UNASSIGNED: While overall head and neck cancer incidence decreases due to reduced tobacco and alcohol consumption, the incidence of HPV negative oral cavity squamous cell carcinoma (SCC) is raising in several industrialized countries, especially in non-smoking and non-drinking patients.
UNASSIGNED: We document a case of gingiva SCC in a 56 years old never-smoker patient reporting low alcohol consumption and unusual occupational solvent exposure. The HPV-negative lesion was surgically removed in 2018, and the patient remains in complete remission 4 years after recurrent surgery in 2019. In 2021, the patient was referred to the occupational cancer consultation. The patient worked as screen printer for 18 years. He reported mouth siphoning every 2-3 days to transfer organic solvents (mainly aromatic hydrocarbons and ketones) from containers into smaller recipients, with regular passage of solvents into his mouth.
UNASSIGNED: According to the literature, the frequency of solvent siphoning using mouth is likely to be underestimated. While our review did not find studies reporting longterm consequences to the oral cavity of mouth siphoning, current evidence supports a positive association of upper aero digestive tract SCC with occupational exposures to organic solvents and printing processes. In absence of major extraprofessional factors, the HPV-negative gingiva SCC of this patient might be attributable to the regular occupational oral solvent exposure. While the available evidence remains limited to formally establish a causal relationship, clinicians should investigate this hazardous work practice in patients with OSCC and history of solvent exposures.

BACKGROUND: Peripheral ossifying fibroma is a nonneoplastic inflammatory hyperplasia that originates in the periodontal ligament or periosteum in response to chronic mechanical irritation. Peripheral ossifying fibroma develops more commonly in young females as a solitary, slow-growing, exophytic nodular mass of the gingiva, no more than 2 cm in diameter. While various synonyms have been used to refer to peripheral ossifying fibroma, very similar names have also been applied to neoplastic diseases that are pathologically distinct from peripheral ossifying fibroma, causing considerable nomenclatural confusion. Herein, we report our experience with an unusual giant peripheral ossifying fibroma with a differential diagnostic challenge in distinguishing it from a malignancy.
METHODS: A 68-year-old Japanese male was referred to our department with a suspected gingival malignancy presenting with an elastic hard, pedunculated, exophytic mass 60 mm in diameter in the right maxillary gingiva. In addition to computed tomography showing extensive bone destruction in the right maxillary alveolus, positron emission tomography with computed tomography revealed fluorodeoxyglucose hyperaccumulation in the gingival lesion. Although these clinical findings were highly suggestive of malignancy, repeated preoperative biopsies showed no evidence of malignancy. Since even intraoperative frozen histological examination revealed no malignancy, surgical resection was performed in the form of partial maxillectomy for benign disease, followed by thorough curettage of the surrounding granulation tissue and alveolar bone. Histologically, the excised mass consisted primarily of a fibrous component with sparse proliferation of atypical fibroblast-like cells, partly comprising ossification, leading to a final diagnosis of peripheral ossifying fibroma. No relapse was observed at the 10-month follow-up.
CONCLUSIONS: The clinical presentation of giant peripheral ossifying fibromas can make the differential diagnosis from malignancy difficult. Proper diagnosis relies on recognition of the characteristic histopathology and identification of the underlying chronic mechanical stimuli, while successful treatment mandates complete excision of the lesion and optimization of oral hygiene. Complicated terminological issues associated with peripheral ossifying fibroma require appropriate interpretation and sufficient awareness of the disease names to avoid diagnostic confusion and provide optimal management.

Neonatal alveolar lymphangioma (NAL) is a rare benign condition most often reported in the oral cavity of neonates of African descent.
The authors present a case report of bilateral NAL, including follow-up images at 6 months and 17 months. The clinical differential diagnosis includes dental lamina cyst, hemangioma, congenital epulis of the neonate, and melanotic neuroectodermal tumor of infancy. Key differences to assist in making the distinction are described.
The distinguishing characteristics of NAL make a clinical diagnosis possible. Accurate clinical recognition of NAL is essential because biopsy is not typically indicated. Because this is a clinical diagnosis, follow-up for 1 year or until complete resolution; biopsy is suggested if clinically indicated for residual or recurrent lesion.

BACKGROUND: Odontogenic fibroma (OF) is a rare benign tumor of mesenchymal odontogenic tissue with variable global incidence. Two variants comprising extraosseous /peripheral (POF) and intraosseous /central (COF) have been identified based on the anatomical origin of the tumor. OF isslow growing, and often asymptomatic with characteristic radiographic appearance of a unilocular or multilocular cyst frequently associated with unerupted or displaced teeth while histologically, varying amount of inactive odontogenic epithelium embedded in a mature relatively dense collagenous stroma is diagnostic. The absence of odontogenic epithelium does not exclude the diagnosis of odontogenic fibroma.
METHODS: A 15 months old female presented with 3 months history of left lower jaw swelling which showed aggressive growth following incisional biopsy. Parents said lesion was noticed for the first time as pinpoint on the submandibular area and was too small to warrant any worries. Initial radiograph revealed absence of bony involvement but the second showed evidence of bony involvement. An impression of soft tissue sarcoma was made and incisional biopsy was sent for pathological diagnosis which revealed peripheral odontogenic fibroma. The rapidity of growth following incisional biopsy bellied a benign lesion.
CONCLUSIONS: An aggressive odontogenic fibroma with multifocal points in a 15months old girl.
BACKGROUND: Le fibrome odontogène (FO) est une tumeur bénigne rare du tissu odontogène mésenchymateu dont l’incidence globale est variable. Deux variantes comprenant des fibromes extra-osseux /périphérique (POF) et intra-osseuse / centrale (COF) ont été identifiés en fonction de l’origine anatomique de la tumeur. La POF est à croissance lente, et souvent asymptomatique avec l’apparence radiographique caractéristique d’un kyste uniloculaire ou multiloculaire fréquemment associé à des dents non érigées ou déplacées alors que histologiquement, une quantité variable d’épithélium inactif noyé dans un stroma collagène mature relativement dense est le diagnostic. L’absence d’épithélium odontogène n’exclut pas le diagnostic de fibrome odontogène.
UNASSIGNED: Une femme de 15 mois a présenté un gonflement de la mâchoire inférieure gauche depuis 3 mois de gonflement de la mâchoire inférieure gauche qui a montré une croissance agressive après une biopsie incisionnelle. Les parents ont déclaré que la lésion avait été remarquée pour la première fois sur forme de point d’épingle dans la zone submandibulaire et était trop petite pour justifier une quelconque inquiétude. La radiographie initiale a révélé l’absence d’atteinte osseuse mais la seconde a montré des signes d’atteinte osseuse. Une impression de sarcome des tissus mous et une biopsie incisionnelle a été envoyée pour un diagnostic pathologique qui a révélé un fibrome odontogène périphérique. La rapidité de la croissance après la biopsie incisionnelle a fait croire à une lésion bénigne.
CONCLUSIONS: Un fibrome odontogène agressif avec des points multifocaux chez une jeune fille de 15 mois.
UNASSIGNED: Fibrome odontogène, Périphérique, Central, Agressif.

Fibrous sclerosing tumours and hypertrophic lesions in IgG4-related disease (IgG4-RD) are formed in various organs throughout the body, but disease in the oral region is not included among individual organ manifestations. We report a case of ossifying fibrous epulis that developed from the gingiva, as an instance of IgG4-RD.
A 60-year-old Japanese man visited the Department of Oral and Maxillofacial Surgery, Gunma University Hospital, with a chief complaint of swelling of the left mandibular gingiva. A 65 mm × 45 mm pedunculated tumour was observed. The bilateral submandibular lymph nodes were enlarged. The intraoperative pathological diagnosis of the enlarged cervical lymph nodes was inflammation. Based on this diagnosis, surgical excision was limited to the intraoral tumour, which was subsequently pathologically diagnosed as ossifying fibrous epulis. Histopathologically, the ossifying fibrous epulis exhibited increased levels of fibroblasts and collagen fibres, as well as infiltration by numerous plasma cells. The IgG4/IgG cell ratio was > 40%. Serologic analysis revealed hyper-IgG4-emia (> 135 mg/dL). The patient met the comprehensive clinical diagnosis criteria and the American College of Rheumatology and European League Against Rheumatism classification criteria for IgG4-RD. Based on these criteria, we diagnosed the ossifying fibrous epulis in our patient as an IgG4-related disease. A pathological diagnosis of IgG4-related lymphadenopathy was established for the cervical lymph nodes. Concomitant clinical findings were consistent with type II IgG4-related lymphadenopathy.
A routine serological test may be needed in cases with marked fibrous changes (such as epulis) in the oral cavity and plasma cells, accompanied by tumour formation, to determine the possibility of individual-organ manifestations of IgG4-related disease.

OBJECTIVE: Metastasis to oral and maxillofacial region (MOMFR) is an unusual finding; representing between 1 and 1.5% of all malignancies in the maxillofacial region. A systematic review is presented to determine trends in presentation, diagnostic features, and patient outcome.
METHODS: Searches of databases were carried out for papers reporting MOMFR. The variables were demographics, patient symptoms, tumor location, tumor size, histopathology, origin of the tumor, immunohistochemical studies, follow-up and survival.
RESULTS: 696 cases were identified; 391 males, and 305 females. The most common race was white. The most common primary tumor for females was from breast 31.1% (n = 95), for males from lung 20.5% (n = 143). The most common location was the mandible 44.9% (n = 313), followed by gingival soft tissue 16.8% (n = 117). A frequent clinical symptom was pain with 17.5% (n = 122). The most common clinical presentation was a mass or tumor 37.4% (n = 260). The mean age was 58.8 years. The average time before diagnosis was 10.3 months, the mean follow-up after diagnosis was 13.1 months, and the average survival was 9.8 months.
CONCLUSIONS: MOMFR shows a strong predilection for the posterior mandible, with a mass or tumor being the most common clinical presentation. They are frequently painful, and demonstrate a poor prognosis.

Congenital granular cell epulis (CGCE) is a rare benign soft tissue lesion that usually originates from the neonatal gingiva and can lead to difficulty in breathing and feeding upon birth. This current case report describes a female newborn with a gingival mass that was identified by prenatal fetal ultrasonography. At birth, the oral mass was observed to protrude from the mouth, which adversely affected feeding. The lips could not be closed. The breathing was unaffected. Through a multidisciplinary team approach involving several healthcare professionals, the mass was successfully removed under general anaesthesia during an uncomplicated surgical procedure. Postoperative histopathological examination confirmed that the mass was a CGCE of the newborn. The infant recovered well after the operation.

Odontogenic gingival epithelial hamartoma (OGEH) is a rare odontogenic epitheliallesion. Distinguishing OGEH from peripheral ameloblastoma can pose a differential diagnostic challenge. We report a case of OGEH affecting the lower gingiva in a 78-year-old male patient. The lesion presented as a large, painless, pedunculated mass with a pleomorphic histological appearance. A diagnosis of OGEH was made after the exclusion of peripheral ameloblastoma and peripheral adenomatoid odontogenic tumour. Previous studies have reported differences in sex and age between patients with OGEH and peripheral ameloblastoma; this suggests that peripheral ameloblastoma is not derived from OGEH. Histological findings suggest that the lesions are independent entities.

Metastatic lesions represent approximately 1% of all the intraoral lesions. They most commonly originate from lung and breast carcinomas, while the third most common source is the renal cell carcinoma. In this paper, we present the rare case of metastases of renal cell carcinoma in the mandibular gingiva of a 53-year-old male patient.

BACKGROUND Odontogenic fibroma is a rare mesenchymal odontogenic tumor. It can be classified as central odontogenic fibroma (COF) or peripheral odontogenic fibroma (POF) based on clinical features. There are several variants of COF, including amyloid, ossifying, and giant cell. It grows slowly and exhibits painless cortical expansion of the jawbone. Radiographically, COF appears as a radiolucent unilocular or multilocular lesion with well-defined borders. COF may be associated with unerupted or displaced teeth and root resorption. CASE REPORT A 35-year-old man was referred to our hospital for submandibular swelling. Panoramic radiography and contrast-enhanced computed tomography revealed a unilocular area of bone resorption with a well-defined border and equal enhancement from the canine to first molar on the right side of the mandible. Root resorption of the first premolar and root separation of the first and second premolars were also evident. The lesion was asymptomatic. Histopathological examination of a biopsy of the lesion was suggestive of OF. Enucleation of the tumor, curettage of the bone around the tumor, and extraction of the premolar were then performed. Histopathological examination of the tumor showed fibrous connective tissue with inactive-looking odontogenic epithelial islands and strands, amyloid deposit, intercalation of Langerhans cells into the tumor epithelium, and no calcification. The final diagnosis of amyloid variant of COF was made. The postoperative course is going well. CONCLUSIONS Herein we describe and discuss the clinical, radiological, and pathological features of the amyloid variant of COF. This report will enhance understanding of this extremely rare variant.