A rare case of dentinogenic ghost cell tumor with concomitant odontoma.

Key Clinical Message: A case of dentinogenic ghost cell tumor occurring simultaneously with a clinically diagnosed odontoma. The occurrence of epithelial and mesenchymal tumors at the same site is very rare, but should be kept in mind during pathological diagnosis. Abstract: Dentinogenic ghost cell tumor (DGCT) is a rare and benign odontogenic tumor composed of ghost cells, calcified tissue, and dentin. We present an extremely rare case of a 32-year-old female who was clinically diagnosed with an odontoma presenting with a painless swelling in her maxilla. Radiographic examination showed a well-defined radiolucent lesion with tooth-like calcified areas. The tumor was resected under general anesthesia. No recurrence was noted at the 12-month follow-up. Histopathological examination of the surgically resected tumor yielded a diagnosis of DGCT with odontoma.

Fenestration is a logical and effective treatment for a large primordial cyst with cholesterol granuloma: a case report.

A cholesterol granuloma (CG) is characterized by the presence of cholesterol crystals that cause a chronic granulomatous reaction in an enclosed space. It occurs most commonly in the head and neck region, particularly in the middle ear. Although CGs in the maxilla have also been reported, odontogenic cysts in conjunction with CGs in the maxilla are very rare. We herein present a case of a 72-year-old man who developed a large primordial cyst with a maxillary CG that extended into the maxillary sinus, nasal cavity, and infraorbital region, causing left-sided facial swelling and discomfort. We successfully controlled the symptoms and reduced the size of the lesion using the treatment approach for a common odontogenic cyst: fenestration followed by complete excision. This case suggests that fenestration is an effective technique to treat odontogenic cysts with CGs. Although the mechanisms underlying the pathogenesis and growth of CGs are still unknown, our report highlights a potential therapeutic approach for these lesions.

Gossypiboma penetrating into the small intestine similar to Meckel's diverticulum: a report and literature review.

Foreign body granuloma due to retained surgical sponge (gossypiboma) with penetration into the small intestine is very rare. Cases of gossypiboma in the abdominal cavity have sometimes been reported, yet the correct incidence has not been determined, only estimated to occur in one of every 1,000 to 1,500 intra-abdominal operations. Acute abdomen may be observed in some cases, requiring treatment. We herein introduce the case of a 70-year-old woman with gossypiboma penetrating into the small intestine presented with anemia and hematochezia. She had a history of emergency laparotomy for ectopic pregnancy several decades ago. A dynamic contrast-enhanced computed tomography revealed a hypovascular mass containing air bubbles that continued to the small intestine approximately 50 mm in size in the right lower abdomen. In addition to describing the presentation and outcome of our patient, we review the image findings of gossypiboma.

A case of cellular variants of Sclerosing epithelioid fibrosarcoma resembling Plasmacytoma/Myeloma: Diagnostic difficulty in the fine needle aspiration.

INTRODUCTION: Sclerosing epithelioid sarcoma (SEF) is a rare fibroblastic sarcoma. It is classically composed of cords of epithelioid cells embedded in a hyalinized stroma; however, cases of cellular variants also exist. A cellular variants of SEF can mimic Plasmacytoma/Myeloma (PM) and myoepithelioma. Hence, accurate diagnosis of SEF is important for cytologists and pathologists. PRESENTATION OF CASE: We present the case of a 75-year-old female patient diagnosed with a cellular variant of SEF occurring in the erector spinal muscle. Immunostaining of MUC4 and fluorescence in situ hybridization of EWSR1 (break-apart signal) were used for diagnosis. CONCLUSION: The cellular variants of SEF presented diagnostic difficulties in fine needle aspiration. Moreover, it could not be distinguished from PM.

Xp11.2 translocation renal cell carcinoma with SFPQ/PSF-TFE3 fusion gene: A case report with unusual histopathologic findings.

Xp11.2 translocation renal cell carcinoma (Xp11tRCC) is a subtype of renal cell carcinoma (RCC) characterized by chromosomal rearrangement of the region harboring the transcription factor for immunoglobulin heavy-chain enhancer 3 (TFE3). Xp11tRCCs comprises 20% to 40% of RCCs of children and adolescents and is generally associated with good prognosis. However in adult, the incidence of this tumor is relatively low (1% to 4%), suggesting a more aggressive course. TFE3 gene is fused by translocation to numerous partner genes, and definitive molecular characteristics can be difficult to verify. In this case report, we presented a case of Xp11tRCC with the SFPQ/PSF-TFE3 chimeric gene. The fusion gene was detected by 5'-rapid amplification of cDNA ends (5'RACE). The tumor was found to be in an advanced stage with multiple lymph node metastases. The histological characteristics of the tumor were different from those of XP11tRCC with other more frequently detected fusion genes.

[Surgical Procedure of Buccal Mucosal Carcinoma - Reconstruction of Mouth Angle].

Surgical resection of the buccal mucosal carcinoma often induces soft tissue defect. The treatment plan should be considered to preserve oro-facial function and morpho-esthetics. This retrospective study reports the surgical reconstruction procedures in buccal mucosal carcinoma patients. We evaluated 4 cases(2 men, 2 women, mean age: 81.8 year-old)treated in Department of Oral and Maxillofacial Surgery, Shimane University Hospital between June 2007 and January 2017. The average size of primary tumor was 4.9 cm2. And the average size of facial skin defect in the mouth angle was 3.1 cm2. The facial local skin flaps and/or other pedicled flap were used for the reconstruction of the mouth angle. Severe contraction of the scar was manifested in 2 cases. Though reconstruction using the local pedicled flaps for full thickness skin defect in the mouth angle would be feasible, special attention is considered regarding the postoperative contraction of the scar.

Diagnostic accuracy of oral cancer cytology in a pilot study.

BACKGROUND: Recently, cytology has been applied to the diagnosis of oral lesions. We aimed to explore the diagnostic accuracy of oral cytology based on the histological diagnosis. METHODS: Histological diagnoses of 327 cases were classified as Negative, Borderline lesion -, Borderline lesion +, oral intraepithelial neoplasia/carcinoma in situ (OIN/CIS), or Positive. Cytological diagnoses were classified as NILM (negative for intraepithelial lesion or malignancy), LSIL (low-grade squamous intraepithelial lesion), HSIL (high-grade squamous intraepithelial lesion), or SCC (squamous cell carcinoma). The cytology slides were evaluated by 10 raters and the results were compared with the histology results. RESULTS: In 142 cases that were histologically negative, the number of NILM, LSIL, HSIL, and SCC and other malignancy was 77 (54.2%), 47 (34.3%), 8 (5.6%), and 10 (7.0%), respectively. Among 32 cases of Borderline lesion -, the number of NILM, LSIL, HSIL, and SCC and other malignancy was 11 (34.3%), 11 (34.3%), 9 (28.1%), and 1 (3.1%), respectively. Also, in 4 cases of Borderline lesion +, the number of NILM, LSIL, HSIL, and SCC and other malignancy was 2 (50.0%), 0 (0.0%), 0 (0.0%), 2 (50.0%), respectively. Among 12 cases of OIN/CIS, the number of NILM, LSIL, HSIL, and SCC and other malignancy was 1 (8.3%), 2 (16.7%), 4 (33.3%), and 5 cases (41.7%), respectively. Among 137 cases with a histological diagnosis of Positive, the number of NILM, LSIL, HSIL, and SCC and other malignancy was 7 (5.1%), 22 (16.1%), 19 (13.9%), and 89 (65.0%), respectively. Sensitivity, specificity, and positive predictive and negative predictive values were 93.5, 50.6, 62.4, and 89.8%, respectively, when the cytological diagnosis of Negative was assumed to be NILM; they were 77.8, 83.9, 81.0 and 81.1%, respectively, if the cytological diagnosis of Negative was assumed to be NILM and LSIL. The number of false-positive and false-negative diagnosis affected cases with LSIL and HSIL may indicate the difficulty in the cytological diagnosis of borderline lesions. While the negative predictive value was relatively high (89.8%) when cytological Negative was assumed to be NILM only. CONCLUSION: Histopathological examination should be recommended in cases with cytological diagnoses of LSIL, HSIL, and SCC.

EEG-EMG polygraphic study of dystonia and myoclonus in a case of Creutzfeldt-Jakob disease.

We report on a patient with sporadic Creutzfeldt-Jakob disease (CJD) who showed dystonia, periodic myoclonus, and periodic sharp wave complexes (PSWCs) on EEG. The EEG-EMG polygraphic study revealed that dystonia appeared without relation to periodic myoclonus and PSWCs and that dystonia EMGs were strongly suppressed after periodic myoclonus EMGs. These findings suggest that dystonia has a pathogenesis different from that of periodic myoclonus and PSWCs, but dystonia and periodic myoclonus may be generated through the sensorimotor cortex in CJD.

Tumor-induced lymphangiogenesis in cervical lymph nodes in oral melanoma-bearing mice.

BACKGROUND: Metastasis via the lymphatic system is promoted by lymphangiogenesis. Alterations of the lymphatic channels during the progression of metastasis to regional lymph nodes (LNs) remain unexplored. To examine whether tumor-induced LN lymphangiogenesis controls metastasis to regional LNs, we investigated cervical LN metastasis in a mouse model of oral melanoma. METHODS: Injection of B16F10 melanoma cells into mouse tongues replicated spontaneous cervical LN metastasis. We performed histological, immunofluorescent, and histomorphometric analyses of tumor-reactive lymphadenopathy and lymphangiogenesis in tumor-associated LNs. We investigated the expression of vascular endothelial growth factor (VEGF)-C and its receptor, VEGF receptor-3 (VEGFR-3), in tumor cells and tissues, and LNs by reverse transcription polymerase chain reaction and immunofluorescence. RESULTS: Tumor-associated LNs comprised sentinel LNs (SLNs) before and after tumor cell invasion (tumor-bearing SLNs), and LNs adjacent or contralateral to tumor-bearing SLNs. Extensive lymphangiogenesis appeared in SLNs before evidence of metastasis. After metastasis was established in SLNs, both LNs adjacent and contralateral to tumor-bearing SLNs demonstrated lymphangiogenesis. Interaction between VEGF-C-positive melanoma cells and VEGFR-3-positive lymphatic vessels was evident in tumor-associated LNs. CONCLUSIONS: LN lymphangiogenesis contributes a progression of tumor metastasis from SLNs to other regional LNs.

Induction of epithelial migration of lymphocytes by intercellular adhesion molecule-1 in a rat model of oral mucosal graft-versus-host disease.

To elucidate the involvement of intercellular adhesion molecule-1 (ICAM-1) in the migration of lymphocytes to the oral mucosal epithelium in a rat model of acute graft-versus-host disease (AGVHD), we investigated (1) ICAM-1 and major histocompatibility complex (MHC) class II expression by keratinocytes (KCs) and their role in the epithelial infiltration of CD8+ cells, (2) the tissue expression of interferon-γ (IFN-γ) mRNA and expression of IFN-γ receptor by KCs, and (3) the ability of KCs to direct CD8+ cells into the epithelial layers. We classified the oral mucosal lesions into three consecutive temporal phases on the basis of increased epithelial ICAM-1 expression: basal- (phase I), parabasal- (phase II), and pan-epithelial except for the cornified cell layer (phase III). Basal ICAM-1 expression by KCs preceded that of MHC class II molecules, infiltration of CD8+ cells and epithelial histological changes. Tissue expression of IFN-γ mRNA and expression of IFN-γ receptor on KCs evidenced by immunohistochemistry were detected in early lesions (phase I), indicating that locally produced IFN-γ induced ICAM-1 expression by KCs. CD8+ cells were bound to KCs in frozen sections of epithelial lesions, whereas no lymphocyte attachment was observed in normal KC. Adherence could be inhibited by pretreating CD8+ cells with lymphocyte function-associated antigen-1 (LFA-1) antibody and/or by pretreating sections with ICAM-1 antibody. Our data suggest that in the early phase of acute oral mucosal GVHD, the induction of ICAM-1 expression on KCs leads to the migration of CD8+ cells into the epithelium and that this is mediated in part by the ICAM-1/LFA-1 pathway.

Solitary neurofibroma of the gingiva with prominent differentiation of Meissner bodies: a case report.

BACKGROUND: Oral neurofibromas are peripheral nerve sheath tumors, similar to schwannomas. Histological variations in oral neurofibromas are relatively uncommon. CASE PRESENTATION: Here, we present a case of unique variation in the observed characteristics of a neurofibroma, with no relation to neurofibromatosis type-1 or von Recklinghausen disease of the skin. The neurofibroma was observed in the right mandibular gingiva of a 32-year-old Japanese woman. Histologically, it differed from conventional neurofibromas in that the tumor was composed of a mixture of fine fibrillary collagen in sheets and/or cords of neoplastic Schwann cells containing numerous clusters of Meissner bodies. Histologically, these bodies were in contact with neoplastic Schwann cells. The Meissner bodies were immunopositive for S-100 protein, neuron-specific enolase, and vimentin, but were negative for calretinin. CD34-positive spindle cells were observed around the Meissner bodies. No recurrence or signs of other tumors have been observed in the patient for 5 years after tumor resection. CONCLUSION: To the best of our knowledge, no formal descriptions of sporadic, solitary neurofibromas containing numerous Meissner bodies occurring in the oral cavity are available in literature. We believe that an uncommon proliferation of Meissner bodies, as seen in the present case, may result from aberrant differentiation of neoplastic Schwann cells.