Solitary synovial osteochondromatosis (SSO) is a rare variant of synovial osteochondromatosis. SSO is thought to represent a solitary juxta-articular mass arising from synovial cells located within joints, bursae, or tendon sheaths. This lesion can grow and show clinical and radiological features of neoplasm. Differentiation from temporomandibular joint diseases is important. Some reports around the knee, foot, nape of the neck, wrist, elbow, and buttocks have described SSO, but SSO of the temporomandibular joint is thought to be uncommon. We report herein a case of giant solitary synovial osteochondromatosis of the temporomandibular joint. While extremely rare, SSO should be considered when treating pathologies of the temporomandibular joint.
Osseous choristoma is a rare entity in the oral and maxillofacial region. The most common site is the tongue, followed by the buccal mucosa. Osseous choristoma of the muscle is unusual and few cases have been described in the literature. No reports have described osseous choristoma arising on the medial side of the lateral pterygoid muscle. Herein, we report the first such case. A 61-year-old woman was referred to our facility for treatment of temporomandibular joint disorder. Computed tomography (CT) revealed two osseous lesions on the medial side of the lateral pterygoid muscle. Preoperatively, a three-dimensional (3D) life-sized model of the skull was fabricated from the CT imaging data. The 3D skull model was used to clarify the 3D positional relationships of the osseous lesions relative to the surrounding anatomical structures. Surgery to remove the osseous lesions was performed under general anesthesia. Unfortunately, only the larger of the two masses was able to be resected because of the risk of nerve and vascular injury. Histological examination revealed that the resected lesion comprised mature lamellar bone with regular osteocytes and no atypia. The diagnosis was multiple osseous choristomas based on the histological and clinical findings.
In this paper, we report the application of bimaxillary surgery for mandibular prognathism in an acromegalic patient with obstructive sleep apnea syndrome (OSAS). The patient, a 21-year-old male, was referred to our clinic for the treatment of malocclusion with open bite and mandibular protrusion. He was 187cm tall and weighed 68kg. The serum growth hormone (GH) and insulin like growth factor I (IGF-I) showed high scores of 5.86ng/ml and 604ng/ml, respectively. His apnea and hypopnea index was also out of the normal range (16.5). MRI revealed a tumor in the sella turcica. Under the diagnosis of acromegaly caused by pituitary adenoma, he underwent removal of the pituitary tumor by neurosurgeons. Ten months later, IGF-I reduced to the normal range. GH and AHI also decreased. Morphological analysis of the pharynx by using lateral cephalograms revealed widening of his upper airway tract after removal of the pituitary adenoma. Le FortI and bilateral sagittal splitting ramus osteotomy along with tracheotomy was successfully performed at the age of 24 years. The postoperative course was uneventful. He was followed up for 2 years without skeletal relapse, deterioration of OSAS, or recurrence of pituitary adenoma.
Myoepithelioma is a rare form of salivary gland tumor composed entirely of myoepithelial cells. This tumor was formerly considered to be a subtype of pleomorphic adenoma; however, in the 1991 World Health Organization classification, it is listed as an independent entity. The most favorable site of occurrence of myoepithelioma is the parotid gland. Here, we report an extremely rare case of myoepithelioma of the upper lip. A 56-year-old woman presented with a painless mass on her upper lip. Magnetic resonance imaging revealed a 23 mm × 18 mm well-defined ovoid tumor. A benign minor salivary gland tumor was clinically suspected, and the patient underwent complete resection of the tumor under general anesthesia. The tumor was histopathologically diagnosed as a benign myoepithelioma of the minor salivary gland. Immunohistochemically, the tumor cells were positive for S-100 protein, AE1/AE3, CAM5.2, CK7, vimentin, and calponin, confirming the morphologic diagnosis of myoepithelioma. The patient's postoperative clinical course was uneventful, and satisfactory results were obtained both functionally and esthetically. To the best of our knowledge, this is the sixth case of myoepithelioma of the upper lip reported in English-language research.
The transcription factor p53 plays a crucial role in coordinating the cellular response to various stresses. Therefore, p53 protein levels and activity need to be kept under tight control. We report here that diacylglycerol kinase ζ (DGKζ) binds to p53 and modulates its function both in the cytoplasm and nucleus. DGKζ, one of the DGK family that metabolizes a lipid second messenger diacylglycerol, localizes primarily to the nucleus in various cell types. Recently, reports have described that excitotoxic stress induces DGKζ nucleocytoplasmic translocation in hippocampal neurons. In this study, we found that cytoplasmic DGKζ attenuates p53-mediated cytotoxicity against doxorubicin-induced DNA damage by facilitating cytoplasmic anchoring and degradation of p53 through a ubiquitin–proteasome system. Concomitantly, decreased levels of nuclear DGKζ engender down-regulation of p53 transcriptional activity. Consistent with these in vitro cellular experiments, DGKζ-deficient brain exhibits high levels of p53 protein after kainate-induced seizures and even under normal conditions. These findings provide novel insights into the regulation of p53 function and suggest that DGKζ serves as a sentinel to control p53 function both during normal homeostasis and in stress responses.
上皮筋上皮癌は,全唾液腺腫瘍の1%を占めるまれな唾液腺悪性腫瘍である。その大部分は耳下腺に好発する。今回われわれは,顎下腺に生じた上皮筋上皮癌と,肺癌の重複癌の1例を経験したので報告する。患者は57歳の女性。約10年前からの左顎下部の腫脹を主訴に当科を受診した。左顎下腺部に拇指頭大の弾性硬の腫瘤を触知した。CTにて,左顎下腺体内ならびに左上葉肺野に腫瘍性病変を認めた。左顎下腺腫瘍と左転移性肺癌の診断にて,2008年4月に左顎下腺腫瘍摘出術を施行,その後同年5月に当院呼吸器外科にて左肺上葉切除術が施行された。術後の病理検査では,顎下腺腫瘍は導管上皮成分と淡明筋上皮成分からなる二層構造の特徴的な上皮筋上皮癌と診断された。また左肺腫瘍は細気管支肺胞上皮癌の診断で,相互の関連性はなかった。術後経過に異常なく,術後3年目であるが,経過は良好で腫瘍の再発も認められない。
Dental technicians use various materials, particularly metal alloys and monomers and polymers based on methylmethacrylate. Environmental hygiene issues surrounding the work of dental technicians have been recognized. Despite the need for observance of occupational environmental controls and work management, compliance among dental laboratories appears to be low. We evaluated the relationship between genotoxic effects in dental technicians and occupational environmental controls and work management. We used cytokinesis-blocked micronuclei (CB-MN) frequencies in peripheral lymphocytes and metal levels in scalp hair. We also assessed nutritional factors related to anti-genotoxic effects using a self-administered brief diet history questionnaire. Study subjects were 54 male dental technicians and 38 male clerical workers. The parameters on the micronuclei (MN) frequency in dental technicians and clerical workers were analyzed by forward stepwise multiple regression analyses. Age (P<0.01, β=0.561), occupation as a dental technician (P<0.01, β=0.636) and aluminum levels in scalp hair (P<0.05, β=0.213) were risk factors that significantly increased MN frequency. The significant parameters on the MN frequency in dental technicians were observance of hand-washing as work management (P<0.01, β=−0.304), work period (P<0.01, β=0.509), germanium levels in scalp hair (P<0.01, β=−0.314) and workplace (P<0.05, β=0.235). To avoid genotoxic effects, observance of occupational environmental controls and work management is necessary for dental technicians.
SOX2 anophthalmia syndrome characteristically presents as anophthalmia or microphthalmia, with various extraocular symptoms, such as hypogonadotropic hypogonadism, brain anomaly, and esophageal abnormalities. In this report, we describe a patient with SOX2 anophthalmia syndrome complicated with a dental anomaly, multiple supernumerary impacted teeth, and persistence of deciduous teeth. Multiple supernumerary teeth are usually not solitary symptoms, but indicate systemic syndrome such as cleidocranial dysplasia. In odontogenesis, many transcriptional factors, such as BMPs, FGFs, and Wnts, play significant roles and SOX2 is known to interact with some of them. The role of SOX2 in dental development remains unknown, however, multiple supernumerary teeth can be considered as extraocular symptoms of SOX2 anophthalmia syndrome, rather than the coincidence of two rare diseases. © 2010 Wiley‐Liss, Inc.
American Journal of Medical Genetics Part AVolume 152A, Issue 9 p. fm i-fm v Table of ContentsFree Access Table of Contents, Volume 152A, Number 9, September 2010 First published: 20 August 2010 https://doi.org/10.1002/ajmg.a.33697AboutPDF ToolsRequest permissionExport citationAdd to favoritesTrack citation ShareShare Give accessShare full text accessShare full-text accessPlease review our Terms and Conditions of Use and check box below to share full-text version of article.I have read and accept the Wiley Online Library Terms and Conditions of UseShareable LinkUse the link below to share a full-text version of this article with your friends and colleagues. Learn more.Copy URL Share a linkShare onFacebookTwitterLinked InRedditWechat Volume152A, Issue9September 2010Pages fm i-fm v RelatedInformation
Single-tooth alveolar osteotomy followed by distraction osteogenesis was applied to move an infrapositional ankylosed tooth to the normal position. A maxillary right canine of a 16-year-old female patient was at 6 mm infraposition compared with the maxillary left canine. Single-tooth alveolar osteotomy was performed under general anesthesia. After suturing the wound, an extrawound distraction device was attached to the crown of the ankylosed canine. The distance of movement was 7 mm. After the distraction movement, the fragment was fixed with wires and resin. The consolidation period was 2 months. During the treatment period, 2 complications associated with the roots occurred: injury of the root of the neighboring tooth during osteotomy and progressive inflammatory root resorption of the ankylosed tooth. The root fragments of the neighboring tooth fused spontaneously and the tooth did not show any symptoms of root resorption. The progressive inflammatory root resorption, which occurred at the cervical portion, was treated by removing the granulation in the resorption-lacuna and restoration with composite resin. It was suggested that root injury under sterile conditions probably promoted periodontal healing. Careful postoperative observation was necessary to detect and promptly treat the progressive inflammatory root resorption.
Since a nutrition support team (NST) began to work in our hospital in March, 2003, we constructed our original nutrition assessment system that supports the prescription formulation of total parenteral nutrition (TPN). However, in daily NST activities, the re-evaluation of this system became necessary because of a high incidence of enteral nutrition (EN) and marked revisions in the dietary reference intakes in Japanese (7th revision). Therefore, we improved this system and added a prescription formulation support function that is also applicable to EN, and also added a function that automatically calculates the necessary doses of nutrients that tend to become deficient in patients with decubituses. This new system allowed the selection/evaluation of EN solutions in a short time with consideration of the 7th revision, and readily identified deficient nutrients and their levels in decubitus patients. We used this system in patients with high-level malnutrition complicated by decubituses and observed certain treatment effects.
Von Recklinghausen's disease is characterized by multiple neurofibromas of the skin and nervous system, with unique systemic pigmented skin lesions (café au lait spots). While these lesions are seen systemically, mandibular deformation is rare. We describe a 13-year-old girl in whom von Recklinghausen's disease was diagnosed at the age of 2 years 5 months. Her brother and sister also have von Recklinghausen's disease. Café au lait spots were seen on the right forearm, left thigh, right lower thigh, left lower prolabium, and left mandibular angle. Two Lisch nodules were detected in the right iris. The patient had borderline to mild mental retardation. Palatal deviation of the right maxillary second premolar was the only abnormality apparent in the oral cavity. A panoramic X-ray film revealed a crescent-shaped bone defect with a clear border at the left mandibular angle. Compressive resorption due to a tumor was suspected. However, magnetic resonance imaging and computed tomography showed no evidence of a tumor or cyst. Parts of the masseter muscle and parotid gland were observed in the defect. There was no resorption of cortical bone. The left maxillary third molar was subsequently extracted because of periodontitis. The postoperative course of the patient has been favorable.
The enamel matrix derivative Emdogain (EMD) has been found to promote regeneration of lost periodontal tissues. We have studied the effects and distribution of EMD in the periodontal tissues of maxillary rat molars transplanted to a subcutaneous position in the abdominal wall. The molars were transplanted with or without EMD either immediately after extraction or after drying for 30 min. After 2 days, 1, 2 or 4 weeks the rats were killed and the teeth were examined by means of light microscopy and immunohistochemistry with anti-amelogenin antibodies. Teeth transplanted immediately after extraction showed formation of alveolar bone separated from the dental roots by a periodontal space, regardless of the use of EMD. Among the teeth that were transplanted with EMD after drying for 30 min, new alveolar bone was formed in five out of eight teeth after 2 and 4 weeks. None of the teeth that were dried for 30 min and transplanted without EMD showed alveolar bone formation. Only one tooth transplanted with EMD showed root resorption after drying, while resorption was noted in all teeth transplanted without EMD. All teeth that were transplanted with EMD and none of the teeth that were transplanted without EMD showed an immunohistochemical reaction for amelogenin. After 2 days, amelogenin was precipitated on all surfaces exposed at the transplantation procedure. Later, the immunoreactive material was redistributed to cells at the root surface, where it was still demonstrable after 4 weeks. In conclusion, EMD is accumulated in cells at the root surface and promotes regeneration of the periodontal tissues of the transplanted teeth. It also seems to promote healing of root resorption.
The effects of wire and miniplate fixations on mandibular stability and temporomandibular joint (TMJ) symptoms were analyzed in 145 patients in whom mandibular prognathism had been corrected by the bilateral sagittal split osteotomy (BSSO). The subjects were divided into two groups based on the method of fixation. Group I consisted of 105 patients treated with intraosseous wiring and group II consisted of 40 patients treated with titanium miniplates. The mean maxillomandibular fixation periods for groups I and II were 48 days and 23 days, respectively. Lateral cephalograms were taken immediately before surgery, within a week after surgery, and at least a year after surgery. Changes in the positions of the incisal edge of the lower central incisors, point B, pogonion, and menton were examined. TMJs were examined clinically for pain, sounds, movements, and limitations before treatment and at least a year after surgery. In both groups, the mean of the horizontal relapses was correlated to the magnitude of their surgical movements. In group II, the mean horizontal relapse was smaller, but not significantly more than that of group I. There was no statistical difference in the pre‐ and postoperative incidence of TMJ signs and symptoms between the two groups. It can be concluded that miniplate fixation for the BSSO procedure has the advantages of shortening the maxillomandibular fixation period and maintaining the postoperative stability of the mandible without causing adverse effects on the TMJ.
Seventeen patients with 23 surgically treated ankylotic temporomandibular joints were studied clinically. Including 2 patients with recurrence, high operations, low operations, and gap surgery were performed on 12, 8, and 6 joints, respectively, in patients 6 to 60 years of age. Joints were reconstructed by costochondral grafts and artificial joints in 3 patients (4 joints). Interpositional materials were used in 7 joints in 5 patients. With the exception of 3 patients who underwent joint reconstruction, all patients started postoperative mouth opening exercises within a week after surgery. The maximum mouth opening increased by 6 to 30 mm 4 months to 7 years 9 months postoperatively. The average postoperative maximum mouth opening was greater in patients younger than 16 years of age than in those older than 20 years of age, and there was no re-ankylosis in the former group. Intermaxillary fixation was maintained for 3 weeks postoperatively in the patients who un-derwent reconstruction. Postoperative open bite was encountered in 5 patients. Open bite was controlled by mouth closing exercises, the use of a chin cap and class II elastics, and occlusal adjustment by prosthetic devices. In conclusion, surgical treatment of ankylotic joints should be performed as early as possible in young patients.
We describe a patient who underwent autotransplantation of a tooth to the maxillary molar region, which lacked adequate bone thickness from the alveolar crest to antral floor. The patient was 34-year-old man with a missing left maxillary second molar. The right mandibular premolar, which had to be extracted because of orthodontic reasons, was transplanted to the region. The apical half of the root of the transplanted tooth projecting into the maxillary sinus was covered with the sinusmucoperiostium, which was carefully detached from the floor, to preserve blood-circulation to periodontal cells. The pocket-depth, mobility, and percussion-sound of the transplanted tooth were normal, and the periodontal space and newly formed bone were observed radiographically around the root 6 months after transplantation. The findings indicated that a tooth can be successfully autotransplanted to a maxillary-molar region that lacks adequate alveolarbony thickness withont the use of a bone graft.
The relationship between recipient condition and the period required for periodontal healing of autotransplanted teeth was studied. The period for periodontal healing was evaluated on the basis of regeneration of the periodontal space and lamina dura on dental radiographs. Age, sex, site of transplantation, period after tooth extraction at the recipient site, and the timing of occlusal function were the recipient factors examined. The mean periods required for regeneration of the periodontal space and lamina dura were 3.7 months and 7.6 months, respectively. Regeneration of the periodontal space was not influenced by any recipient factor, whereas regeneration of lamina dura was associated with the period after tooth extraction, site of transplantation, and timing of occlusal function. It was suggested that regeneration of the lamina dura may not totally depend on that of the periodontal space.
The relationship between root canal filling and the periodontal healing of autotransplantated teeth was studied clinically and radiographically in 21 autotransplanted teeth with complete root formation that were followed up for more than 6 months. The teeth were classified into three types according to the status of root canal filling immediately after transplantation: type A) teeth with root canal (s) wide enough to be detected on radiographs and root canal filling (s) to the apex (es); type B) teeth having incomplete root canal filling (s) because of narrow root canal (s); and type C) teeth having root canal (s) detectable on radiographs but with incomplete filling (s). The periodontal healing was examined more than 6 months postoperatively. Ten teeth classified as type A had no clinical symptoms and showed regeneration of the periodontium on radiographs. Eight teeth classified as type B had no clinical symptoms but the periodontal spaces were indistinct in 5 teeth. No clinical symptoms were associated with 2 of 3 teeth classified as type C, but the periodontal spaces were not clearly seen in the apical regions. The presence of apical lesions and inflammatory resorption of the root was observed on radiographs of the other tooth. These findings suggest that periodontal healing is associated with the width of the root canal and the status of root canal filling.