Congenital Disseminated Pyogenic Granuloma: Characterization of an Aggressive Multisystemic Disorder.

OBJECTIVE: To describe the clinical, radiologic, and histopathologic features of "congenital disseminated pyogenic granuloma" involving various organs with high morbidity related to cerebral hemorrhagic involvement. STUDY DESIGN: We searched the database of the Vascular Anomalies Center at Boston Children's Hospital from 1999 to 2019 for patients diagnosed as having multiple vascular lesions, visceral vascular tumors, congenital hemangiomatosis, multiple pyogenic granulomas, or multiple vascular lesions without a definite diagnosis. A retrospective review of the medical records, photographs, histopathologic, and imaging studies was performed. Only patients with imaging studies and histopathologic diagnosis of pyogenic granuloma were included. RESULTS: Eight children (5 male, 3 female) had congenital multifocal cutaneous vascular tumors. Lesions also were found in the brain (n = 7), liver (n = 4), spleen (n = 3), muscles (n = 4), bone (n = 3), retroperitoneum (n = 3), and intestine/mesentery (n = 2). Less commonly affected were the spinal cord, lungs, kidneys, pancreas, and adrenal gland (n = 1 each). The mean follow-up period was 21.8 months. The cerebral and visceral lesions were hemorrhagic with severe neurologic sequelae. The histopathologic diagnosis was pyogenic granuloma with prominent areas of hemorrhage and necrosis. The endothelial cells had enlarged nuclei, pale cytoplasm and were immunopositive for CD31 and negative for D2-40 and glucose transporter 1. CONCLUSIONS: Congenital disseminated pyogenic granuloma is a distinct multisystemic aggressive disorder that primarily affects the skin, brain, visceral organs, and musculoskeletal system. Differentiation of this entity from other multiple cutaneous vascular lesions is critical because of possible cerebral hemorrhagic involvement.

Does Fee-for-Service Medicare Reimbursement Incentivize Volume Over Value Among Oral and Maxillofacial Surgeons?

PURPOSE: National health care payment models have begun transitioning from fee-for-service to value-based reimbursement because of criticism of the former incentivizing quantity over quality. However, there has yet to be an evaluation of the influence of fee-for-service incentives among oral and maxillofacial surgery services. This study characterized service intensity among oral and maxillofacial surgeons (OMSs) serving Medicare beneficiaries in 2017 to determine if higher Medicare income among OMSs arises from higher service intensity or a higher volume of patients treated. PATIENTS AND METHODS: This cross-sectional study was composed of Medicare Provider Utilization and Payment Data from 2017. Providers were included if their specialty type was listed as "maxillofacial surgery." The predictor variables included service intensity, defined as the number of health care services administered per Medicare beneficiary, and Medicare beneficiary volume. The primary outcome variable was Medicare income. Descriptive statistics and pair-wise comparisons were computed at an α level of .05. RESULTS: The analysis cohort was composed of 696 distinct OMSs. A total of 69,959 services were recorded for 53,245 Medicare beneficiaries, with a mean service intensity of 1.12 services per beneficiary. A statistically significant difference in service intensity was found between Medicare payment deciles (P = .002). The magnitude of this difference was less relative to the difference in all medical specialties. CONCLUSIONS: There is a statistically significant difference in service intensity between low and high earners in oral-maxillofacial surgery; however, the magnitude of the difference is unlikely to be clinically or economically meaningful. Variation in service intensity is lower in oral-maxillofacial surgery relative to all medical specialties in aggregate. Given the changing reimbursement landscape in medicine and surgery, it is important to evaluate existing billing practices within the specialty to advocate for the profession in discussions of payment reform and ensure that patients are receiving only necessary services.

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

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

What is the Prevalence of Undiagnosed Nevoid Basal Cell Carcinoma Syndrome in Children With an Odontogenic Keratocyst?

PURPOSE: Odontogenic keratocysts (OKCs) can occur in isolation or as part of nevoid basal cell carcinoma syndrome (NBCCS). Patients with NBCCS are younger at OKC diagnosis than those with nonsyndromic OKC (NS-OKC). The purpose of this study was to determine the prevalence of undiagnosed NBCCS in children who present with an OKC and to assess differences in demographic and presenting features between children with NBCCS and those with NS-OKC. MATERIALS AND METHODS: This study is a retrospective case series of children with an OKC presenting to Boston Children's Hospital (Boston, MA) from 2007 through 2018. To be included, patients had to be no older than 18 years and have a newly diagnosed OKC. Patients were excluded if they had previous or recurrent OKC or NBCCS diagnosis. Records were reviewed for age at presentation, gender, number and location of OKCs, treatment, recurrence, family history, and clinical features consistent with NBCCS. Descriptive data were summarized. RESULTS: The sample included 50 patients (27 boys) diagnosed with an OKC at a mean age of 11.7 years (range, 2 to 18 yr); 36% (n = 18) with NBCCS and 64% (n = 32) with NS-OKC. NBCCS diagnosis was made in 8 of 18 patients (44%) because of a family history at presentation, and in 10 patients (56%) the diagnosis was made by genetic testing or documentation of diagnostic criteria. Eight of 18 patients (44%) with undiagnosed NBCCS presented with a single OKC. Patients with NBCCS were significantly younger at presentation (NBCCS, 9.3 yr; NS-OKC, 13.0 yr), had more cysts at time of diagnosis (NBCCS, 1.7; NS-OKC, 1.0; P < .05), had more maxillary cysts (NBCCS, 13; NS-OKC, 11), and had a higher recurrence rate (P < .05). CONCLUSION: Given the prevalence of undiagnosed NBCCS in children with OKC, clinicians should have a low threshold for referral for complete examination or genetic testing in children with even a single OKC.

How Often Do Oral Maxillofacial Surgeons Lose Malpractice Cases and Why?

PURPOSE: Although oral maxillofacial surgeons (OMSs) carry high legal risk in malpractice lawsuits, data elucidating the reason behind those claims and their outcomes are scarce. The purpose of the present study was to characterize the trends, analyze the payouts, and determine the etiology of malpractice cases against OMSs. MATERIALS AND METHODS: A retrospective case series study was performed using the Westlaw database to access medical malpractice cases filed against OMSs from 1980 to 2017. The inclusion criterion was that the defendants had included an OMS. Two of us independently collected the demographic data, verdicts, payouts, and etiology of the litigation. RESULTS: The study sample included 183 cases adjudicated from 1980 to 2017. The results highlighted the temporal and geographic trends, payout information, and etiology of the cases. The total number of malpractice cases had decreased by 60% from 2011 to 2015 compared with the previous 5 years. The greatest incidence of malpractice cases filed per 100 practicing OMSs was in New York, followed by California and Massachusetts. Ruling in favor of the defendant OMS was noted in 55% of the cases, of the plaintiffs in 40% of the cases, and had reached a settlement before trial in 3% of the cases. In the cases in which the ruling had favored the plaintiff, the average payment was $812,449.08, with a median payment of $250,000.00 (range, $13,750.00 to $14,887,525.95). Extraction cases represented 53% of all malpractice litigations. Of these, 65% had been third molar extractions that had resulted in lingual nerve injury (26%), postoperative infection (17%), wrong site extractions (15%), and death or brain damage (10%), among other injuries. CONCLUSIONS: Approximately one half of the malpractice cases favored the defendant OMS. Most cases were third molar extractions resulting in injuries ranging from lingual nerve injury to death. Complementary data from insurance companies would be helpful to provide more specific analysis of the etiology and trends of the malpractice cases.

The Impact of Surgical Volume on Outcomes and Cost in Cleft Repair: A Kids' Inpatient Database Analysis.

BACKGROUND: Centralization of specialist services, including cleft service delivery, is occurring worldwide with the aim of improving the outcomes. This study examines the relationship between hospital surgical volume in cleft palate repair and outcomes. METHODS: A retrospective analysis of the Kids' Inpatient Database was undertaken. Children 3 years or younger undergoing cleft palate repair in 2012 were identified. Hospital volume was categorized by cases per year as low volume (LV; 0-14), intermediate volume (IV; 15-46), or high volume (HV; 47-99); differences in complications, hospital costs, and length of stay (LOS) were determined by hospital volume. RESULTS: Data for 2389 children were retrieved: 24.9% (n = 595) were LV, 50.1% (n = 1196) were IV, and 25.0% (n = 596) were HV. High-volume centers were more frequently located in the West (71.9%) compared with LV (19.9%) or IV (24.5%) centers (P < 0.001 for hospital region). Median household income was more commonly highest quartile in HV centers compared with IV or LV centers (32.3% vs 21.7% vs 18.1%, P < 0.001). There was no difference in complications between different volume centers (P = 0.74). Compared with HV centers, there was a significant decrease in mean costs for LV centers ($9682 vs $,378, P < 0.001) but no significant difference in cost for IV centers ($9260 vs $9682, P = 0.103). Both IV and LV centers had a significantly greater LOS when compared with HV centers (1.97 vs 2.10 vs 1.74, P < 0.001). CONCLUSIONS: Despite improvement in LOS in HV centers, we did not find a reduction in cost in HV centers. Further research is needed with analysis of outpatient, long-term outcomes to ensure widespread cost-efficiency.

Treatment of Craniosynostosis: The Impact of Hospital Surgical Volume on Cost, Resource Utilization, and Outcomes.

The authors aim to quantify the impact of hospital volume of craniosynostosis surgery on inpatient complications and resource utilization using national data. Children <12 months with nonsyndromic craniosynostosis who underwent surgery in 2012 at academic hospitals in the United States were identified from the Kids' Inpatient Database (KID) developed by the Healthcare Cost and Utilization Project (HCUP). Hospital craniosynostosis surgery volume was stratified into tertiles based on total annual hospital cases: low volume (LV, 1-13), intermediate volume (IV, 14-34), and high volume (HV, ≥35). Outcomes of interest include major complications, blood transfusion, charges, and length of stay (LOS). In 2012, 154 hospitals performed 1617 total craniosynostosis surgeries. Of these 580 cases (35.8%) were LV, 549 cases (33.9%) were IV, and 488 cases (30.2%) were HV. There was no difference in major complications between hospital volume tertiles (4.3% LV; 3.8% IV; 3.1% HV; P = 0.487). The highest blood transfusion rates were seen at LV hospitals (47.8% LV; 33.9% IV; 26.2%; P < 0.001). Hospital charges were lowest at HV hospitals ($55,839) compared with IV hospitals ($65,624; P < 0.001) and LV hospitals ($62,325; P = 0.005). Mean LOS was shortest at HV hospitals (2.96 days) compared with LV hospitals (3.31 days; P = 0.001); however, there was no difference when compared with IV hospitals (3.07 days; P = 0.282). Hospital case volume may be an important associative factor of blood transfusion rates, LOS, and hospital charges; however, there is no difference in complication rates. These results may be used to guide quality improvement within the surgical management of craniosynostosis.

Interest in global surgery rotations among oral and maxillofacial surgical residents in the United States.

PURPOSE: This study sought to assess interest in global surgery rotations among current United States (US)-based oral and maxillofacial surgery (OMS) residents. METHODS: An anonymous 23-question survey was distributed to 633 current OMS residents in the US to examine resident interest in global surgery rotations during residency. The primary outcome variable was resident interest in participating in global OMS rotations during residency training, whereas the primary predictor variable was the presence of residency faculty involved in global OMS work. Descriptive statistics were calculated for all study variables and univariate/multivariate logistic regression analyses were conducted to identify predictors of interest in global OMS rotations. RESULTS: A total of 120 residents with an average age of 30.4 ± 3.2 years responded to the survey. At present, 22 (18.5%) residents stated that their residency programs offer some sort of global OMS rotation and 21 (95.5%) of these claimed they were willing to participate in global OMS rotations at their residency program. Out of the residents who stated their program did not offer a global OMS rotation, 86 (87.8%) respondents stated they would be interested in adding a dedicated global OMS rotation to their residency curriculum. The presence of OMS residency faculty involved in global OMS work (p = 0.030) and a resident's willingness to dedicate vacation time to participate in a global surgery rotation (p = 0.005) were associated with increased interest in a global surgery rotation. CONCLUSION: The majority of respondents would welcome a dedicated global OMS rotation during their residency training.

The global reach of social media in oral and maxillofacial surgery.

PURPOSE: Social media use among oral and maxillofacial surgeons (OMSs) has grown in recent years, serving as an important resource for the dissemination of medical/surgical knowledge, research, education, diplomacy, and advocacy. However, no studies have attempted to characterize the global reach of social media in OMS. METHODS: This study examined the profile activity, content performance, and demographic characteristics of followers from a single OMS-related Instagram account. Variables assessed include the total number of followers since the account's inception, profile views over the selected time period, and unique media content posts, as well as likes, comments, saves, impressions, and reach for all media content posts. The top 45 countries, cities, and languages based on each follower's geolocation and user settings were also included. RESULTS: There were 9569 followers of which 6208 (64.9%) were listed as public accounts. Of the 6208 followers with public accounts, 2496 (40.2%) were female. The countries with the most followers included the United States (31.7%), India (12.5%), Malaysia (5.3%), Mexico (4.0%), and Pakistan (3.6%). The cities with the most followers included New York, New York (8.9%), Boston, Massachusetts (5.2%), Cairo, Egypt (4.3%), Santiago, Chile (3.7%), and Karachi, Pakistan (3.5%). CONCLUSION: OMS-related social media is uniquely positioned to facilitate global collaboration and augment the dissemination of surgical knowledge and expertise. This information is critical in understanding the distribution and demographics of the OMS workforce, trainees, and affiliates around the world.

A multi-disciplinary team approach to pediatric malignant mandibular tumors.

OBJECTIVE: Mandibular tumors in the pediatric population are rare. These malignancies are variable in their histology, and combined with their rarity, has made it difficult to describe their clinical course, and treatment guidelines. The aim of this paper is to describe the experience of Boston Children's Hospital, a pediatric tertiary referral center, with treating malignant mandibular malignancies, as well as provide multi-disciplinary team approach in managing this clinical entity. METHODS: A retrospective search was performed for mandibular malignancies in pediatric patients between 1995 and 2020 via the pathological database at Boston Children's Hospital. Only patients with malignant solid mandibular neoplasms were included, leaving 15 patients for final analysis. RESULTS: The median age at presentation was 10.1 ± 10.3 years. Nine of 15 patients (60%) presented with jaw mass which was the most common clinical presentation. The most commonly identified histological diagnosis was rhabdomayosarcoma and osteosarcoma (n = 4, 26% each). A mandibulectomy was performed in 12 (80%) cases. Reconstruction of the mandible was performed using a fibular free flap in 6 (40%) cases, and a plate in 3 (20%) cases. Mean follow-up was 4.6 ± 4.9 years. CONCLUSION: Malignant tumors most commonly present with a jaw mass, however asymptomatic and incidental presentations follow closely and pathologies can vary greatly. Surgical resection and reconstruction is often indicated, multidisciplinary tumor board review is required to determine when children are best treated with neo-/adjuvant treatment with chemo- and radiotherapy.

Customized repair of fractured mandibular reconstruction plates.

PURPOSE: To describe a novel technique using custom prostheses to repair fractured mandibular reconstruction plates spanning discontinuity defects. MATERIALS AND METHODS: This is a retrospective case series reviewing the design, rapid manufacture, and use of a novel method to repair fractured plates. Three patients who could not undergo autogenous bone grafting procedures or replacement of the entire plate for medical or socioeconomic factors were treated by this method. RESULTS: Three patients with fractured reconstruction plates were treated with a custom prosthesis engaging the reconstruction plate. Continuity and function were restored with a minimally invasive operation and short hospital stay. The custom prosthesis remained in place with stable occlusion in all 3 patients at a minimum of 9 months' follow-up. CONCLUSIONS: A technique using a custom prosthesis to quickly and less invasively restore continuity and function of the mandible after fracture of a reconstruction plate is described.

Assessment of global health programs at accredited dental schools in the United States.

PURPOSE: Oral diseases pose a serious global health challenge. As such, dental education must integrate global health with emphasis on scalable and sustainable solutions. The aim of our study was to capture the scope of global health programs offered to dental students in the United States and identify ways in which dental schools may modify their curricula to improve global health knowledge and accessibility for students. MATERIALS AND METHODS: For this cross-sectional, cohort study, the investigators sent a confidential and electronic survey to 213 faculty members at the 67 accredited dental schools in the United States. This voluntary survey was distributed in September and October of 2020 with answers being self-reported. RESULTS: A total of 40 different schools are represented in this study, representing 59.7% of accredited dental schools in the United States. Using descriptive statistics, the results present existing global health opportunities, barriers preventing schools from expanding global health education, and necessary resources that schools without a program need to establish one. CONCLUSION: Our results demonstrate promising findings, in relation to global health being a vital part of dental education. However, there is an urgent need for enhanced and more structured education in this space. As the global burden of oral disease contributes to the degree and severity of noncommunicable diseases worldwide, the development of a sustainable, preventive primary care approach must integrate oral health, making future dental professionals a crucial component of global health.

Calcifying synovial sarcoma of the tongue with SS18 rearrangement: a rare variant in a rare location.

Synovial sarcoma is a soft tissue malignancy harboring t(X;18) resulting in fusion of two genes SS8 (at 18q11) and SSX (1, 2 or 4 at Xp11) forming the gene fusion product SS18-SSX. It affects adults in their 3rd-4th decades, most frequently in the para-articular regions of the extremities. Less than 10% of the cases occur within the head and neck region and of these, 60% occur in the neck and only 10% occur in the oral cavity. We report a synovial sarcoma of the tongue in a 14-year-old female patient with unusual histology. The patient presented with a mass occupying most of the tongue with extension into the floor of mouth and the lingual gingiva of the anterior mandibular teeth. The tumor was composed of a highly cellular proliferation of spindle cells in a herringbone pattern with many small vessels but without glandular structures, and with extensive calcifications throughout the tumor. Tumor cells were positive for epithelial membrane antigen and transducin-like enhancer of split-1, and fluorescence in situ hybridization studies identified SS18 gene rearrangement. The patient was managed with two debulking procedures followed by chemoradiation and is currently alive with disease.

Desmoid tumors of the head and neck in the pediatric population: Has anything changed?

INTRODUCTION: Pediatric head and neck desmoid tumors are rare neoplasms that can cause significant morbidity due to infiltration of vital anatomic structures. The goal of this study is to review presentation, evaluation, and management of these tumors. METHODS: Retrospective study of children with head and neck desmoid tumors treated from 1999 to 2018 and literature review. RESULTS: 11 patients (5 boys, 6 girls) were included. Presentation included firm neck mass (n = 8), trismus (n = 2) and tongue lesion (n = 1). All patients had preoperative imaging with CT (n = 2), MRI (n = 1) or both (n = 8). Five patients underwent needle biopsy, five had open biopsy and one was diagnosed on pathology from primary excision. Seven patients were treated by primary surgical resection, with positive surgical margins in six cases due to proximity to vital neurovascular structures. None needed chemotherapy, had disease recurrence or progression. Three patients with unresectable disease were treated with chemotherapy. One patient was monitored with imaging without any treatment and did not have disease progression. Follow-up ranged from 6 months to 6 years (median 21 months). Ten patients (7 surgical, 2 chemotherapy, 1 observation) were either disease-free or had stable disease at last follow-up. CONCLUSION: Pediatric head and neck desmoid tumors, though rare and histologically benign, are locally infiltrative and aggressive. When feasible, surgical treatment results in good disease control despite positive margins. A balance between achieving negative margins and minimizing functional deficits should be considered. Chemotherapy can be successfully utilized in patients where surgery entails a high risk of morbidity and mortality.