鵜澤 一弘

Epstein-Barr virus陽性粘膜皮膚潰瘍(EBVMCU)は,口腔咽頭,皮膚,消化管に生じる成熟B細胞腫瘍である。2017年に造血器腫瘍のWHO分類においてEBVMCUの疾病概念が確立したが,口腔内に生じたEBVMCUの報告例は自験例を含め未だ稀である。今回,右側上顎歯肉部の有痛性腫瘤を1ヵ月前から認めた79歳女性の症例について報告する。患者は関節リウマチで11年間メトトレキサートを使用していた。生検標本の病理所見とPET-CTの画像所見よりEBVMCUの診断に至った。EBVMCUは化学療法を行わずとも寛解が期待できる疾患であり,今回,MTXを休薬することで自然寛解に至っており,その後18ヵ月間再発は認めていない。(著者抄録)

2010年1月～2020年12月に当院歯科・顎・口腔外科を受診した12歳以下の小児158名(男児97名、女児61名)を対象とする臨床的観察研究を行い、性別、年齢、受傷時刻、受傷原因、軟組織受傷、歯の外傷、顎顔面骨折の有無を調べた。年齢別にみると1歳と2歳児がそれぞれ20人と最も多く、低年齢児に多くみられ、0から5歳までが全体の60%を占めていた。受傷時刻は12時から18時の時間帯が73名(46.2%)と最も多く、受傷の原因は転倒が79名(50.0%)と大部分を占めていた。軟組織受傷は94名(59.5%)であり、受傷部位は口唇35名(37.3%)、歯肉24名(25.5%)、舌19名(20.2%)となっていた。歯の外傷は58名(36.7%)であり、3歳児に最も多くみられ(15.5%)、以下、4歳(13.8%)と5歳(13.8%)の順であった。顎顔面骨骨折は11名(7.0%)であり、下顎骨骨折が10名、上顎骨骨折が1名であった。

We report a rare case of high-grade MEC developed in the palate. Myoepithelial carcinoma (MEC) is an uncommon malignant tumor, which is also known as malignant myoepithelioma. It generally occurs in major salivary glands. A 79-year-old female presented complaining a painless swelling on the left side palate which grew rapidly in 3 weeks. A soft and pendulous mass with approximately 42 × 30 mm in diameter with ulceration was observed in the palate.Radiological imaging analyses (e-CT, e-MRI and FDG-PETCT) showed an irregular mass of the left palate with bone invasion into the palatal bone and suggested potential metastases in submandibular lymph nodes. A biopsy was performed and diagnosed as MEC of the palate. Surgical tumor resection and radical neck dissection were performed under general anesthesia. Pathologic evaluation showed that the lesion was mainly consisted of atypical cells with abundant eosinophilic cytoplasm including a mixture of spindled or epithelioid cells. Immunohistochemically, the lesion included specifically and diffusely positive for S-100, CD10, calponin, GFAP, and p63. Cell proliferation activity assessed by the Ki67 labeling index was 55 %. From these findings, the lesion was finally diagnosed as MEC of the palate. The patient has been followed for 6years without a recurrence.

Basal cell adenoma (BCA) is classified in benign salivary gland tumors by WHO in 1991. Incidence rate of BCA is 1.0 %–3.7 % of all salivary gland tumors. BCA tend to occur in parotid glands and those of minor gland origin is rare. We present a case of BCA of minor salivary gland in the alveolus of the anterior maxilla. A 41-year-old man was referred to our department because of a swelling in the left alveolus of the anterior maxilla. On the basis of clinical findings, the diagnosis was a benign tumor of the alveolus of the anterior maxilla and the tumor was removed under general anesthesia. The tumor was well-defined, no invasion of surrounding tissue, and was easily resected. Immunohistochemically, tumor cells showed positive reaction for S-100 protein, cytokeratin (AE1/AE3), p40, vimentin and epithelial membrane antigen (EMA). On the contrary, Ki-67 labeling index that is said to be an indicator of malignancy was low. Thus, the tumor diagnosed BCA. One year and six months have passed from the operation, there is no sign of recurrence and metastasis.

Herein, we present a sporadic case of white sponge nevus (WSN) on the cheek mucosa in a 27-year-old Japanese man. A definite diagnosis of WSN was obtained using the typical clinical appearance and microscopic features of the lesions. Histopathological and cytological studies should be conducted to differentiate this condition from other oral premalignant white lesions. A molecular mutation analysis revealed a missense mutation in exon 1A of the keratin 4 gene, which is correlated with the development of WSN. Hence, genetic analysis must be performed to obtain an accurate diagnosis in sporadic cases. The temporary regression of the lesion after treatment with oral amoxicillin hydrate (AMPC) 750 mg/day indicates that antibiotics are effective in symptomatic cases.

頭頸部癌に対して分子標的薬であるセツキシマブ(Cmab)が2012年12月に適用承認がされた。今回,再発転移口扁平上皮癌に対して当科にてCmabとシスプラチン(CDDP)とフルオロウラシル(5-FU)の併用化学療法を施行した4症例を検討した。症例は36歳〜64歳,男性3例,女性1例であった。原発巣は舌3例,下顎歯肉1例であった。再発・転移部位は肺4例,頸部リンパ節3例,舌1例,胸椎1例,外腹膜壁1例,甲状腺1例であった(重複あり)。1例は経過中に心筋梗塞を発症し,Cmab併用化学療法を中止し,評価不能であった。2例においては転移腫瘍がやや縮小したが治療効果判定としてはstable disease(SD)であった。1例における治療効果判定はprogressive disease(PD)であった。副作用は皮膚毒性,口内炎,嘔気,心筋梗塞,肺塞栓症,infusion reaction(IR)などが出現した。心筋梗塞,肺塞栓症,IRなどは生命を危険にさらす重大な副作用であるので,Cmab併用化学療法には全身管理が非常に重要である。(著者抄録)

The main symptom of Kimura disease (KD), a rare, chronic inflammatory disorder of unknown etiology, is painless granulation preferentially developing in the head and neck region's soft tissues, especially in the parotid and submandibular glands and neck lymph nodes. KD is characterized clinically by peripheral blood eosinophilia and elevated serum immunoglobulin E (IgE) levels and histopathologically by high capillary vascularization and lymphoid follicle formation with eosinophil infiltration in the connective tissues. Angiolymphoid hyperplasia with eosinophilia (ALHE), a rare benign tumor of unknown etiology, also occurs in the head and neck frequently as single/multiple angiomatous papules/nodules varying from light brown to pink. Histopathologically, accelerated endothelial cell proliferation and high lymphocyte and eosinophil infiltration are observed. Due to KD's and ALHE's similar clinical and pathological symptoms, differential diagnosis is challenging. Their manifestations include a painless head/neck subcutaneous swelling. Here, we have described a case involving a 44-year-old man who presented with an upper lip mass that increased in size for 1 year. Laboratory studies revealed leukocytosis and eosinophilia. Computed tomography and magnetic resonance imaging examinations revealed a submucosal lip mass. Suspect minor salivary gland malignancies and lymphomas were surgically excised. Histopathologically, we observed marked lymphoid follicle proliferation with inflammatory cell, including eosinophil infiltration, capillary proliferation, and a developed vessel not normally present in the lip. Because differentiating between KD and ALHE is difficult, clinical differentiation was our focus. The Asian male patient with lymphadenopathy, eosinophilia, and high IgE levels was finally diagnosed with KD that has not recurred in 1 year.

Langerhans cell histiocytosis (LCH） is a rare disease caused by excessive proliferation of Langerhans cells in various regions of the body. Previous studies have reported that 10% of LCH occurs in the oral and maxillofacial regions. In the oral and maxillofacial regions, swelling and pain are the typical symptoms, and jaw fracture and tooth loss are often seen in advanced cases. In this study, we present the results of a literature review of LCH that developed in the oral and maxillofacial regions. The history, pathological conditions, diagnosis, treatment, and progress of LCH are discussed.

Semaphorin family members (Semaphorins SEMAs) consist of a large family that includes both secreted and membrane-associated proteins that are originally found to provide axon guidance in selected areas for neural development. The SEMAs have diverse biologic activities such as neuronal cellular migration, axon guidance, vasculogenesis, branching morphogenesis, and cardiac organogenesis. Recent studies have reported that SEMAs also play crucial roles in various carcinomas. However, the association and their role in oral squamous cell carcinoma (OSCC) are still poorly understood. In this study, we evaluated SEMAs mRNA expression in OSCC-derived cell lines using quantitative reverse transcriptase-polymerase chain reaction (qRT-PCR) analysis and revealed the altered gene expression profile of the SEMAs in OSCC cell lines compared with human normal oral keratinocytes. We found that tendency for up-regulation of SEMA3E, 4C, 5A, 6B, and 7A in OSCC-derived cell lines. Especially, SEMA3E and SEMA7A mRNA were significantly up-regulated in all OSCC-derived cell lines. On the other hands, SEMA3A, 3C, 3F, and 6A mRNA were significantly down-regulated in all OSCC-derived cell lines. The expression and role of SEMAs in OSCC depend on the forms and may provide novel insights and therapeutic target for OSCCs.

Laugier-Hunziker-Baran (LHB) syndrome is an acquired, macular hyperpigmentation of the lips, and oral mucosa, often associated with pigmentation of the nails, palms, and fingertips. LHB syndrome is considered to be a benign disease with no systemic manifestation or malignant potential. Normally, no treatment is required for this condition, unless for esthetic reason, mainly due to pigmentation on the lip mucosa. Here we present a case of LHB syndrome, 32-year-old male, whose pigmentations on his tongue, buccal mucosa, palate, and ventral surface of fingertip, diagnosed by clinical and dermoscopic findings.

口腔扁平上皮癌は高齢者に多く発生し、年齢や内科的疾患を理由に手術不適応となる場合も多い。今回、手術不適応の患者にサイパーナイフ治療を行った32例(初発20例、再発12例)の成績を報告した。完全奏効(CR)が16例、部分奏効(PR)が7例、安定(SD)が3例、増悪(PD)が6例であり、初発例ではCRが12例、PRが3例、SDが2例、PDが3例、再発例ではCRが4例、PRが4例、SDが1例、PDが3例であった。奏効(CR+PR)率は全体で72%、初発例で75%、再発例で67%であった。早期有害事象を25例(78%)、晩期有害事象を6例(19%)に認めた。早期有害事象は口内炎が主症状であり、Grade 2までの事象であった。晩期有害事象の内訳は放射線性骨髄炎5例、皮膚瘻孔1例であった。治療効果に影響している因子を明らかにするため、「性別」「年齢」「PS分類」「原発部位」「病理組織型」「TNM分類」「Stage分類」「照射時期」「PTV」などについて多変量解析を行った結果、有意な因子として「T分類」「Stage分類」「照射時期」「PTV」が抽出された。

© 2019 Japanese Stomatological Society Basaloid squamous cell carcinoma (BSCC) is a histologically distinct variant of squamous cell carcinoma (SCC). In this report, we present a case of a 67-year-old man diagnosed with BSCC in the mandibular gingiva. The patient underwent resection of the gingival tumor with a partial mandibulectomy and a functional neck dissection. The mandibular gingiva was reconstructed with forearm free flap. The patient remained in good general health and free of disease for 44 months.

© 2019 Japanese Stomatological Society Ameloblastic carcinoma (AC) is an odontogenic malignant tumor which combines the histological features of ameloblastoma and cytologic atypia. The key point of the diagnosis and clinical characteristics of AC have not been well defined, as it is a rare type of tumor. Hence, in this study, we present’ for better clarity the diagnostic flowchart for AC, ameloblastoma, and metastasizing ameloblastoma.

Tetraspanin 15 (Tspan15) is a member of the tetraspanin family, which is associated with various biological events and several diseases, however, its role in human oral squamous cell carcinoma (OSCC) remains unknown. The current study aimed to clarify the role of Tspan15 in OSCC. The mRNA and protein expression levels of Tspan15 were up-regulated in OSCC cases and OSCC-derived cell lines. Significant up-regulated Tspan15 expression was found in the advanced OSCC cases; primary tumoral size (P = 0.042), regional lymph node metastasis (P = 0.036) and TNM classification (P = 0.024). The decreased expression of Tspan15 did not significantly affect cellular proliferation, whereas tumoral invasion and migration activities were suppressed in Tspan15-down-regulated cells, suggesting that Tspan15 might activate metastasis-related signaling. Moreover, in the Tspan15-down-regulated cells, the expression of a disintegrin and metalloproteinase (ADAM) 10 was also down-regulated and the cells secreted less soluble N-cadherin compared with control cells. And weak immunoreactivity of β-catenin in the nucleus was detected in Tspan15-down-regulated cells compared with the control cells. These findings suggested that overexpression of Tspan15 positively regulates development of OSCC, and that ADAM10, N-cadherin, β-catenin might be involved in the Tspan15-mediated pathway. These unusual conditions of cell adhesion molecules may lead to high metastasis rate found in Tspan15-overexpressing cases.

40歳女。パノラマX線写真における左側下顎部の透過像の精査を主訴とした。パノラマX線写真では上下顎骨内に複数の透過性病変を認め、嚢胞と近接した歯の根尖部では歯槽硬線がやや不明瞭であった。左下6番より生検を施行し、病理組織学的に単純性骨嚢胞と診断した。全身麻酔下に上下顎すべての骨透過性病変に対して掻爬術を施行した。病理組織学的にはいずれの嚢胞腔内からも上皮成分は認めなかったが、左上3番部の嚢胞腔から線維性結合組織を介在するセメント質様の硬組織を認め、セメント質骨性異形成症の診断とした。

症例は68歳女性で、先天性第V因子欠乏症の既往歴があった。今回、左側舌縁部の潰瘍を指摘され、精査加療目的で当科紹介となった。造影CTで、左側舌縁部に28×23mm大の造影効果を示す境界不明瞭な腫瘤を認めた。また、左側レベルIIAに内部造影性不均一な12×12mm大の腫大したリンパ節を認めた。臨床診断は左側舌癌(T2N1MO、stage III)で、生検により扁平上皮癌の診断を得た。周術期の対応について当院血液内科に対診を行い、新鮮凍結血漿(FFP)の試験投与を施行した。FFP投与終了1時間後に、第V因子活性は12%から32%まで補正され、それに伴ってPTが13.8sec、APTT値が55.2secまで改善した。全身麻酔下に舌部分切除術、左側機能的頸部郭清術、気管切開術を施行した。手術は、術後出血を考慮し再建術は行わず、舌部分切除後は縫縮した。また、気管切開術は咽頭浮腫や後出血による気道閉塞を考慮し施行した。帰室後もFFPを6単位投与し、術後1日目から3日目までFFPを毎日6単位ずつ投与した。術後3日目に第V因子活性は75%まで上昇し、創部からの出血も認められなかったためFFPの投与を中止した。術後4日目には持続吸引ドレーン量が5mL以下となり、後出血なく経過したため、術後6日目に第V因子活性を確認してドレーンを抜去した。病理組織学的診断は、舌扁平上皮癌(pT4aN2bM0、stage IV A)であった。術後47日目より放射線治療を施行し、術後4年2ヵ月が経過した現在も再発所見はなく経過観察中である。