Incorporating Cleft Lip Simulation Into a "Bootcamp-Style" Curriculum.

INTRODUCTION: The traditions of surgical education have changed little over the years. However, the increasing focus on patient safety and duty hour restrictions mandates that residents start developing complex skill sets earlier to ensure they graduate with procedural competency. Surgical training is poised to exploit high-fidelity simulation technology to mitigate these pressures. METHODS: By revisiting principles of adult learning theory, the authors created a "bootcamp-style" cleft lip curriculum that sought to (1) maximize educational impact and (2) pilot a high-fidelity procedural trainer permitting resident operative autonomy as part of that curriculum. Trainees participated in small group educational sessions comprised of a standard cleft didactic lecture, augmented by instructional video. Participants immediately processed knowledge from the lecture/video by "operating" on the simulator, allowing opportunities for questions and self-reflection, completing the learning cycle. A self-assessment survey was taken before and after each component of the session, including a self-confidence survey to conclude the session. Anthropometric measures of lip/nasal symmetry were assessed. RESULTS: Sixteen trainees participated in the program. Little increase in self-assessed knowledge/skill was seen after the lecture, but significant increases in most aspects of cleft lip repair were seen after simulation. The greatest increase in self-assessment was seen for the program as a whole, with significant differences across all aspects of the self-assessment. Higher levels of training were associated with both higher self-assessment scores and better lip symmetry. Regardless of level of training, all participants strongly agreed that simulation helped them actively engage in learning and should be a required aspect of training, whereas 94% (n = 15) thought simulation was much more effective than standard preparation alone. CONCLUSIONS: This pilot curriculum illustrates a mechanism to incorporate lessons from adult learning theory into plastic surgery training using a high-fidelity simulator for deliberate practice of cleft lip repair. Further evaluation is warranted to determine whether this didactic model can accelerate the acquisition of the complex skill set required for cleft lip repair and other surgical procedures.

Does the Nostril Shape Change After Le Fort I Advancement in Patients With Unilateral Complete Cleft Lip?

PURPOSE: Patients with unilateral cleft lip and palate (UCLP) may require Le Fort I advancement to correct maxillary hypoplasia after reaching skeletal maturity. The underlying cleft anatomy, previous operations, and scarring can affect nostril changes after maxillary advancement. The purpose of the present study was to determine whether Le Fort I advancement affects the nostril configuration (ie, width, axis, shape) in patients with UCLP. The specific aims were to (1) compare cleft and noncleft nostrils in patients with UCLP after maxillary advancement and (2) compare the changes in nostril configuration in patients with UCLP with those in noncleft controls after Le Fort advancement. PATIENTS AND METHODS: A retrospective case-control study of nonsyndromic, skeletally mature patients with UCLP and a case-matched control group without UCLP who had undergone single-piece Le Fort I advancement with alar cinch suture from 2010 to 2014. Patients were included if they had undergone pre- and postoperative 3-dimensional photogrammetry without intervening nasal revision. Three-dimensional anthropometry was used to evaluate changes in nostril axis and width, soft triangle angle, columellar show, and nasal width after orthognathic correction. RESULTS: The present study included 19 patients with UCLP (11 males; mean age, 18.0 years) and 19 noncleft controls (11 males; mean age, 18.7 years; P = .276). The mean sagittal advancement in the patients with UCLP and noncleft controls was 7.5 mm and 6.3 mm, respectively (P = .143). On average, the nostrils widened, the soft triangles flattened, and the columellar show increased. No significant difference was found in the changes to the nostril configuration between the cleft and noncleft sides in the patients with UCLP. No significant differences were found in the nostril changes between patients with UCLP and noncleft controls. CONCLUSIONS: Baseline nostril asymmetry is not altered by Le Fort osteotomy in patients with UCLP because both nostrils respond similarly to the deforming forces of maxillary advancement. Similarly, no differences were found in the nostril changes between the cleft and noncleft controls. These findings can aid proper surgical planning for cleft nasal revisions.

Can Robin Sequence Be Predicted From Prenatal Ultrasonography?

PURPOSE: Prenatal diagnosis of Robin sequence (RS) could promote safe delivery and improve perinatal care. The purpose of this study was to evaluate the correlation between prenatal ultrasonography (US) and magnetic resonance imaging (MRI) studies for assessing micrognathia to determine if US alone can be used to reliably screen for RS. MATERIALS AND METHODS: This was a retrospective case-control study of fetuses evaluated in the Advanced Fetal Care Center at Boston Children's Hospital from 2002 to 2017. To be included, 1) prenatal MRI and US must have been performed during the same visit, 2) the infant must have been live-born, and 3) the diagnosis must have been confirmed postnatally. Patients with images of inadequate quality for analysis were excluded. Patients were divided into 4 groups based on postnatal diagnosis: 1) RS (micrognathia, glossoptosis, and airway obstruction) (RS group), 2) micrognathia without RS (micrognathia group), 3) cleft lip and palate (CLP) without micrognathia (CLP group), and 4) gestational age-matched controls with normal craniofacial morphology (control group). The inferior facial angle (IFA) was measured using both imaging modalities and compared. Receiver operating characteristic curves were applied to identify a threshold for the diagnosis of RS from US. The sensitivity, specificity, positive predictive value, negative predictive value, and odds ratio were calculated. RESULTS: A total of 94 patients were included (mean gestational age at imaging, 24.9 ± 5.2 weeks), with 25 in the RS group (26.6%), 29 in the micrognathia group (30.9%), 23 in the CLP group (24.5%), and 17 in the control group (18.1%). The IFA was significantly smaller in the RS group than in all other groups on both US and MRI (P < .001). A moderate correlation was found between IFA measurements on US and MRI (intraclass correlation coefficient, 0.729). An IFA threshold on US of 45.5° maximized sensitivity (84%) and specificity (81%) for the diagnosis of RS. CONCLUSIONS: We suggest incorporating the IFA into routine prenatal US and referring patients for confirmatory MRI when the US IFA is lower than 45.5°.

Schneiderian Membrane Thickness Is Increased in Patients With Cleft Lip and Palate.

OBJECTIVE: Patients with cleft lip and palate (CLP) are more likely to have sinusitis. The purpose of this study is to determine whether patients with CLP have thickening of the Schneiderian membrane. Specific aims were to (1) compare Schneiderian membrane thickness in patients with CLP to noncleft controls, (2) evaluate whether membrane thickening is associated with cleft side in patients with unilateral cleft lip and palate (UCLP), and (3) evaluate if age and sex are predictors of mucosal thickening. DESIGN: Case-control study. SETTING: Tertiary care center. PATIENTS: Patients with CLP and controls. MAIN OUTCOME MEASURE: The primary outcome variable was maximum Schneiderian membrane thickness measured on cone beam computed tomography. The primary predictor variable was the presence of a cleft. Additional variables were cleft phenotype, age, and sex. RESULTS: There were 225 patients with CLP and 93 controls. Median mucosal thickness was 2.4 mm in cleft group and 0.0 mm in controls (P = .006). In cleft group, 56.7% of sinuses had mucosal thickness >2.0 mm compared to 38.2% in controls (P = < .004). Pathologic membrane thickening (>4.0 mm) was significantly higher in cleft group (P = .005). No statistically significant difference in mucosal thickness between cleft and noncleft sides in patients with UCLP. Linear regression showed no association between age or sex and Schneiderian membrane thickness. CONCLUSION: Schneiderian membrane thickening is more common in patients with CLP and is not associated with the side of the cleft in patients with UCLP.

Retropalatal Cross-Sectional Area Is Predictive of Obstructive Sleep Apnea in Patients With Syndromic Craniosynostosis.

OBJECTIVE: There is a high rate of obstructive sleep apnea (OSA) in patients with syndromic craniosynostosis (SCS). Little is known about the airway anatomy in this population. The purpose of this study is to characterize the 3 dimensional (3D) upper airway in patients with SCS with and without OSA. DESIGN: This is a retrospective study of patients with SCS treated at Boston Children's Hospital from 2000 to 2015. Patients were divided into OSA and no-OSA groups based on polysomnography. Predictor variables included age, sex, body mass index (BMI), and 3D upper airway measurements. The primary outcome variable was the presence or absence of OSA. Secondary outcome variables were apnea-hypopnea index and oxygen saturation nadir. Descriptive and bivariate statistics were computed, and significance was set as P < .05. RESULTS: There were 24 patients: 16 in the OSA group and 8 in the no-OSA group. The 2 groups did not differ significantly by age, BMI, or syndromic diagnosis. The presence of OSA was associated with a smaller minimum retropalatal cross-sectional area (minRPCSA; P < .001). In a logistic regression model controlling for age, sex, and upper airway length, minRPCSA was the primary predictor of OSA (P ≤ .002). Receiver operating characteristic analysis determined minRPCSA = 55.3 mm2 to be the optimal diagnostic threshold for OSA, with sensitivity = 100% and specificity = 87.5% (P < .001). CONCLUSION: A minRPCSA ≤55.3 mm2 is predictive of the presence of OSA in patients with SCS.

Is Obstructive Apnea More Severe in Syndromic Than Nonsyndromic Patients With Robin Sequence?

PURPOSE: Patients with syndromic Robin sequence (RS) are known to have poorer outcomes from operations to relieve obstructive apnea (OA) compared with those with nonsyndromic RS. The reason for this is unknown. The purpose of this study was to compare OA in unoperated syndromic and nonsyndromic patients with RS. We hypothesized that the syndromic group wouldhave more severe OA and that this could explain the inferior operative results. MATERIALS AND METHODS: This was a retrospective cohort study of patients with RS at Boston Children's Hospital from 1994-2018. Patients with at least 1 polysomnogram (PSG) before any airway operation were included. Patients were separated into syndromic and nonsyndromic groups. The apnea-hypopnea index (AHI) measured from PSGs was the primary outcome variable. Descriptive statistics and logistic regression were calculated, and statistical significance was set at P < .05. RESULTS: The study sample included 79 patients: 44 in the syndromic group (56%) and 35 in the nonsyndromic group (44%). The mean age at PSG was 7.4 ± 10.2 months, and 56% were female patients. The mean AHIs for the syndromic and nonsyndromic groups were not statistically significantly different: 11.9 ± 20.6 and 12.5 ± 13.9, respectively (P = .839). Among patients who underwent both an airway operation (tongue-lip adhesion, mandibular distraction, or tracheostomy) and a postoperative PSG (n = 42), the mean postoperative AHI was 5.7 ± 15.7 for the syndromic group (74% decrease, n = 20) and 1.3 ± 2.2 for the nonsyndromic group (92% decrease, n = 22, P = .038). CONCLUSIONS: In unoperated patients with RS, there is no significant difference in the severity of OA between syndromic and nonsyndromic patients. The inferior results seen after operations to relieve OA in syndromic patients may be a result of other factors related to the syndromic diagnosis rather than the level of preoperative OA.

Impact of Obstructive Sleep Apnea on Optic Nerve Function in Patients With Craniosynostosis and Recurrent Intracranial Hypertension.

PURPOSE: Assessment of combined impact of intracranial hypertension (ICH) and obstructive sleep apnea (OSA) on optic nerve function in children with craniosynostosis (CS). DESIGN: Retrospective cross-sectional study. METHODS: Patients treated at Boston Children's Hospital for CS who had an ophthalmic examination that included pattern reversal (pr)VEP (2013-2014) and history of ICH based on direct measurement, papilledema, or classic features on neuroimaging and during cranial vault expansion were included. History of OSA was determined by polysomnography and associated conditions, including apnea and (adeno)tonsillectomy. Subjects were divided into 4 groups: group 1, resolved ICH absent history of OSA; group 2, resolved ICH with history of OSA; group 3, recurrent ICH absent history of OSA; and group 4, recurrent ICH with history of OSA. Predictor variables included latency of P100 component of pattern-reversal visual evoked potential, best-corrected visual acuity, optic nerve appearance, visual fields, and global retinal nerve fiber layer. Primary outcome was association of prolonged P100 latency with resolved vs recurrent ICH and OSA. RESULTS: Twenty-eight children met inclusion criteria (mean age 11.6 ± 6.9 years): group 1 (n = 3), group 2 (n = 6), group 3 (n = 8), and group 4 (n = 11). P100 latencies were not prolonged in groups 1 and 2. Three of 8 in group 3 and 9 of 11 in group 4 had prolonged P100 latency. Group 4 was significantly worse than group 3 (P = .005). CONCLUSIONS: History of OSA, in addition to recurrent ICH, is associated with greatest risk of optic neuropathy with CS. Ophthalmologists should encourage early management of OSA as well as ICH to optimize ophthalmic outcomes.

Craniofacial Anthropometry: Normative Data for Caucasian Infants.

PURPOSE: Craniofacial anthropometry is a valuable tool for characterization of facial dysmorphology and evaluation of treatment outcomes. Databases of normal anthropometric ranges are limited for infants. The aim of this study is to establish normative data for craniofacial anthropometric measurements in Caucasian infants. METHODS: This is a prospective cross-sectional study including Caucasian infants (≤12 months old) that were recruited from a pediatric medicine practice and Boston Children's Hospital. Infants with craniofacial deformities, trauma or operations were excluded. The sample was stratified by age (in months) into 4 groups: 0 to 3, 3.1 to 6, 6.1 to 9, and 9.1 to 12. Three dimensional (3D) photographs were obtained for all subjects. Forty-five standard anthropometric points were plotted, and 37 measurements were made on the 3D photographs. Two evaluators independently performed all measurements. One examiner repeated the measurements on 25% of the subjects. Intraclass correlation coefficients (ICC) were calculated to assess inter-rater and intra-rater agreement. RESULTS: Thirty-three subjects were enrolled in the study. The mean age for the entire sample was 6.3 ±â€Š3.0 months, and 17 subjects (52%) were female. The mean ages (months) for each group were: 1.9 ±â€Š0.7 for group 1 (n = 6); 4.4 ±â€Š0.7 for group 2 (n = 8); 7.5 ±â€Š1.1 for group 3 (n = 11); and 9.9 ±â€Š1.0 for group 4 (n = 8). Descriptive statistics are presented for each group. Inter- and intra-rater agreements were acceptable (ICC >0.6) for 21 anthropometric measurements. CONCLUSIONS: This study generated normative craniofacial anthropometric measurements for Caucasian infants. These data can be used in the interpretation of measurements for research studies evaluating craniofacial anomalies in this population.

Is It Cost Effective to Add an Intraoral Scanner to an Oral and Maxillofacial Surgery Practice?

PURPOSE: Intraoral scanners (IOSs), which create digital "impressions" of dental arches, have become popular for prosthetic and orthodontic applications. Adoption in oral and maxillofacial surgery (OMS) practices has been slower, likely because of high implementation costs and low-volume use. The purpose of this study was to evaluate costs for introduction of an IOS into an OMS practice. The authors hypothesized that digital impressions would be more efficient in time and cost compared with conventional impressions and that implementation costs would be offset within 1 year. MATERIALS AND METHODS: This was a prospective study that included patients who had digital impressions during the first year after introduction of an IOS to the practice. Conventional alginate impressions obtained at the same visit were included for comparison. Variables included time for each step in each impression process, IOS experience of the operator obtaining the impression, and associated costs. Per-arch costs for each technique were calculated using time-driven activity-based costing methodology. RESULTS: Sixty-three digital impressions and 31 conventional impressions were included. Mean total times for digital and conventional impressions were 14.1 ± 1.3 and 19.4 ± 4.0 minutes per arch, respectively. On a per-patient basis (2 arches for digital impressions and 4 arches for conventional impressions because of the inability to create duplicate stone models from each alginate impression), total impression times were 24.8 ± 2.7 minutes for digital and 67.2 ± 14.8 minutes for conventional impressions. Total calculated costs for digital and conventional impressions were $21.42 and $29.40 per arch and $37.66 and $102.10 per patient, respectively. In a practice with 2 patients for impressions per working day (500 per year), it would take 1.04 years to offset the purchase of the IOS; with 5 sets of impressions per day (1,250 per year), it would take 5 months. CONCLUSION: Digital impressions are more efficient and cost effective than standard impressions, and implementation costs can be offset within the first year.

Do Oral and Maxillofacial Surgeons Over-Prescribe Opioids After Extraction of Asymptomatic Third Molars?

PURPOSE: Opioid abuse is a public health concern. Oral opioids are prescribed after removal of third molars, but the amount needed for adequate postoperative analgesia is unknown. The purpose of this study was to quantify opioid need after third molar extractions. MATERIALS AND METHODS: This is a prospective cohort study of consecutive patients who had asymptomatic third molars extracted with intravenous sedation at the Boston Children's Hospital (Boston, MA) from June through October 2018 by 3 attending surgeons. To be included, patients had to have had 4 third molars removed. Patients were excluded if they had a concomitant procedure, preoperative infection, postoperative inflammatory complication, chronic pain condition, or did not complete the study. Postoperative prescriptions and instructions included 1) oxycodone 5-mg tablets with 1 tablet taken every 6 hours as needed (dispensed, 6); 2) ibuprofen 600-mg tablets with 1 tablet taken every 6 hours as needed (dispensed, 20); and 3) acetaminophen 325-mg tablets with 2 tablets taken every 6 hours as needed (dispensed, 40). Participants reported medication use by electronic questionnaire each day for 7 postoperative days (PODs). Descriptive statistics were calculated. RESULTS: Eighty-one patients (56% female; mean age, 19.4 ± 7.7 yr) were included. The average number of oxycodone tablets used was 0.04 ± 0.24, and the highest daily use of oxycodone was on POD 2 (1.0 ± 0.0 tablet). Oxycodone was taken by 3 patients (4%) on POD 1, 4 (5%) on POD 2, 2 (3%) on PODs 3 and 4, and 0 on PODs 5 to 7. Seventy-five patients (93%) used no postoperative oxycodone; 466 prescribed oxycodone tablets remained unfilled or unused. Ibuprofen 600 mg was used for 4.6 ± 2.2 PODs and acetaminophen 650 mg was used for 3.4 ± 1.9 PODs. CONCLUSIONS: Oral opioid use after third molar extractions is minimal. Caution is necessary to avoid over-prescribing.

Imaging of the Temporomandibular Joint in Juvenile Idiopathic Arthritis: How Does Quantitative Compare to Semiquantitative MRI Scoring?

PURPOSE: Contrast-enhanced magnetic resonance imaging (MRI) is commonly used to evaluate temporomandibular joint (TMJ) disease in patients with juvenile idiopathic arthritis (JIA). However, MRI assessments have been limited by qualitative reporting, the presence of enhancement in normal joints, and a lack of standardized protocols. The purpose of the present study was to compare a quantitative MRI analysis using an enhancement ratio (ER) to a new semiquantitative system developed by Outcome Measures in Rheumatology and Clinical Trials (OMERACT). MATERIALS AND METHODS: We performed a retrospective cross-sectional study of children with JIA who had undergone TMJ MRI with gadolinium enhancement at Boston Children's Hospital. The predictor variables were demographic data (sex, age at MRI) and disease-related findings (JIA subtype and arthritis medication at MRI). The outcome variables were OMERACT scores and ER (ratio of TMJ synovium and longus capitis muscle pixel intensities). The ER was compared with the OMERACT total, inflammatory, and damage scores using Pearson's correlation analysis and multivariable linear regression. Statistical significance was evaluated using the Student t test and analysis of variance with significance set at P < .05. Inter- and intraexaminer reliability were evaluated using intraclass correlation coefficients (ICCs). RESULTS: The sample included 50 subjects (84% female) with a mean age of 12.7 ± 3.9 years and 124 MRI scans. The mean ER was 2.5 ± 0.9 (normal, <1.55). The mean OMERACT total was 4.9 ± 3.3. Controlling for age, sex, JIA subtype, and medication, for every 1-U increase in ER, the OMERACT increased by 3.11 for the total (P < .001), 1.01 for the inflammatory (P < .001), and 0.86 for the damage (P < .001) scores. The intra- and interrater consistency was better for the ER (ICC, 0.83 and 0.96, respectively) than for OMERACT (ICC, 0.77 and 0.35, respectively). CONCLUSIONS: Quantitative evaluation of TMJ synovial enhancement using the ER is easier to score and has superior intra- and interrater agreement; however, it can only assess inflammation. Semiquantitative assessment of TMJ using the OMERACT adds data about chronic changes. Using both systems might provide the most accurate assessment of JIA status.

Early Management of Infants With Robin Sequence: An International Survey and Algorithm.

PURPOSE: There is no widely accepted protocol for management of infants with Robin sequence (RS) who present with airway obstruction and feeding impairment. The purposes of this study were to evaluate diagnostic and treatment preferences of clinicians from the United States and non-US countries and to use these data to propose an algorithm for early management of infants with RS. MATERIALS AND METHODS: A cross-sectional study was implemented using a survey, which was distributed to craniofacial surgeons and non-surgeon physicians involved in management of infants with RS. Predictor variables were nationality, specialty, and surgical volume. Outcome variables included survey responses regarding preferences for evaluation and early treatment of patients with RS. A diagnostic and early treatment algorithm was proposed based on the most common survey responses. Descriptive and analytic statistics were calculated and a P less than .05 was considered significant. RESULTS: A total of 275 responses were received and 82 were excluded, leaving a final sample of 193 participants: 155 (80%) surgeons and 38 (20%) non-surgeon physicians. Thirty-seven US states were represented by 143 (74%) respondents and 50 (26%) participants were from 19 non-US countries. There were 96 (50%) low-volume and 97 (50%) high-volume respondents. There was general agreement in methods for evaluation and early treatment among respondents; the only significant differences were 1) US surgeons operate earlier to address airway obstruction than those from other countries (P = .001), 2) high-volume providers tend to operate earlier (P = .017) and to choose mandibular distraction more frequently (P = .003) than low-volume respondents, and 3) mandibular distraction is the preferred operation in the United States, but several operations for airway improvement are used equally in non-US countries (P < .001). CONCLUSION: Despite variation in the evaluation and treatment of infants with RS, this international and multispecialty survey showed trends that could represent best practices. An algorithm based on these findings is proposed.

Is Tongue-Lip Adhesion or Mandibular Distraction More Effective in Relieving Obstructive Apnea in Infants With Robin Sequence?

PURPOSE: Tongue-lip adhesion (TLA) and mandibular distraction osteogenesis (MDO) are the most common operations for obstructive apnea (OA) in infants with Robin sequence (RS). The purpose of this study was to compare early outcomes of TLA and MDO on resolution of OA. MATERIALS AND METHODS: This is a retrospective cohort study of infants with RS treated with TLA or MDO from 2005 through 2018. To be included, patients had to have polysomnograms pre- and postoperatively unless intubated or tracheostomy dependent. Patients were excluded if the operation occurred after 1 year of age. The primary predictor variable was type of operation (TLA or MDO). The primary outcome variable was "successful resolution of OA," defined as 1) postoperative apnea-hypopnea index (AHI) score lower than 5, 2) postoperative OA severity score of 0 to 1 (no to mild OA), and 3) no need for additional airway intervention during the first year of life. Descriptive, bivariate, and regression analyses were performed. Statistical significance was set at a P value less than .05. RESULTS: Forty-three patients were included (TLA, n = 19 [44%]; MDO, n = 24 [56%]). Eighteen patients (41%) were syndromic. Patients in the TLA group were significantly younger at operation (28.2 ± 23.1 days) compared with those in the MDO group (87.1 ± 81.7 days; P = .002). The MDO group had more severe preoperative OA (AHI score, 20.5 ± 14.9; OA severity score, 4.7 ± 0.8) than the TLA group (AHI score, 17.6 ± 31.0; OA severity score, 3.6 ± 1.4; P < .041). Postoperative AHI score and OA severity score for the TLA group were 11.7 ± 19.5 (33.5% decrease; P = .496) and 2.3 ± 1.8 (improvement by 1.3 ± 2.4 levels; P = .051), respectively. Postoperative AHI score and OA severity score for the MDO group were 1.1 ± 1.8 (94.6% decrease; P < .001) and 0.2 ± 0.4 (improvement by 4.6 ± 0.8 levels; P < .0001), respectively. Successful resolution of OA occurred in 9 patients (47%) in the TLA group and 22 patients (92%) in the MDO group. Postoperative complications were similar. CONCLUSION: MDO is more effective than TLA in relieving OA in infants with RS.

Does Mandibular Distraction Change the Laryngoscopy Grade in Infants With Robin Sequence?

PURPOSE: To review a series of infants with Robin sequence (RS) who underwent mandibular distraction osteogenesis (MDO) at the authors' institution and document changes in pre- and postoperative laryngoscopy grades. MATERIALS AND METHODS: Consecutive patients with RS who underwent MDO from March 2005 to June 2017 were identified. Patients were included if they had a preoperative polysomnogram confirming obstructive sleep apnea, had failed nonoperative airway management, had undergone MDO by the senior author (C.M.R.), and had complete documentation. Patients were excluded if they did not have a laryngoscopy performed (ie, tracheostomy dependent) or if there was insufficient information in their medical record. Variables included demographic data, operative information, and laryngoscopic details at 3 time points: before MDO (T0), at device removal (T1), and at latest follow-up laryngoscopy (T2). Descriptive statistics were computed. Significance was set at a P value less than .05. RESULTS: Twenty-two infants with RS underwent MDO during the study period and 13 of these met criteria for inclusion. Mean age at MDO (T0) was 19.6 ± 38.8 weeks. Mean ages at device removal (T1) and latest follow-up laryngoscopy (T2) were 28.2 ± 41.5 and 62.7 ± 49.6 weeks, respectively. Three patients had Stickler syndrome; the others had nonsyndromic RS. Mean apnea-hypopnea index was 23.6 ± 16.6 preoperatively and 0.3 ± 0.7 after MDO (P = .011). At T0, a grade IIb laryngoscopic view was most common (46.1%) and only 3 patients (23.1%) had a grade I view. At T1, most patients (69.2%) had a grade I view and only 2 (14.4%) had a grade of IIb or worse (P = .011). At T2, all but 1 patient had a grade I view. CONCLUSION: In addition to resolving obstructive apnea, MDO is associated with an improvement in the grade of laryngoscopic view. This could increase airway safety at home and improve ease of intubation for future operations.

Is Amniotic Fluid Level a Predictor for Syndromic Diagnosis in Robin Sequence?

OBJECTIVE: The purpose of this study was to determine whether gestational amniotic fluid level abnormalities were associated with postnatal syndromic status in a series of patients with Robin sequence (RS). DESIGN: Retrospective study of participants with RS at Boston Children's Hospital from 1967 to 2017. Participants were divided into syndromic and nonsyndromic groups. The primary predictor variable was postnatal syndromic diagnosis (yes/no). Additional predictor variables included gestational age at birth, birthweight, sex, presence of cleft palate, and other congenital anomalies. The primary outcome variable was amniotic fluid level (normal, oligohydramnios, or polyhydramnios). Descriptive statistics were computed and logistic regression was used to analyze amniotic fluid level as a predictor for syndromic diagnosis. Statistical significance was set at P < .05. RESULTS: Sixty-five (54%) syndromic and 56 (46%) nonsyndromic RS participants were included. An abnormal amniotic fluid level was seen significantly more frequently in the syndromic group (49.2% vs 25.0%; P = .001). Abnormal amniotic fluid level was associated with a 2.9-fold increased likelihood of a syndromic diagnosis (P = .007). Polyhydramnios, which was seen more frequently than oligohydramnios, predicted a 4.18 times increased likelihood of a syndromic diagnosis (P = .003). CONCLUSIONS: Abnormal amniotic fluid level, particularly polyhydramnios, is associated with an increased likelihood of a syndromic diagnosis in patients with RS.

Temporomandibular Joint Ankylosis After Ramus Construction With Free Fibula Flaps in Children With Hemifacial Microsomia.

PURPOSE: Patients with hemifacial microsomia (HFM) and Kaban-Pruzansky type III mandibular deformities require ramus construction with autologous tissue. The free fibula flap, an alternative to the costochondral graft, has favorable characteristics for this construction but may be associated with temporomandibular joint ankylosis. The purposes of this study were to present a series of patients with HFM who underwent free fibula flap ramus construction, to determine the incidence of ankylosis, and to identify perioperative factors associated with ankylosis. MATERIALS AND METHODS: We performed a retrospective cohort study of patients with HFM who underwent ramus construction with a free fibula flap at Boston Children's Hospital from 2003 to 2015. Patients who had at least 1 year of follow-up and complete medical records were included. The predictor variables included demographic information, HFM severity, surgical history, and operative details. The primary outcome variable was the occurrence of ankylosis. Descriptive statistics were calculated, and significance was set at P < .05. RESULTS: We included 8 patients (75% of whom were female patients) in the study sample. Patients underwent construction at a mean age of 11.4 ± 5.9 years (range, 5 to 21 years). In 5 patients (63%), ankylosis developed during the follow-up period of 7.3 ± 4.8 years. The average time from construction to ankylosis was 4.2 ± 3.7 years. The only predictor variable statistically significantly associated with ankylosis was the use of a contralateral releasing osteotomy, which reduced the rate of ankylosis (P = .035). There was a trend toward a younger age in patients in whom ankylosis developed (8.8 ± 2.6 years) compared with those without ankylosis (15.5 ± 8.1 years, P = .392). CONCLUSIONS: The free fibula flap can be associated with a high rate of ankylosis when used for ramus construction in patients with HFM. Passive flap insertion and/or use of a contralateral releasing osteotomy may reduce this risk.

Evaluation of Andrews' Analysis as a Predictor of Ideal Sagittal Maxillary Positioning in Orthognathic Surgery.

PURPOSE: There is no universally accepted method for determining the ideal sagittal position of the maxilla in orthognathic surgery. In "Element II" of "The Six Elements of Orofacial Harmony," Andrews used the forehead to define the goal maxillary position. The purpose of this study was to compare how well this analysis correlated with postoperative findings in patients who underwent bimaxillary orthognathic surgery planned using other guidelines. The authors hypothesized that the Andrews analysis would more consistently reflect clinical outcomes than standard angular and linear measurements. MATERIALS AND METHODS: This is a retrospective cohort study of patients who had bimaxillary orthognathic surgery and achieved an acceptable esthetic outcome. Patients with no maxillary sagittal movement, obstructive sleep apnea, cleft or craniofacial diagnoses, or who were non-Caucasian were excluded. Treatment plans were developed using photographs, radiographs, and standard cephalometric measurements. The Andrews analysis, measuring the distance from the maxillary incisor to the goal anterior limit line, and standard measurements were applied to end-treatment records. The Andrews analysis was statistically compared with standard methods. RESULTS: There were 493 patients who had orthognathic surgery from 2007 through 2014, and 60 (62% women; mean age, 22.1 ± 6.8 yr) met the criteria for inclusion in this study. The mean Andrews distances were -4.8 ± 2.9 mm for women and -8.6 ± 4.6 mm for men preoperatively and -0.6 ± 2.1 mm for women and -1.9 ± 3.4 mm for men postoperatively. For women, the Andrews analysis was closer to the goal value (0 mm) postoperatively than any standard measurement (P < .001). For men, the linear distance from the A point to a vertical line tangent to the nasion from the McNamara analysis performed best (P < .001), followed by the Andrews analysis. CONCLUSION: The Andrews analysis correlated well with the final esthetic sagittal maxillary position in the present sample, particularly for women, and could be a useful tool for orthognathic surgical planning.

In Utero Glossoptosis in Fetuses With Robin Sequence: Measurements From Prenatal MRI.

OBJECTIVE: Glossoptosis causes airway obstruction in patients with Robin sequence (RS), but little is known about the in-utero tongue. The purpose of this study was to assess shape and position of the fetal tongue on prenatal magnetic resonance imaging (MRI) to determine if this is predictive of postnatal RS. DESIGN: Retrospective case-control study including fetuses with prenatal MRIs performed from 2002 to 2017. Inclusion criteria were (1) prenatal MRI of adequate quality, (2) live born and evaluated postnatally for craniofacial findings. Subjects were divided into groups based on postnatal findings: (1) RS, (2) micrognathia without RS, and (3) a gestational-age-matched control group with normal craniofacial morphology. Outcome variables were based on the prenatal MRI and included fetal tongue height, length, and width, tongue shape index (TSI, ratio of height to length), and observation of tongue touching the posterior pharyngeal wall. RESULTS: A total of 116 subjects with mean gestational age at MRI of 25.6 ± 5.1 weeks were included: RS, n = 27 (23%); micrognathia, n = 35 (30%); control, n = 54 (47%). Tongue length was significantly shorter ( P = .009) and TSI was significantly larger in the RS group ( P < .0001). The tongue touched the posterior pharyngeal wall in 5 (19%) of the RS group and in no subjects in the other groups ( P < .0001). CONCLUSION: In utero tongue shape and position were significantly different in fetuses with postnatal RS compared to those with isolated micrognathia and controls. Prenatal MRI tongue characteristics may be predictors for postnatal RS.

An algorithm for predicting Robin sequence from fetal MRI.

BACKGROUND: Infants with Robin sequence (RS) may present with airway compromise at delivery. Prenatal diagnosis would improve preparation and postnatal care. The purpose of this study was to devise a predictive algorithm for RS based on fetal magnetic resonance imaging (MRI). METHODS: Retrospective case-control study including fetal MRIs from 2002 to 2017. Inclusion criteria were (1) MRI of adequate quality, (2) live-born infant, and (3) postnatal evaluation. Subjects were grouped on the basis of postnatal diagnosis: (1) RS (micrognathia, glossoptosis, airway obstruction), (2) micrognathia without airway obstruction ("micrognathia"), (3) cleft lip and palate ("CLP"), and (4) gestational age-matched controls. A series of possible predictive variables were assessed on MRI. Receiver operator curves were applied to identify cut-off values, and a multivariable algorithm was developed. RESULTS: A total of 162 subjects with mean gestational age at MRI of 25.6 ± 4.9 weeks were included: RS, n = 27 (17%); micrognathia, n = 35 (22%); CLP, n = 46 (28%); control, n = 54 (33%). Three variables were independent predictors of RS: (1) Veau I/II cleft palate (OR = 38.8), (2) tongue shape index (>80%; OR = 8.7), and (3) inferior facial angle (<48°; OR = 14.5). CONCLUSION: MRI findings of cleft palate, TSI >80% and IFA <48° indicate a 98% probability of RS, whereas a lack of all 3 features denotes a likelihood of 1%.

Maxillary Sagittal Position in Relation to the Forehead: A Target for Orthognathic Surgery.

The relationship of the maxillary incisor to forehead position and angulation may be a predictor for the esthetic antero-posterior maxillary position in orthognathic surgery, but this has not been fully explored in the existing literature. Additionally, this analysis requires a lateral smiling photograph, which is not typically included in orthodontic/orthognathic records. This is a bidirectional study of Caucasian orthodontic patients with malocclusions correctable with nonsurgical and nonextraction treatment. Subjects for Group 1 were enrolled prospectively and records included a lateral smiling photograph. Group 2 patients were collected retrospectively with standard records. The analysis described in Andrews' Element II of The Six Elements of Orofacial Harmony was applied to the smiling photographs for Group 1, resulting in "Incisor- goal anterior limit line (GALL)" measurements. A modified analysis utilizing cephalometric tracings was applied for both the groups, and the results were compared. Forty-nine subjects with mean age 11.8 ±â€Š1.2 years were included (n = 14 for Group 1, n = 35 for Group 2). Mean Incisor-GALL distances were 3.71 ±â€Š5.39 mm for females and 4.47 ±â€Š5.06 mm for males. Correlation between analyses was excellent (intraclass correlation coefficient = 0.872). In conclusion, a modified approach to measuring the Incisor-GALL distance using standard orthodontic records correlated closely with the analysis described by Andrews. Normal values for this measurement are presented for Caucasian patients.