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1.  Xanthogranulomatous inflammation of the perimetrium with infiltration into the uterine myometrium in a postmenopausal woman: a case report 
BMC Women's Health  2014;14:82.
Xanthogranulomatous inflammation is an uncommon form of chronic inflammation that is destructive to the normal tissue of affected organs. Although xanthogranulomatous endometritis and xanthogranulomatous salpingitis of the female genital tract has been described previously, to the best of our knowledge, this is the first report of xanthogranulomatous inflammation with infiltration into the uterine myometrium from the perimetrium without endometritis.
Case presentation
A 68-year-old Japanese woman with intermittent lower abdominal pain and low-grade fever who was initially treated with antibiotics underwent hysterectomy due to abscess formation in the posterior wall of the myometrium and perimetrium (the outer serosal layer of the uterus). Histopathological findings revealed that the abscess was caused by xanthogranulomatous inflammation with the granulation tissue and chronic inflammatory cells that consisted of focal and sheets of foam cells. The inflammation destroyed the perimetrial elastic lamina, and the myometrium was deeply infiltrated by the xanthoma cells. Neither endometritis nor salpingitis was coexistent with the xanthogranulomatous inflammation.
The patient was diagnosed as xanthogranulomatous inflammation, most likely arising from the perimetrium. Our findings suggest that the perimetrium, as well as the endometrium and adnexae, is one of the origins of xanthogranulomatous inflammation in female genital tract.
PMCID: PMC4105790  PMID: 25027857
Xanthogranulomatous inflammation; Myometrial infiltration; Perimetritis
2.  Esophageal intramucosal hematoma after peripheral blood stem cell transplantation: case report and review of literature 
Esophageal complications occur after hematopoietic stem cell transplantation (HSCT). There are, however, only limited reports on the etiology or management of esophageal complications. Here, we report the occurrence of intramucosal hematoma presenting continuous esophageal hemorrhage in a 34 year-old man following the second peripheral blood stem cell transplantation for acute myeloid leukemia. His hematemesis started 2 months after HSCT and was repeated in supportive care. On day 156, he underwent total esophagectomy as a result of uncontrollable massive hematemesis. Histopathological testings of the resected esophagus confirmed intramucosal hematoma as a cause of hematemesis. This case highlights intramucosal hematoma as one of the important etiologies of esophageal complications following HSCT.
PMCID: PMC4069927  PMID: 24966988
Esophageal hemorrhage; intramucosal hematoma; esophagectomy; hematopoietic stem cell transplantation
3.  Aortic Dissection Occurring 18 Months after Successful Endovascular Repair in an Anatomically Difficult Case of Abdominal Aortic Aneurysm 
We report an autopsy case of aneurysm dissection that occurred 18 months after the implantation of a Zenith stent graft. A 94-year-old woman, who had undergone an endovascular repair with postoperative reintervention, died of shock due to retroperitoneal hematoma. An autopsy indicated that the stent graft remained firmly fixed to the native aorta, whereas the dissection occurred near the proximal edge of the stent graft but not at the point of attachment between the suprarenal stent hook and the aorta. The luminal surface of the stent graft was almost completely covered with a transparent film with an endothelial cell lining, which might reflect the tissue regeneration observed on histological examination. This was a rare case of acute aortic dissection that occurred 18 months after EVAR, in which the autopsy indicated interesting microscopic findings and the mechanisms underlying the aortic dissection. We believe that aggressive reintervention at the proximal site in elderly women might cause the dissection of the native aorta.
PMCID: PMC3834608  PMID: 24303225
5.  Correction: Heart Wall Is Thicker on Postmortem Computed Tomography Than on Ante Mortem Computed Tomography: The First Longitudinal Study 
PLoS ONE  2013;8(10):10.1371/annotation/7bae7ece-15f5-43ca-9f98-592df8543978.
PMCID: PMC3805600
6.  Heart Wall Is Thicker on Postmortem Computed Tomography Than on Ante Mortem Computed Tomography: The First Longitudinal Study 
PLoS ONE  2013;8(9):e76026.
To evaluate the postmortem changes of the heart wall on postmortem (PM) computed tomography (CT) in comparison with those on ante mortem CT (AMCT), and in comparison with the pathological findings, obtained in the same patients.
Materials and Methods
We studied 57 consecutive patients who had undergone AMCT, PMCT, and pathological autopsy in our tertiary care hospital between April 2009 and December 2010. PMCT was performed within 20 hours after death, followed by pathological autopsy. The cardiac chambers were measured at five sites on both AMCT and PMCT by two board-certified radiologists who were not provided with clinical information. The differences in heart wall thickness between AMCT with and without contrast medium, between AMCT and PMCT, and between PMCT and pathological anatomy were evaluated statistically. Confounding factors of postmortem change such as gender, presence of arteriosclerosis, the organ related to cause of death, age, and elapsed time since death were examined statistically.
No significant differences were observed on AMCT in comparison of contrasted and non-contrasted images. The heart wall was significantly thicker on PMCT than on AMCT (p < 0.0001) at all five measurement sites. The heart wall was significantly thicker on PMCT than on pathology specimens when measured in accordance with pathological standard mensuration. However, no significant difference was observed between PMCT measurements and those of pathology specimens at any site when the papillary muscles and epicardial fat were included. No significant association was found between postmortem change in heart wall thickness and gender, presence of arteriosclerosis, the organ related to cause of death, age, or elapsed time since death.
This is the first longitudinal study to confirm greater thickness of heart wall on postmortem images compared with ante mortem images, in the same patients. Furthermore, the postmortem changes on CT were supported by the pathological findings.
PMCID: PMC3785517  PMID: 24086680
7.  Peritoneal Morphology After Long-Term Peritoneal Dialysis with Biocompatible Fluid: Recent Clinical Practice in Japan 
♦ Background: Morphology changes of the peritoneal membrane after long-term peritoneal dialysis (PD) consist of denudation of peritoneal mesothelial cells, interstitial sclerosis, and hyalinizing vasculopathy. Those changes are considered to be the result of uremia and bioincompatible effects of conventional acidic lactate-buffered dialysate with glucose degradation products (GDPs). In the last decade, biocompatible dialysate with neutral pH and low GDPs has become widely used. Clinical practice has been modified in Japan, especially for anuric patients, and now includes the use of hybrid therapy. The impact on peritoneal morphology has not been well reported.
♦ Objective: The aim of the present study was to investigate the long-term effect on peritoneal morphology and function of biocompatible fluid use and current clinical practice in Japan, including hybrid dialysis therapy.
♦ Methods: We evaluated peritoneal biopsy specimens from patients who had undergone PD for more than 3 years. We used the average peritoneal thickness (APT) of the submesothelial compact zone as a marker of interstitial sclerosis and the lumen/vessel diameter ratio (L/V ratio) at postcapillary venules as a marker of hyalinizing vasculopathy. Demography and other data for the patients, including dialysate-to-plasma (D/P) ratio of creatinine, were obtained at baseline and every 6 months by peritoneal equilibration test.
♦ Results: Between 2002 and 2009, 110 patients started PD therapy with biocompatible dialysate at Tokyo University Hospital. Among them, 11 patients (8 men, 3 women; age: 54.2 ± 11.8 years; 1 with diabetes mellitus) were enrolled into this morphology study. The mean duration of PD in this group was 61 ± 11.3 months, and the mean time to peritoneal biopsy was 58 ± 15.1 months. The median APT was 180 μm (96 – 1424 μm), and the median L/V ratio was 0.66 (0.46 – 0.74). No obvious correlations between APT, L/V ratio, and PD duration were detected. The D/P creatinine of the 11 patients was maintained at a favorably low value, comparable with that of the other 99 patients.
♦ Conclusions: Peritoneal dialysis therapy using biocompatible dialysate in conjunction with modification of clinical practice may minimize the progression of peritoneal interstitial sclerosis and hyalinizing vasculopathy, preserving favorable peritoneal function for more than 3 years.
PMCID: PMC3525415  PMID: 21804136
Peritoneal membrane morphology; peritoneal solute transport
8.  Quantitative Analysis of Viral Load per Haploid Genome Revealed the Different Biological Features of Merkel Cell Polyomavirus Infection in Skin Tumor 
PLoS ONE  2012;7(6):e39954.
Merkel cell polyomavirus (MCPyV) has recently been identified in Merkel cell carcinoma (MCC), an aggressive cancer that occurs in sun-exposed skin. Conventional technologies, such as polymerase chain reaction (PCR) and immunohistochemistry, have produced conflicting results for MCPyV infections in non-MCC tumors. Therefore, we performed quantitative analyses of the MCPyV copy number in various skin tumor tissues, including MCC (n = 9) and other sun exposure-related skin tumors (basal cell carcinoma [BCC, n = 45], actinic keratosis [AK, n = 52], Bowen’s disease [n = 34], seborrheic keratosis [n = 5], primary cutaneous anaplastic large-cell lymphoma [n = 5], malignant melanoma [n = 5], and melanocytic nevus [n = 6]). In a conventional PCR analysis, MCPyV DNA was detected in MCC (9 cases; 100%), BCC (1 case; 2%), and AK (3 cases; 6%). We then used digital PCR technology to estimate the absolute viral copy number per haploid human genome in these tissues. The viral copy number per haploid genome was estimated to be around 1 in most MCC tissues, and there were marked differences between the MCC (0.119–42.8) and AK (0.02–0.07) groups. PCR-positive BCC tissue showed a similar viral load as MCC tissue (0.662). Immunohistochemistry with a monoclonal antibody against the MCPyV T antigen (CM2B4) demonstrated positive nuclear localization in most of the high-viral-load tumor groups (8 of 9 MCC and 1 BCC), but not in the low-viral-load or PCR-negative tumor groups. These results demonstrated that MCPyV infection is possibly involved in a minority of sun-exposed skin tumors, including BCC and AK, and that these tumors display different modes of infection.
PMCID: PMC3386999  PMID: 22768181
9.  Co-localisation of advanced glycation end products and d-β-aspartic acid-containing proteins in gelatinous drop-like corneal dystrophy 
The British Journal of Ophthalmology  2012;96(8):1127-1131.
Gelatinous drop-like corneal dystrophy (GDLD), also known as familial subepithelial corneal amyloidosis, is an autosomal recessive disorder that causes progressive corneal opacity due to accumulation of amyloid fibrils in the corneal stroma. Genetic analyses have revealed that a mutation in membrane component chromosome 1 surface marker 1 gene is responsible for GDLD. However, the mechanism of amyloid formation in the corneal stroma remains unclear. The present study attempted to reveal the role of advanced glycation end products (AGE) and d-amino acids in amyloid formation in GDLD.
Informed consent was obtained from five patients with GDLD, three patients with bullous keratopathy and three patients with interstitial keratitis and all the specimens were analysed. Localisation of amyloid fibrils was analysed using Congo-red and thioflavin T staining. In addition, the localisation of AGE (Nɛ-carboxy(methyl)-l-lysine, pyrraline and pentosidine) and d-β-aspartic acid-containing proteins, a major form of d-amino acid-containing proteins, was analysed immunohistochemically.
In all GDLD specimens, strong immunoreactivity to AGE and d-β-aspartic acid-containing proteins was detected in the subepithelial amyloid-rich region. In contrast, amyloid fibrils, AGE, or d-amino acid-containing proteins were slightly detected in the corneal stroma of patients with bullous keratopathy and interstitial keratitis.
Abnormally accumulated proteins rich in AGE and d-β-aspartic acid co-localise in the amyloid lesions in GDLD. These results indicate that non-enzymatic post-translational modifications of proteins, including AGE formation and isomerisation of aspartyl residues, will be the cause as well as the result of amyloid fibril formations in GDLD.
PMCID: PMC3404710  PMID: 22694960
Advanced glycation end products; biochemistry; cornead-amino acids; d-β-aspartic acid; familial subepithelial corneal amyloidosis; GDLD; gelatinous drop-like corneal dystrophy; M1S1; Nɛ-(carboxy)methyl-l-lysin; optics and refraction; pathology; pentosidine; physiology; pyrraline; treatment surgery; tumour-associated calcium signal transducer 2 (TACSTD2)
10.  Successful type-oriented endoscopic resection for gastric carcinoid tumors: A case report 
The standard treatment in Japan for gastric carcinoid has been gastrectomy with lymphadenectomy. This report describes the possibility of endoscopic treatment as an appropriate option for gastric carcinoid fulfilling certain conditions. A 46 year old woman underwent endoscopic mucosal resection for two 3 mm gastric carcinoids. The patient had hypergastrinemia with pernicious anemia and type A chronic atrophic gastritis, suggesting that the tumors were type I in Rindi's classification. Both tumors were located in the mucosal layer with no cellular polymorphism and were chromogranin A positive. Neither tumor recurrence in the stomach nor distant metastases have been documented during the 5 years of follow-up. Although many type I gastric carcinoids may be clinically indolent, reports on successful endoscopic treatment for this carcinoid have been scanty in the literature in Japan, presumably because of the hitherto surgical treatment stance for the disease. This report discusses how the size, number, depth and histological grading of the type I gastric carcinoid could allow the correct identification of a benign or malignant propensity of an individual tumor and how endoscopic resection could be a treatment of choice when these factors render it feasible. This stance could also obviate unnecessary surgical resection for more benign tumors.
PMCID: PMC3010472  PMID: 21191515
: Endoscopic resection; Gastric carcinoid; Hypergastrinemia; Pernicious anemia; Type A chronic atrophic gastritis.
11.  Aneurysms of Gastroepiploic Artery and Vein with an Arteriovenous Fistula after Partial Gastrectomy in a Patient Presenting with Abdominal Aortic Aneurysm—Report of a Case 
Annals of Vascular Diseases  2008;1(1):52-55.
A case of asymptomatic right gastroepiploic artery and vein aneurysms with an arteriovenous (AV) fistula coexisting with an abdominal aortic aneurysm (AAA) is presented. A 68-year-old man was referred for treatment of AAA (6 cm in diameter) and he was incidentally diagnosed as having right gastroepiploic artery and venous aneurysms (3 cm and 8 cm in diameter, respectively) with an AV fistula. Resection of the aneurysms and closure of the fistula were successfully completed with AAA repair. He had a history of gastrectomy, and these gastroepiploic aneurysms with an AV fistula were considered a late complication due to mass ligation of vessels during gastrectomy.
PMCID: PMC3610221  PMID: 23555340
gastroepiploic artery aneurysm; gastroepiploic venous aneurysm; arteriovenous fistula

Results 1-11 (11)