Computed tomographic imaging features of maxillary and mandibular melanotic neuroectodermal tumor of infancy: Report of 2 cases and systematic review.

OBJECTIVE: The aim of this systematic review was to determine the computed tomographic (CT) imaging characteristics of maxillary and mandibular melanotic neuroectodermal tumor of infancy (MNTI). Two cases from our institution were also presented. STUDY DESIGN: Full-text case reports and case series of histopathologically proven gnathic MNTI with CT figures of diagnostic quality were searched in PubMed, Scopus, Web of Science, Ovid, and Google Scholar databases from July 2021 to February 2022. Descriptive statistics were used to determine the frequency of each CT feature of gnathic MNTI. RESULTS: Fifty-two published studies met the eligibility criteria, providing a total of 53 maxillary and mandibular MNTIs for analysis. In order of frequency, the CT features of gnathic MNTI that were present in over half of the study sample were bone expansion (53, 100%), a well-defined periphery (49, 92.5%), tooth displacement (45, 84.9%), and a bilocular radiolucent internal pattern (32, 60.4%). CONCLUSIONS: The bilocular radiolucent internal pattern has not been recognized as a common CT feature of gnathic MNTI. When associated with a well-defined, expansile mass in the infantile maxilla or mandible, this imaging characteristic can support a radiologic interpretation of MNTI.

Adenoidal-Nasopharyngeal Ratio in Healthy Adults on Cone Beam Computed Tomography.

OBJECTIVE: This study aimed to quantify the adenoidal-nasopharyngeal ratio (ANR) in a cohort of healthy adults on cone beam computed tomography (CT) using the Fujioka method, which is a reproducible measure of adenoid size and nasopharyngeal patency. METHODS: Electronic health records and maxillofacial cone beam CT in 202 consecutive patients aged 16 years and older were retrospectively reviewed. Patients with a history of adenoidectomy, sinonasal disease, lymphoproliferative disorders, and cleft palate were excluded from the study. The midsagittal reconstructed cone beam CT image was used to determine the ANR. Statistical analysis was conducted using 1-way analysis of variance. RESULTS: Of the 202 subjects, 131 were female and 71 were male. The mean ± SD subject age was 45.43 ± 20.79 years (range, 16-91 years). The mean ± SD ANR in all subjects was 0.22 ± 0.13 (range, 0.03-0.75) and in each decade of adult life was as follows: younger than 21 years, 0.39 ± 0.12; 21 to 30 years, 0.29 ± 0.11; 31 to 40 years, 0.21 ± 0.09; 41 to 50 years, 0.20 ± 0.07; 51 to 60 years, 0.16 ± 0.10; 61 to 70 years, 0.13 ± 0.05; 71 to 80 years, 0.12 ± 0.05; 81 to 90 years, 0.11 ± 0.04; and 91 years or older, 0.10 ± 0. The differences in mean ANR among the age subgroups were statistically significant ( P < 0.001). CONCLUSIONS: The mean ANR gradually decreased from 0.39 in the second decade of life to 0.16 in the sixth decade of life and plateaued at approximately 0.10 thereafter.

Targeting the endothelin axis as a therapeutic strategy for oral cancer metastasis and pain.

Metastasis reduces survival in oral cancer patients and pain is their greatest complaint. We have shown previously that oral cancer metastasis and pain are controlled by the endothelin axis, which is a pathway comprised of the endothelin A and B receptors (ETAR and ETBR). In this study we focus on individual genes of the pathway, demonstrating that the endothelin axis genes are methylated and dysregulated in cancer tissue. Based on these findings in patients, we hypothesize that ETAR and ETBR play dichotomous roles in oral carcinogenesis and pain, such that ETAR activation and silenced ETBR expression result in increased carcinogenesis and pain. We test a treatment strategy that targets the dichotomous functions of the two receptors by inhibiting ETAR with macitentan, an ETAR antagonist approved for treatment of pulmonary hypertension, and re-expressing the ETBR gene with adenovirus transduction, and determine the treatment effect on cancer invasion (i.e., metastasis), proliferation and pain in vitro and in vivo. We demonstrate that combination treatment of macitentan and ETBR gene therapy inhibits invasion, but not proliferation, in cell culture and in a mouse model of tongue cancer. Furthermore, the treatment combination produces an antinociceptive effect through inhibition of endothelin-1 mediated neuronal activation, revealing the analgesic potential of macitentan. Our treatment approach targets a pathway shown to be dysregulated in oral cancer patients, using gene therapy and repurposing an available drug to effectively treat both oral cancer metastasis and pain in a preclinical model.

A radiographic study of permanent molar development in wild Virunga mountain gorillas of known chronological age from Rwanda.

OBJECTIVES: While dental development is important to life history investigations, data from wild known-aged great apes are scarce. We report on the first radiographic examination of dental development in wild Virunga mountain gorillas, using known-age skeletal samples recovered in Rwanda. MATERIALS AND METHODS: In 43 individuals (0.0-14.94 years), we collected radiographs of mandibular molars, and where possible, cone beam CT scans. Molar crown and root calcification status was assessed using two established staging systems, and age prediction equations generated using polynomial regression. Results were compared to available data from known-age captive and wild chimpanzees. RESULTS: Mountain gorillas generally fell within reported captive chimpanzee distributions or exceeded them, exhibiting older ages at equivalent radiographic stages of development. Differences reflect delayed initiation and/or an extended duration of second molar crown development, and extended first and second molar root development, in mountain gorillas compared to captive chimpanzees. However, differences in the duration of molar root development were less evident compared to wild chimpanzees. DISCUSSION: Despite sample limitations, our findings extend the known range of variation in radiographic estimates of molar formation timing in great apes, and provide a new age prediction technique based on wild specimens. However, mountain gorillas do not appear accelerated in radiographic assessment of molar formation compared to chimpanzees, as they are for other life history traits. Future studies should aim to resolve the influence of species differences, wild versus captive environments, and/or sampling phenomena on patterns observed here, and more generally, how they relate to variation in tooth size, eruption timing, and developmental life history.

Mandibular changes on panoramic imaging after head and neck radiotherapy.

OBJECTIVES: Changes to the radiographic appearance of the jaws after head and neck radiotherapy have not been thoroughly characterized. This retrospective study examines changes to the appearance of the mandible on panoramic images following intensity-modulated radiotherapy (IMRT) and relates these changes to medical co-morbidities and radiation dose. STUDY DESIGN: The medical and dental charts, and panoramic images of 126 patients who received IMRT at the Princess Margaret Hospital between January 1, 2005, and December 31, 2008, were analyzed independently by three observers. RESULTS: Of the 126 patients, 75 (60%) had post-IMRT changes, as seen on panoramic images; most, 66 (88%), consisted of widened periodontal ligament space (WPLS). The median time to WPLS was 29 months after IMRT. Female gender and radiation dose correlated with decreased time to WPLS. CONCLUSIONS: These results indicate that WPLS is a common radiographic sequela after head and neck radiotherapy, underscoring its clinical significance as a reliable marker of irradiated bone. Furthermore, this type of WPLS needs to be differentiated from odontogenic inflammatory disease and cancer recurrence to avoid unnecessary treatment that may precipitate osteoradionecrosis.

Does Fluorodeoxyglucose Positron Emission Tomography With Computed Tomography Facilitate Treatment of Medication-Related Osteonecrosis of the Jaw?

PURPOSE: There is considerable controversy over the treatment of medication-related osteonecrosis of the jaw (MRONJ) and growing interest and debate related to the timing, type, technique, and goals of surgical intervention. The specific aim was to evaluate the predictive value of fluorodeoxyglucose (FDG) positron emission tomography (PET) with computed tomography (CT) on healing outcomes in patients undergoing surgery for MRONJ of the mandible. MATERIALS AND METHODS: A retrospective cohort study of 31 patients with 33 MRONJ lesions of the mandible who had undergone surgery using FDG PET-CT was conducted. Data were collected on FDG uptake patterns, healing, follow-up, demographics, lesion characteristics, antiresorptive therapy, and adjunctive therapy. Panoramic and/or periapical radiographs were used to identify non-restorable teeth and PET-CT images were used to identify sequestra and FDG uptake. Above the mandibular canal, surgery consisted of marginal resection and/or debridement of clinically involved bone and exposure of clinically uninvolved bone identified by FDG uptake. Below the mandibular canal, mobile segments of bony sequestra were removed, but areas of clinically uninvolved bone with FDG uptake were not. Patients who did not heal underwent segmental resection and reconstruction with rigid fixation and a local or regional soft tissue flap or free fibular flap. The primary predictor variable was the FDG uptake pattern for each patient. The outcome variable was postoperative healing defined by mucosal closure without signs of infection or exposed bone at the time of evaluation. RESULTS: Two risk groups were identified based on FDG uptake pattern. The low-risk group, type A, included 22 patients with activity limited to the alveolus, torus, and/or basal bone superior to the mandibular canal. The high-risk group, type B, included 11 patients with type A FDG activity with extension inferior to the mandibular canal. Treatment of type A MRONJ lesions was more successful than treatment of type B MRONJ lesions (100 vs 27%; P < .001). Seven of the type B failures were successfully retreated by segmental resection and reconstruction (1 patient refused further treatment). CONCLUSION: These results showed that low-risk FDG PET-CT findings predicted successful healing with surgery above the mandibular canal. In contrast, high-risk FDG findings were associated with a greater than 50% risk of failure for treatment that extended below the mandibular canal. Although these failures suggest that FDG uptake indicates infected tissue, further research is needed to identify which high-risk patients are most likely to benefit from a conservative treatment protocol.

Salivary duct carcinoma ex pleomorphic adenoma of the palate: a case report.

Carcinoma ex pleomorphic adenoma is a rare malignancy of the head and neck, particularly in the minor salivary glands. Most cases arise in the major salivary glands, most commonly in the parotid gland, followed by the submandibular gland. The malignant component of the tumor varies, but can be salivary duct carcinoma, adenoid cystic carcinoma, mucoepidermoid carcinoma, squamous cell carcinoma, or adenocarcinoma, not otherwise specified. Primary salivary duct carcinoma is also a rare malignancy of the head and neck. Similar to carcinoma ex pleomorphic adenoma, it is more common in the major salivary glands, with the parotid gland accounting for 88% and the submandibular gland for 10% of cases. To date, only 25 known cases of primary salivary duct carcinoma arising in the minor salivary glands have been documented, with most arising in the palate. Salivary duct carcinoma ex pleomorphic adenoma of the minor salivary glands appears to be even rarer. Our case of salivary duct carcinoma ex pleomorphic adenoma of the palate is the first complete report, to our knowledge, in the English-language scientific literature.

Fluorodeoxyglucose positron emission tomography with computed tomography detects greater metabolic changes that are not represented by plain radiography for patients with osteonecrosis of the jaw.

PURPOSE: Imaging is important to identify subclinical changes and for treatment planning in patients with osteonecrosis of the jaw (ONJ) exposed to antiresorptive therapy. The aim of this study was to compare the findings at radiography with those at fluorodeoxyglucose (FDG) positron emission tomography (PET) with computed tomography (CT) for patients with ONJ related to antiresorptive therapy. MATERIALS AND METHODS: A cross-sectional retrospective analysis of patients with clinically identified ONJ lesions of the mandible was performed. Two imaging modalities were evaluated for each patient: plain radiography (ie, panoramic or periapical) and FDG PET/CT with 1-mm sections. Outcome variables for the radiographic findings were osteolytic and osteosclerotic bone changes. Outcome variables for FDG PET/CT images were localization of FDG uptake. Maximum standard uptake values (SUVmax) of abnormal FDG jaw uptake were recorded, in addition to the mean SUV of the contralateral normal mandible, and used to calculate the target-to-background ratio. Radiographic changes and FDG uptake were classified as local (ie, corresponding to exposed cortical bone) or diffuse (ie, local changes and changes extending beyond the margins of exposed bone) for each imaging technique. Local and diffuse changes detected by each imaging modality were described and the difference in detection was compared with the McNemar test. RESULTS: Twenty-three patients with 25 clinically identified ONJ lesions were analyzed using radiography and FDG PET/CT. Differences were found in how radiography and FDG PET/CT detect local and diffuse changes associated with ONJ. Radiography showed local changes in 17 patients (68%), diffuse changes in 3 patients (12%), and no changes in 5 patients (20%), whereas FDG PET/CT imaging showed local changes in 17 patients (68%) and diffuse changes in 8 patients (32%). The McNemar test indicated that FDG PET/CT imaging was less likely to miss a lesion (P < .001). Mean SUVmax was 6.59, and the mean target-to-background ratio was 5.37. CONCLUSION: The results of this study show that FDG PET/CT detects local and diffuse metabolic changes that may not be represented by plain radiography for patients with ONJ related to antiresorptive therapy. The target-to-background ratio allowed the discrimination between ONJ lesions and background changes. Future studies are necessary to determine whether FDG PET/CT can determine risk and facilitate management of ONJ.

Labial salivary gland involvement in neonatal hemochromatosis: a report of 2 cases and review of literature.

Neonatal hemochromatosis (NH) is a severe disease of fetal or perinatal onset, in which iron deposition occurs within hepatic and extrahepatic sites without involving the reticuloendothelial system. Labial minor salivary gland biopsy has been suggested as a diagnostic adjunct in patients suspected of having NH, as hemosiderin accumulates in acinar epithelial cells. Prior to this salivary gland pathology, a diagnosis of NH was often delayed, rendered only after the usual causes of neonatal liver failure had been excluded. Recent studies have shown that early diagnosis and treatment can improve survival. Few cases of salivary gland hemosiderosis in NH have been reported in the literature. A positive finding of salivary gland siderosis on biopsy will expedite care. We report 2 cases of NH, of which a labial salivary gland biopsy supported the diagnosis. The clinical and histological features are presented. The NH literature pertaining to labial salivary gland pathology is reviewed.