U1-tip) and 5 softtissue (tip of nose or pronasale; Prn, subnasale; Sn, superior labial sulcus; SLS, upper lip anterior or labrale superius; LS, and stomion superius; SIMS) landmarks were digitized and measured. For the outcome analyses, 5 ratios of soft- to hard-tissue changes (Prn/ANS, Sn/A-point, SLS/A-point, LS/U1-most, and SIMS/ U1-tip) were calculated for each group, and associations between hard-and-soft tissue counterparts were assessed using Pearson correlation coefficient (r).
Result(s): Sixteen patients had UCLP, and 10 patients had BCLP. At one-year follow-up (T1-T3), the mean advancement in UCLP and BCLP groups at ANS were 4.4+/-3 and 4.7+/-3.9 mm, from point A were 6.6+/-2.5, 8.8+/- 2.6 mm, respectively. The mean horizontal changes of the corresponding soft tissue anatomy, Prn, were 2.7 +/-1.7, 4.6+/-3.5 mm, from Sn, were 3.9+/-1.9, 6.2+/-2.4. mm, and from SLS were 5.2+/-2.5, 7.4+/-2.8 mm. The mean advancement in at upper incisor most labial were 7.2+/-2.7 and 8.4+/-2.4 mm, and from the upper incisal edge were 7.5+/-2.9 and 8.4+/-2.7. mm. The mean horizontal changes of the soft tissue counterpart, LS, were 5.6+/-2.9, 7.9+/- 3.7 mm, and SIMS were 6.0+/-3.2, 7.3+/- 2.7 mm. All skeletal, dental, and soft tissue advancements from T1-T3 were significant (P< 0.01) except for Sn and LS in both groups and SIMS in UCLP group. For ratio and correlation analyses in UCLP and BCLP groups, Prn/AND were 0.48 (r=0.40) and (r=0.00), Sn/A-point were 0.58 (r=0.79) and 0.70 (r=0.77), SLS/A-point were 0.79 (r=0.82) and 0.85 (r=0.80), LS/U1-most were 0.74 (r=0.92) and 0.96 (r=0.74), and SIMS/U1-tip were 0.78 (r=0.75) and 0.82(r=0.67), respectively. All associations except for Prn/ANS were statistically significant (P< 0.01).
Conclusion(s): This study demonstrated a linear relationship between soft- and hard-tissue changes in the maxillary landmarks following LeFort I advancement in patients with complete cleft lip and palate (UCLP and BCLP)

Background/Purpose: Primary cheiloplasty for infants born with cleft lip and palate has long been preceded and facilitated by oral appliances intended to aid in feeding, narrow the cleft width, or mold the surrounding cartilages. Presurgical infant orthopedic (PSIO) therapy has evolved in both its popularity and focus of treatment since its advent. Nasoalveolar molding (NAM), nasal elevators, the Latham appliance, lip taping, and passive plates are the modern treatment options offered by cleft teams, and each varies in their associated protocols and treatment philosophies. The purpose of this study is to examine trends in the currently available modalities of PSIO care and PSNS for the management of patients with cleft lip and palate. Methods/Description: Methods: An electronic survey comprised of eight questions was distributed to the one hundred and sixty-seven cleft team coordinators listed by the American Cleft Palate Association. The survey reported on team setting, provider availability, PSIO treatment offerings, relative contraindications, and use of postsurgical nasal stenting. Descriptive statistics and analyses were performed using MS Excel (Microsoft, Redmond, WA) and SPSS (IBM, Chicago, IL).
Result(s): One hundred and two survey responses were received from the total one hundred, and sixty-seven sent, resulting in a response rate of 61%. The majority of settings were children's specialty hospitals (66%) or university hospitals (27%). PSIO was offered by 86% of cleft teams, and the majority of those (68%) provided NAM. Nasal elevators and lip taping are offered at 44% and 53% of centers, respectively. Latham and passive plates are both offered at 5.5% of centers. Teams with a dental specialist as the PSIO provider offered NAM significantly more than centers with surgeons as the provider of PSIO. Most centers (45%) had an orthodontist providing treatment. Patients are considered contraindicated for treatment at many centers for reasons such as mild cleft severity (46%), medically-compromised (42%), advanced age at first visit (29%), far commute (35%), and/or financial reasons (16%). The majority of centers use postsurgical nasal stenting (86%), and almost all insert the device immediately in the operating room (88%).
Conclusion(s): NAM is the most popular PSIO technique in North American cleft centers followed by the nasal elevator, suggesting that the nasal molding component of PSIO of critical influence on current treatment practices. With 86% of centers providing PSIO, access to care is improving with an increasing variety of treatment modalities. Postsurgical nasal stenting is also gaining popularity. The use of various PSIO techniques is ubiquitous, but the emphasis on nasal molding may continue to be the driving force for treatment in the future

Background/Purpose: Nasoalveolar molding (NAM) in combination with primary gingivoperiosteoplasty (GPP) may obviate the need for a secondary alveolar bone graft. While the long-term facial growth following GPP has been well documented, no study has evaluated the transverse growth of the cleft-maxilla following NAM and GPP. Here we report the effects of NAM and GPP on the maxillary transverse dimension in patients with complete unilateral cleft lip and palate (UCLP). Methods/Description: A retrospective single-institution review of nonsyndromic patients with complete unilateral cleft lip and palate born between 2005 and 2010 was completed. Patients were divided into four groups based on their interventions: 1) NAM-GPP with adequate bone formation 2) NAM-GPP without adequate bone formation (requiring ABG) 3) NAM-no GPP (requiring ABG), and 4) No NAM-no GPP control (patients who received primary surgeries outside of our institution). Cone-beam computed tomographic scans (CBCTs) taken at the early-mixed dentition stage, prior to orthodontic intervention, were used to assess the anterior and posterior maxillary transverse dimensions. The transverse discrepancy at the affected and non-affected sides was measured at the level of the primary canines (anterior dimension) and the permanent first molars (posterior dimension) to the maxillary midline. Wilcoxon signed-rank tests were used to compare the transverse dimension of the affected versus non-affected sides within each group. Mann-Whitney U tests were used to compare each NAM group with the no NAM-no GPP control group.
Result(s): A total of 85 patients were included in this study (mean age = 8.7). Male patients (50.6%) and the left side (64.7%) were most affected. Of the 85 patients, 26 (30.6%) underwent NAM-GPP with adequate bone formation, 22 (25.9%) underwent NAM-GPP but required ABG, 16 (18.8%) underwent NAM without GPP, and 21 (24.7%) underwent no NAM-no GPP. Median values were significantly different in the anterior maxilla between the affected and nonaffected sides across all four groups (p = 0.001). The transverse dimension at the affected side also revealed a significant difference in both the NAM-GPP (with adequate bone formation) and the NAM-GPP (requiring ABG) groups compared to the no NAM-no GPP group (p= 0.022 and p= 0.001, respectively). There was no significant difference between the NAM-no GPP group compared to the control (p = 0.059). Distances to the molars of the affected and nonaffected sides were not statistically significant within or across any of the groups (p > 0.05).
Conclusion(s): In patients with UCLP, the maxillary primary canine transverse dimension on the affected side is significantly reduced in patients undergoing NAM and GPP compared to the control. However, the position of the maxillary first molars appear to be unaffected by NAM and GPP

Background/Purpose: Gingivoperiosteoplasty (GPP) is a procedure performed at the time of primary cleft lip or palate repair in which the alveolus is repaired without the need for bone graft. Although the success of GPP is reported up to 70%, the associated disparities with regards to access or receipt of GPP has not been studied. This study reports on patient access to GPP reconstruction. Methods/Description: The American College of Surgeons National Surgical Quality Improvement Program Pediatric (ACS NSQIP Peds) was queried from 2014 to 2019. Patients were selected using the Current Procedural Terminology (CPT) codes (Table 1). Patient race, gender, age at time of surgery, 30 day readmission, comorbidities and complications were recorded. Postoperative complications included surgical site infections (SSI), dehiscence and transfusion. Receipt of GPP was analyzed using binary logistic regression to control for variables that could potentially affect access to/ receipt of GPP. For multivariable analysis, Bonferroni correction was used.
Result(s): 23408 patients with a cleft were included in our analysis. 12590 were White, 1732 were Black/African American, 3914 were Hispanic, 2267 were Asian/other Pacific Islander, and 2905 did not have a reported Race. Amongst this cohort, 709 patients underwent GPP (2.25%). Patients who did not report/of unknown Race were less likely to undergo GPP (p = 0.001), while there was no statistically significant difference amongst access to GPP for Black/African American, Hispanic, or Asian/ other Pacific Islander patients. The average age of all patients was 2411 days. White patients had primary cleft repair at a younger age (p = 0.000) than non-White patients. There was no difference in gender or co-morbidities (cardiac risk factors and congenital/chronic lung disease, respectively) amongst all Races (p = 0.291, p = 0.276, p = 0.547). There was no statistically significant difference in unplanned 30-day readmission and 30-day postoperative complication (p = 0.326, 0.934, respectively). Patients with ASA class 3 or 4 and minor or major cardiac risk factors had a statistically significant higher chance of 30-day readmission (p = 0.000, 0.000, 0.000, 0.001, respectively).
Conclusion(s): Amongst reported Races there was no statistically significant difference with regards to access/receipt of GPP, but patients without a reported Race were less likely to undergo GPP. Undergoing GPP did not appear to increase the likelihood of 30-day readmission or postoperative complication. We did find that White patients received cleft lip/palate repair at a statistically significant younger age and Hispanic patients at a later age, which is similar to previous studies. Although there was no difference in access to GPP amongst Races, further studies to evaluate disparities in outcomes for children undergoing GPP needs to be elucidated

PURPOSE/OBJECTIVE:To examine the growth rate discrepancy of the affected and unaffected ramus heights in Pruzansky Type I and Type II mandibles. METHODS:This is a serial retrospective longitudinal growth study of 30 untreated patients (21 males and 9 females) with UCMF (age range from 5 years to 14 years). The mean age of patients was 8.5 years, and the mean follow-up records were 3.7 years. There were 13 patients in group I with a Pruzansky Type I mandible and 17 patients in group II with a Pruzansky Type II. The unaffected side of the mandible served as a control. Eighteen cephalometric parameters were examined at each of the two-time intervals. RESULTS:In patients with Pruzansky Type I mandible, the affected ramus grew on average 1.41 mm per year; the unaffected ramus grew 1.66 mm per year during the same period. In patients with Pruzansky Type II mandible, the affected ramus grew on average 0.84 mm per year; during the same period, unaffected ramus grew 1.79 per year. When the growth rate of the ramus height on the affected side was compared to the unaffected side, there was no statistically significant difference in Pruzansky Type I mandibles (p > .05); however, there was a statistically significant difference in the Pruzansky Type II mandibles (p < .05). CONCLUSION/CONCLUSIONS:The growth rate discrepancy of the affected and unaffected ramus heights was more severe in Pruzansky Type II mandibles than Pruzansky Type I mandibles explaining the progressive nature of facial asymmetry in Pruzansky II mandibles.

AIM/OBJECTIVE:To assess treatment outcome and 1-year stability of LeFort I advancement in patients with complete cleft lip and palate. METHODS:Thirty-five patients (age 20.65 ± 2.20 years) with unilateral (n = 25) or bilateral (n = 10) complete cleft lip and palate who underwent LeFort I advancement were included.Lateral cephalograms before surgery (T1), immediately postsurgery (T2), and at 1-year follow-up (T3) were superimposed, and the position of anterior nasal spine (ANS), A-point, and U1 Tip assessed using an x, y coordinate system. Differences between landmark positions at the 3-time points were analyzed using paired sample t-tests, with a significance defined as α ≤ 0.05. RESULTS:The mean surgical advancement in the horizontal direction (T2-T1) was 6.50 ± 2.62 mm at ANS (P < 0.001) and 7.05 ± 2.51 mm at A-point (P < 0.001). At a 1-year follow-up (T3-T2), the mean horizontal relapse at ANS was -1.41 ± 1.89 mm (P < 0.001) and -0.79 ± 1.48 mm at A-point (P 0.003). Mean horizontal relapse was 21.7% and 11% of surgical advancement when assessed at ANS and A-point, respectively. The central incisor tip position remained stable during the postsurgical period (0.12 ± 2.11 mm, P 0.732). At A-point, the mean vertical surgical change (T2-T1) was -0.96 ± 2.57 mm (P < 0.001). No significant post-treatment (T3-T2) vertical changes were detected at ANS or A-point. Phenotypic stability was excellent, with all patients maintaining positive overjet at 1-year follow-up. CONCLUSIONS:LeFort I advancement in complete cleft lip and palate is stable, with less than a 2 mm relapse after 1-year. Surgical overcorrection by 10% to 20% is recommended to compensate for the expected skeletal relapse.

ABSTRACT/UNASSIGNED:This is a retrospective study to evaluate the postsurgical position of the maxilla and mandible in 5 matured craniofacial patients with unilateral craniofacial microsomia who underwent 2 jaw surgical procedures using computerized surgical planning. The craniofacial surgeon and orthodontist completed the virtual surgical treatment plan with a biomedical engineer's assistance via a web meeting. The treatment plan of each patient included 2 jaw surgery with genioplasty. At the maxillary dental midline, the planned mean advancement was 4 mm; yaw, a rotational correction towards the unaffected side was 4.96 mm; and impaction was 2.74 mm. The mean advancement measured at point B was 10.5 mm, and the rotational correction towards the unaffected side was 6.58 mm. The mean advancement following genioplasty was 8.43 mm, and the mean transverse correction was 6.33 mm towards the midsagittal plane. The intermediate surgical splint, final surgical splint, bone graft templates, and cutting guides were constructed utilizing computer-aided design/computer-aided manufacturing technology. The surgeon executed the treatment plan in the operating room using appropriate computer-generated guides and splints. A postsurgical cone-beam computed tomography scan was obtained and superimposed on the surgical treatment plan using Simplant OMS 10.1 software. The cranial base was used as a reference for superimposition. Three-dimensional color-coded displacement maps were generated to visually and quantitatively assess the surgical outcome. There was a mean error of 0.88 mm (+0.30) for the position of the maxillary anatomical structures from the planned position, and the anterior mandibular anatomical structures were on average 0.96 mm (+0.26) from the planned position.

PURPOSE/OBJECTIVE:The purpose of this study was to measure the association between the magnitude of advancement and dental and skeletal relapse in patients with cleft lip and palate (CLP). METHODS:A single-institution retrospective cohort study of skeletally matured patients with CLP who underwent isolated Le Fort I advancement surgery between 2013 and 2019 was studied. Patients were included if they had lateral cephalograms or cone-beam computed tomography (CBCT) at preoperative (T1), immediately postoperative (T2), and 1-year follow-up (T3). Lateral cephalometric landmarks were digitized and measured. The sample was divided on the basis of the magnitude of skeletal advancement: minor (<5 mm), moderate (≥5 but <10 mm), and major (≥10 mm) advancement groups. The mean advancement and relapse were compared between groups using 1-way ANOVA. Correlation between the amount of surgical advancement and relapse was evaluated. RESULTS:Forty-nine patients with nonsyndromic CLP with hypoplastic maxilla met inclusion criteria and the sample consisted of 36 males and 13 females with the mean age of 19.5 years. In the minor, moderate, and major advancement groups, the mean advancement at point A was +4.1 ± 0.4, + 7.5 ± 1.4, and +11.3 ± 1.3 mm, respectively. At 1-year follow-up, the mean relapse at point A was -1.3 ± 1.2, -1.1 ± 1.2, and -1.7 ± 1.5 mm, respectively. There was no significant difference in the relapse amount between all surgical groups. No correlation between the magnitude of advancement and relapse was found. CONCLUSIONS:This study demonstrated no statistically significant difference in skeletal stability between a minor (<5 mm), moderate (≥5 but <10 mm), and major (≥10 mm) Le Fort I advancement groups in patients with clefts. Regardless of the degree of advancement, mild skeletal relapse was observed in all 3 groups.