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Objectives The aim of this study was to investigate the involvement of human leucocyte antigen (HLA) class 1 and class 2 alleles in resistance to recurrent aphthous stomatitis (RAS). Materials and methods Nucleotide sequence-based typing of HLA-A, -B, -Cw, and -DRB1 alleles was performed on 22 Japanese participants who had never developed RAS, but had a family history of this disease. Results One HLA-B allele, B*5801, and one HLA-Cw allele, Cw*0302, showed a significant positive association with resistance to RAS, while no such correlation was found for HLA-A and HLA-DRB1 alleles. Conclusion Our findings strongly suggest the existence of HLA alleles associated with resistance to RAS, indicating that HLA alleles or closely linked loci may be involved in the development and pathogenesis of RAS.

We describe here, a case with a giant epidermoid cyst in the floor of the mouth that caused severe obstructive sleep apnea syndrome (OSAS). A 37-year-old man was referred to our clinic because of a swelling in the floor of his mouth and sleep apnea syndrome. The occurrence of breathing disorders and daytime drowsiness was monitored to evaluate his OSAS 1 day before and 7 days after surgery. Before surgery, the apnea-hyponea index (AHI) and Epworth Sleepiness Scale (ESS) were 45.7 and 22, respectively. The clinical diagnosis was a dermoid or an epidermoid cyst that caused severe OSAS. Under general anesthesia, the patient underwent intraoral surgical removal of the cyst, along with aspiration to reduce the mass. After surgery, his sleep apnea syndrome was significantly improved. The postoperative AHI and ESS were 5.5 and 7, respectively. As of 2 years after the operation, there was no evidence of recurrence.

Dental hemorrhagic complications, including postoperative bleeding and traumatic hemorrhage as emergency cases, often occur in patients undergoing oral anticoagulant therapy such as warfarin therapy. Recent research recommends that warfarin dosage should be assessed every 12 weeks. Therefore, most physicians generally accept international normalized ratio (INR) monitoring at longer intervals. However, cases are encountered in which the INR prolongation is observed despite of invariable dosage of warfarin. In this report, we present 2 cases of patients with dental hemorrhagic complications undergoing oral anticoagulant therapy who exhibited excessive INR prolongation. These patients exhibited decreased appetite and hypoalbuminemia. We speculate that long-term appetite loss resulted in the increase in the serum concentration of free warfarin and vitamin K deficiency. Our study indicates that we should notice malnourishment when we treat patients who have undergone warfarin therapy with dental surgical procedures. It is recommended that measurement of INR just before a dental surgical treatment.

Masticatory muscle tendon–aponeurosis hyperplasia (MATAPHY) exhibits hyperplasia on tendon and aponeurosis of the masticatory muscles histopathologically. This disease is characterized by a palpable dense band of the anterior border of the masseter muscle on maximum mouth opening, strike root appearance on MR imaging and square mandible configuration. However, the common useful diagnostic markers of this disease are still unknown. Here we performed a preliminary study to identify potential therapeutic targets and diagnostic markers that may indicate disease progression of MATAPHY using a proteomic analysis method, two-dimensional fluorescence difference gel electrophoresis and liquid chromatography coupled with tandem mass spectrometry. We have found that myosin regulatory light chain 2 and myosin regulatory light chain 3 were down-regulated in MATAPHY. This result is consistent with the observation that muscle tissues showed atrophic changes. We have also demonstrated that aortic smooth muscle actin was down-regulated in MATAPHY, suggesting that this down-regulation is due to decreasing in muscle tissues that blood vessels are abundant and increasing in tendon tissues that blood vessels are exiguous. Furthermore, collagen alpha-1(VI) chain (COL6A1) was up-regulated in MATAPHY. COL6A1, which is essential for tendon fibrillogenesis, expressed in the masticatory muscle, indicating that tendon-aponeurosis hyperplasia is due to the overexpression of COL6A1. Facial deformity, which we choose as a control in this study, is sometimes congenital and may be complicated with deficiency of the skeletal proteins. Although this report has some limitations, this is the first report to describe the relationship between the variation of specific molecules and histopathological observation in MATAPHY.

Objectives: The aim of this study was to evaluate the usefulness of phase-contrast radiography for assessing root morphology of mandibular third molars in comparison with conventional radiography. Methods: We studied 37 extracted mandibular third molars. One oral surgeon compared the number of roots and root curvature of the extracted teeth on conventional radiographs with those on phase-contrast images. Results: The number of roots and root curvature on conventional images differed significantly from those on phase-contrast images. Conclusions: Our results suggest the possibility that phase-contrast radiography is more useful than conventional radiography for assessing the root morphology of mandibular third molars.

A new concept of IgG4-related disease characterized by a high serum IgG4 level and tissue infiltration of IgG4-positive plasmacytes that can involve salivary glands has been proposed. In this article, 2 patients with IgG4-related sclerosing sialadenitis involving the submandibular glands are reported. One patient presented with bilateral and painless swelling of the submandibular glands. He had already been treated with systemic prednisolone owing to the occurrence of retrobulbar neuritis. Laboratory examinations showed high serum IgG4 concentrations, and a biopsy of the submandibular gland revealed the infiltration of IgG4-positive plasmacytes. Abdominal computerized tomography demonstrated tumefaction in the tail of the pancreas, thus suggesting localized autoimmune pancreatitis. The other patient also showed bilateral and painless swelling of the submandibular glands, but there was no involvement of any other organs. These patients were effectively treated with corticosteroids, which resulted in a reduction of the swelling of the submandibular gland and increased saliva. When a patient is suspected of having sclerosing sialadenitis, it is important to consider that the patient may have a systemic IgG4-related plasmacytic disease.

A 77-year-old man with a painless and bilateral submandibular swelling was referred to our hospital. He had already been treated with systemic prednisolone for retrobulbar neuritis. Laboratory examinations showed high serum IgG4 concentrations, and a biopsy of the submandibular gland revealed the infiltration of IgG4-positive plasmacytes. Abdominal CT demonstrated tumor formation in the tail of the pancreas. These findings suggested the patient to have Mikulicz's disease associated with localized autoimmune pancreatitis. Treatment with prednisolone improved the swelling of the submandibular gland. As a result, when a paticent is suspected to have Mikulicz's disease, it is important to consider the possibility of systemic IgG4-related plasmacytic disease.

The surgical treatment of a patient with hereditary angioneurotic edema (BANE) is described in the present report. HANE is an autosomal dominant hereditary disease that causes acute and local submucocutaneous edema. It is a very rare disease in Japan and has been reported in only 20 femilies.The subject was a 24-year-old woman with right upper pericoronitis who had been given a diagnosis of HANE at the age of 13 years. Patients with HANE often have fatal laryngeal edema during dental treatment such as extraction of teeth. Therefore, our patient received ginigivectomy after being hospitalized. Postoperatively, she had mild respiratory distress, but it was controlled by intravenous drip infusion of C 1 esterase inhibitor. When treating patients with HANE, we should take sufficient measures to avoid the development of edema, even when treatment is minimally invasive. Furthermore, sufficient preparation for urgent tracheotomy is necessary owing to the risk of laryngeal edema.

Objectives The aim of this study was to investigate the genetic association between recurrent aphthous stomatitis (RAS) and human leukocyte antigen (HLA) class 1 and class 2 alleles. Materials and methods Nucleotide sequence level typing of HLA-A, -B, -Cw, and -DRB1 alleles was performed on Japanese patients with RAS and their families. The subjects that participated in this study consisted of 25 families that had at least one patient suffering from RAS. Results One HLA-Cw allele, Cw*0303, and three HLA-DRB1 alleles, DRB1*0401, DRB1*0803 and DRB1*1101, showed a significant positive association with RAS. No HLA-A and HLA-B alleles showed significant association with RAS. Conclusion The present study strongly suggests the significant positive association of HLA alleles with RAS. No HLA-A and HLA-B alleles showed significant association with RAS. Thus, the present study indicates that HLA alleles, or closely linked loci, may be involved in the pathogenesis of RAS.

Recent studies have indicated that acetylcholine (ACh) plays a vital role in various tissues, while the role of ACh in bone metabolism remains unclear. Here we demonstrated that ACh induced cell proliferation and reduced alkaline phosphatase (ALP) activity via nicotinic (nAChRs) and muscarinic acetylcholine receptors (mAChRs) in osteoblasts. We detected mRNA expression of several nAChRs and mAChRs. Furthermore, we showed that cholinergic components were up-regulated and subunits/subtypes of acetylcholine receptors altered during osteoblast differentiation. To our knowledge, this is the first report demonstrating that osteoblasts express specific acetylcholine receptors and cholinergic components and that ACh plays a possible role in regulating the proliferation and differentiation of osteoblasts.

Recent studies have suggested that nicotine critically affects bone metabolism. Many studies have examined the effects of nicotine on proliferation and differentiation, but the underlying molecular mechanisms remain unclear. We examined cell cycle regulators involved in the proliferation and differentiation of MC3T3-E1 cells. Nicotine induced cell proliferation in association with p53 down-regulation and cyclin D1 up-regulation. In differentiated cells, nicotine reduced alkaline phosphatase activity and mineralized nodule formation in dose-dependent manners. Furthermore, p53 expression was sustained in nicotine-treated cells during differentiation. These findings indicate that nicotine promotes the cell cycle and inhibits differentiation in association with p53 regulation in pre-osteoblastic cells.

Most previous reports on neurilemmoma of the head and neck region describe a solitary tumor. Although most reports on multiple neurilemmoma considered these lesions to belong to von Recklinghausen disease, atypical cases of von Recklinghausen disease were also included in some reports. Recently, these multiple neurogenic tumors have been classfied into neurofibromatosis type 1 and 2 (NF 1, 2). NF 1 is considered as original von Recklinghausen disease, whereas NF 2 refers to atypical von Recklinghausen disease, which has been reported along with multiple neurilemmoma, including other neurogenic tumors. It is well known that NF 2 is distinguished from NF 1 both clinically and genetically. The clinical criteria for NF 1 and NF 2 have also been described. NF 2 is often associated with meningioma and neurilemmoma. The present case had both meningioma and neurilemmoma, which occurred at multiple sites, such as the tongue, mediastinum, and brain. Considering recent reports that the gene of atypical multiple neurilemmoma may be identical to that of NF 2, we regarded the present case to be NF 2.

Recently, mutations of the fibroblast growth factor receptor ( FGFR ) genes have been detected in syndromic craniosynostosis. We examined nucleotide sequences of FGFR2 in Japanese craniosynostosis patients (Crouzon syndrome: 9 cases; Apert syndrome: 6 cases; scaphocephaly: 3 cases as non-syndromic patients) by polymerase chain reaction (PCR) followed by direct sequencing methods. The results demonstrated FGFR2 heterozygous mutations at codons 252, 290 of exon 7, and at codon 342, 354 of exon 9 in Crouzon syndromes. In Apert syndrome patients, Ser252Trp and Pro253Arg were detected in five and one patients, respectively. No mutation was detected in one case of Crouzon, all cases of scaphocephaly and healthy individuals. Thus far sequence analysis of FGFR2 in syndromic craniosynostosis has been reported in many white patients, whereas in Japanese only several cases have been studied. The current study with 18 patients confirmed that a similar series of mutations occur in Japanese patients as in white patients regardless of ethnicity and environment. The frequency of the mutation was 82% (9/11 cases) in Japanese Crouzon patients. The ratio of S252W:P253R was 5 : 1 in Japanese Apert patients. Moreover, in Japanese Apert patients, complication rate of cleft palate was 60% for mutation of Ser252Trp and 0 of 2 patients for Pro253Arg, with their syndactyly score being 4.90 and 5.50, respectively.

52才の男性が筋力低下と呼吸困難を主訴に来診した. 15年前より関節痛とレイノー症状, 5年前より間質性肺炎を指摘され, 3カ月前に筋肉痛と筋力低下を自覚し始めた.知能は正常であるが類宦官症を認め原発性性腺機能低下症があり,染色体は核型47XXYであった.血中筋原性酵素の著明な上昇,筋電図所見,筋肉生検所見より多発性筋炎と診断した.抗核抗体,抗Jo-1抗体が陽性であった.間質性肺炎を伴う重複型多発性筋炎と判断し免疫抑制療法を加え寛解した. Klinefelter症侯群に多発性筋炎を発症した報告は,著者らの検索した範囲では見当らず初めての報告と考える.

Transducing-like enhancer of split 3 (TLE3), one of the Groucho/TLE family members, targets Runx2 transcription and suppresses osteoblast differentiation in bone marrow stromal cells (BMSCs). Here, we identify Wnt responsive elements of the TLE3 promoter region through comparative genomic and functional analyses and show that expression of TLE3 is increased by Wnt signaling, which is important for osteoblast differentiation. We also demonstrated that TLE3 is able to suppress canonical Wnt signaling in BMSCs. Taken together, our data suggest that induction of TLE3 by Wnt signaling is part of a negative feedback loop active during osteoblast differentiation.