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We herein present the case of a 49-year-old female patient presenting with nausea and headache. The patient's medical history included breast cancer with bone and lymph node metastasis. Computed tomography (CT) examination revealed a high-density lesion in the right subdural space, suggesting hematoma. During surgery planned for subdural hematoma drainage, an en plaque subdural yellowish-white tumor was identified, without hematoma. Histopathological examination revealed metastatic breast cancer. The patient was administered predonisolone and her neurological symptoms gradually recovered. However, 12 days after the first operation, the clinical course was complicated by vomiting and rapid loss of consciousness. Emergency CT revealed that the subdural tumor had enlarged and decompression was performed as life-saving surgery. However, the patient's condition progressively deteriorated and she finally succumbed to the disease 2 months after the second operation. The aim of this study was to present the case of a patient with a large en plaque subdural tumor mimicking subdural hematoma and causing rapid loss of consciousness and cerebral herniation.

Patient derived xenografts are essential tools for translational research and preclinical development of novel therapeutic strategies of primary brain tumors. Recent advances in genomics of primary brain tumors revealed molecular classification of primary brain tumors, thus establishment of patient derived xenografts from each subtype of primary brain tumors is urgently needed. However, currently available patient derived xenografts are limited and are from specific subtype of primary brain tumors such as glioblastoma IDH wild type. In this study, we aim to establish patient derived xenografts from primary brain tumors with various molecular characteristics, especially rare primary brain tumors. We got primary brain tumor tissues from patients, dissociated those tissue into single cells, and orthotopically injected those cells into NOD/Shi-scid IL2Rγ KO mouse. We successfully established rare patient-derived xenografts from atypical teratoid rhabdoid tumor and CNS Ewing sarcoma family tumor with CIC alteration, which is recently described as new entity of primitive neuroectodermal tumors of the CNS. We also analyzed histopathological characteristics of these xenografts and found that each xenograft well recapitulated histopathological features of original patients’ resected tumors. These xenografts have advantages for translational research and preclinical development of novel therapeutic strategies for rare primary brain tumors. In addition, further efforts are needed to establish other types of rare primary brain tumors.

Extensive molecular analyses of ependymal tumors have revealed that supratentorial and posterior fossa ependymomas have distinct molecular profiles and are likely to be different diseases. The presence of C11orf95-RELA fusion genes in a subset of supratentorial ependymomas (ST-EPN) indicated the existence of molecular subgroups. However, the pathogenesis of RELA fusion-negative ependymomas remains elusive. To investigate the molecular pathogenesis of these tumors and validate the molecular classification of ependymal tumors, we conducted thorough molecular analyses of 113 locally diagnosed ependymal tumors from 107 patients in the Japan Pediatric Molecular Neuro-Oncology Group. All tumors were histopathologically reviewed and 12 tumors were re-classified as non-ependymomas. A combination of RT-PCR, FISH, and RNA sequencing identified RELA fusion in 19 of 29 histologically verified ST-EPN cases, whereas another case was diagnosed as ependymoma RELA fusion-positive via the methylation classifier (68.9%). Among the 9 RELA fusion-negative ST-EPN cases, either the YAP1 fusion, BCOR tandem duplication, EP300-BCORL1 fusion, or FOXO1-STK24 fusion was detected in single cases. Methylation classification did not identify a consistent molecular class within this group. Genome-wide methylation profiling successfully sub-classified posterior fossa ependymoma (PF-EPN) into PF-EPN-A (PFA) and PF-EPN-B (PFB). A multivariate analysis using Cox regression confirmed that PFA was the sole molecular marker which was independently associated with patient survival. A clinically applicable pyrosequencing assay was developed to determine the PFB subgroup with 100% specificity using the methylation status of 3 genes, CRIP1, DRD4 and LBX2. Our results emphasized the significance of molecular classification in the diagnosis of ependymomas. RELA fusion-negative ST-EPN appear to be a heterogeneous group of tumors that do not fall into any of the existing molecular subgroups and are unlikely to form a single category. Electronic supplementary material The online version of this article (10.1186/s40478-018-0630-1) contains supplementary material, which is available to authorized users.

Gliomas are genetically and histopathologically heterogeneous. Intratumoral heterogeneity in the MGMT promoter methylation status is an important clinical biomarker of glioblastoma. A higher uptake of 11C-methionine in positron-emission tomography (PET) reportedly reflects increased MGMT promoter methylation; however, non-stereotactic comparison of MGMT methylation and 11C-methionine PET images may not be accurate. The present study examined the correlation between 11C-methionine uptake and MGMT promoter methylation in non-enhancing gliomas using stereotactic image-based histological analysis. Data were collected from 9 patients with newly diagnosed non-enhancing glioma who underwent magnetic resonance imaging and 11C-methionine PET during pre-surgical examination. Clinical data were also collected from 3 patients during repeat surgery. The correlation between 11C-methionine uptake and MGMT methylation or cell density was analyzed using histological specimens obtained by multiple stereotactic sampling and an exact local comparison of 11C-methionine PET images and histological specimens was made. A total of 31 stereotactic sample sites were identified. In newly diagnosed cases, the tumor to normal uptake (T/N) ratio revealed a significant positive correlation with MGMT methylation (R=0.54, P=0.009) and a marginal correlation with cell density (R=0.42, P=0.05). In recurrent cases, the T/N ratio demonstrated no correlation with MGMT methylation (R=0.01, P=0.97) or cell density (R=0.15, P=0.70). An increased uptake of 11C-methionine in PET may reflect increased MGMT promoter methylation according to stereotactic image-based histological analysis. 11C-methionine PET could therefore be a useful tool for detecting regional MGMT promoter methylation in non-enhancing primary glioma.

Introduction This study investigates the current state of clinical practice and molecular analysis for elderly patients with diffuse gliomas and aims to elucidate treatment outcomes and prognostic factors of patients with glioblastomas. Methods We collected elderly cases (≥ 70 years) diagnosed with primary diffuse gliomas and enrolled in Kansai Molecular Diagnosis Network for CNS Tumors. Clinical and pathological characteristics were analyzed retrospectively. Various factors were evaluated in univariate and multivariate models to examine their effects on overall survival. Results Included in the study were 140 elderly patients (WHO grade II: 7, III: 19, IV: 114), median age was 75 years. Sixty-seven patients (47.9%) had preoperative Karnofsky Performance Status score of ≥ 80. All patients underwent resection (gross-total: 20.0%, subtotal: 14.3%, partial: 39.3%, biopsy: 26.4%). Ninety-six of the patients (68.6%) received adjuvant treatment consisting of radiotherapy (RT) with temozolomide (TMZ). Seventy-eight of the patients (75.0%) received radiation dose of ≥ 50 Gy. MGMT promoter was methylated in 68 tumors (48.6%), IDH1/2 was wild-type in 129 tumors (92.1%), and TERT promoter was mutated in 78 of 128 tumors (60.9%). Median progression-free and overall survival of grade IV cases was 8.2 and 13.6 months, respectively. Higher age (≥ 80 years) and TERT promoter mutated were associated with shorter survival. Resection and adjuvant RT + TMZ were identified as independent factors for good prognosis. Conclusions This community-based study reveals characteristics and outcomes of elderly glioma patients in a real-world setting. Elderly patients have several potential factors for poor prognosis, but resection followed by RT + TMZ could lengthen duration of survival. Electronic supplementary material The online version of this article (10.1007/s11060-018-2957-7) contains supplementary material, which is available to authorized users.

AIM We evaluated the association between 11C-methionine positron emission tomography (11C-methionine PET) findings, isocitrate dehydrogenase (IDH) gene mutation, and O6-methylguanine-DNA methyltransferase (MGMT) promoter methylation in patients with grade II and III gliomas. MATERIALS AND METHODS Data were collected from 40 patients with grade II and III gliomas who underwent both magnetic resonance imaging (MRI) and 11C-methionine positron emission tomography (PET) as part of their pre-surgical examination. We examined IDH mutation through DNA sequencing, and MGMT promoter methylation through quantitative methylation-specific polymerase chain reaction (PCR). RESULTS A threshold of MGMT promoter methylation of 1.0% was significantly associated with tumor/normal tissue (T/N) ratio. The T/N ratio in samples with MGMT promoter methylation ≥1.0% was higher than that in samples with MGMT promoter methylation CONCLUSION 11C-methionine PET parameters can predict MGMT promoter methylation but not IDH mutation status. 11C-methionine uptake may have limited potential to reflect DNA methylation processes in grade II and III gliomas.

Low-grade glioma (LGG) is often encountered in relatively young individuals, including women of childbearing age. Notably, case series describing pregnant women with LGG have been reported in the literature. The present study reported a case of pilocytic astrocytoma (PA) requiring tumor resection during pregnancy. The patient had a history of γ-knife radiotherapy for a brainstem tumor 17 years previously. The histological diagnosis was unclear. The tumor had remained stable following radiosurgery for 17 years, including during her first pregnancy. However, rapid tumor growth around the fourth ventricle occurred at week 25 of her second pregnancy. Therefore, an urgent tumor resection was performed despite the pregnancy. Partial resection was performed since the tumor had infiltrated the brainstem. The histological diagnosis was PA. The residual tumor volume decreased gradually following the delivery. Immunostaining of the tumor for the expression of progesterone receptor revealed focal staining. It is possible that reproductive factors, including specific hormonal changes during pregnancy, affected the tumor growth. The present study described this rare case of PA, which exhibited rapid growth and required urgent surgery during pregnancy.

We report a case of retroperitoneal cavernous hemangioma. A 77-year-old woman complaining of nausea was admitted to a different hospital in September 2013. Computed tomography (CT) detected a retroperitoneal mass in the left pararenal space. Three years later, repeated CT showed that the tumor had gradually grown in size. On dynamic contrast-enhanced magnetic resonance imaging (MRI), the tumor demonstrated radiographic signs of a liposarcoma. Resection of the mass with left nephrectomy was performed in June 2016, and histopathology showed cavernous hemangioma. Clinical diagnosis of cavernous hemangioma is difficult, and imaging modalities, including CT and MRI, may not be conclusive. The final diagnosis in most cases is established through surgery. This is the 29th case of retroperitoneal cavernous hemangioma to be reported in Japan.

We report a rare case of polypoid leiomyosarcoma of the esophagus that was treated by endoscopic submucosal dissection (ESD). A 63-year-old man with complaints of progressive dysphagia was referred to Hyogo Cancer Center for treatment of esophageal tumor. Esophagoscopy revealed a polypoid tumor 25 mm in diameter on the left side of the upper esophagus. Despite several biopsy specimens, the diagnosis could not be confirmed. Computed tomography showed a protruded, homogeneously enhancing mass in the upper esophagus, but no lymph node enlargement or metastasis. After 1.5 months, the esophagogram showed a filling defect 47 mm in diameter in the upper esophagus. Given this rapid tumor growth, en bloc resection was done by ESD for therapeutic diagnosis. After this treatment, the tumor seemed to grow larger, showing a short stalk and occupying the esophageal lumen. Histopathologically, the tumor comprised pleomorphic spindle cells with mitosis. Tumor invasion involved the lumina propria mucosae and contact with the muscularis mucosae, but not involving the submucosa. Immunohistochemical examination showed positive staining for smooth muscle actin and HHF35, but negative for desmin, caldesmon, CD34, c-kit, DOG1, ALK, S-100 protein and cytokeratin. These histopathological findings were compatible with a diagnosis of esophageal leiomyosarcoma derived from the muscularis mucosae.

The prognosis of leptomeningeal dissemination of recurrent glioblastoma is poor, and chemotherapy results in minimal palliative efficacy. Temozolomide (TMZ) is an established therapy for patients with malignant glioma and the standard of care in parenchymal gliomas; however, few reports have been published with regard to its use for the treatment of leptomeningeal dissemination. Only one report has indicated the radiographic response of leptomeningeal dissemination to a TMZ rechallenge, suggesting a potential causative effect. While bevacizumab is an effective therapy for recurrent glioblastoma, its effect on leptomeningeal dissemination of recurrent glioblastoma remains unclear. The present study reports a case of leptomeningeal dissemination of recurrent glioblastoma in which transient neurological and radiological improvement was observed following chemotherapy with TMZ and bevacizumab. However, five months after the diagnosis of leptomeningeal dissemination the patient succumbed to the disease.

Background: The prognosis of breast cancer patients with brain metastasis (BM) is extremely poor, and the survival after development of BM is very short. We aimed to investigate clinicopathological factors related to significant effects on the prognosis after BM development. Patients and Methods: This is a retrospective study of 75 early breast cancer patients who received the standard of care and subsequently developed BM. Results: Breast cancer subtype was one of the significant predictors for prognosis after BM diagnosis. Luminal HER2 patients had the most favorable prognosis after BM diagnosis (p = 0.011). Favorable performance status (PS) at BM diagnosis (p < 0.001) and a single metastatic brain tumor (p = 0.032) were significantly associated with good prognosis after BM diagnosis. Metastatic time courses of the patients was found not to be significantly associated with survival after BM diagnosis. Univariate and multivariate analysis indicated that luminal HER2 cancer, favorable PS at BM diagnosis, and a single metastatic brain tumor were the independent prognostic factors for survival after BM development, making a decisive influence on local or systemic control. Conclusion: Appropriate treatments for tumor subtypes and to improve the general condition of patients would result in improved outcomes for the patients with BM.

Choroid plexus metastasis (CPM) is extremely rare and originates most frequently from renal cell carcinoma (RCC). We herein report the case of a 58-year-old man who developed a solitary CPM lesion derived from follicular thyroid carcinoma in addition to intraventricular hemorrhage. Computed tomography revealed acute hydrocephalus as a result of the hemorrhage, and we planned endoscopic hematoma evacuation. Since it was too difficult to reach the hematoma, we considered the possibility of a neoplasm and performed a biopsy of the lesion, the results of which led to an accurate diagnosis of CPM in this case. We also review previous reports of CPM originating from thyroid carcinoma compared with RCC.

We report a case in which combination therapy with albumin-bound paclitaxel (nab-paclitaxel) and gemcitabine converted unresectable pancreatic cancer with peritoneal metastases into resectable disease. The patient was a 71-year-old woman with anorexia. Enhanced abdominal computed tomography (CT) showed an atrophic pancreatic body and tail, dilated main pancreatic duct, peritoneal dissemination, portal vein stricture, bile duct stricture and wall thickening, and blockage of the right ureter. She was diagnosed with pancreatic cancer with peritoneal metastases. Curative resection was initially impossible. Combination therapy consisting of nab-paclitaxel and gemcitabine was initiated. The regimen consisted of 28-day cycles of albumin-bound paclitaxel (nab-paclitaxel) (125 mg/m(2) intravenously over 30 min on days 1, 8, 15) and gemcitabine (1000 mg/m(2) intravenously over 30 min on days 1, 8, 15). After 8 cycles of chemotherapy, enhanced CT showed no evidence of the tumor in the pancreatic body and tail or peritoneal metastases. Positron emission tomography with CT (PET-CT) showed no abnormal fluorodeoxyglucose uptake. After pre-operative chemotherapy for 8 months, the patient was underwent distal pancreatectomy with resection of soft tissue that corresponded to the right ureteral tumor seen on enhanced CT. This case showed that chemotherapy consisting of nab-paclitaxel and gemcitabine can be well-tolerated and can convert unresectable pancreatic cancer with peritoneal metastases into resectable disease.

We aimed to analyze the clinical and histological effects of chemotherapy in superficial esophageal squamous cell carcinoma (SESCC). We analyzed tumor samples from five patients with cT1bN1M0 who underwent subtotal esophagectomy following two courses of a new triplet chemotherapy regimen including docetaxel, cisplatin, and 5-fluorouracil (DCF). To assess the histological effects of chemotherapy, resected specimens were analyzed by macroscopic examination, hematoxylin & eosin (HE) staining, immunohistochemical (IHC) staining (p53, Ki-67 and cytokeratin) and periodic acid-Schiff (PAS) staining. All five patients had a pathological T stage of T0/1a-LPM/1a-MM/1b (1/2/1/1) and histological grade of grade1a/1b/2/3 (1/1/2/1). Endoscopic examination revealed substantial shrinkage of lugol-voiding lesions (LVLs) in all cases. One case showed complete LVL disappearance, and resected specimen examination confirmed pathological complete response (pCR). IHC and PAS staining revealed that most initial LVLs were PAS-negative. Obvious viable cells were confirmed in two cases. The other three cases exhibited nuclear atypia and strong expression of p53 and Ki-67 in the basal layer of mucosa or lamina propria mucosae, even though the superficial layer of mucosa showed no obvious LVLs with PAS-positive. p53-positive lesions were also observed in Ki-67-positive. This indicated discordance between the endoscopic findings and histopathological evaluation. DCF chemotherapy alone had a substantial therapeutic effect on SESCC in all cases. However, despite the normal appearance of the mucosal surface, viable cancer cells remained below the basal layer of mucosa. Careful attention should be paid when diagnosing clinical CR, or securing a resection margin of SESCC after DCF chemotherapy.

We elucidated clinicopathological characteristics of chondroblastoma (CB) in Japan, and reliable clinicopathologic parameters predicting local recurrence and/or metastasis. Clinicopathological profiles of 103 CB (80 male, 23 female) in extra-craniofacial bones were retrieved. Numerical scoring of nine pathological and five radiological features was statistically analyzed to determine prognostic significance. Age ranged 8-61 years (average 19.6 years). Frequently involved sites were femur, tibia, calcaneus, patella and humerus. Radiologically, tumors were 2-80 mm (average 31.1 mm) in size. Marginal sclerosis and calcification were common. Histologically, pink cartilage, mitoses, and chicken-wire calcification were often seen. Within a follow-up period [2-260 months (average 53.5 months)], the local recurrence rate was 15.5%. No patient had metastasis. Recurrence was most frequently observed at the femur. By log-rank analysis, only cyst formation in images was significant for predicting recurrence free survival (RFS). By Cox hazard analysis with representative clinico-radiological and pathological features, only age (≥16 years) and cyst formation were significant predictors for RFS. Pathological features were not significant in both uni- and multivariate analyses.

The diagnosis of non-cirrhotic portal hypertension (NCPH), a rare but potentially life-threatening complication in human immunodeficiency virus (HIV)-positive individuals, often occurs only after the emergence of fatal manifestations such as bleeding of esophageal varices. We herein report a female Japanese HIV patient who developed NCPH approximately 4 years after discontinuation of 65 months of didanosine (ddI) administration. The patient presented with severe ascites, bloody bowel discharge, extreme abdominal swelling, and symptoms of portal hypertension but no sign of liver cirrhosis. Examination revealed esophageal varices, oozing-like bleeding from a wide part of the colon, significant atrophy of the right lobe of the liver, and arterio-portal shunting and recanalization from the left medial segment branch of the portal vein to a paraumbilical vein, but no visible obstruction of the main trunk of the portal vein. Treatment for esophageal varices consisted of coagulation therapy with argon plasma after enforcement by endoscopic sclerotherapy and oral administration of β-blockers for elevated portal blood pressure. The patient has not experienced gastrointestinal bleeding in the approximately 5 years since the diagnosis of NCPH. Reviewing this case suggests the importance of suspecting NCPH in HIV patients with liver dysfunction of unknown etiology with a history of ddI and other purine analogs use, as well as the importance of controlling portal hypertension and esophageal varices in the treatment of NCPH.

It is of importance to clarify pathophysiology of diabetic heart diseases such as heart failure and coronary artery disease. We reported a novel clinical phenotype called triglyceride deposit cardiomyovasculopathy (TGCV), showing aberrant TG accumulation in both coronary arteries and myocardium, in a cardiac transplant recipient. Here, we examined autopsied diabetics for TG deposition in cardiovasculature. Consecutive series of hearts from advanced diabetes mellitus (DM) subjects (DM group: DMG, n = 20) and those from age- and sex-matched non-diabetic controls (non DM group: NDMG, n = 20) were examined. The diagnostic criteria of 'advanced DM' was made based on 2014 Clinical Practice Recommendations proposed by the American Diabetes Association. The mean duration of DM was 15.8 years. All DMG suffered from heart diseases including coronary artery diseases and 14 subjects had multi-vessel disease. Tissue TG contents were measured biochemically. Coronary arterial TG contents was significantly higher in DMG compared with NDMG. Spatial distribution of TG in transverse sections of coronary arteries showed TG deposition mainly in smooth muscle cells by Imaging Mass Spectrometry. Abundant TG deposition in coronary artery might be associated with advanced DM.

Background Brain metastases (BM) from esophageal cancer (EC) are rare. Despite of an advance of treatments for the patients with BM from EC, life expectancy and quality of life of these patients are still poor. We present an overview of the patients with BM from EC at a single institute. Methods We retrospectively identified 10 patients with BM from EC treated with surgery, radiation, or a combination of multidisciplinary therapies at Osaka National Hospital between 2003 and 2017 for stages IIb through IV of primary EC (follow-up, > 157 days). Medical records were reviewed to collect demographic and clinical information. Results Median age at diagnosis of BM was 63.5 years (range, 53–79 years). 9 patients were male, and 7 patients had squamous cell carcinoma of EC at the primary esophageal resection. Median overall survival from the commencements of therapy for BM was 156 days (range, 17–5404 days). The interval between the primary esophagectomy and the start of therapy for BM from EC was 298 days (range, 64–860 days). The average score of Karnofsky performance status (KPS) just before a diagnosis of BM was 75 (range, 50–90). On univariate analysis, the patients with the lower score of KPS (P = 0.01) or the shorter interval between the primary esophageal surgery and the start of therapy for BM (P = 0.06) were found to have worsened survival after the therapy for BM from EC. Conclusion The patients who had a poor KPS just before a diagnosis of BM, or the shorter interval between the esophagectomy for the primary EC and the start of therapy for BM, had poor prognosis. Disclosure All authors have declared no conflicts of interest.

We report a case of recurrent breast cancer with involvement of the right adrenal grand, which was resected using laparoscopic surgery. The patient was a 41-year-old woman who underwent duct-lobular segmentectomy for ductal carcinoma in situ. Histopathology showed microinvasion, and tested positive for ER and PR, but negative for HER2, so we applied radiation to the remaining breast and administered tamoxifen. After 9 months, local recurrence was detected and quadrantectomy with axillary lymph node dissection was performed. One year and 10 months later, local recurrence was again detected and a tumorectomy was performed. The adjuvant therapy was changed to an LH-RH analog plus anastrozole and it was administered effectively for 5 years. Left ileal metastasis appeared in the 2nd month after completion of the adjuvant chemotherapy, so radiation was applied and an LH-RH analog plus exemestane administration was started. Three years passed without recurrence, but a right adrenal tumor appeared on computed tomography. The tumor grew over 6 months, so laparoscopic right adrenalectomy was performed. Histopathologically, the tumor tested positive for ER and PR, and negative for HER2 so we diagnosed metastasis of breast cancer, and administered an LH-RH analog plus exemestane. The patient's disease has not progressed in the 3 months since surgery.

We report a case of recurrent esophagogastric junction cancer successfully treated with chemotherapy including trastuzumab. A 66-year-old man reporting black stool was examined, and through upper gastrointestinal endoscopy was found to have a tumor at the esophagogastric junction. Video-assisted thoracoscopic surgery of the esophagus with 3-field lymph node dissection was performed. The final diagnosis was adenocarcinoma(tub1-pap-muc), pT3N3M0, pStage III C. The tumor was at the squamo-columnar junction and classified as Siewert type II . Following surgery, 6 months of adjuvant therapy with S-1 was administered. Computed tomography(CT)confirmed metastases in the lung, liver, and recurrence in the reconstructed stomach. In spite of chemoradiotherapy(FP plus RT)and weekly PTX, these metastases had grown. Pathological examinationshowed overexpressionof humanepidermal growth factor receptor 2(HER2), and treatment with CPT- 11 plus trastuzumab was initiated at postoperative 15 months. A partial response was achieved 3 months later. The lesions in the lung, liver, and reconstructed stomach were controlled with this therapy, but he died from meningeal seeding at postoperative 20 months. These findings suggest that inclusion of trastuzumab in chemotherapy regimens is effective for recurrent HER2-positive esophagogastric junction cancers.

The most frequent relapse site of gastrointestinal stromal tumors(GIST)is the liver. We encountered a patient with longterm survival treated with multidisciplinary treatment, including4 hepatectomies for liver metastases. The patient was a woman aged 69 years at the time of the first medical treatment. She underwent total gastrectomy and S6 segmental hepatec- tomy for a stomach GIST with a hepatic metastasis. The tumor diameter was 24 cm and the mitotic figure was 65/50 HPF. According to the guidelines, it was diagnosed as a high risk GIST based on strong positive immunostaining for CD34 and c-kit. The tumor had metastasized to the liver and greater omentum. She took imatinib in the year following surgery. Because the GIST had spread to the lung1 8 months after the operation, she took imatinib again. Two years after the operation the pulmonary metastasis showed a clinical complete response(cCR)and the CR lasted for 4 years. Six years after the surgery she had a hepatic recurrence in S5, and she underwent an S5/4 partial hepatic resection. Seven years after the first operation, a liver S7 metastasis developed and she underwent S7 partial hepatectomy. Ten years after the first surgery, the GIST relapsed in liver S6 with right adrenal gland permeation. She underwent partial S6/7 liver resection and a right adrenal gland resection. She resumed takingimatinib after this surgical resection. Now, 11 years after the first operation, she is alive with an S1 hepatic recurrence taking sunitinib. Therefore, multidisciplinary treatment with surgical resection prolongs the survival of patients with resectable liver metastases of GIST.

Angiomyolipoma (AML) arising in the liver is rare and usually benign, but it occasionally has malignant potential. A 58-year-old man with a liver tumor identified by a previous doctor with features suggestive of hepatocellular carcinoma on computed tomography (CT) underwent anterior segmentectomy of the liver in 2006. Microscopically, the tumor was composed of exclusively epithelioid cells that were scatteredly positive for human melanoma black 45 on immunohistochemistry. Accordingly, primary hepatic epithelioid AML (eAML) was diagnosed. The patient was subsequently referred to our hospital for follow-up after hepatectomy. He had event-free survival for nearly 7 years. In 2013, two well-defined round nodules were detected in the right lung field by chest CT, and partial pneumonectomy was performed for diagnosis and treatment. Histological examination of the resected lung tissue showed that it was morphologically and immunohistochemically identical to his primary hepatic eAML, leading to the diagnosis of pulmonary metastasis. This paper demonstrates a rare case of malignant hepatic eAML with late recurrence in the lung after hepatectomy.

Amelanotic primary malignant melanoma of the esophagus (PMME) is extremely rare. We report a case of amelanotic PMME that was accurately diagnosed and subsequently treated by radical resection with adjuvant chemotherapy. A 69-year-old woman was admitted to our hospital because of dysphagia. Endoscopic examination revealed a tumor without pigmentation in the upper thoracic esophagus, and melanosis in the lower thoracic esophagus. Tumor biopsy showed proliferation of malignant spindle cells with conspicuous nucleoli. Immunohistochemically, the tumor cells were positive for melanosomes (HMB45). The tumor was diagnosed as PMME. Positron emission tomography and computed tomography showed accumulation of the tracer only in the primary lesion. The preoperative diagnosis according to the extent of the tumor, whether cancer cells have spread to lymph nodes, and whether metastasis has occurred (TNM) classification was malignant melanoma in the upper third of the esophagus, T2N0M0 stage IIA. The patient underwent minimally invasive video assisted esophagectomy in the prone position with three-field lymph node dissection. On the resected specimen, the malignant cells were positive for HMB45, KIT, and melan-A. Masson-Fontana staining demonstrated no melanin pigmentation. The patient was diagnosed with amelanotic PMME and received adjuvant chemotherapy, consisting of dacarbazine, nimustine, cisplatin, and tamoxifen. The patient showed no systemic metastasis, and is alive 18 months after the operation with no evidence of recurrence.

We report 3 cases of extrapulmonary lymphangioleiomyomatosis incidentally found in pelvic and paraaortic lymph nodes in association with uterine cancers. Three women, 47-year-old, 59-year-old, and 71-year-old, respectively, had uterine cancers and underwent hysterectomy, bilateral salpingo-oophorectomy, and pelvic and paraaortic lymph node excision. None of the 3 patients had tuberous sclerosis complex or lymphangioleiomyomatosis in other organs. None had any history of extrinsic hormonal administration. The postoperative pathologic diagnoses were uterine cervical squamous cell carcinoma for the first patient and endometrioid adenocarcinomas for the second and the third patients. Besides these malignant lesions, all 3 patients showed spindle cell proliferation, 2 to 5 mm in size, in 1 to 8 foci of the pelvic and paraaortic lymph nodes. The spindle cells having small polygonal nuclei and inconspicuous nucleoli with palely eosinophilic cytoplasm, reminiscent of immature smooth muscle cells, proliferated in nested and whorling patterns. Neither cellular atypia nor mitotic figures were observed. Immunohistochemically, these spindle cells were positive for α-Smooth Muscle Actin, Desmin, HMB45, Microphthalmia Transcription Factor, Estrogen receptor, and Progesterone receptor. And the network of the vascular-like channels surrounded by these spindle cells was positive for D2-40. From the pathologic and immunohistochemical findings, the spindle cell proliferation in the lymph nodes is best interpreted as lymphangioleiomyomatosis.

Triple-negative breast cancers (TNBCs) do not derive benefit from molecular-targeted treatments such as endocrine therapy or anti-HER2 therapy because they lack those molecular targets. On the other hand, TNBCs have been shown to respond to neoadjuvant chemotherapy (NAC). In this study, we analyzed TNBC patients who were treated with NAC at Osaka National Hospital over a recent 5-year period to clarify the predictive factors for NAC and prognostic factors.Thirty-three TNBC patients underwent sequential NAC with anthracycline (FEC100: 5FU 500 mg/m(2), epirubicin 100 mg/m(2), and cyclophosphamide 500 mg/m(2)/q3w, 4 courses) and taxanes (paclitaxel 80 mg/m(2)/qw, 12 courses or docetaxel 75 mg/m(2)/q3w, 4 courses) from May 2003 to July 2008. Pre-therapeutical and surgical specimens were studied for expressions of ER, PgR, HER-2, EGFR, cytokeratin 5/6, Ki-67, p53 and androgen receptor by immunohistochemistry (IHC). We analyzed clinicopathological factors and molecular markers in regard to the response to NAC and prognosis.Pathological complete response (pCR) was achieved in 12 TNBC patients (36%). The pCR rate in the basal-like phenotype was significantly lower than in the non-basal-like phenotype (23 vs. 64%, respectively: P = 0.02). High pre-operative expressions of Ki-67 (≥50%) and HER-2 (2+) were considered as predictive factors for a better response from NAC. Pre-operative Ki-67 expression showed a significant correlation with disease-free survival (DFS) and a lower expression of Ki-67 (50%) after NAC was favorable for DFS among non-pCR patients.A non-basal-like phenotype and higher expressions of Ki-67 and HER-2 (2+) were favorable factors for NAC. However, a higher expression of Ki-67 on the surgical specimen after NAC was also a poor prognostic factor.

はじめに：悪性腹膜中皮腫はまれな疾患であるが，遭遇する頻度は増加している．症例数が少なく，その経過，診断，治療方針に関して明らかにされていない．今回，我々は12例を経験したので，その臨床病理，石綿ばく露の関与について検討した．方法：1993年1月～2009年3月までに岡山労災病院アスベスト疾患ブロックセンターで診断・加療した悪性腹膜中皮腫12例を対象とした．結果：男性10例，女性2例で，平均年齢は66歳であった．石綿ばく露歴を11例（91.7％）に認め，職種別では造船業，石綿製造業の占める割合が高く，石綿肺や胸膜プラークの合併より石綿ばく露の関与が示唆された．確定診断は，10例（83.3％）が開腹または腹腔鏡による生検で得られた．組織型は上皮型11例（91.7％），二相型1例（8.3％）であった．主な治療は，化学療法が10例（83.4％），手術療法は1例（8.3％）に行われた．予後は，生存期間中央値は12.0か月で，2年生存率は19.3％であった．考察：発生にはアスベストばく露が強く関与すると考えられた．解剖学的に根治手術は困難であり，局所制御や転移を防ぐために，各種治療法の組み合わせによる，治療法の確立を急ぐ必要がある．

目的：悪性腹膜中皮腫の臨床像を胸膜中皮腫と比較し明らかにする．対象：1993～2009年3月までに診断・治療した悪性腹膜中皮腫12例と胸膜中皮腫96例を対象とした．結果：悪性腹膜中皮腫は，胸膜中皮腫と比較して，女性の比率が高く，上皮型が多く，治療は化学療法が中心であった．職業歴，石綿肺や胸膜プラークの合併より石綿ばく露の強い関与が窺われ，胸膜中皮腫と比較しても，短期間に高濃度のばく露例が多かった．予後は，悪性腹膜中皮腫および胸膜中皮腫の生存期間中央値は12.0ヵ月であり，同等に予後不良の疾患であった．結語：悪性腹膜中皮腫は女性が多く，上皮型が主であった．石綿ばく露が強く関与しており，石綿ばく露歴，腹水貯留がある場合には鑑別疾患のひとつと考えられた．治療に関しては，解剖学的に根治手術は困難であり，各種治療法の組み合わせによる，治療法の確立を急ぐ必要があると考えられた．

BACKGROUND: In neoadjuvant chemotherapy for advanced esophageal cancers, complete tumor regression has been difficult to achieve, and tumor often remained after chemotherapy. However, the best method for evaluating the response to chemotherapy based on histopathologic examination of residual tumors has not been established. METHODS: Studied were 74 patients who received neoadjuvant chemotherapy (5-fluorouracil, cisplatin, and doxorubicin), followed by surgery for advanced esophageal squamous cell carcinoma. The correlation between various histopathologic factors and clinical response with survival was examined, including the importance of tumor budding in the invasive front of tumors on clinical response and survival. RESULTS: Among 74 patients, 3 achieved a pathologic complete response, and 29 (41%) of 71 residual tumors demonstrated high-grade budding in the invasive front. The 5-year survival rate of patients with low-grade budding tumors was 49%, compared with 17% for those with high-grade budding (P < .001). Budding correlated inversely with good response, which was observed in 44 (60%) of 74 patients. Univariate analysis showed that pathologic tumor depth, number of lymph node metastases, pathologic stage, lymphatic invasion, budding and clinical response were significant prognostic factors. Multivariate analysis identified budding as the most important prognostic factor followed by number of lymph node metastases. CONCLUSIONS: The results of the current study indicated that tumor budding in the invasive front of tumors correlated significantly with clinical response and prognosis of patients with esophageal squamous cell carcinomas who received neoadjuvant chemotherapy. However, the mechanism of tumor budding in the invasion front of esophageal squamous cell carcinomas treated with chemotherapy was not clarified. Cancer 2009. © 2009 American Cancer Society.

Objective: Determinants of long-term outcome of patients with human epidermal growth factor receptor 2 (HER2)-positive breast cancer who received neoadjuvant chemotherapy (NAC) are not clear. The purpose of this study was to explore the prognostic factors of HER2positive breast cancer patients administered NAC. Methods: A retrospective analysis of 125 HER2-positive breast cancer patients treated by NAC using an anthracycline plus taxane with (HCN group, n ¼ 54) or without trastuzumab (non-HCN group, n ¼ 71) was performed. The clinical parameters, including the pathological complete remission (pCR) rate, disease-free survival (DFS) and organ-specific recurrencefree survival, were measured. Results: According to the results of the univariate analyses, age, clinical stage, pCR and axillary lymph node status were the factors significantly associated with the DFS. The inclusion of trastuzumab in the NAC regimen did not yield a significant difference in the DFS. Only the axillary lymph node status and age were found to be the significant factors affecting the DFS in a multivariate model. There were no significant differences in the patient/tumor characteristics between the HCN and non-HCN groups except for the pCR rate (50% in the HCN group vs. 24% in the non-HCN group) and the median follow-up time (738 days in the HCN group vs. 1579 days in the non-HCN group). Within the first 2 years from the initiation of NAC treatment, the central nervous system (CNS) was the most common site of first recurrence in the HCN group, whereas no cases of CNS metastasis were observed in the non-HCN group. Conclusions: The pathological axillary node status and age were found to be the significant prognostic factors in HER2-positive breast cancer patients who received NAC. The pattern of recurrence may be different between HCN-treated and non-HCN-treated patients.

Purpose: Accurate assessment of metastasis in sentinel lymph nodes (SLN) of breast cancer is important but involves a heavy workload for the pathologist. We conducted a multicenter clinical trial in Japan to evaluate a new automated assay system for cytokeratin 19 mRNA, the one-step nucleic acid amplification (OSNA) assay (Sysmex), to detect lymph node metastasis of breast cancer. Experimental Design: Surgically obtained axillary lymph nodes were sectioned into four pieces, two of which were examined with the OSNA assay. The other two adjacent pieces were examined with H&E and immunohistochemical staining for cytokeratin 19. Serial sections at 0.2-mm intervals were used in trial 1 to determine the specificity of the OSNA assay, and three pairs of sections cut from the sliced surfaces of the pieces were used in trial 2 to compare the accuracy of the OSNA assay with that of a routine pathologic examination for SLNs in Japan. Results: In trial 1, the sensitivity and specificity were 95.0% [95% confidence interval (95% CI), 75.1-99.9%] and 97.1% (95% CI, 91.8-99.4%), respectively, for 124 axillary lymph nodes obtained from 34 patients. In trial 2, the agreement between findings of the assay and of the pathologic examination was 92.9% (95% CI, 90.1-95.1%) for 450 axillary lymph nodes obtained from 164 patients. Conclusion: The OSNA assay can detect lymph node metastasis as accurately as can conventional pathology and thus can be an effective addition to or alternative for rapid intraoperative examination of SLNs.

The purpose of this study was to establish a standard histological classification for intra-operative histological examination of ductal resection margins in cholangiocarcinoma to distinguish between epithelial and intramural lesions and to clarify correlations between the new classification and clinical outcomes. Intra-operative diagnosis of ductal margins was performed for 357 stumps from 216 patients undergoing surgical resection of cholangiocarcinoma at the National Cancer Center, Japan. Three expert pathologists reviewed the materials and established a histological classification defined by grade of atypia. The new classification comprised four categories: ‘insufficient’, insufficient for diagnosis due to distortion of specimen; ‘negative for malignancy’, no atypia suggestive of neoplasia; ‘undetermined lesion’, specimen showing either cellular or structural atypia; and ‘positive for malignancy’, specimen showing both cellular and structural atypia. Each category was defined to distinguish between epithelial and intramural lesions. Validity and reproducibility of the proposed classification were found to be moderate to substantial. Multivariate analyses using the clinicopathological factors identified to be associated with overall survival by univariate analyses indicated that patients diagnosed with ‘positive for malignancy’ in intramural lesions of the proximal margin displayed significant poor prognosis. Meanwhile, in patients diagnosed with ‘positive for malignancy’ or ‘undetermined lesion’ in epithelial lesions of the proximal margin, no difference in overall survival was apparent compared to patients diagnosed with ‘negative for malignancy’. We propose new histological classification for intra-operative histological examination of ductal resection margins in cholangiocarcinoma that shows a correlation with patients’ prognosis and should facilitate the determination of ductal resection margin status for cholangiocarcinoma. (Cancer Sci 2009; 100: 255–260)

方法：1993～2008年の期間に，24例の胸膜中皮腫に胸膜肺全摘術を施行した．そのうち再発した10例を対象とし検討した．結果：性別内訳は，男性9例，女性1例で，平均年齢は62.3歳（54～75歳）であった．組織型は上皮型7例，肉腫型2例，二相型1例であった．臨床病期は，II期が8例，III期が2例で，病理病期は，II期が2例，III期が8例であった．術後補助療法は，化学療法が2例に，放射線療法が1例に行われた．再発までの期間は，平均8.5ヵ月（1～17ヵ月）で，再発部位は，8例は局所再発で，2例が遠隔転移であった．全例に化学療法が行われ，Pemetrexedが3例に，Gemcitabineが7例に使用された．24例の無病生存期間中央値は13.2ヵ月で，再発例は7.0ヵ月であった．再発からの生存期間中央値は9ヵ月であった．結論：胸膜肺全摘術後の再発は局所再発が多く，局所制御法の確立が望まれる．

症例は52歳男性で，慢性B型肝炎を背景とする肝細胞癌に対して肝右葉切除術を施行した．術後病理検査の結果，胆嚢頸部のリンパ節に転移を認めStage IVb進行肝細胞癌と診断された．術後残肝再発に対して肝動脈化学塞栓術を，腹部大動脈リンパ節再発に対して全身化学療法を併用しつつ，放射線療法とリンパ節切除術の組み合わせを施行した結果，初回治療後3年9カ月目の現在，明らかな再発兆候なく経過している．肝細胞癌におけるリンパ節再発は予後不良とされ，治療方針に確立したものはない．本症例では肝内病変が制御されており，肝外病変としてのリンパ節再発に対する集学的治療が奏効したものと考えられた．

目的：アスベスト関連肺癌（以下，石綿肺癌）およびじん肺肺癌について検討した．対象と方法：1998～2008年の期間に当院で診断・治療した症例について検討した．比較対照は，同時期の石綿肺癌，じん肺肺癌を除いた肺癌（以下，その他肺癌）とした．結果：石綿肺癌は98例，じん肺肺癌は28例，その他肺癌は491例であった．石綿肺癌とじん肺肺癌の臨床病理学的比較において，組織型と臨床病期に有意差を認めた．手術症例の検討では，じん肺肺癌は，肺葉切除が少なく，予後も不良であった．石綿肺癌，じん肺肺癌およびその他の肺癌の予後は，生存期間中央値は20.4ヵ月，16.2ヵ月および19.2ヵ月で有意差は認めなかった．全症例を対象とした多変量解析では，石綿ばく露歴およびじん肺の合併は予後因子でなかった．結論：石綿肺癌およびじん肺肺癌は一般的な肺癌と同等に予後不良であると考えられた．

胸膜中皮腫を疑い胸腔鏡を施行した症例の臨床所見，胸水マーカー・細胞診，胸部CT所見，胸腔鏡所見を検討した．対象は2003年から2006年4月までに胸膜中皮腫を疑い，胸腔鏡下胸膜生検を施行した32例で，最低1年間の経過観察をした症例である．診断は胸膜中皮腫19例，良性石綿胸水11例，結核性胸膜炎1例，咽頭癌胸膜播種1例であった．胸水ヒアルロン酸，CYFRA21-1がカットオフ値を超えるものは，強く胸膜中皮腫が疑われた．胸部CTは胸膜中皮腫の検出に優れているが，胸膜中皮腫の19例中3例に胸膜の肥厚のみで不整を認めないものがあり，胸水マーカーを組み合わせることにより診断率を上げることができた．胸腔鏡所見では，隆起型は生検部位の特定や診断が容易であったが，肥厚型は生検部位の特定が困難であり，十分な観察と全層性の組織の生検が必要であると考えられた．

目的：胸膜中皮腫の臨床像について検討した．対象：当院で診断・治療した胸膜中皮腫79例を対象とした．結果：男性70例，女性9例，平均年齢65.5歳．アスベストばく露歴を83.5％に認めた．発見動機に胸部痛を49.4％に認めた．確定診断は，64.6％が胸腔鏡下胸膜生検で得られた．初診から診断が得られるまでに，4.6ヵ月（平均）を要した．治療は22例が手術，46例に化学療法もしくは放射線療法が，11例には支持療法が行われた．全例の生存率は1年，2年，3年がそれぞれ50.7％，26.9％，18.6％で，生存期間中央値は12.0ヵ月であった．予後因子の分析では，単変量解析では，IMIG臨床病期，手術の有無が有意な因子であったが，多変量解析では，IMIG臨床病期のみが有意な因子であった．結論：確定診断には，胸腔鏡下胸膜生検が有用であると考えられた．適正な手術適応の設定，補助療法の開発等が今後の課題であると考えられた．

Background Neoadjuvant chemotherapy (NACT) has been postulated but not yet proven to eradicate micrometastases and improve the prognosis of patients with advanced esophageal squamous cell carcinomas (ESCC). Cytokeratin immunohistochemistry of the lymph nodes of ESCC revealed immunohistochemical micrometastases (IHM) and cytokeratin deposits (CD), which are hyalinized denucleated particles considered to be cadavers of carcinoma cells. Successful chemotherapy should convert cancer cells from IHM to CD and improve the status of ESCC patients from systemic disease to regional disease. Methods Cytokeratin immunostaining of surgically removed lymph nodes was performed for 107 patients with node-positive ESCC, including 32 patients without preoperative treatment (Surgery group) and 75 patients undergoing NACT using CDDP, doxorubicin hydrochroride, and 5-fluorouracil (NACT group). Cytokeratin-positive staining was done for serial hematoxylin-eosin–stained sections and classified as pathologic metastasis, IHM, or CD. Results CD was observed less frequently in the Surgery group than in the NACT group (6% vs 43%, P % vs 24 %, P = . 019). IHM was a poor prognostic factor in both groups, whereas CD was a favorable one in the NACT group. The effect of chemotherapy on IHM was classified as eradicated, IHM(− )/ CD (+); persistent, IHM (+)/ CD (+); no effect, IHM (+)/ CD(− ); or not informative, IHM(− )/ CD(−). This classification correlated well with the clinical response of the primary neoplasm, number of pathologic metastases, and postoperative survival (3- year survival rates : 78 % , 18 %, 0% , and 38 %). IHM / CD was found to be an independent prognostic factor together with the number of pathologic metastases in the multivariate analysis. Conclusions Disappearance of IHM and the emergence of CD suggest the eradication of micrometastases by NACT. The clinical benefit of NACT was apparent for IHM(-)/CD(+) patients with node-positive ESCC.

We report a case of a patient with resection margin involvement gastric carcinoma that recurred 5.5 years after additional resection. A 64-year-old man underwent distal gastrectomy for advanced gastric carcinoma (sig+por2, pSE, pN0, pStage IIB) in January 2008. A total gastrectomy was performed 2 months after the initial gastrectomy because of proximal resection line involvement, and curative resection was obtained. Adjuvant chemotherapy with S-1 was completed, and follow-up surveillance was finished 5 years after the additional surgery. In November 2013, the patient experienced bouts of vomiting, and a computed tomography (CT) scan showed an abdominal abscess that had spread to the liver and communicated with the intestine. Despite abscess drainage and antibiotic therapy, infection control was difficult and the patient died 20 days after hospitalization. An autopsy showed the recurrence lesions had diffusely spread to the peritoneum and was also disseminated around the Roux-Y jejunum. These findings suggest that peritoneal recurrence might lead to penetration of the intestine and abscess formation.

A 70-year-old man was admitted to our hospital with obstructive jaundice. Computed tomography revealed a tumor in the left intrahepatic bile duct extending to the common bile duct without any significant lesions in the liver. Cholangiography showed a filling defect due to an intraductal tumor. Cytology of the bile juice was negative and tumor markers were carcinoembryonic antigen 5.7 ng/ml, carbohydrate antigen 19-9 49 U/ml, alpha-fetoprotein 9 ng/dl, and PIVKA-II 19 200 AU/ml. With a preoperative diagnosis of hilar bile duct carcinoma, a laparotomy was performed. The common bile duct was filled with a tumor and it extended into the bilateral intrahepatic bile ducts. The intraductal tumor was removed together with the extrahepatic bile ducts. An intraoperative histological examination of the tumor showed a well-differentiated hepatocellular carcinoma. No lesions were detected in the liver by ultrasonography, palpation during the operation, or a computed tomography scan after the operation. At 1 year postoperatively, no recurrence has been seen in this patient.

はじめに: 大腸印環細胞癌は極めてまれで予後不良とされているが, 発生頻度が少ないためその特長は明らかでない. 方法: 2003年までのm癌を除いた大腸癌症例3,169例の中で, 印環細胞癌は7例であった. 分化型腺癌, 低分化腺癌および粘液癌と比較し, 予後および臨床病理組織学的検討を行った. 結果: 印環細胞癌において男女比は2: 5, 平均年齢は45.7歳, 占居部位は肛門管を含む直腸4例, 盲腸1例, S状結腸1例, 横行結腸1例であった. 肉眼型は, 潰瘍浸潤型4例, 腫瘤型2例であった. 壁深達度はいずれもss (a1) 以上の進行癌でリンパ節転移は6例中5例で陽性であった. 同時性腹膜転移を7例中3例に認めたが肝転移は認めなかった. 根治度A/Bの4例の再発形式は2例が局所再発, 1例がリンパ節再発, 1例が骨髄転移であった. 7例全例が術後3年以内に死亡しており平均生存日数は319.9日で, 低分化腺癌・粘液癌と比較しても予後は極めて不良であった. 分子生物学的検討においてmicrosatellite instabilityは6例中3例に陽性であった. 考察: 予後の向上のためには, 早期診断および最近の化学療法を含めた集学的治療が重要であると考えられた.

Postoperative compensatory sweating (PCS) is an important problem impacting on quality of life for patients after endoscopic thoracic sympathectomy (ETS). The present study investigated whether intraoperative palmar temperature and blood flow are useful for assessing PCS after ETS. Methods: Retrospectively, results were evaluated for ETS in 27 consecutive patients with primary palmar hyperhidrosis between 1996 and 2002. For all patients, bilateral nerve conduction to the palms was interrupted. The relationship between the range of PCS and intraoperative changes in palmar temperature and blood flow was investigated. Results: PCS developed in all cases. After completion of ETS, mean blood flow and temperature increased respectively. Significant correlations were found between the range of PCS and increases in palmar temperature (p

Patients with adenosquamous carcinoma of the gallbladder tend to present with a bulky tumor that frequently displays involvement with adjacent organs, and diagnosis is usually made at an advanced stage. This may contribute to the poor prognosis of patients with adenosquamous carcinoma of the gallbladder compared to patients with adenocarcinoma. We present herein a patient with advanced adenosquamous carcinoma of the gallbladder who is doing well with no evidence of recurrence more than 5 years after radical resection. A 72-year-old woman complaining of generalized fatigue and loss of appetite was referred to our hospital with suspected cholelithiasis, following ultrasonography done at another hospital. On admission, abdominal ultrasonography and computed tomography revealed an irregularly shaped solid mass extending from the fundus of the gallbladder, and invasion of the stomach was strongly suspected. Gallbladder carcinoma was diagnosed, and radical resection, including partial gastrectomy, was performed. Histopathologically, adenosquamous carcinoma was diagnosed, and it was graded as si, hinf0, binf0, pv0, a0, t4, n0, and stage IVa.