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Background: Cherubism is most commonly caused by rare heterozygous gain-of-function (GOF) missense variants in SH3BP2, which appear to signal through phospholipase C gamma 2 (PLCG2) to cause excessive osteoclast activity leading to expansile lesions in facial bones in childhood. GOF variants in PLCG2 lead to autoinflammatory PLCG2-associated antibody deficiency and immune dysregulation (autoinflammatory PLAID, or PLAID-GOF), characterized by variably penetrant autoinflammatory, autoimmune, infectious, and atopic manifestations. Cherubism has not been reported in PLAID to date., Objective: We determined whether GOF PLCG2 variants may be associated with cherubism., Methods: Clinical, laboratory, and genomic data from 2 patients with cherubism and other clinical symptoms observed in patients with PLCG2 variants were reviewed. Primary B-cell receptor-induced calcium flux was assessed by flow cytometry., Results: Two patients with lesions consistent with cherubism but no SH3BP2 variants were found to have rare PLCG2 variants previously shown to be GOF in vitro, leading to increased primary B-cell receptor-induced calcium flux in one patient's B cells. Variable humoral defects, autoinflammatory rash, and other clinical and laboratory findings consistent with PLAID were observed as well., Conclusion: GOF PLCG2 variants likely represent a novel genetic driver of cherubism and should be assessed in SH3BP2-negative cases. Expansile bony lesions expand the phenotypic landscape of autoinflammatory PLAID, and bone imaging should be considered in PLAID patients., Competing Interests: Disclosure statement Supported by the National Institutes of Health, National Institute of Allergy and Infectious Diseases (R24 AI 17745 to J.D.M.), and the Intramural Research Program of the National Institute of Arthritis and Musculoskeletal and Skin Diseases (Z01-AR041198 to M.J.O.). Disclosure of potential conflict of interest: J. D. Milner is on the scientific advisory board for Blueprint Medicine and receives grant funding from Pharming. W. K. Chung is the on the board of directors of Rallybio and Prime Medicine. S. B. Eisig is cofounder of Epibone. The rest of the authors declare that they have no relevant conflicts of interest., (Copyright © 2024 American Academy of Allergy, Asthma & Immunology. All rights reserved.)

Competing Interests: Disclosures None of the authors reported any disclosures.

Objective: The purpose of this study was to identify factors associated with the management of the cleft dental gap after alveolar bone grafting., Methods: This was a retrospective cohort study of patients with unilateral cleft lip and alveolus or palate who had successful alveolar bone grafting. Our primary study outcome was alveolar cleft management (orthodontic closure or space maintenance). Our secondary study outcome was the rate of fixed dental rehabilitation. Univariate comparisons were made with SAS 9.4., Results: The final study sample consisted of 54 patients. Most patients were treated with orthodontic closure (55.6%). Patients missing multiple teeth ( P < 0.01) were less likely to receive orthodontic closure. Orthodontic closure was not associated with differences in intermaxillary midline coincidence ( P = 0.22) or the need for LeFort advancement ( P = 0.15). Only 41.7% of patients who were managed with space maintenance obtained a fixed prosthesis. Hispanic ethnicity ( P < 0.01) and Medicaid insurance ( P < 0.01) were associated with lower rates of fixed dental rehabilitation., Conclusions: Orthodontic closure was the most common approach, and it did not result in significant maxillary midline distortion. Less than half of patients treated with space maintenance obtained fixed restorations. Socioeconomic barriers are likely preventing access to definitive dental rehabilitation in patients with unilateral cleft lip and alveolus or palate., Competing Interests: The authors report no conflicts of interest., (Copyright © 2023 by Mutaz B. Habal, MD.)

Objective: The pandemic caused by SARS-CoV-2 virus continues to have a profound effect worldwide. However, COVID-19 induced oral facial manifestations have not been fully described. We conducted a prospective study to demonstrate feasibility of anti-SARS-CoV-2 IgG and inflammatory cytokine detection in saliva. Our primary objective was to determine whether COVID-19 PCR positive patients with xerostomia or loss of taste had altered serum or saliva cytokine levels compared to COVID-19 PCR positive patients without those oral symptoms. Our secondary objective was to determine the correlation between serum and saliva COVID-19 antibody levels., Materials and Methods: For cytokine analysis, saliva and serum were obtained from 17 participants with PCR-confirmed COVID-19 infection at three sequential time points, yielding 48 saliva samples and 19 paired saliva-serum samples from 14 of the 17 patients. For COVID-19 antibody analyses, an additional 27 paired saliva-serum samples from 22 patients were purchased., Results: The saliva antibody assay had 88.64% sensitivity [95% Confidence Interval (CI) 75.44%, 96.21%] to detect SARS-CoV-2 IgG antibodies compared to serum antibody. Among the inflammatory cytokines assessed - IL-6, TNF-α, IFN-γ, IL-10, IL-12p70, IL-1β, IL-8, IL-13, IL-2, IL-5, IL-7 and IL-17A, xerostomia correlated with lower levels of saliva IL-2 and TNF-α, and elevated levels of serum IL-12p70 and IL-10 (p < 0.05). Loss of taste was observed in patients with elevated serum IL-8 (p < 0.05)., Conclusions: Further studies are needed to construct a robust saliva-based COVID-19 assay to assess antibody and inflammatory cytokine response, which has potential utility as a non-invasive monitoring modality during COVID-19 convalescence., (© 2023 The Authors.)

Purpose: Underlying psychiatric conditions are thought to influence the presentation, management, and outcomes of facially injured patients. Our study sought to determine if psychiatric diagnoses were associated with the decision to repair facial fractures during the index hospitalization., Methods: This was a cross-sectional review of the 2014 Nationwide Emergency Department Sample. All patients with the primary diagnosis of a facial fracture were included in the study. The primary study predictor was the presence of a documented psychiatric illness. Covariates included patient age, insurance, injury mechanism, primary fracture location, other concomitant injuries, and Injury Severity Score. The study outcome was facial fracture treatment status (reduction or no reduction). A multiple logistic regression model was created to identify and measure independent factor associations for fracture treatment., Results: The final sample included 59,378 patients of whom 10,485 (17.7%) had a documented psychiatric illness. Most of these diagnoses involved substance use (62.5%). Patients with psychiatric illness had significantly higher rates of extra-nasal primary fracture location (56.2 vs 47.1%, P < .01) and a greater mean Injury Severity Score (5.0 vs 3.8, P < .01). In the unadjusted analysis, patients with psychiatric illness had higher rates of fracture repair during their index hospitalization (RR = 2.42, P < .01). After adjusting for covariates in the multiple logistic regression model, psychiatric illness became negatively associated with fracture repair (OR = 0.82, P < .01)., Conclusions: Patients with psychiatric illness experienced higher rates of hospitalization and suffered more severe patterns of injury but had lower odds of fracture repair during their index hospitalization after controlling for injury characteristics., (Copyright © 2021 The American Association of Oral and Maxillofacial Surgeons. Published by Elsevier Inc. All rights reserved.)

Purpose: This study sought to compare radiographic outcomes and resource utilization between recombinant human bone morphogenetic protein-2 (rhBMP-2) and anterior iliac crest bone graft (AICBG) when used for secondary alveolar grafting., Materials and Methods: This is a 14-year retrospective study of patients with alveolar clefts treated at the Morgan Stanley Children's Hospital of New York-Presbyterian/Columbia University Irving Medical Center between January 2006 and January 2020. Patients who had alveolar grafting with either rhBMP-2 or AICBG were included in this study. The primary study predictor was the graft material. The study outcomes were bone height, operating room time, and the number of scrubbed personnel (surgeon and assistants). Graft survival was measured at a minimum of 6 months postoperatively. Bone height was scored according to the Bergland scale, and radiographic success was defined as Bergland types 1 or 2., Results: The study sample included a total of 115 patients with 130 alveolar clefts. Overall, 13.0% of patients had bilateral repairs, and 17.4% were retreatments. The cumulative success rate was 89.5%. There were no differences in success between materials (rhBMP: 90.3%; AICBG: 89.1%; P = .85). Patients presenting for retreatment were more likely to receive rhBMP-2 than AICBG (48.6 vs 3.8%, P < .01). After controlling for other significant confounders, the rhBMP-2 group required less personnel (P < .01) and operating room time (P < .01). Only 1 patient in the rhBMP-2 group was admitted, whereas all AICBG patients were admitted a minimum of 1 night., Conclusions: Compared with AICBG, rhBMP-2 produced a similar height of bone but required less hospital resources. The decision to use harvested ilium or rhBMP-2 is not limited by outcome data at this time. More studies will need to be performed to identify the particular advantages of each graft material. The choice of material is currently both surgeon specific and patient specific and requires thorough informed consent., (Copyright © 2020 American Association of Oral and Maxillofacial Surgeons. Published by Elsevier Inc. All rights reserved.)

Joint disorders can be detrimental to quality of life. There is an unmet need for precise functional reconstruction of native-like cartilage and bone tissues in the craniofacial space and particularly for the temporomandibular joint (TMJ). Current surgical methods suffer from lack of precision and comorbidities and frequently involve multiple operations. Studies have sought to improve craniofacial bone grafts without addressing the cartilage, which is essential to TMJ function. For the human-sized TMJ in the Yucatan minipig model, we engineered autologous, biologically, and anatomically matched cartilage-bone grafts for repairing the ramus-condyle unit (RCU), a geometrically intricate structure subjected to complex loading forces. Using image-guided micromilling, anatomically precise scaffolds were created from decellularized bone matrix and infused with autologous adipose-derived chondrogenic and osteogenic progenitor cells. The resulting constructs were cultured in a dual perfusion bioreactor for 5 weeks before implantation. Six months after implantation, the bioengineered RCUs maintained their predefined anatomical structure and regenerated full-thickness, stratified, and mechanically robust cartilage over the underlying bone, to a greater extent than either autologous bone-only engineered grafts or acellular scaffolds. Tracking of implanted cells and parallel bioreactor studies enabled additional insights into the progression of cartilage and bone regeneration. This study demonstrates the feasibility of TMJ regeneration using anatomically precise, autologous, living cartilage-bone grafts for functional, personalized total joint replacement. Inclusion of the adjacent tissues such as soft connective tissues and the TMJ disc could further extend the functional integration of engineered RCUs with the host., (Copyright © 2020 The Authors, some rights reserved; exclusive licensee American Association for the Advancement of Science. No claim to original U.S. Government Works.)

Purpose: The purpose of the present study was to compare the severity of craniomaxillofacial injuries between accidents involving motorized and nonmotorized standup scooters., Materials and Methods: This is a 20-year cross-sectional study of the National Electronic Injury Surveillance System. Injuries from powered and unpowered standup scooters were included in this study if they involved the head, face, eyeball, mouth, or ear. Study predictors were obtained from both patient and injury characteristics. The study outcome was the probability of hospital admission from the emergency department. A multiple logistic regression model was created to model the probability of admission using all significant univariate predictors., Results: A total of 11,916 records were included in the present study, of which 9.5% involved motorized scooters. The proportion of motorized injuries more than tripled from 2014 (5.8%) to 2018 (22.1%). Motorized injuries occurred more often in older individuals (24.0 vs 8.5 years; P < .01). A greater proportion of motorized injuries involved the head (55.0 vs 36.9%; P < .01) and resulted in concussion (11.5 vs 5.6%; P < .01), fractures (6.7 vs 2.0%; P < .01), and other nonspecified internal organ injuries (31.1 vs 19.6%; P < .01). Motorized scooter injuries had more than triple the admission rate compared to nonmotorized injuries (13.9 vs 3.7%; P < .01). After controlling for potential confounders, injuries from motorized scooters still had double the odds of hospital admission (odds ratio, 2.03; P < .01)., Conclusions: Motorized standup scooters appear to cause more severe injuries than conventional nonmotorized scooters. The recent growth of rentable electric scooters may pose a future public health concern. Ride-sharing companies should ensure that customers are capable of safely and responsibly operating these vehicles., (Copyright © 2020 American Association of Oral and Maxillofacial Surgeons. Published by Elsevier Inc. All rights reserved.)

Objective: The purpose of this study was to determine whether performing mandibular distraction osteogenesis (MDO) during the neonatal period increased inpatient complications as measured through health-care burden., Materials and Methods: This was a retrospective cohort study of the Kids' Inpatient Database from 2000 to 2011. Infants receiving MDO prior to 12 months of age were included. The primary study predictor was distraction age, classified as either neonatal or non-neonatal. Secondary predictors were patient demographics, hospitalization characteristics, diagnoses, and procedures. The outcomes were the number of procedures performed, postoperative length of stay (pLOS), hospital charges, and the discharge transfer rate. Outcomes were compared between the primary predictors using χ 2 and independent 2-sample t tests. Multiple linear and logistic regression models were created using clinically relevant predictors to assess the independent effect of neonatal age on each outcome., Results: The study sample contained 102 patients, of who 50 (49.0%) were distracted in the neonatal period. Neonatal MDO patients were more likely to have a cleft palate (86.0% vs 55.8%; P < .001) and present with feeding difficulties (38.0% vs 19.2%; P = .036) that were treated through total parenteral nutrition (26.0% vs 9.6%; P = .030) but otherwise did not have significantly different characteristics compared to non-neonatal patients. The multiple regression models confirmed that neonatal age did not influence any of the study outcomes, although other secondary predictors were found to influence the pLOS, hospital charges, and number of inpatient procedures., Conclusions: Neonatal MDO was not associated with increased complications. At experienced centers, neonatal status should not be considered a contraindication to treatment.

Purpose: Because Robin sequence (RS) is clinically heterogeneous, there is uncertainty as to whether different presentations yield different perioperative outcomes. The purpose of this study was to evaluate factors associated with postoperative length of stay and readmission rate after mandibular distraction osteogenesis (MDO) for RS., Materials and Methods: This was a 10-year retrospective cohort of patients with RS who had MDO performed from 2007 through 2017 at the Morgan Stanley Children's Hospital of the NewYork-Presbyterian/Columbia University Irving Medical Center (New York, NY). Predictor variables were the presence of multiple anomalies, airway intervention used before MDO, and feeding method used before MDO. Outcome variables were postoperative day (POD) of discharge and number of readmissions from the period of discharge to hardware removal. Patient characteristics and outcomes were compared within predictors using Fisher's exact and 2-tailed Student's t tests. Multiple regression models were calculated for each outcome variable using univariate predictors with P values less than or equal to .25., Results: Twenty-four patients who had MDO performed at a mean age of 18.1 weeks were included in this study. On average, patients were extubated on POD 6.6 and distracted to 13.9 mm. Seven patients (29.2%) were readmitted from the period of discharge to hardware removal. Patients receiving an invasive airway intervention before MDO were significantly older at the time of the operation (55.0 vs 8.4 weeks; P = .01). In addition, these patients had a significantly higher readmission rate (80.0 vs 15.8%; P = .01) with a trend toward longer postoperative stays (45.0 vs 21.6 days; P = .06). Feeding status and presence of multiple anomalies did not appear to influence these outcomes., Conclusions: Baseline airway intervention may be useful for predicting length of stay and readmission rate after MDO., (Copyright © 2019 American Association of Oral and Maxillofacial Surgeons. Published by Elsevier Inc. All rights reserved.)

Purpose: The aims were to report the characteristics of Le Fort fractures and to quantify the associated hospital costs., Materials and Methods: From October 2015 to December 2016, the National Inpatient Sample was searched for patients admitted with a primary diagnosis of a Le Fort fracture. Predictor variables were drawn from demographic, admission, and injury characteristics. The outcome variable was hospital cost. Summary statistics were calculated and compared among Le Fort patterns. Univariate comparisons and multivariate regression analyses were conducted to determine predictors associated with cost., Results: A total of 519 patients were identified in this cohort. Associated injuries included skull fractures (28%), intracranial hemorrhage (13%), cervical spine injury (9.8%), and concussion (9.1%). Seventy-three percent of patients received open reduction and internal fixation (ORIF) for their facial fractures during their admission, 13% received a tracheostomy, and 10% were mechanically ventilated for at least 1 day. The ventilation (P < .01) and tracheostomy (P < .01) rates increased with Le Fort complexity, as did length of stay (LOS; P < .01), costs (P < .01), and charges (P < .01). The mean costs of treating Le Fort I, II, and III fractures were $25,836, $28,415, and $47,333, respectively. Increased cost was independently associated with younger age, male gender, African-American ethnicity, Le Fort II and III patterns, motor vehicle accident etiology, mechanical ventilation requirement, tracheostomy, ORIF, transfer to an outside facility, and increased LOS., Conclusions: The prevalence of head injuries and the need for respiratory support substantially increased with Le Fort complexity. Hospital costs were not markedly influenced by the diagnosis and management of associated injuries. Instead, costs were predominantly driven by fracture complexity and the need for necessary procedures, such as ORIF, tracheostomy, and mechanical ventilation., (Copyright © 2019 American Association of Oral and Maxillofacial Surgeons. Published by Elsevier Inc. All rights reserved.)

Purpose: The purpose of this study was to characterize the sources, amounts, and nature of general industry payments to oral and maxillofacial surgeons (OMSs)., Materials and Methods: The Open Payments database was searched from August 2013 to December 2017 for all general payments made to OMSs. For each payment, data were recorded regarding the date, value, paying organization, and nature of the payment. Analyses of variance were performed to compare payment amount per surgeon, number of payments per surgeon, and amount per payment across payment years., Results: Overall, there were 112,448 payments totaling $28,644,063.20. Although, on average each year, 69% of OMSs received at least 1 payment, the top 10% of earners accounted for 89% ($25,607,781) of all payments. Each OMS received a median total of $93.75 (interquartile range, $33 to $263) and an average of $1,109.68 (SD, $14,297; range, $2-$2,250,345) per year. The mean payment value was $254.73 (SD, $2,727; range, $0.01-$548,126) compared with a median of $28.90 (interquartile range, $15-$82). Most payments were in the form of food and beverage (72%); however, the most dollars were spent on consulting fees ($5,594,087; 20%). From 2014 to 2016, significant decreases occurred in the payment amounts (P < .01) and number of payments (P < .01) per surgeon, as well as the value of each payment (P < .01)., Conclusions: Although OMSs receive a substantial amount of industry payments, the overall figure is driven by a small percentage of top earners. Most individual payments were of nominal value, and their effect on clinical practice remains to be seen., (Copyright © 2018 American Association of Oral and Maxillofacial Surgeons. Published by Elsevier Inc. All rights reserved.)

Purpose: The purpose of this study was to evaluate the readability and quality of online patient educational materials (PEMs) for orthognathic surgery., Materials and Methods: Two internet searches were performed using the search terms orthognathic surgery and jaw surgery. The presence of content related to the risks, benefits, procedure, and postoperative care was recorded. Readability was measured using 4 validated scales: Flesch-Kincaid grade level, Gunning Fog index, Coleman-Liau index, and Simple Measure of Gobbledygook index. Materials were readable if they were written at or below an eighth-grade reading level as recommended by the American Medical Association (AMA) and the National Institutes of Health (NIH). Quality was assessed using 2 metrics: the DISCERN instrument and the Journal of the American Medical Association benchmark criteria. A DISCERN score of 50 was set as the lower limit of acceptable quality. Mann-Whitney U and Fisher exact tests were used to compare the readability, quality, and presence of content between private practice and non-private practice PEMs., Results: Fifty websites were included in the study after removing duplicates and applying exclusion criteria. On average, PEMs were written at a 13.4-grade level (range, 7.8 to 17.3). Nearly every website (n = 49; 98%) mentioned the benefits of surgery; however, very few websites discussed the surgical procedure (n = 12; 24%), postoperative care (n = 10; 20%), and risks or complications (n = 6; 12%). The mean DISCERN score was 25.5 of 80 (range, 18 to 63), and only 2 websites achieved DISCERN scores of acceptable quality. Private practice websites reported less content related to the surgical procedure (P = .03) and had lower DISCERN scores (P = .02)., Conclusions: As a whole, online PEMs for orthognathic surgery failed to meet AMA and NIH readability recommendations and yielded poor quality scores. Increasing the presence of content related to treatment risks and postoperative care will help improve the quality of PEMs., (Copyright © 2018 American Association of Oral and Maxillofacial Surgeons. Published by Elsevier Inc. All rights reserved.)

Purpose: Prospective applicants use residency program websites (PWs) to evaluate programs when deciding where to apply, interview, and rank. The purpose of this study was to evaluate the content of oral and maxillofacial surgery (OMS) PWs., Materials and Methods: This is a cross-sectional study of all OMS programs accredited by the Commission on Dental Accreditation. PWs were identified through the American Association of Oral and Maxillofacial Surgeons Program Directory (AAOMS-PD) and a secondary internet search. The predictor variables were program characteristics (federal service, medical school curriculum, program size, and viability of AAOMS-PD link). The outcome variables were 22 content items related to resident recruitment and resident education and 4 content items related to medical school commitment. Content was recorded as present or absent without judgment on the quality or accuracy. Descriptive statistics were calculated for all outcome variables, and Student t tests were used to compare program characteristics with the amount of content reported., Results: Ninety-six of 101 programs had dedicated PWs. On average, programs included 39% (range, 0 to 77%) of the recruitment and education items assessed. Most programs did not address crucial aspects of training, such as faculty research interests (22%), resident selection criteria (22%), alumni rosters (19%), surgical case load (13%), and dedicated scholarly time (8.8%). Less than half the 6-year programs discussed a tuition responsibility (44%) or tuition assistance (21%). Four-year programs provided significantly less recruitment content (P < .01) and 4-year (P < .01) and federal (P < .01) programs reported significantly less resident education content., Conclusions: As a whole, OMS PWs are under-reporting content related to resident recruitment and resident education. Program directors should update their PWs to provide adequate information for prospective applicants., (Copyright © 2018 American Association of Oral and Maxillofacial Surgeons. Published by Elsevier Inc. All rights reserved.)

After discectomy, interpositional implants (IPIs) are believed to provide temporomandibular joint stability and protect against degenerative joint space remodeling. Alloplastic IPIs gained popularity in the late 1970s because the practice showed early success without donor site morbidity. Unfortunately, these implants were subject to substantial fragmentation resulting in an exuberant foreign body giant cell response that progressively eroded adjacent structures. Most of these alloplastic implants were removed in the years following their recall by the US Food and Drug Administration in 1991, however some remained in circulation. This report describes a case of a failed Proplast/Teflon IPI 27 years after its placement. This case highlights the considerations for managing a patient with a Proplast/Teflon IPI and serves to remind providers that, although increasingly rare, these implants are still present and subject to delayed failure., (Copyright © 2018 American Association of Oral and Maxillofacial Surgeons. Published by Elsevier Inc. All rights reserved.)

The Comprehensive Basic Science Examination (CBSE) is the entrance examination for oral and maxillofacial surgery, but its implementation among dental students is a relatively recent and unintended use. The aim of this study was to examine the relationship between pre-admission data and performance on the CBSE for dental students at the Columbia University College of Dental Medicine (CDM). This study followed a retrospective cohort, examining data for the CDM Classes of 2014-19. Data collected were Dental Admission Test (DAT) and CBSE scores and undergraduate GPAs for 49 CDM students who took the CBSE from September 2013 to July 2016. The results showed that the full regression model did not demonstrate significant predictive capability (F[8,40]=1.70, p=0.13). Following stepwise regression, only the DAT Perceptual Ability score remained in the final model (F[1,47]=7.97, p<0.01). Variations in DAT Perceptual Ability scores explained 15% of the variability in CBSE scores (R 2 =0.15). This study found that, among these students, pre-admission data were poor predictors of CBSE performance.

Objectives/hypothesis: A candidate variant (p.Val496Ala) of the ACSS2 gene (T > C missense, rs59088485 variant at chr20: bp37 33509608) was previously found to consistently segregate with nonsyndromic cleft lip and/or palate (NSCLP) in three Honduran families. Objectives of this study were 1) to investigate the frequency of this ACSS2 variant in Honduran unrelated NSCLP patients and unrelated unaffected controls and 2) to investigate the frequency of this variant in Colombian unrelated affected NSCLP patients and unrelated unaffected controls., Study Design: Case-control studies., Methods: Sanger sequencing of 99 unrelated Honduran NSCLP patients and 215 unrelated unaffected controls for the p.Val496Ala ACSS2 variant was used to determine the carrier frequency in NSCLP patients and controls. Sanger sequencing of 230 unrelated Colombian NSCLP patients and 146 unrelated unaffected controls for the p.Val496Ala ACSS2 variant was used to determine the carrier frequency in NSCLP patients and controls., Results: In the Honduran population, the odds ratio of having NSCLP among carriers of the p.Val496Ala ACSS2 variant was 4.0 (P = .03), with a carrier frequency of seven of 99 (7.1%) in unrelated affected and four of 215 (1.9%) in unrelated unaffected individuals. In the Colombian population, the odds ratio of having NSCLP among carriers of the p.Val496Ala ACSS2 variant was 2.6 (P = .04), with a carrier frequency of 23 of 230 (10.0%) in unrelated affected and six of 146 (4.1%) in unrelated unaffected individuals., Conclusions: These findings support the role of ACSS2 in NSCLP in two independent Hispanic populations from Honduras and Colombia., Level of Evidence: NA Laryngoscope, 127:E336-E339, 2017., (© 2017 The American Laryngological, Rhinological and Otological Society, Inc.)

Facial deformities require precise reconstruction of the appearance and function of the original tissue. The current standard of care-the use of bone harvested from another region in the body-has major limitations, including pain and comorbidities associated with surgery. We have engineered one of the most geometrically complex facial bones by using autologous stromal/stem cells, native bovine bone matrix, and a perfusion bioreactor for the growth and transport of living grafts, without bone morphogenetic proteins. The ramus-condyle unit, the most eminent load-bearing bone in the skull, was reconstructed using an image-guided personalized approach in skeletally mature Yucatán minipigs (human-scale preclinical model). We used clinically approved decellularized bovine trabecular bone as a scaffolding material and crafted it into an anatomically correct shape using image-guided micromilling to fit the defect. Autologous adipose-derived stromal/stem cells were seeded into the scaffold and cultured in perfusion for 3 weeks in a specialized bioreactor to form immature bone tissue. Six months after implantation, the engineered grafts maintained their anatomical structure, integrated with native tissues, and generated greater volume of new bone and greater vascular infiltration than either nonseeded anatomical scaffolds or untreated defects. This translational study demonstrates feasibility of facial bone reconstruction using autologous, anatomically shaped, living grafts formed in vitro, and presents a platform for personalized bone tissue engineering., (Copyright © 2016, American Association for the Advancement of Science.)

Objective: There are limited clinical treatments for temporomandibular joint (TMJ) pathologies, including degenerative disease, disc perforation and heterotopic ossification (HO). One barrier hindering the development of new therapies is that animal models recapitulating TMJ diseases are poorly established. The objective of this study was to develop an animal model for TMJ cartilage degeneration and disc pathology, including disc perforation and soft tissue HO., Methods: New Zealand white rabbits (n = 9 rabbits) underwent unilateral TMJ disc perforation surgery and sham surgery on the contralateral side. A 2.5 mm defect was created using a punch biopsy in rabbit TMJ disc. The TMJ condyles and discs were evaluated macroscopically and histologically after 4, 8 and 12 weeks. Condyles were blindly scored by four independent observers using OARSI recommendations for macroscopic and histopathological scoring of osteoarthritis (OA) in rabbit tissues., Results: Histological evidence of TMJ condylar cartilage degeneration was apparent in experimental condyles following disc perforation relative to sham controls after 4 and 8 weeks, including surface fissures and loss of Safranin O staining. At 12 weeks, OARSI scores indicated experimental condylar cartilage erosion into the subchondral bone. Most strikingly, HO occurred within the TMJ disc upon perforation injury in six rabbits after 8 and 12 weeks., Conclusion: We report for the first time a rabbit TMJ injury model that demonstrates condylar cartilage degeneration and disc ossification, which is indispensible for testing the efficacy of potential TMJ therapies., (Copyright © 2014 The Authors. Published by Elsevier Ltd.. All rights reserved.)

Purpose/aim: The primary objective was to evaluate the effect of a bupivacaine mandibular nerve block on intraoperative blood pressure (BP) and heart rate (HR) in response to surgical stimulation and the need for systemic analgesics postoperatively. We hypothesized that a mandibular nerve block would decrease the need for systemic analgesics both intraoperatively and postoperatively., Materials and Methods: Fourteen adult male Yucatan pigs were purchased. Pigs were chemically restrained with ketamine, midazolam, and dexmedetomidine and anesthesia was maintained with isoflurane inhalant anesthesia. Pigs were randomized to receive a mandibular block with either bupivacaine (bupivacaine group) or saline (control group). A nerve stimulator was used for administration of the block with observation of masseter muscle twitch to indicate the injection site. Invasive BP and HR were measured with the aid of an arterial catheter in eight pigs. A rescue analgesic protocol consisting of fentanyl and lidocaine was administered if HR or BP values increased 20% from baseline. Postoperative pain was quantified with a customized ethogram. HR and BP were evaluated at base line, pre-rescue, 10 and 20 min post-rescue., Results: Pre-rescue mean BP was significantly increased (p = .001) for the bupivacaine group. Mean intraoperative HR was significantly lower (p = .044) in the bupivacaine versus saline group. All other parameters were not significant., Conclusion: Addition of a mandibular nerve block to the anesthetic regimen in the miniature pig condylectomy model may improve variations in intraoperative BP and HR. This study establishes the foundation for future studies with larger animal numbers to confirm these preliminary findings.

Treacher Collins syndrome is a disorder of craniofacial development with high penetrance and variable expressivity. Its incidence is approximately 1 in 50,000 live births. In this article, we describe the orthodontic treatment of an 11-year-old boy with Treacher Collins syndrome., (Copyright © 2014 American Association of Orthodontists. Published by Elsevier Inc. All rights reserved.)

Recent technologic advances allow clinicians to place dental implants using computer-generated templates. However, there are limited data regarding treatment outcomes for implants placed using these techniques. The purpose of this retrospective study was to report the 2- to 4-year prosthetic outcomes and survival of dental implants placed by postdoctoral residents with a flapless surgical protocol using computer-based planning and stereolithographic surgical templates. Thirty-six patients were treated using the NobelGuide concept, comprising an image-based three-dimensional implant planning software and flapless implant surgery with stereolithographic templates.

Objectives/hypothesis: Orofacial clefts are the most common craniofacial birth defects in humans, with the majority of orofacial clefts occurring as nonsyndromic cleft lip with or without cleft palate (NSCLP). We previously demonstrated associations between single-nucleotide polymorphisms (SNPs) in the IRF6 gene and NSCLP in the Honduran population. Here we investigated other candidate genes and chromosomal regions associated with NSCLP identified from genome-wide association studies (GWAS), including MAFB, ABCA4, 8q24, 9q22, 10q25, and 17q22 in two independent Hispanic populations., Study Design: Case-control and family-based association testing., Methods: Honduran families with two or more members with NSCLP (multiplex) were identified. DNA was collected from affected and unaffected family members (488) and 99 gender-matched controls. NSCLP Colombian families were identified; DNA was collected from 26 proband-parent trios. All participants were genotyped for 17 SNPs in six chromosomal regions. Case-control association and family-based association testing (FBAT) analyses were conducted., Results: Seven SNPs demonstrated association in at least one model in the Honduran population. In the Colombian families, five SNPs demonstrated significance in FBAT when patients with isolated cleft palate (CP) were included; four overlapped with SNPs demonstrating significance in the Honduran population, two with the same allele. One SNP retained significance with CP excluded., Conclusions: This study supports the previous GWAS findings and is the first to suggest a role for FOXE1, ABCA4, and MAFB in orofacial clefting in two separate Hispanic populations., (Copyright © 2012 The American Laryngological, Rhinological, and Otological Society, Inc.)

Objective: A preliminary study was conducted to investigate feasibility of using an oral cancer chemopreventive agent (-)-epigallocatechin-3-gallate (EGCG), the most biologically active component in the green tea extract, in a form of 'swish-and-spit' mouthwash. Such application of EGCG is beneficial as it maximizes exposure of the oral mucosa to the agent but minimizes systemic side effect., Study Design: The study was conducted on individuals suspected to have oral field cancerization who are at a high risk for developing recurrent oral precancerous and carcinomatous lesions. EGCG was used as a daily mouthwash for 7 days. EGCG's ability to modulate target molecules implicated in oral carcinogenesis was assessed by measuring the change in expression level of biomarkers., Results: Immunohistochemical expression of phosphoactivated epidermal growth factor receptor (pEGFR), cyclooxygenase-2 (cox-2) and ki-67 were evaluated at baseline and at the endpoint (day 8). Although not statistically significant, overall decrease in expression levels of pEGFR (27.5%), cox-2 (15.9%) and ki-67 positive cells (51.8%) were observed following EGCG treatment. Moreover, a detectable level of EGCG was found in saliva but not in plasma after the one-week treatment regime, demonstrating local availability of EGCG in oral mucosa without significant systemic absorption., Conclusion: To best of our knowledge this is the first study to explore use of oral cancer chemopreventive agent in a form of mouthwash in patients with oral field cancerization. Although a definitive conclusion was not reached due to limited sample size, if proven effective, EGCG therapy may offer a non-invasive preventive modality for oral field cancerization.

Dental pulp has intrinsic capacity for self-repair. However, it is not clear whether dental pulp cells can be recruited endogenously for regenerating pulp tissues, including mineralizing into dentin. This work is based on a hypothesis that dental pulp stem/progenitor cells can be induced to migrate by chemotactic cytokines and act as endogenous cell sources for regeneration and mineralization. Dental stem cells (DSCs) were isolated from adult human tooth pulp and seeded on the surfaces of 3D collagen gel cylinders that were incubated in chemically defined media with stromal-derived factor-1α (SDF1), basic fibroblast growth factor (bFGF), or bone morphogenetic protein-7 (BMP7). Significantly more cells were recruited into collagen gel by SDF1 or bFGF than without cytokines in 7 days, whereas BMP7 had little effect on cell recruitment. BMP7, however, was highly effective, equally to dexamethasone, in orchestrating mineralization of cultured DSCs. Cell membrane receptors for SDF1, bFGF, and BMP7 were up-regulated in treated DSCs. Upon in vivo delivery, bFGF induced re-cellularization and re-vascularization in endodontically treated human teeth implanted into the dorsum of rats. Thus, endogenous dental pulp cells, including stem/progenitor cells, may be recruited and subsequently differentiated by chemotaxis of selective cytokines in the regeneration of dental pulp.

Purpose: To determine the efficacy of preoperatively administered nicotine nasal spray (3 mg) for analgesia after third molar (TM) surgery., Materials and Methods: A single-center, prospective, randomized, double-blind, crossover trial was conducted. The study population consisted of 20 nonsmoking patients referred to the Department of Oral and Maxillofacial Surgery of Columbia University College of Dental Medicine for extraction of all 4 TMs. Each patient received nicotine nasal spray or placebo spray before TM surgery. At a subsequent visit the contralateral TMs were removed with prior administration of the alternate treatment. For an hour postoperatively, subjects reported information on pain and nausea, and hemodynamic variables were recorded at 15-minute intervals. Telephone follow-up was recorded for 5 days postoperatively, where patients reported information on pain, nausea, and use of hydrocodone/acetaminophen as rescue analgesia., Results: Nicotine treatment was associated with a highly significant decrease in pain reported during the 5 days after TM surgery. There was no difference in the amount of hydrocodone/acetaminophen used or amount of nausea reported. There was a small but significant increase in heart rate after nicotine treatment compared with placebo during the first hour after surgery. There was no difference in blood pressure between groups., Conclusion: Pain is well controlled by hydrocodone/acetaminophen in most patients after TM surgery. However, there is significant variability in pain reported. Nicotinic agonists represent a new class of analgesic that can be considered for patients who are expected to have significant opioid-resistant pain after TM surgery. Caution should be used with patients in whom a small increase in heart rate would be deleterious., (Copyright © 2011 American Association of Oral and Maxillofacial Surgeons. Published by Elsevier Inc. All rights reserved.)

The face distinguishes one human being from another. When the face is disfigured because of trauma, tumor removal, congenital anomalies, or chronic diseases, the patient has a strong desire for functional and esthetic restoration. Current practice of facial reconstruction using autologous grafts, synthetic fillers, and prostheses is frequently below the surgeon's and patient's expectations. Facial reconstruction is yet to take advantage of recent advances in seemingly unrelated fields of stem cell biology, chemical engineering, biomaterials, and tissue engineering. "Biosurgery," a new concept that we propose, will incorporate novel principles and strategies of bioactive cues, biopolymers, and/or cells to restore facial defects. Small facial defects can likely be reconstructed by cell homing and without cell transplantation. A critical advantage of cell homing is that agilely recruited endogenous cells have the potential to harness the host's innate capacity for regeneration, thus accelerating the rate of regulatory and commercialization processes for product development. Large facial defects, however, may not be restorable without cell delivery per our understanding at this time. New breakthrough in biosurgery will likely originate from integrated strategies of cell biology, cytokine biology, chemical engineering, biomaterials, and tissue engineering. Regardless of cell homing or cell delivery approaches, biosurgery not only will minimize surgical trauma and repetitive procedures, but also produce long-lasting results. At the same time, caution must be exercised against the development of products that lack scientific basis or dogmatic combination of cells, biomaterials, and biomolecules. Together, scientifically derived biosurgery will undoubtedly develop into new technologies that offer increasingly natural reconstruction and/or augmentation of the face.

Platelet-rich plasma (PRP) has been gaining increasing popularity in orthopedics and oral and maxillofacial surgery because of its potential efficacy in enhancing bone regeneration. To maximally augment bone healing using PRP and to control the bioavailability of the relevant growth factors, we have designed an alginate hydrogel-based PRP-delivery system. The bioactivity of the growth factors released from PRP carriers was evaluated by determining the ability of these factors to induce osteogenic differentiation of human mesenchymal stem cells (hMSCs). Specifically, monolayers of hMSCs were incubated with the PRP-containing hydrogel carriers over a two-week culture period. Osteoblast-like cells treated with the hydrogel carriers served as controls. The growth and osteogenic differentiation (alkaline phosphatase activity and mineralization) of hMSCs was determined. The results showed that PRP-derived growth factors released from hydrogel carriers stimulated the osteogenic differentiation of hMSCs and most significantly, the cellular response was carrier type-dependent. Future studies will focus on in vitro and in vivo testing of the efficacy of hydrogel-based PRP release systems.

Purpose: This study evaluates the temporal sequence and growth factor release from platelet-rich plasma (PRP) combined with different bone substitutes (BS), to identify an optimal substrate for extended growth factor retention., Materials and Methods: PRP was clotted with bovine thrombin or thrombin receptor activator peptide-6 (TRAP). In addition, PRP was clotted using Allogro (Ceramed, Lakewood, CO), BioGlass (Mo-Sci, Rolla, MN), or BioOss (Osteohealth, Shirley, NY). The effects of media exchange and BS on platelet-derived growth factor (PDGF) and transforming growth factor-beta (TGF beta) release were quantified via enzyme-linked immunosorbent assay., Results: At day 1, the thrombin group released 36% more PDGF than the TRAP group and 80% more than the BS groups. At 7 days, PDGF release was the greatest for the TRAP group. PDGF release was minimal for all groups at day 14, with BS groups retaining 60% more PDGF than thrombin clots. Similarly, the thrombin group released the greatest amount of TGF beta (81.4% of the total), whereas TRAP and BS groups released significantly less TGF beta at day 1. Compared with thrombin, TRAP retained 39.2% more TGF beta, whereas BS groups retained even greater levels (Allogro, 54.3%; BioOss, 45.8%; BioGlass, 67.0%). No significant difference in TGF beta release was observed among the substitutes after day 1. The BS groups continued to retain TGF beta after 14 days, whereas all TGF beta in the thrombin clots was depleted., Conclusions: PRP preparation with thrombin results in a large, immediate release of growth factors that could be lost into the interstitium in vivo. TRAP-BS may prove more efficacious than thrombin in sustaining growth factor levels critical for the cascade of events leading to bone formation.

Purpose: This study proposes an alternative preparation method of platelet-rich plasma (PRP). Specifically, we compare the use of thrombin receptor agonist peptide-6 (TRAP) and bovine thrombin as a clotting agent in the preparation of PRP., Materials and Methods: PRP was prepared by centrifugation and clotted with thrombin or TRAP. In vitro clotting times were monitored as a function of TRAP concentration, and clot retraction was determined by measuring clot diameter over time. Following the optimization of TRAP concentration, experiments were repeated with the addition of several commercially available bone substitutes. The release of PRP-relevant growth factors as a function of PRP preparation was also determined., Results: The most rapid polymerization of PRP takes place with the addition of thrombin, followed by TRAP/Allogro (Ceramed, Lakewood, CO), TRAP/BioGlass (Mo-Sci, Rolla, MN), TRAP/BioOss (Osteohealth, Shirley, NY), and TRAP alone. Thrombin caused considerable clot retraction (43%), whereas TRAP alone resulted in only 15% retraction. TRAP/Allogro, TRAP/BioOss, and TRAP/BioGlass all exhibited minimal retraction (8%)., Conclusions: The use of TRAP to activate clot formation in the preparation of PRP may be a safe alternative to bovine thrombin. It results in an excellent working time and significantly less clot retraction than the currently available methods of PRP production.

Surgery of the cranial base presents significant challenges for the surgeon and patient alike. The goal of ablative surgery-to obtain disease-free margins-contrasts with the patient's functional and esthetic needs. This comes into sharp juxtaposition in cranial base surgery. The technical and technologic advances of the last two decades have made surgical treatment of cranial base lesions possible. This article reviews the pathology, anatomy, and lateral and midline approaches to the cranial base.

Purpose: Augmentation of the alveolar ridge before implant placement is frequently performed. The purpose of this study was to compare the amount and quality of bone formation under Micro Titanium Augmentation Mesh (M-TAM) when used alone for guided tissue regeneration or in combination with a porous hydroxyapatite (HA) bone graft substitute., Materials and Methods: Nine adult female dogs underwent extraction of premolars and molars and had a knife-edge ridge created. Three months later, the ridges were augmented with either M-TAM alone or M-TAM with a nonresorbable porous HA (Interpore 200; Interpore Cross International, Irvine, CA). Six months after augmentation, the dogs were killed, and the mandibles were harvested and imaged using 3-dimensional computed axial tomography. Statistical analysis was performed from the data obtained from the scans. The mandibles were then fixed, sectioned, and examined by light microscopy., Results: Dehiscence occurred in 22 of 32 experimental sites. Seven of these 22 dehisced sites showed increased ridge width. Ridge width increased in both the HA and non-HA groups. The HA group showed a greater increase in ridge width., Conclusion: A high rate of dehiscence was observed in this animal study using M-TAM for guided tissue regeneration. In animals that did not dehisce, increased width was greater when nonresorbable porous HA was used as a bone graft substitute., (Copyright 2003 American Association of Oral and Maxillofacial Surgeons J Oral Maxillofac Surg 61:347-353, 2003)

Purpose: This article discusses the use of the 2-piece Le Fort I osteotomy to gain access to the cranial base and the authors' experiences with this technique., Methods: Medical records of patients who required surgical access to the cranial base were retrospectively reviewed, and demographic data, procedures done, postoperative results, and complications were recorded., Results: Nine patients underwent a 2-piece Le Fort I osteotomy. Complications included development of a pseudoaneurysm of the internal carotid artery in 1 patient, 2 cerebrospinal fluid leaks, both of which resolved, and 1 patient who lost 2 teeth and some associated bone., Conclusion: The 2-piece Le Fort I osteotomy provides excellent access to the clival region of the cranial base with minimal complications.

The purpose of this study was to compare soft tissue prediction accuracy of model surgery combined with computer software prediction with that of computer software prediction alone and to assess surgical accuracy by comparing the immediate postsurgical cephalogram with the planned movement of skeletal hard tissue. The predicted and actual soft tissue changes and the corresponding skeletal changes of 16 patients were compared using the Quick Ceph Image cephalometric treatment simulation software. A custom analysis was created to measure the hard tissue and soft tissue changes that occurred as a result of the surgical procedure. On average, the predictions were not significantly different from the actual postsurgical profile changes. Surgical changes of hard tissues from presurgery to postsurgery were accurate as planned except for the position of N-ANS. All lower soft tissue points moved significantly during treatment. Quick Ceph Image offers a rapid and reliable method of profile prediction that does not require artistic skill. If predictions are interpreted with caution and transferred accurately to the model surgery, they can provide an excellent visual aid during presurgical treatment planning and patient presentation.

Purpose: This study was performed to compare the dimensions of the nasopharynx, oropharnynx, and hypopharynx of persons with hyperdivergent and normodivergent facial types, and to determine whether any variations exist., Patients and Methods: Lateral cephalometric records of a population with a normodivergent facial pattern (n = 23) and a group with a hyperdivergent facial pattern (n = 27) as evidenced by increased mandibular plane angle were used to compare the soft tissue airway dimensions. Statistical analysis consisted of Student's t-tests, Wilcoxon rank sums, and chi2. Statistical significance was set .05., Results: Overall the hyperdivergent group had a narrower anteroposterior pharyngeal dimension than the normodivergent control group. This narrowing was specifically noted in the nasopharynx at the level of the hard palate and in the oropharynx at the level of the tip of the soft palate and the mandible. In addition, the posterior pharyngeal wall had a thinning at the level of the inferior border of the third cervical vertebrae, and there was a more obtuse palatal angle. The tongue was also positioned more inferiorly and posteriorly in the hyperdivergent group, as evidenced by the increased distance between the hyoid bone and the mandibular plane and the increased distance between the soft palate tip and the epiglottis. The hyperdivergent group had more retruded maxillary and mandibular apical bases and a higher Class II skeletal discrepancy., Conclusions: The narrower anteroposterior dimension of the airway in hyperdivergent patients may be attributable to skeletal features common to such patients, that is, retrusion of the maxilla and the mandible and vertical maxillary excess. Other features, such as an obtuse soft palate and low-set hyoid, also may be contributory factors. The relatively thin posterior pharyngeal wall observed in hyperdivergent patients might be a compensatory mechanism.

Miles, Lovas, and Cohen first described hemimaxillofacial dysplasia in two patients in 1987. This disorder consists of facial asymmetry, facial hypertrichosis, unilateral maxillary hyperplasia, and hypoplastic teeth. We report two additional cases with similar findings.

The purpose of this investigation was to compare two sedation techniques for use in outpatient third molar surgery. Forty ASA class I or II volunteers were randomly allocated to two groups. All subjects received supplemental oxygen via a nasal hood, sublimaze (.0007 mg/kg [corrected] intravenous bolus), and midazolam (.5 mg/min) titrated to effect. Using an incremental bolus technique, group A then received methohexital, while group B received propofol. Both groups maintained stable mean arterial pressure, oxygen saturation, and end-tidal CO2 throughout the perioperative period. However, group A had a dramatic increase in heart rate (26.7% versus 13.9% for group B [P < .05]). Better postoperative psychomotor performance (P < .05) as measured by the Trieger Dot analysis was demonstrated by patients who received propofol. It was concluded that propofol is superior to methohexital for intravenous sedation.

T2-weighted magnetic resonance imaging (MRI) presents paranasal sinus pathology with remarkable clarity. However, it has yet to be demonstrated that all MRI findings represent true pathology and not minor or incidental findings of no consequence. In an effort to resolve this question, we have analysed retrospectively 263 consecutive T2-weighted MRI examinations of the head performed for indications not associated with possible sinus pathology. We examined these studies for abnormally increased signal in the paranasal sinuses and the sites of involvement. Mucoperiosteal thickening, mucus retention cysts, air-fluid levels or total sinus opacification were recorded. Of the 263 studies examined, 65 (24.7%) demonstrated abnormalities in the paranasal sinuses. We conclude that because of its great sensitivity MRI will often detect abnormalities in the paranasal sinuses which are unrelated to the patients' presenting problems.