The TNM staging system suggests that advanced oral squamous cell carcinoma (OSCC) with T4b is often unresectable and its outcome is poor. The purpose of this multicenter retrospective study is to assess treatment outcomes of OSCC with T4b. Factors that influence treatment outcome were also analyzed. Clinical and pathological data from 99 consecutive patients with resected T4b OSCC (years from 2000 through 2010) were reviewed and analyzed. The 5-year survival and control rates were the main outcome measures, and prognostic factors were identified by univariate and multivariate analyses. All of the resected T4b tumors involved the masticator space, pterygoid plates, or both without carotid artery encasement and skull base extension. The 5-year overall survival, primary/neck control, and distant metastasis rates were 54.3%, 63.6%, and 18.1%, respectively. Multivariate analysis showed an independent and significant impact of the Eastern Cooperative Oncology Group performance status, mode of tumor invasion, and number/ level of metastatic lymph nodes on overall survival (Cox proportional-hazards model, p<0.01). Postoperative radiotherapy and adjuvant chemotherapy had a marginal impact (p=0.06). The results of this study suggested that T4b OSCC involving only the masticator space, pterygoid plates, or both was resectable and expected to have a favorable outcome. Strategies to overcome highly invasive tumors and multiple lymph node metastases are required.
Case reports on malignant lymphoma in the oral and maxillofacial region are rare, and most describe extranodal lymphomas. We report a case of diffuse large B-cell lymphoma (DLBCL) that invaded into the right mental nerve of a 78-year-old woman, who initially presented with right lower lip palsy and pain. We diagnosed severe marginal periodontitis of the right lower first premolar, and planned to extract the tooth in 3 months because she had been taking bisphosphonates for more than 3 years. Two months after the first visit, her palsy and pain became more severe. The right lower lip was red and swollen, and a bead-like mass had developed around the mental foramen. Biopsy of the swollen mental nerve showed DLBCL. Systemic screening results showed that the stage was Ⅳ A. The patient was given chemotherapy with rituximab, tetrahydropyranyl adriamycin, and cyclophosphamide (R-THP-COP) combined with radiotherapy to the left cervical lesion. The redness, swelling, and paralysis of the right lower lip disappeared, and there have been no signs of recurrence in the 13 months after treatment.
Methotrexate (MTX) is widely used as an anchor drug for rheumatoid arthritis (RA). As one of the adverse events, there are several reports of MTX-associated lymphoproliferative disorders (MTX-LPD). However, the majority of reports were cases of B-cell malignant lymphoma, histopathologically. We describe our experience with a rare case of T-cell dominant MTX-LPD of the upper gingiva. A 87-year-old man was first referred to our clinic for pain of the anterior gingiva of the maxilla. He was a RA patient and had received MTX therapy for 12 years and bisphosphonate for 5 years. An ulcer with necrosis and bone exposure of the maxillary gingiva were partly seen. Histopathologically, biopsy specimens were diagnosed as T-cell dominant lymphoproliferative disease' and MTX-LPD was suspected because the patient had received MTX. Therefore, both MTX and bisphosphonate withdrawal resulted in disappearance of the lesion after 96 days. To date, there has been no evidence of recurrence.
Recently, the occurrence of methotrexate-associated lymphoproliferative disorders (MTX-LPD) in the oral area is reported one after another. We report a case of MTX-LPD of the maxillary gingiva associated with rheumatoid arthritis (RA). A 80-year-old man was referred to our clinic because of severe pain associated with a ulcer on the left side of the maxillary gingiva. On blood tests, soluble interleukin-2 receptor (sIL-2R) was high, and EBV infection was detected. The histopathological diagnosis was EBV-positive diffuse large B cell lymphoma (DLBCL). He had been receiving methotrexate for RA. We therefore requested discontinuation of methotrexate, and the lesion was decreased and symptoms disappeared. Examination after 21 months showed no evidence of recurrence. We reviewed reports documenting 17 cases of oral MTX-LPD in Japan. The age of onset was 40-84 years old, the male-to-female ratio was 4:13, and the methotrexate treatment period was 1-114 months. The affected sites were the maxillary and mandibular gingiva in 14 cases, the tongue in 1 case, and the mouth floor in 1 case. On histopathological classification, 14 cases were non-Hodgkin’s lymphoma, and 1 case was Hodgkin's lymphoma. As for the course of treatment, 11 cases had remission with cessation of methotrexate. Symptoms improved in 1-2 weeks, and lesions disappeared in 1-4 months.
Systemic amyloidosis occurs with multiple myeloma at the rate of 5% to 20%, and most cases in the oral and maxillofacial region develop in the tongue and lips. We describe a case of amyloidosis with multiple myeloma in which the tongue grew rapidly, leading to macroglossia. The patient was a 81-year-old woman. In January 2013, she noticed a mass from the right side to the tip of the tongue and consulted an oral surgeon of a certain hospital. A biopsy of the tongue was performed at the hospital, and the patient was given a diagnosis of a hypertrophic scar. However, her tongue was getting bigger slowly, and she was referred to our hospital in April 2013. Based on diagnostic imaging, a second biopsy was done at the tip of the tongue, and it revealed amyloidosis. Then, a puncture test of bone marrow was performed by an oncolo-hematologist, and multiple myeloma was diagnosed (IgG λ type, ISS III stage). Chemotherapy for the myeloma was started at once, but amyloidosis of tongue and digestive organs progressed very rapidly. The conditions were not able to be controlled, and the patient died 5 months after diagnosis.
Although pleomorphic lipoma may contain a small number of atypical cells and resembles an atypical lipomatous tumor histologically, it is a tumor characterized by a clinically benign course. Pleomorphic lipoma typically arises in the posterior cervical region, shoulders, and back, with relatively few reported cases of the tumor in the oralmaxillofacial region. We report on a 60-year-old man with a pleomorphic lipoma that developed from the neck to the submandibular region and was left untreated for more than 40 years because the patient was asymptomatic. The tumor was removed together with its fibrous capsule. Twenty months after surgery, the patient remains recurrence-free and has a good clinical course. Among 39 reports of this tumor in Japan that we searched this time, only one case similat to ours was reported, but reports of pleomorphic lipoma arising in the cervical region are rare in Japan.
We report a case of paracoccidioidomycosis that was difficult to differentiate from sarcoidosis. Paracoccidioidomycosis is a deep fungal infection caused by Paracoccidioides brasiliensis. It causes a granulomatous systemic mycosis and is endemic to rural areas of Latin America. A 57-year-old man visited our clinic because of swelling and slight pain in the lower lip. He had been previously been given a diagnosis of sarcoidosis after a histological examination of the tongue and pulmonary lesions, and had received steroid therapy at the respiratory department of a general hospital. We perform biopsy which specimens were taken from the lower lip and tongue. The initial pathological diagnosis was sarcoidosis, based on the presence of granulomas without caseous necrosis. Several months later, when the pain and swelling of lower lip and tongue had worsened, it was found that the patient had grown up in a rural area in Paraguay and that his relatives had suggested the possibility of fungal disease. This led us to suspect endemic fungal disease and additional pathological examination of the same specimen was performed. Giemsa staining and periodic acid-Schiff staining were negative, but Grocott-Gomori’s methenamine silver staining was positive and showed the characteristic features of paracoccidioidomycosis, associated with a so-called Mickey Mouse ears appearance.
Platelet transfusion recipients occasionally become alloimmunized to foreign HLA and HPA antigens, impairing their responses to further platelet transfusions. Anti-HLA and anti-HPA antibodies, which are produced during pregnancy and blood transfusion, often cause platelet transfusion refractoriness (PTR). We report a case of PTR occurring after platelet transfusion for tooth extraction. A 78-year-old woman was referred to our department because of tooth pain. She had chronic hepatitis and thrombocytopenia, as well as a history of blood transfusion on two occasions. We planned extraction of the left mandibular premolars and left maxillary second molar after the platelet transfusion. After a preoperative platelet transfusion of 20 units was performed, her platelet count decreased. Tests for anti-HLA antibody suggested the possibility of PTR. She had a good response to transfusion of HLA-matched platelets, and the tooth extraction was carried out successfully.