A Case of a Granular Cell Tumor Arising in a Patient with Long-segment Barrett's Esophagus.

Esophageal cell tumors are rare. Esophagogastroduodenoscopy performed on a 48-year-old woman revealed an elevated esophageal lesion and the presence of long-segment Barrett's esophagus. Endoscopic ultrasonography showed a 15 mm homogeneous hypoechoic tumor extending from the lamina propria mucosa to the submucosa. Pathological examination of the biopsy tissue revealed a sheet-like cluster of histiocytoid cells with an abundant eosinophilic granular cytoplasm. Immunohistochemical examination revealed S-100 (+) and CD68 (+), thus suggesting the diagnosis of a granular cell tumor. The tumor was resected by endoscopic submucosal dissection. Pathologically, the background mucosa was Barrett's mucosa. This is the first reported case of an esophageal granular cell tumor in long-segment Barrett's esophagus.

Primary anastomosing hemangioma as a preoperative diagnostic mimicker of retroperitoneal cavernous hemangioma: A case report.

Anastomosing hemangioma (AH) is rare and a newly recognized variant of capillary hemangioma that is mostly found in the genitourinary tract. Additionally, AH is sometimes difficult to diagnose without pathological specimens. It is difficult to diagnose preoperatively due to the lack of specific clinical and radiologic appearance. The present report describes the imaging features from a radiological perspective and outlines the clinicopathologic features and treatment options. A 67-year-old woman was referred to Dokkyo Medical University Saitama Medical Center (Koshigaya, Japan) for a retroperitoneal tumor that was identified at a medical checkup 4 years prior. The patient had no symptoms, no abnormal physical signs and no past medical or specific family history. Routine blood tests were all within the normal ranges. A nonenhanced CT scan showed a circular, homogenous, well-circumscribed retroperitoneal tumor that was ~32×23 mm in size, between the abdominal aorta and the inferior vena cava, and just below the left renal vein. On a contrast-enhanced multidetector CT scan, the tumor showed heterogeneous septal enhancement in the arterial phase and persistent enhancement in the portal phase. The tumor was diagnosed as a benign neurogenic tumor or a retroperitoneal cavernous hemangioma at the time, and the patient was intended to be followed up at the outpatient clinic. However, it gradually increased to a maximum diameter of 35 mm over 4 years. Finally, it was completely resected by open laparotomy and pathologically diagnosed as AH. Retroperitoneal hemangioma is extremely rare in adulthood and has been confirmed in only 1-3% of all retroperitoneal tumors. To the best of our knowledge, only 6 cases of para-aortic AH have been reported. The incidence of this variant is very low. However, AH may be included in the differential diagnosis when a slowly progressing heterogeneous mass appears in the para-aortic region that exhibits a CT-enhanced pattern similar to a typical cavernous hemangioma.

Two tumor types in a unilateral testis in a patient with severe oligozoospermia and a history of cryptorchidism surgery: A case report.

Testicular cancer, the most common cancer among young male adults, is associated with infertility. A 38-year-old male patient was admitted to Dokkyo Medical University Saitama Medical Center, Japan, with infertility associated with severe oligozoospermia. Scrotal ultrasonography revealed two distinct tumors in the left testis: A mass with abundant blood flow on the cranial side and a mass with poor blood flow on the caudal side. Additional analysis revealed mild elevation of intact human chorionic gonadotropin (hCG) levels (tumor marker level assessment), high testosterone and low luteinizing hormone and follicle-stimulating hormone levels (hormonal level assessment) and severe oligoasthenozoospermia (semen assessment). The preoperative diagnosis was left-sided testicular cancer and severe oligoasthenozoospermia and the patient underwent left high orchiectomy and oncological testicular sperm extraction. Based on the pathological assessment, the cranial tumor was diagnosed as a seminoma with syncytiotrophoblastic cells, whereas the caudal tumor had only scar tissue with germ cell neoplasia in situ in the adjacent parenchyma. Following surgery, intact hCG and hormone levels of the patient were normalized, and the semen parameters (semen volume, sperm density, and motility) improved dramatically. To the best of our knowledge, the present case is the first report of two types of testicular tumor in a unilateral testis in a patient with a history of cryptorchidism surgery. The present case demonstrated that scrotal ultrasonography should be performed in patients with abnormal semen results to rule out testicular tumors.

Hepatic invasion of a gastric ulcer confirmed by endoscopy and surgery.

A 66-year-old woman who had been suffering from chronic anorexia for two years was transported to the hospital after being unable to consume food for three days. She had no hematemesis or abdominal pain and had no history of taking nonsteroidal anti-inflammatory drugs. Blood tests showed marked anemia with hemoglobin of 3.3 g/dL, and esophagogastroduodenoscopy revealed a large ulcer lesion in the lesser curvature of the gastric body and a liver-like mass protruding from the ulcer base. Biopsy of the mass showed proliferation of cells showing irregular cord-like structures, suggestive of normal liver tissue or hepatocellular carcinoma. Computed tomography scan showed no obvious free air in the abdomen. Despite conservative treatment, the patient developed hematemesis and progressive anemia, and surgery was performed (total gastrectomy with partial hepatectomy). Surgical specimen showed an ulcer lesion with fibrosis and loss of wall structure in all layers of the stomach, and liver adhesion with fibrosis deep in the ulcer, but no malignant findings. With the advent of powerful gastric acid secretion inhibitors, gastric ulcer invasion into the liver is now very rare, and this case is thus a valuable example showing very clear images.

Comparison of the risk of ovarian malignancy algorithm and Copenhagen Index for the preoperative assessment of Japanese women with ovarian tumors.

OBJECTIVE: To compare the risk of ovarian malignancy algorithm (ROMA) and Copenhagen Index (CPH-I) in their ability to distinguish epithelial ovarian cancer (EOC) and malignant ovarian tumors (MLOT) from benign ovarian tumors (BeOT) in Japanese women. METHODS: Patients with pathologically diagnosed ovarian tumors were included in this study. The study validated the diagnostic performance of ROMA and CPH-I. RESULTS: Among the 463 Japanese women included in this study, 312 had BeOT, 99 had EOC, and 52 had other MLOT. The receiver-operator characteristic (ROC) area under the curve (AUCs) of ROMA (0.89) and CPH-I (0.89) for distinguishing EOC from BeOT were significantly higher than that of CA125 (0.82) (CA 125 vs. ROMA; p = 0.002, vs. CPH-I; p < 0.001). The ROC-AUCs of ROMA (0.82) and CPH-I (0.81) for distinguishing MLOT from BeOT were significantly higher than that of CA125 (0.75) (CA 125 vs. ROMA: p = 0.003, vs. CPH-I: p < 0.001). The sensitivity (SN)/specificity (SP) of ROMA and CPH-I for distinguishing EOC from BeOT at standard cut-off points were 69%/90%, and 69%/90%, respectively, those for distinguishing MLOT from BeOT were 54%/90%, and 55%/90%, respectively. CONCLUSION: ROMA and CPH-I performed comparably well and better than CA125 in distinguishing EOC from BeOT in Japanese women. ROMA and CHP-I should be used with caution in practical situations, where all histological possibilities for must be considered, because the SNs of ROMA and CPH-I were only 54% and 55%.

Clinical utility of artificial intelligence assistance in histopathologic review of lymph node metastasis for gastric adenocarcinoma.

BACKGROUND: Advances in whole-slide image capture and computer image analyses using deep learning technologies have enabled the development of computer-assisted diagnostics in pathology. Herein, we built a deep learning algorithm to detect lymph node (LN) metastasis on whole-slide images of LNs retrieved from patients with gastric adenocarcinoma and evaluated its performance in clinical settings. METHODS: We randomly selected 18 patients with gastric adenocarcinoma who underwent surgery with curative intent and were positive for LN metastasis at Chiba University Hospital. A ResNet-152-based assistance system was established to detect LN metastases and to outline regions that are highly probable for metastasis in LN images. Reference standards comprising 70 LN images from two different institutions were reviewed by six pathologists with or without algorithm assistance, and their diagnostic performances were compared between the two settings. RESULTS: No statistically significant differences were observed between these two settings regarding sensitivity, review time, or confidence levels in classifying macrometastases, isolated tumor cells, and metastasis-negative. Meanwhile, the sensitivity for detecting micrometastases significantly improved with algorithm assistance, although the review time was significantly longer than that without assistance. Analysis of the algorithm's sensitivity in detecting metastasis in the reference standard indicated an area under the curve of 0.869, whereas that for the detection of micrometastases was 0.785. CONCLUSIONS: A wide variety of histological types in gastric adenocarcinoma could account for these relatively low performances; however, this level of algorithm performance could suffice to help pathologists improve diagnostic accuracy.

Duodenal gastric-type neoplasm with uncertain malignant potential in a lipoma.

The pathologic diagnosis of duodenal tumors is a developing field; however, its overview remains unclear. We describe a rare case of a duodenal gastric-type neoplasm in a 50-year-old woman. She visited her primary care doctor with complaints of upper abdominal pain, tarry stools, and shortness of breath on exertion. She was admitted owing to a stalked polyp with erosion and hemorrhage in the descending part of the duodenum. Endoscopic mucosal resection (EMR) was performed on the polyp. Histologically, the resected polyp was a lipomatous lesion in the submucosal layer, composed of mature adipose tissues. Scattered irregular lobules of Brunner's gland-like structures with well-preserved construction but mildly enlarged nuclei and occasional conspicuous nucleoli of the constituent cells were observed. The resection margin was negative. EMR findings of the duodenal polyp showed a gastric epithelial tumor within a lipoma, a rare histological type that has not been reported previously. This tumor may be classified as a "neoplasm with uncertain malignant potential" in a lipoma, an intermediate category between adenoma and invasive adenocarcinoma. There is no consensus on treatment, and careful follow-up is recommended. This is the first report of a duodenal gastric-type neoplasm with uncertain malignant potential in a lipoma.

A case of eosinophilic pancreatitis in a patient with ulcerative colitis.

Eosinophilic pancreatitis (EP) is very rare and characterised by infiltration of eosinophils into the pancreatic parenchyma. A 40-year-old man was diagnosed with total-colitis-type ulcerative colitis at the age of 15 years. He was then diagnosed with steroid-dependent ulcerative colitis. He was given golimumab, which resulted in remission. Ten months after beginning golimumab, he was urgently hospitalised with a diagnosis of acute pancreatitis. Hence, endoscopic ultrasound-guided fine needle biopsy was performed to obtain a definitive diagnosis. Pathologically, abundant infiltration of eosinophils was observed in the edematous intralobular stroma of the pancreas. He was diagnosed with EP, and treated with corticosteroids.

Endoscopic Treatment for Submucosal Heterotopic Gastric Gland in a Case Observed over Nine Years from Development to Enlargement.

A 70-year-old Japanese man with a submucosal gastric mass that continued to increase in size underwent endoscopic submucosal dissection using the pocket creation method. Histologically, some epithelial cell nuclei were enlarged, but there was little atypia overall and no sign of malignancy, suggesting a diagnosis of submucosal heterotopic gastric gland (SHGG). SHGG that enlarges over time has been associated with gastric cancer, but a preoperative diagnosis is difficult. This case was very valuable, as it enabled us to follow the course of SHGG over a period of about nine years, from the onset to enlargement.

Unique membranous gastrin receptor expression of parietal cells and its distribution pattern in the gastric oxyntic mucosa and fundic gland polyps.

The aim of this study was to clarify the correlation between gastrin receptor (GR) expression in the gastric oxyntic mucosa and fundic gland polyps (FGPs) and the histological and immunohistochemical findings of the mucosa as well as the history of proton pump inhibitor (PPI) administration. The unique membranous linear positivity of GR in parietal cells was reproducibly observed by immunohistochemistry, which was also validated by immunofluorescence. Further histological and immunohistochemical examination of 34 oxyntic mucosae and 43 FGPs revealed the following: 1) parietal cells (PCs) with membranous linear GR expression (mGR) were observed to be limited to the isthmus-neck region in the normal state; 2) appearance of PCs with mGR in the deep oxyntic gland regions was significantly related to the PPI medication history; 3) PCs with mGR were more frequently observed in the deep oxyntic gland regions when the oxyntic mucosa showed derangement of mucosal component cell compartmentalization revealed by MUC5AC and MUC6 immunohistochemistry, which was also significantly related to the PPI use; and 4) PCs with intense membranous linear positivity of GR were observed to be diffusely distributed in all of the cases of FGPs. In conclusion, the distribution of unique GR membranous linear expression in PCs of the oxyntic mucosa under PPI medication and FGPs could reflect the pathologic mucosal state characterized by derangement of the compartmentalization of mucosal component cells, which could be another basis for evaluating physiologic and/or pathophysiologic conditions of the gastric mucosa.

Cerebellar tonsillar herniation in sudden death of an adolescent anorexia nervosa patient: a case report.

BACKGROUND: Anorexia nervosa not only results in severe malnutrition but also carries a high risk of sudden death. Although fatal arrhythmias are the most common cause of sudden death, it is often unclear what exactly causes them. To the best of our knowledge, there have been no reports of cerebellar tonsillar herniations in patients with anorexia nervosa. CASE PRESENTATION: A 17-year-old girl with anorexia nervosa and autism spectrum disorder was admitted to the pediatric ward for extreme weight loss. Since she refused to take oral nutrition or tube feeding, we started continuous intravenous fluids. Eight hours after admission, she suddenly went into cardiopulmonary arrest and died despite resuscitation. A postmortem autopsy revealed the unexpected findings of generalized severe cerebral edema and cerebellar tonsillar herniation. CONCLUSION: Intracranial hypertension may need to be considered when the condition of patients with anorexia nervosa suddenly worsens during refeeding periods. Postmortem autopsy and autopsy imaging are recommended to determine the exact cause of sudden death. LEVEL OF EVIDENCE: Level IV: Evidence obtained from multiple time series analysis such as case studies. (NB: Dramatic results in uncontrolled trials might also be regarded as this type of evidence).

Expression of SATB2 in Neuroendocrine Carcinomas of the Lung: Frequent Immunopositivity of Large Cell Neuroendocrine Carcinoma with a Diagnostic Pitfall.

Small cell lung carcinoma (SCLC) and pulmonary large cell neuroendocrine carcinoma (LCNEC) are both classified as lung neuroendocrine carcinoma (NEC). It has recently been reported that the special AT-rich sequence-binding protein 2 (STAB2), known as a colorectal cancer marker, is also expressed in NECs occurring in various organs including the lung. However, few studies have examined any differences of SATB2 immunopositivity between SCLC and LCNEC. We investigated SATB2 expression in 45 SCLC and 14 LCNEC cases using immunohistochemistry as well as the expression of caudal-type homeobox 2 (CDX2) and keratin (KRT) 20. The LCNEC cases were more frequently positive for SATB2 (ten out of 14, 71%) than the SCLC ones (seventeen out of 45, 38%) with a statistically significance (P = 0.035). Furthermore, two LCNEC cases were positive for CDX2 while no positive findings were observed for any SCLC cases, the difference of which, however, was not statistically significant (P = 0.053). KRT20 was negative in all LCNEC and SCLC cases. These results require our attention when we use SATB2 and CDX2 as colorectal cancer markers because their expression in pulmonary NECs can lead to a misdiagnosis that the tumor is of metastatic colorectal adenocarcinoma, especially when the patient has a past history of colorectal cancer. Analyzing the relationship between the demographic/clinical variables and the SATB2 expression in the SCLC cases, just high Brinkman index (≥ 600) was significantly related to the positivity of SATB2 (P = 0.017), which is interesting considering the strong relationship between SCLC and smoking.

Does mirabegron deteriorate spermatogenesis? A lesson from spinal cord injury cases.

OBJECTIVES: To evaluate whether the long-term usage of mirabegron, which was reported to have potential side effects on male reproductive organs in animal studies, was harmful to spermatogenesis in human testis. METHODS: Thirty consecutive patients with spinal cord injury (20-48 years old) who performed clean intermittent catheterization were involved in this study. Ten patients were treated with mirabegron (50 mg/d) for more than 2 years and refrained from using an antimuscarinic agent due to the side effects of constipation and dry mouth. Twenty patients were treated with neither anticholinergic agents nor mirabegron. All underwent conventional testicular sperm extraction. The sperm recovery rate and histopathologic findings of the retrieved testicular tissue were compared between both groups. RESULTS: We found no difference in the sperm recovery rate (P = .083) between both groups. Spinal cord injury patients treated with mirabegron had better spermatogenesis than those not treated with mirabegron (P < .05). CONCLUSIONS: From these data, we conclude that the therapeutic dose of mirabegron had no harmful effect on spermatogenesis in spinal cord injury patients of reproductive age.

Histopathologic Change of a Case of Gastric Oxyntic Neoplasm (Gastric Adenocarcinoma of Fundic Gland Mucosa Type) Through 5 Years With Concurrent Other Oxyntic Gland Lesions.

Some gastric epithelial neoplasms show predominant chief cell differentiation (oxyntic gland neoplasms), in which the entity of "gastric adenocarcinoma of fundic gland type" was firstly designated, whereas a possible more aggressive subgroup "gastric adenocarcinoma of fundic gland mucosa type" (GA-FGM) was subsequently proposed. However, the histopathologic progression mode of these neoplasms has not been sufficiently reported. In this article, we describe a case of GA-FGM in which we could observe its progression during 5 years. The tumor was removed by endoscopic submucosal dissection 5 years after the first biopsy, which had already shown a feature of oxyntic gland neoplasm. During the follow-up period, the endoscopy revealed little change in the tumor appearance. However, the histology of endoscopic submucosal dissection showed submucosal extension with its histological progression. Besides, other oxyntic gland neoplasms of the stomach were observed metachronously or synchronously, giving an implication about a common pathogenetic basis of these lesions.

SRY-Positive 46, XX Testicular Disorder of Sexual Development With Leydig Cell Tumor.

The risk of a gonadal tumor is high in testicular disorder of sexual development (DSD) with the Y chromosome, but cases of DSD without the Y chromosome are extremely rare. We reported a gonadal tumor in a phenotypically male individual with 46, XX testicular DSD. A testicular tumor was incidentally found in a 32-year-old phenotypic male who was presented to the hospital with male infertility. A diagnosis of 46, XX testicular DSD was made by the presentation of karyotype analysis of 46, XX with the sex-determining region of the Y chromosome (SRY) positive and gonadal tissue without female gonads. Surgery was performed due to a gradually growing tumor. The partial orchidectomy was performed with the diagnosis of a benign Leydig cell tumor in frozen biopsy.

Myxoid stroma is associated with postoperative relapse in patients with stage II colon cancer.

BACKGROUND: Fibrosis surrounding cancer cells has been shown to affect cancer cell metastatic behavior. The present study aimed to explore the utility of myxoid stroma as a predictive factor for postoperative relapse in patients with stage II colon cancer. METHODS: The present study retrospectively investigated 169 patients who underwent curative surgical resection of stage II colon cancer. The fibrotic stroma was classified according to Ueno's criteria, and the patients were divided into the myxoid (MY) group and the non-MY (NMY) group. We also recorded tumor budding (TB) and investigated the combination of MY and TB for postoperative relapse. Postoperative survival was also explored. RESULTS: Thirty-two (18.9%) patients had MY. MY was significantly associated with tumor budding (TB) and postoperative relapse (p <  0.001 and p <  0.001, respectively). The 5-year RFS rates in MY group and NMY group were 52.1 and 94.6% (p < 0.0001), and the 5-year OS rates in MY group and NMY group were 74.6 and 93.3% (p = 0.001). Multivariate analysis showed that both MY and TB were significant risk factors for postoperative relapse (p < 0.001 and p = 0.02, respectively), and that only TB was a significant risk factor for OS (p = 0.043). Furthermore, compared with patients with either one of MY or TB, patients with both MY and TB had postoperative relapse more frequently (11.4% vs. 53.8%). CONCLUSIONS: The present study suggests that MY is a predictive marker for postoperative relapse in patients with stage II colon cancer.

Normal bile duct findings using peroral cholangioscopy-guided probe-based confocal laser endomicroscopy (with video).

We encountered a case of wall thickening of the bile duct in a 69-year-old man. endoscopic retrograde cholangiopancreatography (ERCP) was performed for detailed examination. When an area considered to be the healthy bile duct was examined by peroral cholangioscopes (POCS) (SPYGlass DS), the vascular network was observed. POCS-guided, probe-based confocal laser endomicroscopy (pCLE; CholangioFlex, Cellvizio; Mauna Kea Technologies, Paris, France), performed using the fluorescein-dripping method on this area, showed a reticular network of thin dark branching bands, which were presumed to be a collagen bundle or lymphatic vessels according to the Miami Classification. However, 8 µm-diameter objects thought to be red blood cells were observed inside the bands, which were considered to correspond to the vascular network observed on POCS. A biopsy specimen of this site was taken. The histological examination demonstrated capillaries just beneath the bile duct epithelium. Thus, the histology also suggested the presence of the vascular network. In this study, we obtained findings that cannot be explained in terms of the Miami Classification, which we describe here with a video.

Open Surgical Repair for a Giant Common Hepatic Artery Aneurysm with Difficulty in Proximal Arterial Clamping: A Case Report.

Hepatic artery aneurysm has been considered as a rare, life-threatening disease. In this study, we report on a patient requiring surgical treatment for a giant hepatic artery aneurysm by aneurysmectomy without revascularization. A 70-year-old woman who complained of epigastric pain was referred to our hospital. Enhanced computed tomography scan has revealed a giant (11×9 cm) common hepatic artery aneurysm. She then underwent emergency surgery; the intra-aortic balloon occlusion technique was applied in order to control the blood inflow into the aneurysm. The aneurysm was then incised, and direct closure of the inflow and outflow orifices was performed safely without evidence of ischemic change in the liver.