Language：English   Publisher：(一社)日本脳神経血管内治療学会  

症例は33歳女性で、全身性エリテマトーデスであり、昏睡状態で発見された。MRIで右内頸動脈閉塞と右中大脳動脈領域の広範な虚血性脳卒中が認められたため、血管内血栓回収術を施行した。術後1日目に意識が回復したが、頭部CTで浮腫と出血の悪化がみられたため、同日に減圧開頭術を行った。術後の全身CTで肺塞栓症と腎梗塞が明らかになった。抗リン脂質抗体症候群(APS)が疑われたが、ループスアンチコアグラントのための臨床検査の結果は陰性であった。患者はステロイドを長期間使用していたことからステロイド誘発性血栓症が疑われた。5日目に抗凝固療法を開始した結果、12日目に急性心筋虚血と左下肢の深部静脈血栓症を発症した。最初のイベントから25日後に左頭頂葉の脳梗塞に起因する意識障害を示した。患者は短期間に多臓器塞栓を発症し、多臓器不全が確認された。劇症型APS疑いと考え、集中治療と血漿交換療法を開始したところ回復し、退院となった。

Language：Japanese   Publisher：Surgical Neurology International  

Background: In this study, we examined the impact and degree of lumbar stenosis on cerebrospinal fluid (CSF) protein concentration. Methods: In this retrospective study, we analyzed protein concentrations in CSF samples of 61 patients with lumbar spinal stenosis (LSS) obtained during pre-operative myelography. Patients were divided into two groups: those showing no block to contrast (Group A) versus those showing medium block to contrast below the lumbar puncture level (Group B). Results: The CSF protein concentration in Group B (104.3 ± 59 g/dL) patients with medium block was significantly greater than that in Group A (65.1 ± 33 g/dL) patients without medium block. Conclusion: A higher average CSF protein concentration was seen in Group B patients with significant lumbar stenosis versus Group A patients without significant lumbar stenosis. Theoretically, damage to the cauda equina in patients with LSS may cause these elevated CSF protein levels.

DOI： 10.25259/SNI_610_2024

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PubMed

Language：Japanese   Publisher：World Neurosurgery  

Objective: Spondylotic changes in the cervical spine cause degeneration, leading to cervical spinal canal stenosis. This stenotic change can affect cerebrospinal fluid (CSF) dynamics by compressing the dural sac and reducing space in the subarachnoid space. We examined CSF dynamics at the craniovertebral junction (CVJ) using time-spatial labeling inversion pulse magnetic resonance imaging (Time-SLIP MRI) in patients with cervical spinal canal stenosis. Methods: The maximum longitudinal movement of the CSF at the CVJ was measured as length of motion (LOM) in the Time-SLIP MRI of 56 patients. The sum of ventral and dorsal LOM was defined as the total LOM. Patients were classified into 3 groups depending on their spinal sagittal magnetic resonance imaging findings: control (n = 27, Kang classification grades 0 and 1), stenosis (n = 14, Kang classification grade 2), and severe stenosis (n = 15, Kang classification grade 3). Results: Time-SLIP MRI revealed pulsatile movement of the CSF at the CVJ. The mean total, ventral, and dorsal LOM was 14.2 ± 9, 8.1 ± 5.7, and 3.8 ± 2.9 mm, respectively. The ventral LOM was significantly larger than the dorsal LOM. The total LOM was significantly smaller in the severe stenosis group (6.1 ± 3.4 mm) than in the control (16.0 ± 8.4 mm) or stenosis (11 ± 5.4 mm) groups (P < 0.001, Kruskal-Wallis H-test). In 5 patients, postoperative total LOM was improved after adequate decompression surgery. Conclusions: This study demonstrates that CSF dynamics at the CVJ are influenced by cervical spinal canal stenosis. Time-SLIP MRI is useful for evaluating CSF dynamics at the CVJ in patients with spinal canal stenosis.

DOI： 10.1016/j.wneu.2024.02.020

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PubMed

Language：Japanese   Publisher：(一社)日本脳卒中学会  

症例は73歳男性.起床時に左共同偏視,右片麻痺および全失語に気付かれ,救急搬送された.超急性期脳梗塞の診断で,機械的血栓回収療法を施行する際に左ICA無形成が判明した.左後交通動脈(posterior communicating artery:PcomA)の閉塞を認め,同血管を主要な側副血行路として,左大脳半球領域は灌流されていると判断した.左VAを経由して,閉塞していた左PcomAへアプローチし,同血管の再開通を得て,症状の改善が得られた.ICA無形成は稀な破格で0.01%未満の発症頻度とされる.通常は無症候性だが,主要な側副血行路の閉塞により症状が顕在化する.頭部CTによる頸動脈管の有無の確認や,側副血行路の把握が重要である.通常とは異なる経路でのデバイス誘導が必要な場合があり,デバイス選択の配慮,また潜在的な動脈瘤や動脈硬化性病変の存在に注意した慎重な操作が求められる.(著者抄録)

Language：Japanese   Publisher：Surgical Neurology International  

Background: Utilizing computed tomography (CT) studies, we correlated cervical spinal canal diameters (SCDs) with pedicle size between the C3 and C7 levels to more safely perform posterior cervical surgery. Methods: We retrospectively analyzed CT studies for 71 patients with cranial or spinal disorders and correlated the cervical SCD with the pedicle outer width (POW) between the C3 and C7 levels. Patients were divided into normal (SCD ≥12 mm at any level, n = 30) and stenosis groups (SCD <12 mm at any level, n = 41). Results: C7 exhibited the largest SCD and POW values, while C3 and C4 exhibited the smallest SCD and POW values. Moderate correlations (r = 0.3, P = 0.002) were observed at the C3 and C4 levels but no significant correlations were observed from the C5 to C7 levels. For SCD values, the normal group demonstrated significantly greater values between the C3 and C7 levels versus the stenosis group. For POW values, only the C4 level differed significantly between the two groups (P = 0.014, Mann–Whitney U-test). Conclusion: Preoperative pedicle size evaluation remains an essential manoeuvre before performing cervical C3–C7 pedicle screw placement. In 71 cervical CT studies, we found no consistent correlation between POW and SCD values, indicating that it is difficult to estimate POW values based on spinal canal size.

DOI： 10.25259/SNI_590_2024

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PubMed

Language：English   Publisher：The Japanese Society for Neuroendovascular Therapy  

<p><b>Objective:</b> Catastrophic antiphospholipid syndrome (CAPS) is a disease characterized by a poor prognosis and a high mortality rate, leading to systemic thrombosis. Approximately two-thirds of CAPS cases are associated with conditions such as infections, malignancies, surgical interventions, and events linked to the disease activity of systemic lupus erythematosus (SLE). Herein, we present a case of CAPS with multiorgan ischemia following ischemic stroke.</p><p><b>Case Presentation:</b> In this case report, a 33-year-old woman with a history of SLE and prolonged steroid use manifested impaired consciousness. Detection of the right internal carotid artery (ICA) occlusion led to successful ICA recanalization through endovascular thrombectomy. Postoperatively, she experienced pulmonary embolism and renal infarction. Although antiphospholipid syndrome (APS) was suspected, APS-related antibodies were negative. Anticoagulation therapy was initiated, presuming corticosteroid-induced thrombosis. However, she developed multiorgan thrombosis, culminating in multiple organ failure. Based on her clinical course, a diagnosis of CAPS was established. Intensive care and plasma exchange therapy were instrumental in her recovery, and she was discharged with a modified Rankin Scale score of 4.</p><p><b>Conclusion:</b> When encountering multiorgan ischemia following ischemic stroke in a young adult patient with an autoimmune disease, the consideration of CAPS as a differential diagnosis is crucial, even if APS-related antibodies test negative.</p>

DOI： 10.5797/jnet.cr.2024-0023

PubMed

CiNii Research

Language：Japanese   Publisher：Surgical Neurology International  

Background: Although rare, cases of hypophysitis resembling a pituitary abscess (PA) have been reported. Differential diagnosis between hypophysitis and PA is crucial as the two diseases require different treatments. Case Description: A 38-year-old woman with headaches underwent head magnetic resonance imaging (MRI), which revealed an 11-mm mass lesion in the sella turcica. Due to breastfeeding, contrast-enhanced MRI was avoided. Pituitary adenomas and Rathke’s cleft cyst (RCC) were suspected, and she was initially treated conservatively. Five months later, she acquired syndrome coronavirus two infections, and while the fever subsided with acetaminophen, the headache persisted. One month later, the headache worsened, followed by fever and diabetes insipidus. MRI revealed a pituitary cystic mass with ring-shaped contrast enhancement on T1-weighted MRI and increased signal intensity on diffusion-weighted imaging (DWI). PA was suspected, and emergency endoscopic transsphenoidal surgery was performed. The microbiological examination of the yellowish-brown content drained from the cystic mass was negative. Microscopically, the cystic lesion was covered with ciliated columnar epithelium and stratified squamous epithelium, with a dense inflammatory cell infiltrate consisting mainly of lymphocytes and plasma cells observed around the cyst. This supported the diagnosis of secondary hypophysitis associated with RCC without PA. Conclusion: We report a case of hypophysitis secondary to RCC resembling PA with ring-shaped contrast enhancement on MRI and increased signal intensity on DWI. This case emphasizes the need for cautious diagnosis of secondary hypophysitis due to RCC in individuals with MRIs and clinical manifestations resembling an abscess.

DOI： 10.25259/SNI_947_2023

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PubMed

Language：English   Publisher：The Japan Neurosurgical Society  

<p>Internal carotid artery aplasia or hypoplasia above the cervical bifurcation is rare, occurring in less than 0.01% of the general population. Unilateral neurocristopathy complicated by unilateral internal carotid artery agenesis or hypogenesis has been reported, but bilateral internal carotid artery hypoplasia is rare and scarcely reported. Herein, we report a novel case of Treacher Collins syndrome complicated by bilateral internal carotid artery hypoplasia. A 94-year-old woman presented with complaints of headache and vomiting. Computed tomography revealed a subarachnoid hemorrhage and dysplasia of the bilateral zygoma, mandible, and external auditory meatus. The patient had severe hearing loss and visual impairment. Computed tomography angiography revealed bilateral internal carotid artery hypoplasia and multiple aneurysmal changes in the intracranial arteries. We diagnosed the patient with a ruptured anterior inferior cerebellar artery aneurysm and performed coil embolization. The patient's unique facial features were consistent with neurocristopathy, especially Treacher Collins syndrome. Developmental anomalies of neural crest cells can present as vascular abnormalities and craniofacial malformations. Special care is required for endovascular treatment and airway management in cases of neurocristopathy because of the specific craniofacial anomalies.</p>

DOI： 10.2176/jns-nmc.2022-0267

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CiNii Research

Language：Japanese   Publisher：Neurosurgical Review  

To assess the use of indocyanine green (ICG) fluorescence endoscopy to evaluate pituitary blood flow in craniopharyngioma resection and its possible impact on intraoperative decisions regarding pituitary stalk processing. Patients with craniopharyngiomas who had undergone transsphenoidal surgery since March 2021, when an ICG endoscope was introduced at the Kagoshima University Hospital, were included in the study. When targeted tumor removal was approaching completion, 10 mg of ICG was administered intravenously to evaluate blood flow in the pituitary stalk and gland. ICG signals and endocrinological status before and after surgery were evaluated retrospectively. Pituitary stalk and gland blood flow were evaluated as positive (++), weakly positive (+), and no signal (−).Ten patients with craniopharyngiomas underwent transsphenoidal surgery using an ICG endoscope (mean age 56.6 ± 14.2 years; 40% male). Among the eight patients in whom the pituitary stalk was preserved, pituitary function with positive signal on the stalk was intact in two. Two other patients with weakly positive stalk and positive pituitary gland signals showed intact function or minimal pituitary dysfunction. Four patients had impairments in more than three axes with poor ICG signals in the stalk or pituitary gland. Two patients underwent pituitary amputation because of high tumor invasion and lack of ICG signal in the stalk after tumor removal, resulting in panhypopituitarism. A negative ICG signal in the pituitary stalk is likely to indicate postoperative pituitary function loss. Craniopharyngioma surgery using ICG endoscopy may be useful for predicting endocrine prognosis and improving tumor outcomes.

DOI： 10.1007/s10143-023-02223-w

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PubMed

Language：English   Publisher：(一社)日本脳神経外科学会  

症例は94歳女性で、先天性頭蓋顔面異形成であった。1日前から頭痛および嘔吐を呈していた。重度の難聴および視覚障害があり、コミュニケーションは困難であった。CTにて、くも膜下出血、両側頬骨、下顎骨、外耳道の異形成が認められた。CT angiographyにて、頸動脈低形成および頭蓋内動脈に多発性動脈瘤性変化を認めた。前下小脳動脈瘤破裂と診断し、コイル塞栓術を施行した。患者の特異的顔貌はTreacher Collins症候群と一致していた。術前に誤嚥性肺炎を発症しており、術後6日目まで呼吸管理を行った後、抜管した。その後、意識状態の改善は認められず、下顎骨および鼻腔異形成による気道閉塞のため呼吸状態は悪化した。患者の家族は侵襲的蘇生を望まず、術後14日目に死亡した。

Language：Japanese   Publisher：Pituitary  

Purpose: Delayed postoperative hyponatremia (DPH) is a unique complication of transsphenoidal surgery (TSS) in pituitary tumors. Growth hormone (GH) enhances renal sodium reabsorption; however, the association between postoperative GH reduction and DPH in acromegaly is unclear. This study was performed to clarify the incidence of and the predictive factors for DPH in patients with acromegaly who underwent TSS. Methods: Ninety-four patients with active acromegaly were examined retrospectively. During the postoperative course, patients with serum sodium levels ≤ 134 mEq/L were classified into the DPH group. We compared basic clinical characteristics, tumor characteristics, and preoperative and postoperative examination findings between the DPH and non-DPH groups. Results: DPH occurred in 39 patients (41.5%), and the lowest serum sodium levels were generally observed during postoperative days (PODs) 7–9. They needed a 3-day longer hospital stay than those without DPH. The DPH group had lower preoperative body weight and body mass index. In addition, a transient increase in body weight during PODs 5–7 occurred with a transient decrease in urinary volume in the DPH group. Preoperative and postoperative GH and insulin-like growth factor-1 levels did not differ between the two groups. Conclusion: The findings suggested that lower preoperative weight and a postoperative transient gain in body weight are associated with an increased risk of DPH in acromegaly patients undergoing transsphenoidal surgery.

DOI： 10.1007/s11102-022-01288-y

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Language：Japanese   Publisher：Neuro-Oncology Advances  

Background: Glioblastoma (GBM) is a malignant brain tumor, with radiological and genetic heterogeneity. We examined the association between radiological characteristics and driver gene alterations. Methods: We analyzed the driver genes of 124 patients with IDH wild-type GBM with contrast enhancement using magnetic resonance imaging. We used a next-generation sequencing panel to identify mutations in driver genes and matched them with radiological information. Contrast-enhancing lesion localization of GBMs was classified into 4 groups based on their relationship with the subventricular zone (SVZ) and cortex (Ctx). Results: The cohort included 69 men (55.6%) and 55 women (44.4%) with a mean age of 66.4 ± 13.3 years. EGFR and PDGFRA alterations were detected in 28.2% and 22.6% of the patients, respectively. Contrast-enhancing lesion touching both the SVZ and Ctx was excluded because it was difficult to determine whether it originated from the SVZ or Ctx. Contrast-enhancing lesions touching the SVZ but not the Ctx had significantly worse overall survival than non-SVZ lesions (441 days vs. 897 days, P = .002). GBM touching only the Ctx had a better prognosis (901 days vs. 473 days, P < .001) than non-Ctx lesions and was associated with EGFR alteration (39.4% vs. 13.2%, P = .015). Multiple contrast lesions were predominant in PDGFRA alteration and RB1-wild type (P = .036 and P = .031, respectively). Conclusions: EGFR alteration was associated with cortical lesions. And PDGFRA alteration correlated with multiple lesions. Our results suggest that clarifying the association between driver genes and tumor localization may be useful in clinical practice, including prognosis prediction.

DOI： 10.1093/noajnl/vdad110

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PubMed

Language：Japanese   Publisher：Neuro-Oncology Advances  

Background: Telomerase reverse transcriptase promoter (TERTp) mutations are a biological marker of glioblastoma; however, the prognostic significance of TERTp mutational status is controversial. We evaluated this impact by retrospectively analyzing the outcomes of patients with isocitrate dehydrogenase (IDH)- and TERTp-wild-type glioblastomas. Methods: Using custom next-generation sequencing, we analyzed 208 glioblastoma samples harboring wild-type IDH. Results: TERTp mutations were detected in 143 samples (68.8%). The remaining 65 (31.2%) were TERTp-wild-type. Among the TERTp-wild-type glioblastoma samples, we observed a significant difference in median progression-free survival (18.6 and 11.4 months, respectively) and overall survival (not reached and 15.7 months, respectively) in patients with and without phosphatase and tensin homolog (PTEN) loss and/or mutation. Patients with TERTp-wild-type glioblastomas with PTEN loss and/or mutation were younger and had higher Karnofsky Performance Status scores than those without PTEN loss and/or mutation. We divided the patients with TERTp-wild-type into 3 clusters using unsupervised hierarchical clustering: Good (PTEN and TP53 alterations; lack of CDKN2A/B homozygous deletion and platelet-derived growth factor receptor alpha (PDGFRA) alterations), intermediate (PTEN alterations, CDKN2A/B homozygous deletion, lack of PDGFRA, and TP53 alterations), and poor (PDGFRA and TP53 alterations, CDKN2A/B homozygous deletion, and lack of PTEN alterations) outcomes. Kaplan-Meier survival analysis indicated that these clusters significantly correlated with the overall survival of TERTp-wild-type glioblastoma patients. Conclusions: Here, we report that PTEN loss and/or mutation is the most useful marker for predicting favorable outcomes in patients with IDH- and TERTp-wild-type glioblastomas. The combination of 4 genes, PTEN, TP53, CDKN2A/B, and PDGFRA, is important for the molecular classification and individual prognosis of patients with IDH- and TERTp-wild-type glioblastomas.

DOI： 10.1093/noajnl/vdad078

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Language：Japanese   Publisher：Cancer Medicine  

Background: We aimed to evaluate the mutation profile, transcriptional variants, and prognostic impact of the epidermal growth factor receptor (EGFR) gene in isocitrate dehydrogenase (IDH)-wildtype glioblastomas (GBMs). Methods: We sequenced EGFR, evaluated the EGFR splicing profile using a next-generation sequencing oncopanel, and analyzed the outcomes in 138 grade IV IDH-wildtype GBM cases. Results: EGFR mutations were observed in 10% of GBMs. A total of 23.9% of the GBMs showed EGFR amplification. Moreover, 25% of the EGFR mutations occurred in the kinase domain. Notably, EGFR alterations were a predictor of good prognosis (p = 0.035). GBM with EGFR alterations was associated with higher Karnofsky Performance Scale scores (p = 0.014) and lower Ki-67 scores (p = 0.005) than GBM without EGFR alterations. EGFRvIII positivity was detected in 21% of EGFR-amplified GBMs. We identified two other EGFR variants in GBM cases with deletions of exons 6–7 (Δe 6–7) and exons 2–14 (Δe 2–14). In one case, the initial EGFRvIII mutation transformed into an EGFR Δe 2–14 mutation during recurrence. Conclusions: We found that the EGFR gene profiles of GBM differ among cohorts and that EGFR alterations are good prognostic markers of overall survival in patients with IDH-wildtype GBM. Additionally, we identified rare EGFR variants with longitudinal and temporal transformations of EGFRvIII.

DOI： 10.1002/cam4.4939

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PubMed

Language：Japanese   Publisher：Surgical Neurology International  

Background: Solitary fibrous tumor/hemangiopericytomas (SFT/HPCs) are rare mesenchymal tumors of nonmeningothelial origin that comprises <1% of all central nervous system tumors. Case Description: A 45-year-old male presented with sleep apnea (apnea-hypopnea index was 17.1 events/hour) and dysesthesias of the right upper and lower extremities. The magnetic resonance demonstrated a heterogeneous intradural extra-axial C1 mass with syringobulbia and syringomyelia. The right vertebral angiography revealed a hypervascular mass (i.e., intense tumor staining). With the preoperative diagnosis of a spinal hemangioblastoma, the patient underwent tumor removal. However, intraoperative findings demonstrated that the ventral component of the tumor was intramedullary without a dural attachment. Further, the histological diagnosis was consistent with SFT/HPC (HPC phenotype). The postoperative course was uneventful, and the patient’s symptoms and the syrinxes spontaneously regressed. Conclusion: A 45-year-old male presented a rare spinal intradural lesion at C1 appeared to be a spinal hemangioblastoma, but proved to be SFT/HPC (HPC phenotype) with intramedullary invasion.

DOI： 10.25259/SNI_655_2022

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Language：Japanese   Publisher：Interdisciplinary Neurosurgery: Advanced Techniques and Case Management  

Background: Spinal sagittal malalignment affects a patient's quality of life, and the condition has gained increasing importance. The cranial center of the gravity-sagittal vertical axis (CCG-SVA) helps in assessing sagittal alignment. Two major landmarks of the CCG are used for measuring the CCG-SVA: the midpoint of the nasion-inion line (MNI) and the external auditory canal (EAC). However, the correspondence between these two points has not been proved. In this study, we radiologically examined the positional relationship between them. Methods: We obtained lateral skull radiographs from 87 patients aged between 21 and 91 years. We evaluated the following: the vertical distance between the MNI and the upper margin of the EAC (Distance A), the horizontal distance between the plumb line from the MNI and the anterior margin of the EAC (Distance B), and the horizontal distance between the plumb line from the MNI and the center of the EAC (Distance C). Results: The average values of Distances A, B, and C were 19.6 ± 5.0, 1.65 ± 5.7, and 5.45 ± 5.5 mm, respectively, indicating that the plumb lines dropped from the MNI and the EAC did not match exactly. The measured values showed no sex-specific differences (P > 0.05, Mann–Whitney U test). Conclusions: The present study demonstrated that the MNI and EAC locations had minimal horizontal differences. The CCG-SVA should be measured by considering the differences between the MNI and the EAC.

DOI： 10.1016/j.inat.2021.101299

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Language：Japanese   Publisher：一般社団法人 日本内分泌学会  

DOI： 10.1507/endocrine.97.s.hpt_52

CiNii Research

Language：Japanese   Publisher：一般社団法人 日本内分泌学会  

DOI： 10.1507/endocrine.97.s.hpt_89

CiNii Research

Language：Japanese   Publisher：(一社)日本内分泌学会  

術後残存腫瘍、再発腫瘍に対してガンマナイフ療法を行った下垂体腺腫40例を対象として、その効果と安全性について検討した。非機能性下垂体腺腫28例の照射線量中央値は単回照射15Gy、分割照射27.5-30Gyで、経過中の腫瘍制御率は100%であり、照射後新たにホルモン補充を要した3例のうち1例は照射半年後に副腎不全に陥った。機能性下垂体腺腫は先端巨大症11例、Cushing病1例で、照射線量中央値は25Gyであり、先端巨大症の腫瘍制御率は100%で、照射後8例でIGF-1が正常化したが、Cushing病は難治性で再手術、再照射を要した。また、機能性下垂体腺腫では内頸動脈狭窄3例と動眼神経麻痺1例を認めた。下垂体腺腫に対するガンマナイフ療法の有効性は明らかであったが、機能性下垂体腺腫では治療適応や照射線量、照射方法を再検討する必要がある。

Language：Japanese   Publisher：(一社)日本内分泌学会  

当院で手術を行った下垂体卒中32例(男性16例、女性16例、平均年齢51.7±18.5歳)の画像所見と臨床経過について検討した。その結果、術前症状は頭痛が71.9%と最多で、病理診断は非機能性下垂体腺腫が71.9%と最も多かった。頭部CTにおけるトルコ鞍内の高吸収域は50%にみられ、MRI T1強調像高信号域は56.3%、鏡面像は28.1%、副鼻腔粘膜肥厚は34.3%で認められた。また、術中所見で血腫は62.5%で認められた。尚、退院時ホルモン補充の要否と上記画像所見、あるいは術中所見との間に有意な関連は認められなかったが、頭痛発症例ではホルモン補充を要する傾向があった。

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