Primary Sjören’s syndrome （SS） is an autoimmune disorder characterized by lymphocytic infiltrates and destruction of the salivary and lacrimal glands, leading to clinical symptoms of dryness of the mouth and eyes. Autoreactive T cells bearing CD4 molecule may recognize unknown self antigen, triggering autoimmunity in the salivary and lacrimal glands. Recent evidences suggest that the apoptotic pathway plays a central role in tolerazing T cells to tissue-specific self antigen including 120KD α-fodrin, and may drive the autoimmune phenomenon. Cleavage of certain autoantigens during apoptosis may reveal immunocryptic epitopes that could potentially induce autoimmune response. It is now evident that the interaction of Fas with FasL regulates a large number of pathophysiological process of apoptosis including autoimmune diseases. Our data imply that tissue-specific apoptosis and caspase-mediated α-fodrin proteolysis are involved in the progression of autoimmune lesions in SS. We found that retinoblastoma-associated protein RbAp48 overexpression induces apoptosis in the salivary glands caused by estrogen deficiency. The studies reviewed the pathology and molecular mechanisms on the development of SS.
In the radiotherapy for salivary gland malignant tumors the postoperative radiotherapy is often used to prevent locoregional recurrences. In some cases where the surgical operation is impracticable the radiotherapy can be performed alone. And the elective neck radiotherapy effectively prevents nodal relapses and is advised for select patients at high risk for regional failure.
In some researches the prognostic importance of a large number of patients has been studied. In these researches the postoperative radiotherapy for salivary gland malignant tumors is a strong prognostic factor for locoregional control and disease-specific survival. In order to enhance the locoregional control rate the postoperative radiotherapy is strongly recommended for cases of perineural invasion, close or incomplete margin, bone invasion, large （T3-） tumor, and recurrence.
The recommended dose is over 60 Gy and the doses higher than 66 Gy enhanced the locoregional control rate （＞60 ％）.
Complications due to the radiotherapy for salivary gland malignant tumors, including xerostomia, hearing loss,and mastoiditis influence the quality of patient’s life. New modality of the radiotherapy, such as intensity-modulated radiotherapy and particle beam radiotherapy, will reduce the risk of the complications due to the radiotherapy for salivary gland malignant tumors.
The objective of this study was to examine clinical and immunological factors influencing the efficacy ofcevimeline hydrochloride hydrate （cevimeline） for the treatment of xerostomia in patients with Sjögren’s syndrome （SS）. Thirty patients with primary SS who received cevimeline were enrolled in this study. Whole stimulated sialometry （WSS） was performed before and after treatment （4 weeks after oral cevimeline was begun）, and the increment rate of WSS was calculated. Multiple regression analysis was employed to examine the relative contributions of clinical and immunological factors, including age, pretreatment WSS, duration of disease, sialography, minor salivary gland biopsy, anti-Ro/SS-A antibodies, anti-La/SS-B antibodies, and antibodies to muscarinic type 3 receptors to the posttreatment results of WSS. Patients with normal sialographic findings, negative results of minor salivary gland biopsy, and absence of anti-La/SS-B antibodies had significantly higher increment rates of WSS than those with positive findings （p ＝ 0.042, 0.002, and 0.018, respectively）. The results of multiple regression analysis showed that sialography （coefficient ＝− 0.867, p ＝ 0.004） and minor salivary gland biopsy （coefficient ＝− 0.869, p ＝ 0.003） were significantly associated with the posttreatment WSS findings. Our preliminary results demonstrated a relation between the effect of cevimeline on saliva secretion and the degree of salivary gland destruction as evaluated by sialography and histopathological findings in the labial minor salivary glands. These diagnostic approaches could provide useful prognostic information on the efficacy of cevimeline in SS patients.
The present report reviews education systems and clinical practices of Oral-Maxillofacial Surgery in China, with special reference to the contents of Chinese textbooks. After World War II the 1st generation textbook of “Oral Surgery” by Xia Liong Cai appeared in 1959 in Beijing, and the second textbook was “Oral-Maxillofacial Surgery” by Zhang Xize and Qiu Wei-Lie, published in Shanghai in 1980 （2nd generation textbook）. This textbook was used for about 20 years in Chinese stomatology. The 3rd generation textbook “Theory and Practice of Oral-Maxillofacial Surgery”, edited by Qiu WL, was published in 1998. Another textbook “Oral and Maxillofacial Surgery”, an operation text series by Zhou Shu-Xia of the 4th Military Medical University, was published in 1996. A new Book, “Qiu WL’s Oral-Facial Surgery”, was published in 2008. It had 66 authors from Shanghai Ninth People’s Hospital and consisted of 110 chapters （total 1530 pages）. This new textbook covers full-scale surgery for oral and maxillofacial diseases, including head and neck, vascular, and skull-base surgery.University hospitals in Chinese provinces have many patients and many rare cases, which will probably promote new trends in Chinese stomatology. The present paper gives an overview of recent trends in oral and maxillofacial surgery in China from the viewpoint of these textbooks.
Mucinous cystadenoma commonly occurs in the grandular system, including the appendix, ovary, kidney, and pancreas. It is considered a benign tumor. In the head and neck region, it is rare but sometimes occurs in the parotid glands and the minor salivary glands.
We report a case of mucinous cystadenoma occurring in the retromolar region. A 73-year-old woman was referred to our hospital in August 2005 because of a mass in the retromolar region. Intraoral examination revealed an enlarged mass measuring about 20 × 15 mm and covered with normal mucosa in the retromolar region. A benign tumor was suspected clinically. The mass was resected under general anesthesia. Histopathologically, it was diagnosed as a mucinous cystadenoma. There was no evidence of recurrence 60 months after operation.
Osteoporotic bone marrow defect （OBMD） is a rare benign jaw lesion consisting of hematopoietic bone marrow, which results in a focal bone defect. The present report describes a case of OBMD in the mandibular molar region of a 53 year-old-woman. A panoramic radiograph showed the presence of an undefined round,thumb-sized radiolucent lesion in the lower left first molar region. The density inside of the lesion was −37.7 Hounsfield units on CT scanning, suggesting fat tissue. The biopsy specimens revealed fat tissue with thin bone trabeculae and hematopoietic cell infiltration. The clinical course has been uneventful without any treatment for 4 years.
Malignant lymphomas in the head and neck region rarely occur in the oral cavity, but frequently develop in the cervical lymph nodes or Waldeyers’ring initially. We report a case of a malignant lymphoma of the margin of the tongue.
The patient was an 82-year-old woman with a painless tumor arising in the right margin of the tongue. Clinical examination showed that the lesion was covered with normal mucosa. The tumor was elastic hard with a clearmargin and was movable. We diagnosed the lesion as a benign tumor of the right margin of the tongue and extirpated the tumor under local anesthesia. The specimen was histopathologically diagnosed as a non-Hodgkins’lymphoma, diffuse large B cell type. Consequently, we transferred the patient to the department of internal medicine,where she received chemotherapy with R-THP-COP, leading to remission. The remission was maintained, and there was no evidence of abnormalities. However, the patient died of acute cardiac insufficiency 1 year 9 months after the completion of chemotherapy.
A case of squamous cell carcinoma originating in Stensens’duct is presented. A 62-year-old man visited our hospital because of a swelling in his right cheek. At presentation, palpation of the lesion revealed a hard, elastic spherical mass 30 mm in diameter in the right cheek. Adhesion to the skin was found. However, the mobility of the surrounding tissue was poor. The buccal mucosa showed no abnormalities macroscopically. However, salivary flow was slightly poor. An aspiration biopsy revealed atypical keratinocytes, leading to the diagnosis of Class V,and squamous cell carcinoma was suspected. MRI revealed a 15-mm mass located mainly on Stensen’s duct. Neck dissection and resection of the parotid gland lamina superficialis resulted in complications which required further resection of the superficial layer of the masseter muscle and part of the skin of the cheek region. A platysma cervical flap was used to repair the defect. Histopathological examination of the resected tissue revealed a welldifferentiated squamous cell carcinoma. The patient received adjuvant postoperative radiotherapy at a total dose of 40 Gy. As of 14 months after operation, there has been no recurrence or sequelae.
Blood coagulation factor V is a common factor involved in both intrinsic and extrinsic pathways of coagulation; its congenital deficiency is rare. We report a case of congenital factor V deficiency detected because of bleeding after tooth extraction.
A 60-year-old man visited our department because of continued bleeding after tooth extraction. We were able to stop the bleeding by local hemostatic measures. However, prothrombin time （PT） was found to be prolonged on a screening blood test. After further evaluation at the Department of Hematology/Oncology, prolongations of both PT and activated partial thromboplastin time （APTT） were confirmed, and the factor V activity was 4.7 ％, which is very low. After correction of the PT and APTT values by a cross-mixing test, the possibility of an acquired coagulation factor inhibitor was ruled out. The patient was then given a diagnosis of congenital factor V deficiency.
He visited our department again to undergo extraction of the right upper first premolar. After consultation with the Department of Hematology/Oncology, tooth extraction was performed under local anesthesia concurrently with transfusion of fresh frozen plasma.
Factor V activity was corrected to 22.3 ％ from 3.8 ％, and the PT and APTT values improved. Postoperative bleeding was controlled by local hemostatic measures using oxidized cellulose and a gingival pack.
Extranodal NK/T cell lymphoma, nasal type rarely arises in the oral cavity. We report the case of a 55 year-old man who was referred to our hospital because of facial swelling. At presentation, a necrotizing ulcer was found on the left side of the anterior palatal gingiva. Computed tomography showed a soft tissue density in the left sinus, but no evidence of bone destruction. Because the necrotizing ulcer expanded rapidly for a few days, a malignant tumor was suspected. Immunohistochemical analyses of a biopsy specimen revealed that the tumor cells were positive for CD3ε, CD56, granzyme B, perforin, and TIA-1, and negative for CD20, CD79a, and AE1/AE3. Epstein-Barr virus-encoded small RNA -1 was detected on in situ hybridization. Based on these findings, the patient was given a diagnosis of extranodal NK/T cell lymphoma, nasal type. The patient received chemoradiotherapy. Treatment was effective, and a complete response was obtained temporarily. However, recurrence occurred, and the patient received secondary chemotherapy with CHOP and allogeneic hematopoietic stem cell transplantation. Thereafter, his general condition deteriorated rapidly, and the patient died approximately 3 months after recurrence.
We report the case of a giant epidermoid cyst of the submandibular region with tracheal deviation.
The patient was a 24-year-old man with an asymmetric face caused by enlargement of the left side of the neck. Magnetic resonance imaging （MRI） showed a high-signal intensity, clear-bordered mass （greatest dimension, 210 × 160 mm） on T2-weighted images, displacing the trachea. Needle aspiration yielded a bean-curd, refuseshaped, keratinized substance and yellowish white viscous fluid. A dermoid cyst or epidermoid cyst was suspected. A cystectomy was performed via a submandibular incision under general anesthesia . Although the cyst had a thick capsule surrounded by mild adhesions, it was extirpated after aspiration and reduction of the contents of the cyst because its major axis was 230 mm, making en bloc removal difficult. The cystic wall was composed of keratinizing stratified squamous epithelium, and did not include a dermal appendage. The pathological diagnosis was an epidermoid cyst.
The postoperative course has been good, and respiratory discomfort has also improved. About 1 year postoperatively, the face is symmetric, and there has been no evidence of recurrence.
Central giant cell granuloma （GCG） is a relatively rare lesion.
We present a case of central giant cell granuloma of mandible in which segmental resection was performed, immediately followed by reconstruction with titanium mesh and transplantation of iliac particulate cancellous bone and marrow （PCBM）.
The patient was a 49-year-old man. He presented with a sharp pain in the right lower third molar region. Orthopantomography revealed multilobular radiolucent areas from the right mandibular angle to the ramus, with partially absorbed dental roots of the third molar. The tentative diagnosis on an incisional biopsy was giant cell tumor （GCT） or GCG of the mandible. Segmental resection of the mandible and immediate reconstruction with a titanium mesh plate and iliac PCBM transplantation were a performed. A histological specimen showed proliferations of spindle-shaped cells and multinuclear giant cells, which were scattered heterogeneously. The final diagnosis of the resected specimen was GCG. Excellent bone regeneration in the reconstructed region was observed, with no recurrence on postoperative follow-up at 14 months.
This report describes a case of styloid process syndrome （Eagle’s syndrome） with ossification of the stylohyoid ligament. A 58-year-old woman was referred to our department because of right submandibular pain in December 2009. She presented with a 2-month history of spreading pain of the right submandibular region. She had spontaneous pain of the submandibular region, which spread to the right side of the face. On examination, the patient reported some discomfort when she turned her head right. She could open her mouth, but complained of painful limitation of mandibular opening at 39 mm. Bernfeld’s digital examination elicited pain of the right posterior region of the ramus of the mandible. She had no throat pain on swallowing. On oral examination, there was no abnormality; however, palpation detected a bony structure and elicited pain of the right tonsillar fossa. A right elongated styloid process was demonstrated on a panoramic radiography and 3D-CT. These radiographic findings showed a mineralized stylohyoid ligament, approximately 70 mm in length, posterior to the right ramus of the mandible. Although glossopharyngeal neuralgia should be included in the differential diagnosis, a clinical diagnosis of styloid process syndrome was made on the basis of radiographic findings, the nature of the patient’s symptoms, and positive results of the digital examination. The patient was given NSAIDs for 1 month, but the symptoms did not resolve. In April 2010, the patient underwent a styloid process resection by a transcervical approach under general anesthesia. The right elongated process was removed and measured 45 mm in length. The postoperative course was uneventful. The patient has been free of symptoms since the operation. Histologic examination of the mineralized tissue showed normal mature bone with a central cavity, containing viable marrow and cartilage.
Our findings support the concept that the transcervical resection of an elongated styloid process is safe and effective, especially in patients with complete ossification of the stylohyoid ligament.
We describe our experience with 3 patients who had sequelae of facial foreign bodies associated with silicone injection. All 3 patients received facial silicon injections in their twenties. Swelling occurred at the site of silicone injection about 40 years later. No patient voluntarily declared a history of silicon injection. One patient selected surgical treatment, and 2 selected the conservative drug therapy. After treatment, swelling improved in each patient. No recurrence of symptoms has occurred.
Neurofibromatosis （NF） type 1, known as von Recklinghausen’s disease, is an autosomal dominant inherited disorder caused by a mutation of a gene on the long arm of chromosome 17. Malignant tumors are often observed in patients with NF, whereas oral carcinoma concurrent with NF is very rare. This paper describes a case of squamous cell carcinoma of the lower gingiva adjacent to a neurofibroma in a patient with NF type 1.
A 57-year-old man with NF type 1 was referred to our hospital. Intraoral examination revealed two masses: one was a firm nodule on the buccal shelf of the lower left posterior region covered by ulcerated mucosa, and the other was a soft to firm nodule adjacent to the other one in the left cheek region and was covered by normal mucosa.
Excisional biopsy of the two nodules was done. Histological examination showed that the nodule in the buccal region was squamous cell carcinoma （T2N0M0）, and the other was a usual type of neurofibroma. The patient underwent chemotherapy and radiotherapy, followed by local resection of the carcinoma. Local recurrence developed 1 month later. Mandibular segmental resection including the region of neurofibroma, followed by neck dissection and reconstruction with a rectus abdominus free flap was done. The patient has not had tumor recurrnce or metastasis for 8 years after mandibular resection.
Ameloblastic fibroma is a relatively rare benign tumor that consists of odontogenic ectomesenchyme and epithelial strands resembling an enamel organ. The variable distributions of epithelial and mesenchymal components within the tumor often make diagnosis challenging. We report a case of mandibular ameloblastic fibroma in which fibrous components occupied the main portion of the tumor.
An 8-year-old boy underwent a panoramic X-ray examination for abnormal deciduous tooth eruption in the maxilla, and a well-defined multilocular radiolucency in the mandible was detected. Since the biopsy specimen showed proliferation of fibroblast-like cells, the histopathological diagnosis was odontogenic fibroma. A pale white, lobulated solid lesion was resected under general anesthesia. No epithelial components were observed in the middle to upper parts of the tumor, and fascicular proliferation of fibroblast-like cells was observed. However, dental laminalike epithelial components forming cord- or islet-like structures were observed in the deep part of the tumor near the impacted tooth, and ameloblastic fibroma was diagnosed. The clinical course has been good, without recurrence for 2 years postoperatively.
It is necessary to be aware that the histopathological diagnosis of ameloblastic fibroma may be complicated by an uneven distribution of epithelial and mesenchymal components.
Behçet’s disease is an autoimmune disorder with various symptoms caused by systemic vasculitis. We report a case of squamous cell carcinoma arising at the dorsum of the tongue in a patient with Behçet’s disease.
The patient was a 57-year-old woman who had been given a diagnosis of Behçet’s disease at a department of internal medicine. She had had recurrent aphthas every 2 or 3 months, which were resolved by steroid ointment. She had been aware of a refractory ulcer at the dorsum of the tongue for 3 months, resulting in loss of mobility of the tongue, and was referred to our department. An ulcer measuring 4.8 × 2.5 cm was found at the site, and the induration covered nearly the entire tongue.
Based on a clinical diagnosis of tongue carcinoma T4N0M0, we performed tracheotomy, bilateral neck dissection, total glossectomy, marginal mandibulectomy, and reconstruction using a rectus abdominis flap. The carcinoma was diagnosed as a well-differentiated squamous cell carcinoma. Eight years after surgery, she has had no recurrence or metastasis.