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Abstract Fibrous dysplasia (FD) is a non-malignant condition caused by post-zygotic, activating mutations of the GNAS gene that results in inhibition of the differentiation and proliferation of bone-forming stromal cells and leads to the replacement of normal bone and marrow by fibrous tissue and woven bone. The phenotype is variable and may be isolated to a single skeletal site or multiple sites and sometimes is associated with extraskeletal manifestations in the skin and/or endocrine organs (McCune-Albright syndrome). The clinical behavior and progression of FD may also vary, thereby making the management of this condition difficult with few established clinical guidelines. This paper provides a clinically-focused comprehensive description of craniofacial FD, its natural progression, the components of the diagnostic evaluation and the multi-disciplinary management, and considerations for future research.

PURPOSE: The purpose of this study was to evaluate porcine bone marrow-derived progenitor cell (pBMPC) proliferation and penetration into a novel 3-dimensionally printed scaffold. MATERIALS AND METHODS: Four different tissue engineering scaffolds to evaluate pBMPC proliferation and penetration were examined. Scaffolds were fabricated from polycaprolactone (PCL) or the combination of [beta]-tricalcium phosphate ([beta]-TCP) and PCL (50:50), with 2 separate channel sizes (1 mm [small (S)] vs 2 mm [large (L)]). Scaffolds were fabricated into 20 x 20 x 7-mm blocks by use of a TheriForm machine (Integra Life Sciences, Akron, OH). Four groups of scaffolds were examined for pBMPC proliferation and penetration: group 1, [beta]-TCP/PCL S; group 2, [beta]-TCP/PCL L; group 3, PCL S; and group 4, PCL L. Nonparametric mean (Kruskal-Wallis) and multiple comparisons tests were used to compare the 4 groups. RESULTS: No shrinkage or deformation was noted in any of the scaffold groups after 2 weeks of culture. Mean surface cell counts ranged from 13.4 to 87.8 cells/0.57 mm(2), with group 1 ([beta]-TCP/PCL S) having statistically significantly higher counts than the other groups (P < .001). Mean interior cell counts ranged from 10.9 to 75.6 cells/0.57 mm(2), with group 1 having the greatest interior cell count (P < .001). Total collagen formation ranged from 0.2% to 86%, with group 1 having the highest collagen formation (P < .001). CONCLUSIONS: The 3-dimensionally printed scaffold ([beta]-TCP/PCL) with 1-mm channels showed greater cellular proliferation, penetration, and collagen formation after a 2-week in vitro culture than the other scaffolds evaluated. [beta]-TCP/PCL S scaffolds warrant further evaluation for bone tissue engineering in vivo. [ABSTRACT FROM AUTHOR]

PURPOSE: Current devices for mandibular distraction osteogenesis (DO) are complex and require significant patient or family skill during active distraction. Successful development of an automated, continuous distraction device would eliminate the need for patient participation in this process. The purpose of this study was to comprehensively review devices currently in development for continuous DO and to identify and evaluate the achieved successes and remaining problems. MATERIALS AND METHODS: A PubMed search of the English language literature in October 2008 using the keywords automatic or automated or continuous or hydraulic or motor or magnetic or spring and distraction osteogenesis was performed. The search included all technical notes, animal studies, and human studies describing the use of any automated continuous distraction device for the mandible. Excluded were studies using distraction devices employing hydraulics, motors, or springs that did not distract automatically and continuously and devices used for bones other than the mandible. RESULTS: The search returned 97 matches. Of these, 12 articles were selected as relevant to this review based on the inclusion and exclusion criteria detailed above. Eight distinct devices for automated, continuous DO were described in these reports and evaluated in this review. These included motor-driven, spring-mediated, and hydraulically powered distractors. CONCLUSIONS: The abundance of research currently underway to develop a continuous distractor highlights the clinical demand for, and usefulness, of such a device. Despite many advances and promising results, significant problems remain to be overcome before any of these devices gain widespread clinical acceptance. [ABSTRACT FROM AUTHOR]

PURPOSE: To document histologic and immunohistochemical changes in the anterior digastric muscle during distraction osteogenesis (DO). MATERIALS AND METHODS: Nineteen Yucatan minipigs with mixed dentition were used for these experiments. Group A (n = 16) underwent unilateral mandibular distraction at a rate of 1 mm/day (no latency) for 12 days. Animals were killed at mid-DO (n = 5), end-DO (n = 5), mid-fixation (n = 4), and end-fixation (n = 2). Group B (n = 2) underwent acute 12-mm advancement, and group C (n = 1) dissection and osteotomy. Animals from groups B and C were killed at the end-DO time point. Digastric muscles from treatment and contralateral sides of all animals were harvested and embedded in paraffin. Specimens were stained with hematoxylin/eosin or immunohistochemically for proliferating cell nuclear antigen (PCNA; total cell proliferation), paired Box-7 gene protein (Pax7; satellite cells), or myogenic differentiation 1 protein (MyoD; differentiating myoblasts). Descriptive and bivariate statistics were computed to compare groups (P <= .05 statistically significant). RESULTS: All animals survived the operation and observation period; there were no device failures. Two animals (1 at mid-DO, 1 at mid-fixation) were eliminated from the study because of postoperative infection. There was minimal digastric inflammation, fibrosis, and muscle fiber size variability during active DO. Immunohistochemical analysis showed statistically significant increases in PCNA (cellular proliferation), Pax7 (satellite cells), and MyoD (differentiating myoblasts) positive nuclei in digastrics at mid-DO and end-DO. CONCLUSIONS: Results of this study indicate that there are minimal pathologic changes but significant increases in PCNA, Pax7, and MyoD positive nuclei during active distraction. This supports the hypothesis that the digastric muscle response to DO consists of proliferation and hypertrophy. [ABSTRACT FROM AUTHOR]

PURPOSE: Most reports on airway anatomy after maxillomandibular advancement (MMA) for obstructive sleep apnea (OSA) have focused on changes in airway diameter, ie, posterior airway space (PAS), rather than airway length. The purpose of this study was to evaluate changes in cephalometrically measured upper airway length (UAL) after MMA. MATERIALS AND METHODS: This was a retrospective cohort study. The sample was composed of subjects who underwent MMA for OSA. The primary outcome measurement was change in UAL measured on lateral cephalograms. Predictor variables included patient demographic and cephalometric characteristics. Secondary outcome measurements were changes in sleep disturbance. Descriptive, bivariate, and regression statistics were computed. For all analyses, P <= .05 was considered statistically significant. RESULTS: The sample included 23 adult subjects (mean age, 39.3 ± 12.1 years; 6 women). Subjects had average maxillary and mandibular advancements of 9.8 ± 2.0 and 10.8 ± 2.2 mm, respectively. Mean pre- and postoperative UALs were 75.8 ± 7.0 and 67.0 ± 5.7 mm, respectively (P < .001). Mean pre- and postoperative respiratory disturbance indexes were 53.2 ± 22.4 and 19.0 ± 12.0 events/hour, respectively (P = .003). All patients had improvement in OSA symptoms, but 3 (13%) required continuous positive airway pressure. CONCLUSION: The results of this study suggest that UAL decreases as a result of MMA. In addition, these results support the observation that MMA is associated with objective and subjective improvements in patients with OSA. [ABSTRACT FROM AUTHOR]

In this report, we explore the little known role of Dr Nathan Cooley Keep in the dissemination of ether anesthesia in Boston. Keep was a prominent Boston dentist who, for a short time, taught and employed both William Morton and Horace Wells. He used ether anesthesia for a variety of dental and other surgical procedures requiring pain control. Keep administered ether to anesthetize Henry Wadsworth Longfellow's wife during the delivery of their daughter. This was the first use of ether for obstetric anesthesia. Dr Keep was also the first Dean of the Harvard Dental School and convinced the Massachusetts General Hospital to appoint a dentist to the staff of the hospital for the first time. [ABSTRACT FROM AUTHOR]

Purpose: The relationship between giant cell lesions (GCLs) of the maxillofacial (MF) skeleton and those of the axial/appendicular (AA) skeleton has been long debated. The present study compared the clinical and radiographic characteristics of subjects with MF and AA GCLs. Materials and Methods: This was a retrospective cohort study of patients treated for GCLs at Massachusetts General Hospital from 1993 to 2008. The predictor variables included tumor location (MF or AA) and clinical behavior (aggressive or nonaggressive). The outcome variables included demographic, clinical, and radiographic parameters, treatments, and outcomes. Descriptive and bivariate statistics were computed, and P Results: The sample included 93 subjects: 45 with MF (38 with aggressive and 7 with nonaggressive) and 48 with AA (30 with aggressive and 18 with nonaggressive). Comparing the patients with MF and AA GCLs, those with MF lesions presented younger (P < .001), and the lesions were more commonly asymptomatic (P < .001), smaller (P < .001), and managed differently (P < .001) than AA lesions. When stratified by clinical behavior, aggressive tumors were diagnosed earlier than nonaggressive tumors (P < .001). Controlling for location and clinical behavior, patients with MF aggressive lesions were younger (P < .001) than those with AA aggressive lesions. MF nonaggressive lesions were more commonly asymptomatic (P = .04), smaller (P = .05), and less commonly locally destructive (P = .05) than AA nonaggressive lesions. Conclusions: These results suggest that MF and AA GCLs represent a similar, if not the same, disease. Comparing the aggressive and nonaggressive subgroups, more similarities were found than when evaluating without stratification by clinical behavior. The remaining differences could be explained by the likelihood that MF tumors are diagnosed earlier than AA tumors because of facial exposure and dental screening examinations and radiographs. [ABSTRACT FROM AUTHOR]

PURPOSE: Nonsteroidal anti-inflammatory drugs are commonly prescribed to reduce inflammation and pain. However, little is known about the direct effect of these drugs on the differentiation of bone marrow-derived progenitor cells into osteoblasts. The purpose of this study was to determine the effect of ibuprofen on osteoblast differentiation and proliferation in a minipig model. MATERIALS AND METHODS: Bone marrow was aspirated from the minipig ilium, and porcine bone marrow-derived progenitor cells (pBMPCs) were isolated and expanded in standard culture medium. The pBMPCs were replated and differentiated into osteoblasts by use of osteogenic supplements (OS). Five groups were studied: negative control-pBMPCs in standard medium only; positive control-pBMPCs, standard culture medium, and OS; and 3 experimental groups-pBMPCs, standard culture medium, OS, and ibuprofen added in doses of 0.1, 1.0, and 3.0 mmol/L. Cell cultures were evaluated quantitatively by alkaline phosphatase (ALP) stain, von Kossa stain, and deoxyribonucleic acid (DNA) content. RESULTS: pBMPCs cultured with OS and low-dose ibuprofen (0.1 mmol/L) showed ALP stain, von Kossa stain, and DNA content similar to pBMPCs cultured in OS (positive control). pBMPCs cultured in higher doses of ibuprofen (1.0 and 3.0 mmol/L) produced significantly less positive staining of ALP and von Kossa and decreased DNA content. CONCLUSION: The results indicate that high-dose ibuprofen has a deleterious effect on pBMPC differentiation into osteoblasts whereas low-dose ibuprofen does not. The low dose of 0.1 mmol/L is the typical serum level when prescribed for clinical use. [ABSTRACT FROM AUTHOR]

Background: The impact of cleft and craniomaxillofacial surgeons (CCMFSs) on oral and maxillofacial surgery (OMS) resident training is not well known., Purpose: The purpose of this study was to measure the association between resident exposure to OMS faculty with CCMFS training, clinical experience, and scholarly activity., Study Design, Setting, Sample: An anonymous survey of OMS residency directors in the United States, distributed electronically by the American Association of Oral and Maxillofacial Surgeons, was used to conduct a cross-sectional study. Survey consisted of 3 following sections: 1) CCMFS faculty and program-related information, 2) CCMFS curriculum in residency programs, and 3) academic productivity related to CCMFS., Predictor Variable: Primary predictor variable was CCMFS exposure: presence or absence of an OMS faculty with CCMFS fellowship or equivalent., Main Outcome Variable: Primary outcome variables were: 1) number of CCMFS cases with OMS resident involvement, 2) number of OMS residents who pursued CCMF surgery fellowship, and 3) CCMFS-related academic productivity of faculty and residents by abstract or publication., Covariates: Covariates include clinical experience of OMS residents in CCMFS cases and CCMFS-related academic productivity., Analyses: Descriptive statistics and continuous/ordinal variables were summarized. Fisher exact and Wilcoxon Rank Sum tests were completed. 2-sided P values of <.05 were considered statistically significant., Results: There were 81 completed surveys (91% response rate; excluded military programs). Almost half of OMS training programs have at least one attending surgeon who completed a CCMF surgery fellowship (n = 36/81, 44.4%). OMS departments with a CCMFS-trained faculty performed more operations on patients with congenital craniofacial differences (P value <.001) and were more likely to publish at least one manuscript related to CCMF surgery in the last 5 years (P value = .029)., Conclusion and Relevance: Nearly half of OMS training programs have at least one attending surgeon who completed a CCMF surgery fellowship or equivalent. CCMF surgery faculty may increase opportunities for OMS resident involvement in caring for patients with congenital craniofacial differences. There is academic productivity in programs with CCMF surgeons. This should improve as the cohort of CCMF trained oral and maxillofacial surgeons increases., (Copyright © 2024. Published by Elsevier Inc.)

Background: Idiopathic condylar resorption (ICR) is a rare condition of unknown etiology characterized by progressive decrease in volume and pathologic remodeling of a previously normal mandibular condyle. Juvenile idiopathic arthritis (JIA) affecting only the temporomandibular joint (TMJ-only JIA) is characterized by synovitis and destruction of TMJ tissues without involvement of other joints. It is often difficult to differentiate the 2 conditions because they exhibit similar phenotypes., Purpose: To compare histology of resected condylar specimens from patients with ICR and TMJ-only JIA. Specific aims were as follows: 1) to correlate longitudinal clinical data with histopathology of resected condyle specimens and 2) to compare resorption patterns between the 2 disease processes., Study Design, Setting, Sample: This was a retrospective cohort study of patients treated at the Massachusetts General Hospital from 1999 through 2023., Predictor/exposure/independent Variable: Primary predictor variable was the diagnosis (ICR or JIA). Secondary predictor variables included age, gender, race, putative contributing factors, and laboratory studies., Main Outcome Variable(s): Primary outcome variable was presence or absence of inflammatory infiltrates in bone and synovial specimens. Secondary outcome variables were structural integrity and morphologic characteristics of the condylar cartilage and bone., Analyses: Spearman correlation was used to assess the relationship between histological scores and age, gender, and possible associated contributing factors. A P value < .05 was considered statistically significant., Results: Thirty-five subjects (67 specimens) were included in group 1 (ICR). Eight subjects (15 specimens) were included in group 2 (TMJ-only JIA). The histopathologic findings in ICR consisted of severe and irregular cartilage surface disruption, fibrocartilage degeneration, and subchondral bone with no inflammatory infiltrate. Degeneration was observed to begin at the anterior pole of the condyle and progress eventually to total resorption to the sigmoid notch. TMJ-only JIA was notable for more severe condylar degeneration and inflammation in the bone and synovia. There was no specific pattern of degeneration. For both groups, the subject's age, gender, or putative contributing factors did not correlate with the histopathologic scores., Conclusion and Relevance: These results support the hypothesis that "ICR" and TMJ-only JIA are distinct clinical entities and can be distinguished by histopathologic findings in the mandibular condyles and synovia., (Copyright © 2024 American Association of Oral and Maxillofacial Surgeons. Published by Elsevier Inc. All rights reserved.)

Bone scans, reflecting blood flow and metabolic activity in a region of interest, are frequently used to evaluate mandibular growth disorders. Increased uptake is a non-specific finding and can occur as a result of multiple causes. The correlation between radioactive tracer uptake and growth activity has not been consistently demonstrated. The aim of this study was to assess the accuracy of planar skeletal scintigraphy (SS), single-photon emission computed tomography (SPECT), and SPECT with computed tomography (CT) images (SPECT/CT) in detecting abnormal mandibular growth activity compared to clinical and radiographic/tomographic methods (reference standard) and histologic findings. A systematic review was conducted following the PRISMA guidelines. Sensitivity, specificity, and accuracy were calculated for planar SS, SPECT, and SPECT/CT. Compared to the reference standard, SPECT/CT had the best diagnostic accuracy (76.5% sensitivity, 90.4% specificity, 83.2% accuracy), followed by planar SS (81.8% sensitivity, 84.5% specificity, 83.0% accuracy) and SPECT (77.7% sensitivity, 72.4% specificity, 74.5% accuracy). The results of this study indicate that SPECT/CT has the best clinical correlation, but the certainty of the evidence is low. The differences in sensitivity and specificity between the three index tests were not clinically significant. The three tests can be useful, with only a small difference in their diagnostic value. Histopathology was found not to be satisfactory as a reference standard., Competing Interests: Declaration of Competing Interest Dr. Zachary Peacock received a travel stipend from KLS Martin group in 2022. This has no relevance or relationship to the current topic of study in the submitted manuscript. No other authors have conflicts of interest., (Copyright © 2023 International Association of Oral and Maxillofacial Surgeons. Published by Elsevier Inc. All rights reserved.)

Context: Pain is a poorly managed aspect in fibrous dysplasia/McCune-Albright syndrome (FD/MAS) because of uncertainties regarding the clinical, behavioral, and neurobiological underpinnings that contribute to pain in these patients., Objective: Identify neuropsychological and neurobiological factors associated with pain severity in FD/MAS., Design: Prospective, single-site study., Patients: Twenty patients with FD/MAS and 16 age-sex matched healthy controls., Intervention: Assessments of pain severity, neuropathic pain, pain catastrophizing (pain rumination, magnification, and helplessness), emotional health, and pain sensitivity with thermal quantitative sensory testing. Central nervous system (CNS) properties were measured with diffusion tensor imaging, structural magnetic resonance imaging, and functional magnetic resonance imaging., Main Outcome Measures: Questionnaire responses, detection thresholds and tolerances to thermal stimuli, and structural and functional CNS properties., Results: Pain severity in patients with FD/MAS was associated with more neuropathic pain quality, higher levels of pain catastrophizing, and depression. Quantitative sensory testing revealed normal detection of nonnoxious stimuli in patients. Individuals with FD/MAS had higher pain tolerances relative to healthy controls. From neuroimaging studies, greater pain severity, neuropathic pain quality, and psychological status of the patient were associated with reduced structural integrity of white matter pathways (superior thalamic radiation and uncinate fasciculus), reduced gray matter thickness (pre-/paracentral gyri), and heightened responses to pain (precentral, temporal, and frontal gyri). Thus, properties of CNS circuits involved in processing sensorimotor and emotional aspects of pain were altered in FD/MAS., Conclusion: These results offer insights into pain mechanisms in FD/MAS, while providing a basis for implementation of comprehensive pain management treatment approaches that addresses neuropsychological aspects of pain., (© The Author(s) 2023. Published by Oxford University Press on behalf of the Endocrine Society. All rights reserved. For permissions, please e-mail: journals.permissions@oup.com.)

High energy trauma has been considered a risk factor for blunt cerebrovascular injuries (BCVI). The purpose of this study was to determine the incidence and risk factors for BCVI specifically in patients with maxillofacial fractures in an urban level I trauma center. A retrospective cohort study of patients aged ≥ 18 years, admitted to Massachusetts General Hospital (MGH) between 2007 and 2017, was implemented. There were 23,394 patients treated and entered into the MGH Trauma Registry: 22,287 sustained blunt trauma. Of the total blunt trauma patients, 68 (0.3%) had BCVI. There were 2421 patients with CMF fractures from blunt trauma (mean ± standard deviation age, 53 ± 22 years; 29.9% female included as study subjects, of whom 24 (1.0%) had BCVI). In a multivariate model, all mandible fracture (odds ratio (OR) 4.3, 95% confidence interval (CI) 1.6-11.6, P = 0.004), crush injury, defined as blunt compression injury (OR 11.1, 95% CI 2.1-58.1, P = 0.004), and cervical spine injury (OR 10.1, 95 CI 3.7-27.5, P < 0.001) were independent risk factors for BCVI. Mortality was 4.3 times higher in craniomaxillofacial fracture patients with BCVI versus those without BCVI; complications of BCVI (stroke) contributed to the majority of deaths. Appropriate screening and treatment of BCVI in patients with maxillofacial fractures is important., Competing Interests: Competing interests None., (Copyright © 2022 International Association of Oral and Maxillofacial Surgeons. Published by Elsevier Inc. All rights reserved.)

Involvement of the temporomandibular joint (TMJ) is common in juvenile idiopathic arthritis (JIA). TMJ arthritis can lead to orofacial symptoms, orofacial dysfunction, and dentofacial deformity with negative impact on quality of life. Management involves interdisciplinary collaboration. No current recommendations exist to guide clinical management. We undertook this study to develop consensus-based interdisciplinary recommendations for management of orofacial manifestations of JIA, and to create a future research agenda related to management of TMJ arthritis in children with JIA. Recommendations were developed using online surveying of relevant stakeholders, systematic literature review, evidence-informed generation of recommendations during 2 consensus meetings, and Delphi study iterations involving external experts. The process included disciplines involved in the care of orofacial manifestations of JIA: pediatric rheumatology, radiology, orthodontics, oral and maxillofacial surgery, orofacial pain specialists, and pediatric dentistry. Recommendations were accepted if agreement was >80% during a final Delphi study. Three overarching management principles and 12 recommendations for interdisciplinary management of orofacial manifestations of JIA were outlined. The 12 recommendations pertained to diagnosis (n = 4), treatment of TMJ arthritis (active TMJ inflammation) (n = 2), treatment of TMJ dysfunction and symptoms (n = 3), treatment of arthritis-related dentofacial deformity (n = 2), and other aspects related to JIA (n = 1). Additionally, a future interdisciplinary research agenda was developed. These are the first interdisciplinary recommendations to guide clinical management of TMJ JIA. The 3 overarching principles and 12 recommendations fill an important gap in current clinical practice. They emphasize the importance of an interdisciplinary approach to diagnosis and management of orofacial manifestations of JIA., (© 2022 The Authors. Arthritis & Rheumatology published by Wiley Periodicals LLC on behalf of American College of Rheumatology.)

Here, we trace the history of oral and maxillofacial surgery (OMS) education from the mid-19th century to the present. We consider the effects of separation of dentistry and medicine, discovery of anesthesia, antisepsis, antibiotics, and wars on surgical progress and training. In the 19th century, apprenticeships with well-known surgeons were the norm. In the 20th century, training evolved from nonintegrated dental school and hospital experiences to 3- and then 4-year integrated hospital programs. After World War II individual oral surgeons pursued the MD degree after residency. The formal dual degree OMS paradigm began in the 1970s., (Copyright © 2022 Elsevier Inc. All rights reserved.)

This article outlines a conceptual approach to the reconstruction of jaw deformities associated with abnormalities in the mandibular condyle. The authors describe a hierarchy of reconstruction, emphasizing use of the least invasive and progressing to the most complex and invasive techniques, depending on the nature and severity of the underlying deformity, prior operations, patient age, and stage of growth. Consider joint preservation orthognathic surgical correction, followed by biological techniques for replacement of the condyle, and avoid replacing a functional temporomandibular joint based only on radiographic remodeling and concerns about potential future flare-ups of disease based on anecdotal data., (Copyright © 2021 International Association of Oral and Maxillofacial Surgeons. Published by Elsevier Inc. All rights reserved.)

Competing Interests: Declaration of competing interest The authors declare that they have no known competing financial interests or personal relationships that could have appeared to influence the work reported in this paper.

The purpose of this study was to assess skeletal stability and predictors of relapse in patients undergoing an isolated Le Fort I osteotomy. A retrospective cohort study of 92 subjects undergoing Le Fort I osteotomy for Class III malocclusion was implemented. Predictor variables were demographic and perioperative factors. The primary outcome variable was postoperative skeletal position with relapse defined as >2mm sagittal and/or vertical change at A-point on serial lateral cephalograms at immediate postoperative, 1 year, and latest follow-up time points. Mean advancement at A-point was 6.28±2.63mm and mean lengthening was 0.92±1.76mm. Eight subjects (8.70%) had relapse (>2mm) in the sagittal plane, and two subjects (2.17%) in the vertical plane. No subjects required reoperation for relapse as overbite and overjet remained in an acceptable range due to dental compensation. In regression analysis, magnitude of maxillary advancement was an independent predictor of relapse in the sagittal plane (P=0.008). There were no significant predictors of relapse in the vertical plane. This study suggests that isolated Le Fort I osteotomy for correction of skeletal Class III malocclusion is a stable procedure and that greater advancement is an independent risk factor for sagittal relapse., (Copyright © 2021 International Association of Oral and Maxillofacial Surgeons. Published by Elsevier Inc. All rights reserved.)

Denosumab has been advocated as a potential treatment for the rare skeletal disorder fibrous dysplasia (FD); however, there is limited data to support safety and efficacy, particularly after drug discontinuation. We report a case of successful treatment of aggressive craniofacial FD with denosumab, highlighting novel insights into the duration of efficacy, surrogate treatment markers, and discontinuation effects. A 13-year-old girl presented with persistent pain and expansion of a maxillary FD lesion, which was not responsive to repeated surgical procedures or bisphosphonates. Pre-treatment biopsy showed high RANKL expression and localization with proliferation markers. Denosumab therapy was associated with improved pain, decreased bone turnover markers, and increased lesion density on computed tomography scan. During 3.5 years of treatment, the patient developed increased non-lesional bone density, and after denosumab discontinuation, she developed hypercalcemia managed with bisphosphonates. Pain relief and lesion stability continued for 2 years following treatment, and symptom recurrence coincided with increased bone turnover markers and decreased lesion density back to pre-treatment levels. This case highlights the importance of considering the duration of efficacy when treating patients with FD and other nonresectable skeletal neoplasms that require long-term management., (© 2021. This is a U.S. government work and not under copyright protection in the U.S.; foreign copyright protection may apply.)

Purpose: The natural history of fibrous dysplasia (FD) is poorly understood. The purpose of this study was to identify differences in demographic, clinical, and radiographic characteristics among patients with craniofacial FD, including McCune-Albright syndrome (MAS), polyostotic fibrous dysplasia (PFD), and monostotic fibrous dysplasia (MFD). We hypothesized that patients with MAS would show higher disease severity, have more complications, and undergo more operations than those with PFD or MFD., Patients and Methods: A retrospective cohort study of patients with MAS or FD, evaluated at Massachusetts General Hospital from 2000 to 2018, was implemented. Patients of all ages and genders were identified through Massachusetts General Hospital Data Registries using International Classification of Diseases, Ninth Revision (ICD-9) and International Classification of Diseases, Tenth Revision (ICD-10) codes. Those with adequate clinical and radiographic data were included. Predictor variables were diagnosis of MAS, PFD, or MFD; age; and gender. Outcome variables included severity of disease at initial presentation (aggressive, nonaggressive and slow growing, or quiescent), number of operations, and complications: pain, sensory disturbances, pathologic fracture, airway obstruction, osteomyelitis, and dental findings. Data were analyzed with descriptive statistics and assessed for significance using χ 2 tests and analysis of variance (P < .05)., Results: A total of 229 patients were identified: 114 had craniofacial FD, and 70 of these 114 (61.4%) met the inclusion criteria (48 of whom were female patients). The average age at diagnosis was 23.5 years; mean length of follow-up, 5.8 years. Diagnoses included MAS in 9 patients, PFD in 24, and MFD in 37. Signs and symptoms at initial presentation were pain (n = 29), sensory abnormalities (n = 13), facial deformity or swelling (n = 54), and dental findings (n = 25). At presentation, the biological behavior of disease was 77.8% aggressive, 11.1% nonaggressive, and 11.1% quiescent in the MAS group; 41.7%, 41.7%, and 16.7%, respectively, in the PFD group; and 29.7%, 29.7%, and 40.5%, respectively, in the MFD group. Patients with MAS were younger and were more likely to have pain, pathologic fractures, more bones involved, bilateral disease, and visual symptoms than those with PFD or MFD. MAS patients underwent more operations (mean, 4.2 ± 4.18) than those with PFD (mean, 2.6 ± 2.31; P = not significant) or MFD (mean, 1.7 ± 1.28; P = .010)., Conclusions: The results of this study indicate that patients with MAS, presumably with the same mutation, are more likely to have aggressive disease, complications, and more operations than those with PFD or MFD., (Copyright © 2020 American Association of Oral and Maxillofacial Surgeons. Published by Elsevier Inc. All rights reserved.)

The purpose was to determine whether there are regional differences in temporomandibular joint (TMJ) inflammation in patients with juvenile idiopathic arthritis (JIA). This was a retrospective study of all patients with dynamic, contrast-enhanced magnetic resonance imaging through the TMJs at Massachusetts General Hospital between January 2015 and July 2016. The patient cohort included those with a history of JIA and control patients who underwent MRI for other routine clinical purposes. TMJ inflammation was quantified as the difference between post-gadolinium and pre-gadolinium articular T1 signal intensity normalized to post-gadolinium signal intensity of the longus capitis muscle. TMJ enhancement profiles were generated for the lateral, central, and medial portions of the TMJ. Regional differences in TMJ enhancement were investigated using basic descriptive statistics. Medial edge enhancement of the TMJs was highest in symptomatic JIA joints, followed by asymptomatic JIA, then control joints. Medial edge enhancement was a significant discriminator between symptomatic JIA TMJs and control joints (P = 0.0001), between symptomatic and asymptomatic JIA TMJs (P = 0.0003), and between asymptomatic JIA TMJs and controls (P = 0.0019). A shift in distribution of TMJ enhancement towards the medial edge that was seen uniquely in both asymptomatic and symptomatic JIA TMJs compared to control joints was found. This suggests a pattern of worsening medial edge inflammation with disease., (Copyright © 2020 International Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved.)

Complications in orthognathic surgery are commonly a result of inadequate preoperative planning and communication between the surgeon and orthodontist. Unfavorable outcomes can often be avoided when overall treatment goals along with a surgical and orthodontic plan are developed and agreed upon by the orthodontist, surgeon, and patient before the start of active tooth movement or any surgical procedures. Continuous evaluation of the patient's progress throughout treatment and subsequent communication between the surgeon and orthodontist are recommended to prevent frequent errors, such as inadequate dental decompensation, poor appliance selection or management, and occasional contraindicated orthodontic elastic traction or tooth movements., (Copyright © 2019 Elsevier Inc. All rights reserved.)

The aim of this retrospective cohort study was to determine the frequency and risk factors for cervical spine injury (CSI) in patients with midface fractures. Patients ≥18 years of age entered in the Massachusetts General Hospital Trauma Registry from 2007 to 2017 were identified. Those with a midface fracture, computed tomography and/or magnetic resonance imaging of the cervical spine, and complete medical records were included. There were 23,394 patients in the registry; 3950 (16.9%) had craniomaxillofacial fractures and 1822 (7.8%) had a CSI. Craniomaxillofacial fractures included fractures of the midface (n=2803, 71.0%), mandible (n=873, 22.1%), and midface plus mandible (n=274, 6.9%). The overall frequency of CSI in patients with midface fractures was 11.4% (350/3077). Patients with midface fractures had a higher risk for CSI compared to patients without a midface fracture (odds ratio 2.4, 95% confidence interval 2.1-2.4, P<0.001). In a multivariate model, nasal and orbital fractures, chest injuries, age, injury severity score, and motor vehicle crash or fall as the etiology were independent risk factors for CSI. Mortality was two times higher in subjects with CSI. Early and accurate diagnosis of CSI is a critical factor when planning the treatment of patients with these fractures., (Copyright © 2019 International Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved.)

In the present report, we trace the history of education and training of oral and maxillofacial surgeons as it has evolved from the mid-19th century to the present. We consider the effects of the discovery of ether anesthesia, the separation of medicine and dentistry, and other milestones such as antisepsis (Lister), antibiotics (Fleming) and surgical progress during wartime. The main emphasis, however, is on the background, development, and implementation of current dual-degree oral and maxillofacial surgery training programs, the first 5 programs, the pioneer advocates for this training, and progress and challenges for the future., (Copyright © 2019 American Association of Oral and Maxillofacial Surgeons. Published by Elsevier Inc. All rights reserved.)

The purpose of this study was to summarize the currently published cases of clear cell odontogenic carcinoma (CCOC). The PubMed and Springer databases were used to collect available reports, searching for 'clear cell odontogenic carcinoma', 'CCOC', or 'clear cell ameloblastoma'. The search resulted in 75 reports detailing 107 cases between 1985 and 2018. Clinically the tumor manifests as a swelling in the posterior mandible (n=46), anterior mandible (n=33), and maxilla (n=28). Radiological analysis of 85 cases typically showed a poorly defined expansive radiolucency (n=83). Of the 70 patients with symptoms reported, 44 specified a swelling, 11 tooth mobility, seven gingival/periodontal issues, five numbness, and three decreased jaw opening. One patient presented with a neck mass. The duration of symptoms prior to seeking care was specified for 52 patients: 2 months to 1 year for 34 patients, 1-2 years for seven, 2-4 years for two, 4-7 years for six, and 7-12 years for three. The incidence of recurrence appeared to be 38 of the 88 cases where recurrence was reported. CCOC can be distinguished from other oral cancers by its distinctive histology and immunohistochemical characteristics and less aggressive behavior. Currently, treatment should be early and aggressive resection with clear surgical margins and long-term follow-up. The overall goal is to collect a cohort of patients., (Copyright © 2019. Published by Elsevier Ltd.)

Purpose: Resident interview experiences are crucial for applicants when ranking programs. The purpose of the present study was to evaluate the interview experience among current oral and maxillofacial surgery (OMS) residents to determine the factors that influenced their selection and ranking of training programs and whether these experiences differed between women and men., Materials and Methods: We conducted a cross-sectional survey of OMS residents in 2018. The 12-question survey included demographics, reasons for selecting an interview and ranking programs, and positive and negative experiences during the interviews. Logistic regression models were constructed to evaluate the predictors of unprofessional or negative experiences., Results: A total of 1134 surveys were emailed, with 165 completed questionnaires (14.6%) returned by 35 women (21.2%) and 130 men (78.8%). Their average age was 30.8 years (range, 25 to 42). The racial/ethnic distribution was as follows: white, 75.8%; Asian, 15.8%; and other, 8.4%. Of the 165 respondents, 52% were in MD and 48% in non-MD programs. The top factors in selecting an institution at which to interview were clinical scope and volume, and the reason for ranking a program high was resident friendliness, which was similar among the female and male respondents. Unprofessional behavior or negative experiences were reported by 62 respondents (38%) and occurred by both faculty and residents and during both interviews and social events. Demeaning behavior toward the applicant, residents, and colleagues was the most common negative experience overall, with the women experiencing more gender-specific inappropriate behavior. Female respondents and those who were in dual-degree programs were 2.4 and 2.1 times more likely to experience unprofessional conduct than their peers, respectively (P = .03)., Conclusions: Female and male residents were influenced by the same factors when selecting interviews and ranking residency programs. Unprofessional and inappropriate conduct was reported by 38% of the respondents. Women and dual-degree respondents were 2.4 and 2.1 times more likely to experience unprofessionalism during interviews, respectively. This might have contributed to the low number of current female OMS residents., (Copyright © 2019. Published by Elsevier Inc.)

Purpose: A variety of linear (Harvold) and angular (Steiner) cephalometric analyses are used to identify the skeletal basis for a malocclusion; there is no universally accepted standard. The purpose of this study was to compare the concordance of Harvold and Steiner analyses with the clinicians' impression of maxillary and mandibular jaw position., Patients and Methods: We conducted a retrospective cohort study of patients who underwent orthognathic surgery at Massachusetts General Hospital from 2012 through 2016. Patients were included if they had symmetrical deformities not related to trauma, clefts, or syndromes; complete records; and a clinical diagnosis documented at initial consultation. The predictor variables were Harvold- and Steiner-derived diagnoses of jaw position (hypoplasia, neutral, or hyperplasia). The outcome variables were maxillary and mandibular clinical impressions (hypoplasia, neutral, or hyperplasia). The concordance, sensitivity, specificity, positive predictive value (PPV), and negative predictive value relative to clinical diagnoses were calculated., Results: During the study period 388 patients had orthognathic surgery and 222(112 females, mean age 26.4±9.9 years) met the inclusion criteria. Harvold and Steiner analyses were 82% and 33% concordant with the maxillary clinical impression, respectively (P < .001), and 62% and 52% concordant with the mandibular clinical impression, respectively (P = .044). Steiner analysis had greater concordance in females (P < .001). For maxillary hypoplasia, the maxillary unit length had a sensitivity of 87%, specificity of 36%, and PPV of 92% and the sell-nasion-A (SNA) point had 28%, 84%, and 93%, respectively. For mandibular hypoplasia, the mandibular unit length had a sensitivity of 52%, specificity of 96%, and PPV of 94% and the sell-nasion-B (SNB) point had 52%, 98%, and 97%, respectively. For mandibular hyperplasia, the mandibular unit length had a sensitivity of 46%, specificity of 93%, and PPV of 40% and the SNB point had 73%, 73%, and 23%, respectively., Conclusions: Harvold analysis was significantly more consistent with the clinical impression of the maxillary and mandibular sagittal position than Steiner analysis. Both analyses were highly specific and had high PPVs to confirm the clinical impression. Clinicians should consider incorporating Harvold analysis during treatment planning for orthognathic surgery., (Copyright © 2019 American Association of Oral and Maxillofacial Surgeons. Published by Elsevier Inc. All rights reserved.)

Maxillofacial central giant cell lesions (CGCLs) are often locally aggressive tumors in young patients that may be histologically very similar to or quite distinct when compared with giant cell tumors (GCTs) of long bones. It has been well established that GCTs express high levels of receptor activator of nuclear factor-kappa B ligand (RANKL) and are amenable to treatment with denosumab. To assess the predictive value of morphology, we evaluated CGCLs with GCT-like or non-GCT-like histology for RANKL expression by RNA in situ hybridization. Tumors were classified by clinical and radiographic criteria as aggressive or nonaggressive and histopathologically as resembling GCT or non-GCT-like. RNA in situ hybridization for RANKL mRNA was performed and scored semiquantitatively based on the magnification at which the signal was first detected. There were 17 patients (M:F=8:9) with a median age of 15 years. Nine patients were children under 18 years of age. In 10 patients, tumors were characterized as GCT-like and in 7, non-GCT-like; 6 occurred in the setting of a known associated syndrome. Of the sporadic tumors, 9/11 (82%) were classified as aggressive. Fifteen of 17 (88%) tumors strongly expressed RANKL (8/9 aggressive, 2/2 nonaggressive; 10/10 GCT-like and 5/7 non-GCT-like). Two patients with clinically aggressive CGCL, GCT-like histology and high tumor RANKL expression were identified as candidates for a trial of denosumab with notable clinical response. CGCLs demonstrate strong and diffuse RANKL mRNA expression in mononuclear stromal cells, regardless of histology or presence of an associated syndrome. Denosumab may be clinically beneficial in aggressive CGCLs.