A giant peripheral ossifying fibroma of the maxilla with extreme difficulty in clinical differentiation from malignancy: a case report and review of the literature.

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. CASE PRESENTATION: 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.

Congenital Epulis: A Rare Head And Neck Tumor in the Newborn.

Congenital epulis is a rare tumor; when voluminous and ulcerated, appearance can generate concern. Nevertheless, it is a benign lesion that is easily treated with surgery. This is a retrospective study, with the presentation of 2 cases treated by the same team from 2016 to 2020 and a bibliographic review (PubMed and Elsiever). This surgical team is based in Buenos Aires, working in the private medical system. Congenital epulis is an infrequent, benign tumor affecting females predominantly, appearing at birth on the alveolar median ridge of the maxilla. Its pathogenesis is uncertain. Voluminous tumors interfere with feeding, but respiratory interference is exceptional. Histologically, this tumor is similar to the granular cell tumor but has different clinical characteristics and Periodic Acid Schiff stain. Prenatal diagnosis with ecographic or magnetic resonance imaging is ideal; if undiagnosed prenatally, clinical diagnosis is done based on typical features. Surgical excision is the treatment of choice.

Congenital Epulis Diagnosed Antenatally.

Background: Congenital epulis is a benign gingival tumor whose differential diagnosis includes other oral-facial masses such as teratoma, hemangioma, lymphatic malformation and dermoid cysts. This tumor can cause obstruction of the airway or feeding problems in the newborn. Surgical excision is the treatment of choice. Case Report: We present a case of congenital epulis, diagnosed prenatally with ultrasonography. Conclusion: Although difficult, a defined prenatal image of congenital epulis is possible by means of accurate high-resolution ultrasonography. It facilitates the narrowing down of differential diagnosis. The confirmatory final diagnosis relies on histopathological examination.

Prognostic factors for lymph node metastasis from upper gingival carcinomas.

OBJECTIVES: This study sought to identify tumor characteristics that associate with regional lymph node metastases in squamous cell carcinomas originating in the upper gingiva. MATERIALS AND METHODS: Data from 113 patients from Osaka University Dental Hospital were included. We measured each primary tumor's width, length, depth, and the extent of bone invasion. Additionally, tumor signal intensity for T1 and T2-weighted images as well as the center of the tumor's location and T classification was assessed, and a histopathological analysis was performed. RESULTS: Tumor signal intensity was not found to be a significant prognostic factor. However, bucco-lingual width, histopathological classification as well as the tumor's location were significantly different between metastatic and non-metastatic groups in both univariate and multivariate analysis. Superior-inferior depth and T classification were significant only in the univariate (and not the multivariate) analysis. CONCLUSIONS: Bucco-lingual width, histopathological grading as well as the tumor's location are likely to be important predictors for the occurrence of LN metastasis in upper gingival carcinoma patients and should be considered when managing care for these patients.

Determination of diagnostic and predictive parameters for vertical mandibular invasion in patients with lower gingival squamous cell carcinoma: A retrospective study.

Vertical mandibular invasion of lower gingival squamous cell carcinoma (LGSCC) determines the method of resection, which significantly affects the patient's quality of life. Therefore, in mandibular invasion by LGSCC, it is extremely important to monitor progression, specifically whether invasion is limited to the cortical bone or has progressed to the bone marrow. This retrospective study aimed to identify the diagnostic and predictive parameters for mandibular invasion, particularly vertical invasion, to enable appropriate selection of the method of mandibular resection. Of the patients who underwent surgery for LGSCC between 2009 and 2017, 64 were eligible for participation in the study based on tissue microarrays (TMA) from surgical specimens. This study analyzed morphological features using computed tomography (CT), and metabolic characteristics using maximum standardized uptake value (SUVmax), peak value of SUV (SUVpeak), metabolic tumor volume (MTV), and total lesion glycolysis (TLG). Moreover, immunohistochemical analysis of proteins, including parathyroid hormone-related protein (PTHrP), interleukin-6 (IL-6), E-cadherin, and programmed cell death-1 ligand 1 (PD-L1), was performed. Statistical analysis was performed using univariate logistic regression analysis with the forward selection method. The present study showed that MTV (≥2.9 cm3) was an independent diagnostic and predictive factor for positivity of mandibular invasion. Additionally, TLG (≥53.9 bw/cm3) was an independent diagnostic and predictive factor for progression to bone marrow invasion. This study demonstrated that in addition to morphological imaging by CT, the volume-based parameters of MTV and TLG on fluorine-18 fluorodeoxyglucose positron emission tomography were important for predicting pathological mandibular invasion in patients with LGSCC. A more accurate preoperative diagnosis of vertical mandibular invasion would enable the selection of appropriate surgical procedure for mandibular resection.

Prenatal diagnosis and multidisciplinary management: a case report of congenital granular cell epulis and literature review.

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.

Congenital granular cell epulis of newborn: importance of prenatal diagnosis.

Congenital granular cell epulis is a rare benign lesion usually arising as single mass from the alveolar ridge of maxillary bone of female newborns, composed of polygonal granular cells that typically stain negative for S-100, in contrast to the adult counterpart. Larger lesions can disturb breathing and breast-feeding, requiring surgery. Prenatal diagnosis is achieved in few cases, even if this would be important for best management of delivery and therapy. Here we present a case of multiple CGCE in a female newborn discovered at birth, together with a brief review of pathogenesis, differential diagnoses and treatment implications of early diagnosis.

Extraosseous odontogenic fibroma with a clear cells component: a 5-year follow-up.

The extraosseous/peripheral odontogenic fibroma (E/POF) is a benign mesenchymal odontogenic tumour found on the gingival surface with clinical characteristics identical to those of reactive lesions. A histopathological analysis is the only method for determining the difference between reactive lesions and these neoplastic lesions, whose recurrence potential varies between 38.9% and 50%, highlighting the importance of correct diagnosis. The following report describes an E/POF case with a clear cells component, as well as a long-term follow-up treatment, which we emphasise due to its potential for recurrence.

Extra-axial chordoma of the gingiva.

BACKGROUND: Extra-axial chordomas are exceedingly rare malignant tumours. Herein, we present the first case of an extra-axial chordoma in the gingivae. METHODS AND RESULTS: A 21-year-old man presented with a suspected chordoma in the upper right gingiva. Diagnosis was difficult; however, owing to strong expression of the chordoma marker brachyury, extra-axial chordoma was ultimately diagnosed. The tumour was completely resected without performing a facial incision. To ensure its safety and effectiveness, the surgical procedure was simulated several times before its performance using a three-dimensional (3D) model. Twenty-four months after surgery, the patient remains disease-free. CONCLUSION: A diagnosis of extra-axial chordoma can be confirmed by immunohistochemical staining for brachyury.

Late Diagnosis of Oral Melanoma / Diagnóstico Tardío de Melanoma Oral

ABSTRACT: Oral melanoma (OM) is an extremely rare and aggressive malignancy. A 67-year-old patient presented with complains of a slightly symptomatic spot in the mouth since the past 2 years. Extraoral examination revealed left cervical lymphadenopathy, and intraoral examination a blue-black multinodular sessile mass, with irregular margins, involving the attached gingiva of teeth 27 and 28, extending to vestibular sulcus and hard palate, measuring approximately 3.5 cm. The lesion presented focal areas of ulceration. Panoramic radiograph did not show bone involvement. The main diagnostic hypothesis was oral melanoma. Microscopic findings of the incisional biopsy revealed a proliferation of densely pigmented pleomorphic cells, invading the subepithelial connective tissue in sheets or nests showing an organoid pattern. Immunopositivity for S-100, Melan-A and HMB-45 confirmed the diagnosis of melanoma. The patient was referred to an oncology hospital in which multiple metastases were detected, and the patient was subjected to palliative care. Herein we report an OM in advanced clinical stage, and discuss the clinical, morphological and immunohistochemical diagnostic criteria with emphasis on the importance of early diagnosis.

RESUMEN: El melanoma oral (MO) es una malignidad extremadamente rara y agresiva. Un paciente de 67 años acudió a consulta con la queja de una mancha intraoral ligeramente sintomática, presente desde hace dos años. Al examen clínico extraoral, se encontró adenopatía cervical del lado izquierdo, y al examen intraoral, se observó una masa sésil multinodular de color negro azulado, focalmente ulcerada, con bordes irregulares, afectando la encía de los dientes 27 y 28, extendiéndose hasta el surco vestibular y el paladar duro, midiendo aproximadamente 3,5 cm. La radiografía panorámica no mostró involucramiento óseo. La principal hipótesis diagnóstica fue MO. Los hallazgos microscópicos de la biopsia incisional revelaron una proliferación de células pleomórficas densamente pigmentadas, invadiendo difusamente el tejido conectivo en forma de sábanas o nidos con patrón organoide. La positividad inmunohistoquímica para S-100, Melan-A y HMB-45 confirmó el diagnóstico de melanoma. El paciente fue referido a un hospital oncológico, en el cual se le detectaron múltiples metástasis y fue sometido a cuidados paliativos. Este es el reporte de un caso de MO diagnosticado en estado avanzado, en el que se discuten los criterios clínicos, morfológicos e inmunohistoquímicos para su diagnóstico, haciendo énfasis en la importancia del diagnóstico temprano.

[Carcinoma cuniculatum: An unusual oral tumor]. / Carcinome cuniculatum : une tumeur orale inhabituelle.

We report on a case of carcinoma cuniculatum (CC) of the maxillary gingival mucosa. A 60-year-old woman presented with an exophytic gingivo-palatal mass with slow growth and osteolytic evolution. A first performed biopsy was negative for malignancy. The diagnosis of CC was established on the surgical representative biopsy. CC is a rare low-grade variant of squamous cell carcinoma that is usually found in the foot or in oral cavity. The pathognomonic microscopic feature of CC is an endo- and/or exophytic lesion composed by a well differentiated squamous epithelium infiltrating into underlying stroma forming a complex pattern of keratin cores and keratin filled "rabbit warren" crypts. CC is a locally evolutive carcinoma with a usually good prognosis usually without lymph node or distant metastatic evolution.

Comparison of Computed Tomography, Magnetic Resonance Imaging, and Positron Emission Tomography and Computed Tomography for the Evaluation Bone Invasion in Upper and Lower Gingival Cancers.

PURPOSE: Preoperative detection of bone invasion is important in cases of gingival cancer. The aim of this study was to compare the diagnostic value of 3 imaging methods for the detection of bone invasion in upper and lower gingival cancer: computed tomography (CT), magnetic resonance imaging (MRI), and positron emission tomography (PET) and CT. MATERIALS AND METHODS: This retrospective cohort study enrolled patients who underwent a maxillectomy or a mandibulectomy for gingival cancer. Each preoperative image (CT, MRI, or PET/CT) was reviewed for the presence of bone invasion, and the possibility for bone invasion was graded. These results were verified with pathology reports. Sensitivity, specificity, positive predictive value, and negative predictive value for the detection of mandibular involvement in alveolar bone were calculated, and a receiver operating characteristics (ROC) curve analysis was performed. RESULTS: Forty patients (27 men and 13 women) were enrolled. Pathologic examination disclosed bone invasion in 25 of the 40 patients. Of these patients, 13 had maxillary and 12 had mandibular alveolus involvement. The diagnostic accuracy of CT (90.0%) was highest among the 3 modalities for the detection of bone invasion. In the ROC curve analysis, values for the area under the curve for upper gingival cancer were lower than those for lower gingival cancer. CONCLUSIONS: The 3 imaging methods were less sensitive for the detection of bone invasion in upper gingival cancer than in lower gingival cancer. Cases of upper gingival cancer should be evaluated more carefully for bone invasion before surgery.

Squamous Cell Carcinoma of Maxillary Gingiva Progressing to Disseminated Carcinomatosis of Bone Marrow.

Disseminated carcinomatosis of the bone marrow (DCBM) is characterized by diffuse metastasis to bone marrow and sudden mortality. To the best of our knowledge, no studies to date have reported progression of oral squamous cell carcinoma to DCBM. Herein, we report a case of squamous cell carcinoma in the maxillary gingiva suspected of progressing to DCBM. A 64-year-old woman presented with white lesions on the left maxillary gingiva. The lesions were diagnosed as squamous cell carcinoma (T2, N0, M0), and partial maxillectomy performed. Two years and 5 months after surgery, metastasis was noted in the left cervical lymph node and left radical neck dissection carried out. The subsequent diagnosis was right cervical lymph node metastasis and multiple bone metastases. The patient also presented with thrombocytopenia, anemia, and elevated levels of alkaline phosphatase, probably due to metastatic bone disease. Although various antitumor therapies were administered, the patient died 6 months after diagnosis of multiple bone metastases.

Oral carcinoma: Clinical evaluation using diffusion kurtosis imaging and its correlation with histopathologic findings.

PURPOSE: In this study, we aimed to determine the usefulness of diffusion kurtosis imaging (DKI) as a noninvasive method for evaluation of the histologic grade and lymph node metastasis in patients with oral carcinoma. MATERIALS AND METHODS: Twenty-seven patients with oral carcinoma were examined with a 3-T MR system and 16-channel coil. DKI data were obtained by a single-shot echo-planar imaging sequence with repetition time, 10,000 ms; echo time, 94 ms; field of view, 250 × 204.25 ms; matrix, 120 × 98; section thickness, 4 mm; four b values of 0, 500, 1000, and 2000 s/mm2; and motion-probing gradients in three orthogonal directions. Diffusivity (D) and kurtosis (K) were calculated using the equation: S = S0 ∙ exp(-b ∙ D + b2 ∙ D2 ∙ K/6). Conventional apparent diffusion coefficient (ADC) was also calculated. The MR images were compared with the histopathologic findings. RESULTS: Relative to the histologic grades (Grades 1, 2, and 3) of the 27 oral carcinomas, D values showed a significant inverse correlation (r = -0.885; P < 0.001) and K values showed a significant positive correlation (r = 0.869; P < 0.001), whereas ADC values showed no significant correlation (r = -0.311; P = 0.115). When comparing between metastatic and non-metastatic lymph nodes, significant differences in the D values (P < 0.001) and K values (P < 0.001), but not the ADC values (P = 0.110) became apparent. CONCLUSIONS: In patients with oral carcinoma, DKI seems to be clinically useful for the evaluation of histologic grades and lymph node metastasis.

Linfoma primario difuso de células B grandes de mucosa gingival, en paciente VIH y EBV negativos / Primary gingival diffuse large b-cell lymphoma in HIV/EBR negative patient abstract

El linfoma difuso de células B grandes (LDCBG) es el linfoma más frecuente. La presentación clínica puede ser nodal o extranodal y sus síntomas dependen de la localización tumoral; en la mayoría de los casos están asociados a algún tipo de inmunodeficiencia. Referiremos un caso de LDCBG de presentación atípica en una localización muy infrecuente. Es importante tener en cuenta estas situaciones, ya que pueden simular otros procesos patológicos, retrasando así su correcto diagnóstico y por lo tanto un adecuado tratamiento. (AU)

Diffuse large cell lymphoma B (LDCBG) is the most common type of lymphoma. It´s clinical presentation can be nodal or extranodal and it's symptoms depend where the tumor is located and whether is associated or not with an immunodeficiency disease. We present an atypical presentation of a LDCBG in a very unusual location. It´s important to consider these kind of appearance, as they can mimic other oral pathological processes, delaying their correct diagnosis and therefore an appropriate treatment. (AU)

Successful management of rare gingival metastasis from gastric adenocarcinoma: a case report and literature review.

BACKGROUND: Gastric cancer rarely metastasizes to the oral cavity, especially to gingiva. Only 18 cases have been reported worldwide to date. This paper herein presents the nineteenth case of gingival metastasis from gastric cancer. CASE PRESENTATION: A 75-year-old man who underwent a radical gastrectomy for gastric adenocarcinoma was admitted to clinical oncology center for gingival mass which was originally diagnosed as epulis. The subsequent positron emission tomography-computed tomography (PET-CT) and histopathological examination revealed a gingival metastatic adenocarcinoma originated from gastric carcinoma. Then three-dimensional conformal radiotherapy (3D-CRT) with synchronization and sequential chemotherapy demonstrated clinical benefit in this patient. Furthermore, this research reviewed the records of 18 cases of gingival metastasis from gastric carcinoma in English, Japanese, and Chinese literature, and summarized the clinicopathologic features of the disease based on previously published papers. CONCLUSION: This case suggests that gingival metastasis from gastric cancer is worthy of vigilance. Biopsy and immunohistochemical (IHC) staining should be used for the final diagnosis. Moreover, the patient with uncommon gingival metastatic lesion can be successfully treated by radiotherapy with adjuvant chemotherapy.

Clinicopathologic evaluation of malignancy adjacent to dental implants.

OBJECTIVE: The aim of this study was to describe a new case series of peri-implant malignancy, review the literature, and discuss the implications of malignancies resembling peri-implantitis. STUDY DESIGN: This study was a retrospective analysis of cases from 2000 to 2016. RESULTS: Seven patients (two males and five females), aged 44 to 89 years, were included, representing 1.5% of oral malignancy cases. Five cases were squamous carcinoma, one of basal cell carcinoma, and one of carcinoma of metastatic origin. Six cases presented with nonulcerated overgrowth, with bone loss in three and massive osteolysis in one. Misinterpretation as peri-implantitis delayed diagnosis in six cases. Risk factors included previous oral malignancy (2), potentially malignant conditions (2), and smoking (1). Of the 47 cases in the English language literature, 85% were squamous cell carcinoma and 8.5% had distant metastasis. Most cases had one or more risk factors. CONCLUSIONS: Peri-implant malignancy may represent up to 1.5% of oral malignancy cases. Clinical features imitating peri-implantitis may delay diagnosis. Lesions failing to respond to treatment, especially in patients with pre-existing risk factors, should significantly increase suspicion. Histopathology is crucial for diagnosis.

Metastasis of Mammary Gland Malignant Phyllodes Tumor to the Mandibular Region: A Case Report and Review of the Literature.

Phyllodes tumor is a rare breast tumor described by Müller (1938) as a lesion comprising leaflike stromal fibrous components and narrow cysts. The frequency of distant metastasis from this entity is reportedly approximately 20%, and no effective therapy has been established, so the prognosis is poor. This report describes the case of a 60-year-old woman with a history of left lung resection who showed metastasis of a mammary gland malignant phyllodes tumor to the oral cavity. Intraoral examination showed an elastic, hard mass measuring 28 × 27 mm in the gingiva around the left mandibular second molar. Biopsy examination showed growth of giant cells and roughly circular cells showing positivity for S-100, p63, and vimentin on immunohistochemical staining. The authors diagnosed metastasis of the mammary gland malignant phyllodes tumor to the left mandible and performed cyber knife irradiation (44 Gy in 5 fractions) of the left mandible. The mass in the oral cavity disappeared after cyber knife irradiation, but the patient died of direct invasion to the spine.