Mandibular Osteomyelitis as the Earliest Clinical Manifestation of Maxillary Sinus Lymphoma.

Extranodal natural killer/T-cell lymphoma is a distinct subtype of non-Hodgkin lymphoma that originates from natural killer cells or cytotoxic T cells. Its diagnosis is challenging due to the rarity and lack of awareness, especially in cases where osteomyelitis of the jawbone is the initial symptom. This paper reports a case of extranodal natural killer/T-cell lymphoma presenting primarily with oral ulcers. Through analyzing the clinical and pathological characteristics, differential diagnosis, treatment and prognosis, and reasons for misdiagnosis of the disease, this study aims to provide references for clinical diagnosis and treatment.

Solitary primary intraosseous xanthoma of the mandible in a 15-year-old boy: a case report.

BACKGROUND: A xanthoma is a rare bone condition consisting of a predominant collection of lipid-rich, foamy histiocytes. The central xanthoma of the jaws is a unique benign tumor. CASE REPORT: A 15-year-old Caucasian male has been presented to our department. He had radiological changes in the area of the left mandibular angle, with an area of diffuse osteolysis of 3.0 cm by 2.0 cm. Computed tomography reveals an area of diffuse osteolysis that starts from the distal root of the lower second molar and reaches the ascending process. A bone biopsy was performed, which revealed a benign proliferative process composed of histiocytic cells involving and infiltrating trabecular bone in a background of loose fibrous connective tissue devoid of any other significant inflammatory infiltrate. The size of the formation was 2.9 cm by 2.0 cm. Immunohistochemical staining for CD68 was strongly positive and negative for S-100 and CD1a. From routine blood tests, cholesterol, triglycerides, and blood sugar are within normal values, which excludes systemic metabolic disease. Subsequent to the surgical intervention, the patient underwent postoperative assessments at intervals of 14, 30, 60 days, and a year later, revealing the absence of any discernible complications during the aforementioned observation periods. CONCLUSION: The diagnosis of primary xanthoma of the mandible is rare and can often be confused with other histiocytic lesions. A differential diagnosis should be made with nonossifying fibroma and Langerhans cell histiocytosis, as in our case. In these cases, immunohistochemistry with CD 68, S-100, and CD1a, as well as blood parameters, are crucial for the diagnosis.

Central giant cell granuloma in the posterior region of mandible mimicking a fibro-osseous lesion and hemangioma: a case report.

BACKGROUND: A central giant cell granuloma (CGCG) is a benign, proliferative, intraosseous, and non-odontogenic lesion occurring primarily in children and young adults. On the histological level, it is characterized by numerous multinucleated giant cells scattered randomly throughout a sea of spindle-shaped mesenchymal stromal cells which are dispersed throughout the fibrovascular connective tissue stroma containing areas of haemorrhage. When it comes to radiographic features, CGCG can have an array of variations, ranging from well-defined expansile lesions to ill-defined and destructive lesions, with or without expansion. CASE PRESENTATION: This case report reviews an 11-year-old Caucasian patient with a chief complaint of slow-growing swelling involving the right posterior mandibular region. The cone beam computed tomography (CBCT) revealed an ill-defined mixed lesion mimicking both fibro-osseous lesion and hemangioma. However, microscopic examination revealed multinucleated giant cells in a fibrous stroma suggestive of central giant cell granuloma. CONCLUSION: Our intent in reporting this case is to highlight the importance of thorough clinical, radiographical and histopathological examination for accurate diagnosis and therapeutic interventions as well as to emphasize the importance of taking different possibilities into consideration when examining bony swellings in the head and neck region.

Bony Canal Method of Dexamethasone Injections in Aggressive Form of Central Giant Cell Granuloma-Case Series.

Central Giant Cell Granuloma constitutes approximately 7% of benign tumors of the jaws. The aggressive form of CGCG clinically behaves like a classic semi-malignant neoplasm. In the literature, the suggested method of treatment of aggressive forms of CGCG is curettage or resection with the margin of 0.5 cm. Surgical treatment, especially in the developmental age, entails disturbances in the growth and differentiation of tissues and deforms and disturbs the functioning of the stomatognathic system. Alternative treatment methods of the CGCG presented in this article lead to the patient avoiding a mutilating procedure and improve their quality of life. The aim was to present alternative method of treatment of aggressive forms of Central Giant Cell Lesion of the jaws-injections of dexamethasone into the tumor mass through drilled bony canals. Here, we present the three cases of aggressive forms of CGCG of jaws treated with dexamethasone injections into the tumor mass. Two cases resulted in regression of the tumor, which was confirmed in histologic evaluation after remodeling surgery. Those two patients were uneventful and showed no signs of tumor recurrence at 8 and 9 years of thorough follow-up, respectively. The third patient was qualified for the mandible resection due to the enlargement of the lesion and destruction of the cortical bone. According to our observations, if the proper patient discipline, and thorough, careful clinical and radiological examinations are provided, the dexamethasone injections could be a recommended method of treatment of intraosseous giant cell granuloma. The indication is restricted to the cases with preserved bony borders despite deformation. Additionally, leaving vital teeth in the lesion is also possible.

Comparison of the histopathological characteristics of diffuse sclerosing osteomyelitis of the mandible, chronic suppurative osteomyelitis, and craniofacial fibrous dysplasia.

BACKGROUND: There are relatively few reports on the histopathological characteristics of diffuse sclerosing osteomyelitis of the mandible (DSOM), which is difficult to distinguish from chronic suppurative osteomyelitis (CSO) and craniofacial fibrous dysplasia (CFD). This study aimed to summarize and compare the histopathological characteristics of DSOM, CFD, and CSO. MATERIALS AND METHODS: In this study, hematoxylin and eosin-stained sections of patients with DSOM, CSO, and CFD at the Peking University Hospital of Stomatology from 2015 to 2020 were retrieved. The histopathological characteristics were summarized, including new bone formation, inflammatory cell infiltration, bone trabecular morphology, osteoclasts, sequestrum, bacterial mass, and calcified spherules, similar to cementicles. The histopathological characteristics of DSOM, CSO, and CFD were compared, and the results were statistically analyzed. RESULTS: In total, 50, 13, and 10 patients with DSOM, CSO, and CFD were included in this study, respectively. In terms of new bone formation, both DSOM and CSO showed reactive bone formation (p = 1), whereas CFD mainly showed fiber osteogenesis (p < 0.001). The inflammatory cells of DSOM were mainly lymphocytes and plasma cells, whereas those of CSO were mainly lymphocytes and neutrophils (p < 0.001), and there was usually no inflammatory cell infiltration in the CFD specimens (p < 0.001). DSOM, CSO, and CFD showed irregular bone trabeculae (p = 0.045, p = 0.703) and active osteoclasts (p1 = 0.189, p2 = 0.256). DSOM showed a small amount of bacterial mass but no sequestrum; neither of which was found in CFD (p = 1, p = 1), but it was common in CSO (p = 0.011 and p = 0.025). DSOM and CSO showed smooth and regular basophilic lines (p = 0.308), whereas CFD showed a rough and irregular basophilic line (p < 0.001). CONCLUSIONS: The histopathological characteristics of the three diseases were partly similar, but there were evident differences. The main differences are the type of new bone formation, types and distribution of inflammatory cells, and presence of sequestrum and bacterial masses. These differences will help clinicians diagnose DSOM.

Different presentations of the Buccal Bifurcation Cyst: A case series.

The buccal bifurcation cyst (BBC) is an uncommon odontogenic inflammatory cyst affecting the vestibular aspects of the first or second mandibular molar of pediatric patients. Its etiopathogenesis is not fully understood, but it is hypothesized that food and detritus impacting buccal periodontal pockets in titled tooth would be responsible for inflammation of the pericoronal tissues, leading to proliferation of epithelial rests and subsequent cystic formation. The true prevalence of the BBC is not known, but it is estimated to be less than 1% of all the inflammatory cysts. Most cases are unilateral but bilateral cases may account for up to 30% of all BBCs, which can generate confusion to unfamiliar clinicians. Maxillary cases are extremely uncommon, and to our knowledge, there are no cases published in the English literature. In this case series, we present five BBC cases; two unilateral, two bilateral, and one affecting the maxilla. We included clinical, imaging, and histopathological information to highlight the different presentations that this cyst might have, with the final aim to aid clinicians in its diagnosis and ultimately, its treatment.

Mandibular Buccal Bifurcation Cyst: Report of Two Cases.

Mandibular buccal bifurcation cyst is a rare inflammatory odontogenic cyst. We reported two cases who complained of painful swelling of extraoral soft tissue. Intraoral examination revealed the partially erupted mandibular first molar. Cone beam computed tomography showed a well-defined cystic lesion surrounding the first molar. Histopathologic images showed the cyst wall was infiltrated by a large number of plasma cells, neutrophils and eosinophils, and lined with a thin layer of non-keratinized stratified squamous epithelium. Finally, the two patients were diagnosed as mandibular buccal bifurcation cyst and treated with cyst enucleation and curettage.

Clinical and radiologic spectrum of glandular odontogenic cysts: A multicenter study of 92 cases.

OBJECTIVE: The purpose of this multicenter retrospective study was to report the clinical and radiologic features of 92 glandular odontogenic cysts (GOCs) diagnosed over a 20-year period. STUDY DESIGN: Histologically confirmed cases of GOC were retrospectively reviewed from 4 oral pathology laboratories in South Africa and Brazil to categorize the clinical and radiologic spectrum of GOCs. RESULTS: The mean age of patients was 46 years (range 17-87) with a male-to-female ratio of 1.2:1. GOCs had a mandibular predilection (68%), with 42% of all cases located anteriorly. Additionally, 42% of cases crossed the midline. Radiologically, most lesions were unilocular (53%) and uniformly radiolucent (97%), with well-demarcated borders (93%). Cortical expansion (62%), loss of cortical integrity (71%), and maxillary sinus (67%) and nasal cavity encroachment (72%) were common findings. Significant differences in lesions between the 2 countries were discovered in sex predilection, clinical signs and symptoms, and lesion locations within the mandible and maxilla. CONCLUSION: GOCs present with a wide spectrum of clinical and radiologic features, ranging from cysts with typical GOC-like presentations to more aggressive lesions. The need for advanced imaging in the surgical planning of GOCs exhibiting radiologic signs of aggression is justified based on the high recurrence rate.

The W9 peptide inhibits osteoclastogenesis and osteoclast activity by downregulating osteoclast autophagy and promoting osteoclast apoptosis.

The W9 peptide has been shown to act as a receptor activator for nuclear factor-κB ligand (RANKL) antagonist and tumor necrosis factor (TNF)-α antagonist, which can promote bone formation and inhibit bone resorption. Studies on the W9 peptide at the cellular level have mainly focused on osteoblasts, and little research on the mechanism by which the W9 peptide regulates osteoclasts has been reported, which was the aim of this work. In this study, a rat mandibular defect model was established in vivo and implanted with hydrogel containing the W9 peptide for 2 weeks and 4 weeks, and histochemical staining was used to evaluate the formation of new bone and the changes in osteoclasts. RAW264.7 cells were cultured in vitro for osteoclast induction, and different concentrations of W9 peptide were added. Tartrate resistant acid phosphatase staining, monodansylcadaverine staining, TdT-mediated dUTP Nick-End Labeling assay, real-time PCR and Western blot were used to detect osteoclast differentiation, autophagy and apoptosis. Our results showed that the W9 peptide could reduce osteoclastogenesis and osteoclast activity induced by RANKL, and these effects were partly due to the inhibition of osteoclast autophagy. On the other hand, the W9 peptide could promote mature osteoclast apoptosis, in which autophagy might play an antagonistic role. Taken together, these results suggest that the W9 peptide inhibits osteoclastogenesis and osteoclast activity by downregulating osteoclast autophagy and promoting osteoclast apoptosis. Our results will benefit the development and application of new small molecule peptides for the treatment of bone resorption diseases.

Central giant cell granuloma of the head & neck: A case report and systematic review.

PURPOSE: The purpose of this paper is to describe a recent case of central giant cell granuloma (CGCG) that rapidly progressed post corticosteroid treatment while also providing a review of the existing literature on CGCG of the head and neck (HNCGCG), with particular emphasis on extra-mandibular and maxillary cases. MATERIALS AND METHODS: The investigators designed and implemented a 32-year review of literature, using the online databases: PubMed, Google Scholar, Medline, and Proquest. The total number of cases analyzed was 55 (42 case reports; 3 case series; 8 comparative studies; 1 retrospective cohort). CASE PRESENTATION: We present a case of a CGCG in a 10-year old male. The lesion originated in the right anterior mandibular body and progressed after corticosteroid treatment. Diagnosis was made using a combination of imaging and histology. A timely debulking procedure of the hemi-mandible was performed and there was no recurrence of the lesion at follow up. RESULTS: The average age at the time of diagnosis of CGCG was 27.5 years. HNCGCG was most commonly detected in the jaw (43.1%), but was also found in the temporal bone (33.3%). The most frequently employed treatment modality was complete surgical excision (76.9%). 93.2% of patients were alive with no evidence of disease at follow-up, while 6.8% of patients exhibited recurrence at follow-up. The median follow up was 13 months. CONCLUSION: It is important for clinicians to recognize that CGCGs are capable of manifesting outside of the jaw. CGCG should be considered in the differential diagnosis of non-odontogenic radiolucent lesions, especially in young patients. CGCGs also need to be distinguished from brown tumor of hyperparathyroidism (BTH) and giant cell tumors, which are histologically similar.

Predictive factors of osteoradionecrosis necessitating segmental mandibulectomy: A descriptive study.

OBJECTIVE: The objective of this study was to assess characteristics of patients with mandibular osteoradionecrosis (ORN) of severity necessitating segmental mandibulectomy and osteocutaneous free flap reconstruction. STUDY DESIGN: This study is a retrospective review of patients who underwent free flap reconstruction of the mandible at the UCLA Medical Center between January 2016 and February 2020 secondary to ORN. RESULTS: Twenty-nine charts with detailed dental and medical records were identified. Hypertension was reported in 14 of 29 patients, diabetes in 2 of 29, osteoporosis in 2 of 29, antiresorptive use in 3 of 29, tobacco use in 15 of 29, and alcohol use in 19 of 29. Twenty-three patients initially had stage III-IV cancer. The median radiation dose was 68 Gy and median time to ORN was 5.2 years. Chemotherapy was given in 21 patients and 4 had previous mandibular surgery. Twelve of 29 patients had surgical procedures identified as the causative factor and 17 of 29 occurred spontaneously. Median decayed, missing, and filled teeth score was 17 and 17 of 29 patients had grade II-IV periodontitis. Periodontitis was present in 8 of 17 of spontaneous and 1 of 12 of surgery cases. Twenty-five of 29 cases occurred in the same oral sextant as the tumor. CONCLUSION: Severe ORN occurred at doses >60 Gy in most cases. Location of the primary tumor was predictive of site of ORN and only molars were involved when precipitated by tooth extraction. Risk of ORN persists indefinitely.

Central mucoepidermoid carcinoma arising directly from a glandular odontogenic cyst of the mandible: a case report.

BACKGROUND: Central mucoepidermoid carcinoma (MEC) is a rare salivary gland tumor that affects the jawbone. Glandular odontogenic cyst (GOC) is also a rare odontogenic developmental cyst with glandular differentiation. GOC shares some histological features with central MEC, and a pre-existing GOC can develop into central MEC. Here, we present a rare case of central MEC developed directly from a pre-existing GOC of the mandible. CASE PRESENTATION: A 67-year-old Japanese man presented with a cystic lesion in the right third molar region. Histologically, the biopsy specimen demonstrated both typical findings of a GOC component lined with non-keratinized squamous epithelium and a recognizable component of central MEC consisting of polycystic nests with mucous cells, intermediate cells, and epidermoid cells in the cyst wall. The results from the immunohistochemistry for cytokeratin (CK) profiling demonstrated that, while both central MEC and GOC expressed CKs 7, 14, 18, and 19, CK13 was interestingly exclusively expressed in GOC. Fluorescence in-situ hybridization (FISH) revealed the rearrangement of the Mastermind like (MAML)-2 gene in both the MEC and GOC components. CONCLUSIONS: Our case suggests that central MEC and GOC may be in the same spectrum of diseases caused by the rearrangement of the MAML-2 gene. However, given that the expression profile of CK13 was completely different between central MEC and GOC, they can be considered as separate tumors. Overall, we demonstrated a rare case in which central MEC may have originated directly from the GOC.

Sine Qua Non: Dentigerous Cyst.

Dentigerous cysts, also known as follicular cysts, are among the most common developmental cysts of the gnathic bones. The majority of cases are clinically asymptomatic and discovered incidentally on panographic radiographs during routine dental care. The cyst appears as a radiolucency, classically unilocular, associated with the crown of an unerupted or impacted tooth. Usually diagnosed in the 2nd-3rd decade, third molars of the mandible are the most commonly affected teeth. Histologically, dentigerous cysts demonstrate a fibrous or fibromyxoid connective tissue wall lined by squamous epithelium, classically lacking rete ridges. Inflammation may introduce histologic changes, however. The differential diagnosis includes hyperplastic dental follicle, periapical or radicular cyst, unicystic ameloblastoma, odontogenic keratocyst, and other odontogenic cysts and tumors. While the findings are generally classic and pose no diagnostic dilemma, the diagnosis is best made in the context of the appropriate clinical and radiographic setting. Submitted tissue with a lack of history, to include a detailed relationship with the affected tooth, may result in misdiagnosis and subsequent confusion for the clinician. So, despite its simple features, dentigerous cysts are not uncommonly mischaracterized. Therefore a review of a classic case of dentigerous cyst is presented.

Developmental Odontogenic Lesions Associated with the Crown of an Impacted Tooth: A Guide to the Distinct Histologic Features Required for Classification.

Odontogenic cysts are categorized as inflammatory and developmental. Of the developmental odontogenic cysts, the dentigerous cyst is the most common and by definition is attached to the cervical region of an unerupted tooth. The cyst envelops the crown forming a sac. However, there are other developmental cysts, and rarely, odontogenic tumors, that can have a similar clinical and radiographic presentation as dentigerous cyst, including odontogenic keratocyst, orthokeratinized odontogenic cyst and ameloblastoma, unicystic type. Understanding the key histologic differences of these cysts will aid the pathologist to correctly diagnose these lesions, ensuring appropriate clinical management.

Local treatment of experimental mandibular osteomyelitis with an injectable biomimetic gentamicin hydrogel using a new rabbit model.

Mandibular osteomyelitis (OM) is a challenging disease. Our objective was to assess a new OM model in rabbits induced by arsenic trioxide and to assess the efficacy of local treatment of OM using injectable gentamicin-collagen hydrogels (GNT-COLL). OM was induced unilaterally by controlled confinement of arsenic trioxide paste to the root canal of lower incisors of rabbits, while OM progression was characterized for 16 weeks. On the other hand, two injectable COLL hydrogels functionalized with GNT were prepared and characterized for physicochemical properties; a simple GNT-COLL and a nanohydroxyapatite (nHA)- loaded hydrogel (GNT-COLL/nHA). The two hydrogels were evaluated to treat OM model, while a multidose intramuscular GNT solution served as positive control. Outcomes were assessed by standard methods at 4 and 12 weeks post-surgery. The clinical, radiographical, and histopathological findings provided evidence for the validity of the arsenic-induced OM. The results demonstrated that a single intra-lesional injection of the two hydrogels was more suppressive to OM compared to multidose systemic GNT. The composite GNT-COLL/nHA hydrogel proved to induce early preservation of alveolar bone (ridge) length and higher amount of bone area\total area at 4 weeks (40.53% ± 2.34) followed by GNT-COLL (32.21% ± 0.72). On the other hand, the positive control group revealed the least ridge length and bone area\total area (26.22% ± 1.32) at 4 weeks. Both hydrogels successfully arrested OM with no signs of recurrence for up to 12 weeks. Therefore, results support the greater advantages of the composite hydrogel as an osteogenic/antibiotic delivery system in OM treatment.

Unique case of glandular odontogenic cyst showing mucoepidermoid carcinoma-like islands in cystic wall.

Glandular odontogenic cyst (GOC) is a very rare jaw cyst accounting for 0.2% of all odontogenic cysts. It presents usually in adults with a slight male predominance. It shows radiological, histopathological and even immunohistochemical overlap with low grade intraosseous mucoepidermoid carcinoma (MEC) but their distinction is crucial. A 57-year-old woman with bilocular radiolucency in the anterior mandible crossing the midline is described here. Microscopy features were consistent with glandular odontogenic cyst but multiple MEC-like islands were seen in the capsule, creating a diagnostic head trip with low grade intraosseous MEC. However, the absence of cellular atypia and epidermoid and intermediate cells led to a final diagnosis of GOC, with close follow-up of the patient recommended. This rare finding shows the relation between GOC and MEC or the origin of MEC from GOC.

Glandular Odontogenic Cyst with Metaplastic Cartilage: Report of an Unusual Case and Literature Review.

Glandular odontogenic cysts are rare odontogenic cysts with a wide range of histopathological features. In this paper we describe the clinical and pathological features of an unusual case of a glandular odontogenic cyst with metaplastic cartilage. The previous literature of odontogenic cysts presenting with metaplastic cartilage is reviewed alongside a discussion of the differential diagnoses. To our knowledge this is the first reported case of a glandular odontogenic cyst with metaplastic cartilage.

Concurrent High Condylectomy and Orthognathic Surgery for Treatment of Patients With Unilateral Condylar Hyperplasia.

BACKGROUND: Facial asymmetry from unilateral condylar hyperplasia (UCH) may be definitively treated in the presence of active disease (with high condylectomy and concurrent orthognathic surgery) or after waiting for disease inactivity (orthognathic surgery alone). There is currently no consensus on the standard of care. In this study, we sought to compare functional and esthetic outcomes, as well as treatment duration, between these 2 management options. METHODS: Patients who underwent treatment for UCH were identified through retrospective review. Pre- and postoperative 3-dimensional (3D) images were obtained. Short- and long-term operative outcomes of those treated during the active (group 1) were compared to those treated in the inactive phase (group 2). Total treatment time, operative time, and length of hospital stay were evaluated. Facial asymmetry was also assessed by laypersons using a Likert scale. RESULTS: Fifteen patients (mean 25.6 years, range 14-56) were included: 6 in group 1 and 9 in group 2. All surgical outcomes were statistically independent of procedure type. Treatment time was significantly longer in the group 2 (P = 0.03). Both groups demonstrated significant improvement in facial asymmetry scores postoperatively with no significant difference in pre- or postoperative asymmetry between groups (P = 0.64). CONCLUSIONS: In patients with active UCH, high condylectomy and orthognathic surgery is a procedure that restores facial symmetry and improves jaw function while halting mandibular growth. Good esthetic and functional outcomes, as well as reduced treatment time and disease burden, support the use of this treatment option for this population.