Publications by authors named "Masayuki Shintaku"

108 Publications

Esophageal Intramural Pseudodiverticulosis: A Histopathological and Immunohistochemical Study of 2 Cases.

Case Rep Gastroenterol 2022 Jan-Apr;16(1):270-277. Epub 2022 Apr 28.

Department of Gastroenterology, Japan Community Health Care Organization, Hoshigaoka Medical Center, Hirakata, Japan.

Esophageal intramural pseudodiverticulosis (EIPD) is a rare disorder characterized by an abnormal, cyst-like dilatation of the excretory ducts of esophageal submucosal glands. We aimed to elucidate the histopathological features and immunohistochemical properties of the epithelial lining of the cyst-like lesions in EIPD. We performed a histopathological and immunohistochemical study of 2 cases (one autopsy and one surgical) of EIPD. The ductal walls consisted of inner ductal cells, which were cytokeratin (CK) 7-positive and CK5/6-negative, and outer basal cells, which were CK7-negative and CK5/6-positive. The ductal epithelium frequently showed squamous metaplasia and rarely simulated a false diverticulum. Immunohistochemistry for CK7 was useful for distinction between the conditions because the surface epithelium was negative for CK7. We also confirmed that myoepithelial cells in the acinar portion of submucosal glands were well-preserved in EIPD, the finding that explained the periodic opening and closing movements of orifices of cyst-like lesions in this disorder. The immunohistochemical properties of the epithelial lining of cyst-like lesions in EIPD were essentially similar to those of the normal ducts of submucosal glands.
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http://dx.doi.org/10.1159/000524496DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC9149551PMC
April 2022

Disseminated mucormycosis with protracted clinical course and formation of a large intra-ventricular mural thrombus.

Int J Clin Exp Pathol 2022 15;15(5):233-237. Epub 2022 May 15.

Department of Hematology, Hikone Municipal Hospital Shiga, Japan.

We report the autopsy findings of a case of disseminated mucormycosis caused by , a rare pathogenic fungus of the family . The patient was a 49-year-old woman with B-lymphoblastic leukemia with hyperdiploidy, who died of progressive heart failure 4 months after induction chemotherapy successfully brought about complete remission of the leukemia. Granulocyte colony-stimulating factor (G-CSF) had been administered along with anti-neoplastic drugs, and her blood neutrophil count was markedly elevated. Autopsy revealed disseminated mycotic thromboembolism and abscess formation in the heart, lung, liver, kidney, and spleen. The most marked feature was a large mural thrombus in the left ventricle containing numerous fungal hyphae. In the myocardium and disseminated foci in visceral organs, giant cell-rich, fibrotic reactions to the mycotic infection were observed. Both the formation of a large intra-ventricular mural thrombus and giant cell reactions are rare findings in mucormycosis. We considered that the recovery and marked increase in neutrophil count induced by chemotherapy and G-CSF administration prolonged the clinical course and pathologically elicited an atypical, giant cell reaction to the mycotic infection. The prolonged clinical course also contributed to the formation of an unusually large intra-ventricular mural thrombus.
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http://www.ncbi.nlm.nih.gov/pmc/articles/PMC9187921PMC
May 2022

Pathological evidence of demyelination in the recurrent laryngeal, phrenic, and oculomotor nerves in Charcot-Marie-Tooth disease 4F.

eNeurologicalSci 2021 Dec 13;25:100358. Epub 2021 Jul 13.

Department of Neurology and Geriatrics, Kagoshima University Graduate School of Medical and Dental Sciences, Kagoshima, Japan.

We present pathology of the peripheral nerves of a patient with Adult-onset Charcot-Marie-Tooth disease 4F caused by periaxin gene mutation p.D651N. The patient was a 72-year-old woman. She had hoarseness and underwent continuous positive airway pressure therapy at night due to sleep apnea. The patient died abruptly. Remarkable demyelination with tomacula formation was found in the phrenic nerve, vagal nerve, recurrent laryngeal nerve, and oculomotor nerves. The cause of death could have been insufficient reactivity to the aspiration or sudden onset of bilateral vocal cord palsy. We must pay attention to respiratory function and cranial nerve palsies in hereditary demyelinating neuropathies.
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http://dx.doi.org/10.1016/j.ensci.2021.100358DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC8713021PMC
December 2021

Occurrence of a Duodenal Polypoid Lesion During Long-Term Acid Suppression Therapy and Its Regression After Drug Discontinuation.

Case Rep Gastroenterol 2021 Sep-Dec;15(3):779-784. Epub 2021 Sep 2.

Department of Gastroenterology, Japan Community Health care Organization, Hoshigaoka Medical Center, Hirakata, Japan.

Formation of multiple fundic gland polyps or hyperplastic polyps in the gastric mucosa is one of the well-known adverse effects of the long-term acid suppression therapy for peptic ulcer disease. However, similar phenomenon has not been reported to occur in the duodenum. We report a case of duodenal polypoid lesion that developed after the long-term use of acid suppressants and disappeared after the cessation of the treatment. The patient was a 76-year-old man with a history of heavy cigarette smoking and excessive alcohol intake who had been treated with medication of gastric acid suppressants, including proton pump inhibitors and potassium-competitive acid blockers, for refractory gastroesophageal reflux disease. After receiving the acid suppression therapy for 3 years, a polypoid lesion of 10 mm in diameter was found at the portion of the duodenal bulb. This polypoid lesion disappeared 1.5 months after the cessation of treatment. We hypothesized that changes in serum gastrin levels caused by acid suppression therapy might have been associated with the development and regression of the duodenal polypoid lesion.
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http://dx.doi.org/10.1159/000518871DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC8460887PMC
September 2021

Development of Epidermoid Metaplasia of the Mucosa in Association with Esophageal Intramural Pseudodiverticulosis and Candidiasis.

Case Rep Gastroenterol 2021 May-Aug;15(2):709-714. Epub 2021 Aug 2.

Department of Diagnostic Pathology, Japan Community Health care Organization, Hoshigaoka Medical Center, Hirakata, Japan.

We report a case of epidermoid metaplasia of the esophageal mucosa that developed in a patient with a long history of esophageal intramural pseudodiverticulosis (EIPD) complicated by candidiasis. The patient, a 69-year-old man, had been treated for about 3 years for EIPD with candidiasis. After candidiasis improved, the development of epidermoid metaplasia of the esophageal mucosa was observed. It comprised longitudinally arranged, multiple, small, whitish flecks with a scaly appearance on the mucosa of the middle to lower esophagus, and pathological examination demonstrated several fine keratohyalin granules in superficial layers of the squamous epithelium. Six months later, candidiasis was almost cured, but these small flecks had slightly increased in size, and pathological examination demonstrated epidermoid metaplasia consisting of a thick, acellular keratin layer and well-developed granular layer beneath it. We considered that chronic candida esophagitis played the principal pathogenetic role in the development of epidermoid metaplasia. EIPD may have provided an environment suitable for the growth of fungi, and mucinous material contaminated by and excreted from the orifices of EIPD may have irritated the mucosa and induced epidermoid metaplasia.
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http://dx.doi.org/10.1159/000518023DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC8436600PMC
August 2021

Multiple Gastric Carcinomas Associated with Epstein-Barr Virus and Helicobacter pylori: A Thought-Provoking Case.

Am J Case Rep 2021 Jun 23;22:e931668. Epub 2021 Jun 23.

Department of Surgery, Shiga General Hospital, Moriyama, Shiga, Japan.

BACKGROUND Epstein-Barr virus (EBV) and Helicobacter pylori (HP) infections are associated with gastric carcinoma (GC). We present a thought-provoking case of multiple GCs associated with EBV and HP infections. CASE REPORT HP infection was incidentally detected in an asymptomatic 60-year-old man. Upper endoscopy revealed gastric "kissing" ulcers. The lesions were located in the body of the stomach and measured 25 and 27 mm, respectively. They were diagnosed on pathology as moderately differentiated tubular adenocarcinoma. Imaging revealed no enlarged lymph nodes or distant metastatic lesions. Distal gastrectomy with lymphadenectomy was performed and surgical cure was obtained. The multiple GCs were categorized on pathology as infß ly0 v0 pT1b(SM)UL1N0M0H0P0CY0 pStage IA according to the Japanese classification and as T1bN0M0 Stage IA according to the tumor, node, metastasis classification. Pathological examination revealed remarkable lymphocytic infiltration into the stroma, as shown by in situ hybridization of EBV. These lymphocytic infiltrations were observed only at the sites of GC. In the immunohistochemical examination, in situ hybridization of EBV was positive for EBV-encoded small ribonucleic acid. The patient's postoperative course was uneventful. Hence, an unexpected relationship between EBV infection and multiple GCs was suggested by pathology. Quantitative determination of EBV DNA in peripheral blood was normal postoperatively. Adjuvant chemotherapy was not recommended. HP eradication therapy was successful. The patient remained asymptomatic and developed no recurrence or metastasis for 3 years after surgery. CONCLUSIONS This thought-provoking case suggests that coinfection with EBV and HP increases GC occurrence.
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http://dx.doi.org/10.12659/AJCR.931668DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC8237700PMC
June 2021

Neuropathology of the spinal nerve roots, spinal cord, and brain in the first autopsied case of Charcot-Marie-Tooth disease 4F with a D651N mutation in the periaxin gene.

Neuropathology 2021 Aug 17;41(4):281-287. Epub 2021 May 17.

Department of Clinical Laboratory Medicine and Diagnostic Pathology, Shiga University of Medical Science, Ohtsu, Japan.

Charcot-Marie-Tooth disease (CMT) 4F is an autosomal recessive, hereditary peripheral neuropathy, mostly caused by mutations in the periaxin gene (PRX). This article reports neuropathological findings of the spinal nerve roots, spinal cord, and brain of a patient with CMT4F and a D651N missense mutation in PRX. The patient was a 74-year-old woman who had a history of peripheral neuropathy with onset at the age of 30 years. She also had a history of infantile paralysis at the age of 18 months. The most pronounced autopsy finding was diffuse enlargement of anterior and posterior nerve roots, accentuated at the lumbo-sacral levels. On microscopy, the swollen nerve roots showed a loss of large-diameter myelinated fibers and formation of numerous onion bulbs. Most of the onion bulbs lacked the central, regenerating thin myelin sheaths, and in large-diameter nerve fibers whose axons had been lost, collagen fibers occupied the center of the onion bulbs. Some nerve roots formed glial bundles at the proximal end. The spinal cord showed degeneration of the gracile fascicles, and the lumbar segment anterior horn showed an asymmetric neuronal loss with rarefaction of the neuropil. The brain did not show any notable changes except for multiple foci of a radial microcolumnar arrangement of neurons in the cerebral cortex. Degeneration of the lumbar segment anterior horn is most likely secondary to the anterior radiculopathy, but a localized circulatory disturbance is another possibility.
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http://dx.doi.org/10.1111/neup.12731DOI Listing
August 2021

Olfactory neuroblastoma associated with extensive "in situ" lesion and aberrant glandular and rhabdomyosarcomatous differentiation.

Neuropathology 2021 Aug 5;41(4):273-280. Epub 2021 May 5.

Department of Pathology, Nishinotoin Bukkoji Clinic, Kyoto, Japan.

A case of olfactory neuroblastoma (ONB) associated with extensive intraepithelial neoplastic proliferation, evidenced by an "in situ" lesion, in the overlying olfactory epithelium and aberrant glandular and rhabdomyosarcomatous differentiation is reported. The tumor was a polypoid lesion that involved the upper nasal cavity and ethmoid sinus of a 63-year-old woman and consisted of an ONB surrounded by and mixed with a proliferative lesion of rhabdomyoblastic cells, consistent with an embryonal rhabdomyosarcoma. A few small foci of tubular glands with mucus-producing cells were also observed. In the olfactory epithelium covering the polypoid lesion, a nested or band-like arrangement of primitive-appearing small cells was found, and the tumor cells were immunoreactive for epithelial cell adhesion molecule (detected with Ber-EP4) and low-molecular weight cytokeratin (detected with CAM5.2) but not for synaptophysin or calretinin. The intraepithelial lesion was contiguous with the subepithelial cell nests of ONB and appeared to invade the subjacent stroma and show transition to ONB, and some tumor cell nests of ONB also contained small aggregates of similar primitive-appearing cells. The intraepithelial growth was considered to represent a preinvasive precursor lesion of ONB. Previous descriptions of an "in situ" lesion in ONB are limited. The aberrant glandular and rhabdomyosarcomatous differentiation noted in this case is also an exceptionally rare phenomenon of ONB.
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http://dx.doi.org/10.1111/neup.12730DOI Listing
August 2021

Solitary pancreatic metastasis of occult pulmonary small cell carcinoma diagnosed by EUS-FNA cytology: a case report.

Int J Clin Exp Pathol 2021 15;14(4):514-518. Epub 2021 Apr 15.

Department of Gastroenterology and Hepatology, Shiga General Hospital Japan.

A 74-year-old man presented with symptoms suggestive of acute pancreatitis, and a mass lesion measuring 25 mm was detected in the pancreatic head. Cytological and histopathological examinations of EUS-FNA specimens taken from the lesion demonstrated small cell (neuroendocrine) carcinoma. Tumor cells were immunoreactive for cytokeratin, synaptophysin, chromogranin A, CD56, and TTF-1, and PET-CT of the chest revealed a small tumor in the upper lobe of the left lung. Pulmonary carcinoma, particularly small cell carcinoma, infrequently presents with a solitary metastatic lesion in the pancreas as an initial manifestation and clinically simulates a primary pancreatic neoplasm. Because primary small cell carcinoma of the pancreas is very uncommon, metastasis from the lung should always be considered when evaluating pancreatic neoplasms showing a "small cell" morphology. Immunohistochemistry for TTF-1 is useful for determining the pulmonary origin of this type of neoplasm, and its application to cytology specimens is recommended.
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http://www.ncbi.nlm.nih.gov/pmc/articles/PMC8085835PMC
April 2021

Segmental arterial mediolysis of the middle colic artery: report of a case with special reference to lesions of small arteries and veins.

Int J Clin Exp Pathol 2021 15;14(4):508-513. Epub 2021 Apr 15.

Department of Pathology, Amagasaki Central Hospital Hyogo, Japan.

We report a case of segmental arterial mediolysis (SAM) that involved the middle colic artery, and present some pathologic alterations found in mesenteric small arteries and veins. The patient, a 52-year-old woman, underwent an emergency laparotomy for acute intra-abdominal hemorrhage, and a segment of the transverse colon with hemorrhagic mesocolon was excised. Microscopic examination demonstrated two separate lesions of SAM in the middle colic artery. The involved arterial segments showed a partial to circumferential loss of the media (mediolysis) and intima associated with the formation of a pseudoaneurysm. Smooth muscle cells adjacent to the mediolysis showed various degenerative changes. In addition, we found multiple, tiny foci of degenerative lesions affecting the outer media in the wall of small arteries. Subendothelial vacuoles and nodular intimal proliferation were also noted in small veins. Whereas SAM chiefly affects large or medium-sized arteries, small blood vessels, including veins, are also affected. The present case is unusual in that degeneration of medial smooth muscle cells was clearly observed in the arterial walls, and the small veins were affected by lesions similar to those in arteries. We suspect that a phenotypic modulation of vascular smooth muscle cells induced by some genetic vulnerability plays a role in the pathogenesis of SAM.
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http://www.ncbi.nlm.nih.gov/pmc/articles/PMC8085823PMC
April 2021

Genetic Creutzfeldt-Jakob disease-M232R with the cooccurrence of multiple prion strains, M1 + M2C + M2T: Report of an autopsy case.

Neuropathology 2021 Jun 14;41(3):206-213. Epub 2021 Feb 14.

Department of Neurological Science, Tohoku University Graduate School of Medicine, Sendai, Japan.

Genetic Creutzfeldt-Jakob disease (gCJD) with a methionine to arginine substitution at codon 232 of the prion protein gene (gCJD-M232R) is rare and has only been reported in Japan. We report an autopsy case of gCJD-M232R showing alleles of codon 129 that were homozygous for methionine and the presence of multiple strains of the protease-resistant, abnormal isoform of prion protein (PrP ), M1 + M2C + M2T. The patient, a 54-year-old Japanese man, died after a clinical course of 21 months characterized by slowly progressive dementia and sleep disturbance. At autopsy, the neuropil of the cerebral neocortex showed a widespread and severe spongiform change. Grape-like clusters of large confluent vacuoles were admixed with fine vacuoles. Neuronal loss was moderate, but reactive astrocytosis was mild. The dorsomedial nucleus of the thalamus and the inferior olivary nucleus showed moderate and severe neuronal loss, respectively. Many amyloid plaques were present in the cerebellar molecular layer. PrP deposition pattern was predominantly the synaptic type in the cerebrum and corresponded to the plaques in the cerebellum. Perivacuolar deposition was also seen. Western blot analysis of PrP revealed the predominance of type 2. Moreover, by employing Western blot analysis in combination with the protein misfolding cyclic amplification (PMCA) method, which selectively amplifies the minor M2T prion strain, we demonstrated the presence of M2T, in addition to M1 and M2C strains, in the brain of the patient. PMCA was a powerful method for demonstrating the presence of the M2T strain, although the amount is often small and the transmission is difficult.
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http://dx.doi.org/10.1111/neup.12722DOI Listing
June 2021

Pulmonary carcinoma showing shadow cell differentiation and nuclear β-catenin accumulation: Report of a case.

Pathol Int 2021 Feb 19;71(2):167-169. Epub 2021 Jan 19.

Department of Respiratory Medicine, Hikone Municipal Hospital, Shiga, Japan.

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http://dx.doi.org/10.1111/pin.13055DOI Listing
February 2021

Acute actinomycotic brain abscess in a patient with rheumatoid arthritis.

Neuropathology 2020 Dec 18;40(6):641-645. Epub 2020 Nov 18.

Pathos Tsutsumi, Toyoake, Japan.

An autopsy case of acute actinomycotic brain abscess involving a patient with rheumatoid arthritis (RA) is reported. The patient was a 72-year-old man with a seven-year history of RA and pulmonary complications, who acutely developed dysarthria and dysphagia three days before death. Autopsy revealed a fresh, non-encapsulated abscess in the "late cerebritis" stage, measuring 2 cm in diameter, in the white matter of the right parietal lobe. A small number of tiny "sulfur granules" consisting of numerous filamentous bacilli were found within the abscess. The abscess had ruptured to the lateral ventricle and elicited ventriculitis, and mild acute purulent leptomeningitis was also observed. The lung showed chronic interstitial pneumonia/pulmonary fibrosis with bronchiectasis and emphysema, and large sulfur granules were found in the lumens of a few bronchi. Less than 5% of patients with actinomycotic infection develop central nervous system lesions, and actinomycotic brain abscesses make up only 0.6% of all brain abscesses. Actinomycotic brain abscesses usually pursue a protracted clinical course, and well-formed pyogenic membranes are commonly observed. The present case is exceptional in that the very early stage of the cerebral abscess formation was pathologically captured.
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http://dx.doi.org/10.1111/neup.12699DOI Listing
December 2020

Benign notochordal cell tumor of the lung: Report of a case.

Pathol Int 2020 Nov 22;70(11):871-875. Epub 2020 Aug 22.

Department of Thoracic Surgery, Shiga General Hospital, Shiga, Japan.

A surgical case of a benign notochordal cell tumor of the lung is reported. The patient was an asymptomatic 41-year-old man, who was incidentally found to have a small tumor in the subpleural region of the left lingular segment. Since wedge resection of the tumor, the patient has been free from recurrence. The tumor measured 12 mm in diameter and showed a central cystic change. It consisted of a diffuse proliferation of polygonal cells with abundant, uni- or multi-vacuolated cytoplasm and bland nuclei. The tumor did not show a lobular architecture and lacked a myxoid or fibrous connective tissue containing blood vessels. In the peripheral region of the tumor, a small number of alveolar epithelial cells were entrapped. The nuclei of tumor cells were immunoreactive for brachyury, and the cytoplasm was positive for cytokeratin and S-100 protein. The entrapment of alveolar epithelial cells suggests infiltrative growth of the tumor, and the almost complete absence of blood vessels within the tumor may have restricted tumor growth and induced a cystic change in the central region.
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http://dx.doi.org/10.1111/pin.13005DOI Listing
November 2020

Follicular lymphoma with prominent Mott cell formation.

Pathol Int 2020 04 10;70(4):231-233. Epub 2020 Feb 10.

Department of Hematology and Oncology, Shiga General Hospital, Shiga, Japan.

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http://dx.doi.org/10.1111/pin.12907DOI Listing
April 2020

Amyotrophic lateral sclerosis with appearance of many skein-like inclusions in anterior horn cells.

Int J Clin Exp Pathol 2019 1;12(11):4156-4161. Epub 2019 Nov 1.

Department of Clinical Laboratory Medicine and Diagnostic Pathology, Shiga University of Medical Science Ohtsu, Shiga, Japan.

We report an autopsy case of amyotrophic lateral sclerosis (ALS), in which an abnormally large number of skein-like inclusions (SLIs) was found in anterior horn cells. The patient was a 73-year-old man, who presented with dysarthria. His motor neuron symptoms were predominantly of the upper-neuron type, and cognitive impairment was also noted. He died of septic shock 13 months after onset of the first neurological symptoms. Autopsy revealed marked loss of upper motor neurons, severe degeneration of the pyramidal tract, mild to moderate loss of anterior horn cells, and the appearance of many SLIs, which were immunoreactive for both pTDP-43 (phosphorylated transactivation responsive DNA-binding protein of 43 kDa) and ubiquitin, in anterior horn cells. Intra-axonal pTDP-43-positive granules arranged in a bead-like fashion were also found. The appearance of pTDP-43-positive intracytoplasmic inclusions in the brain was mostly restricted to the motor cortex. An Alzheimer type tau-pathology was found mainly in the hippocampus (Braak stage III), and many argyrophilic grains were distributed in the limbic area. Atypical ALS showing a rapid clinical course associated with cognitive impairment and predominant involvement of the upper motor neurons has recently been reported. The present case shares some clinical and pathologic findings with this type of atypical ALS. The appearance of a large number of SLIs is an unusual finding. Although its pathologic significance remains unknown, it cannot simply be ascribed to the relative preservation of anterior horn cells.
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http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6949794PMC
November 2019

Amyotrophic lateral sclerosis associated with pleuroparenchymal fibroelastosis.

Int J Clin Exp Pathol 2019 1;12(10):3956-3960. Epub 2019 Oct 1.

Department of Neurology, Shiga General Hospital Moriyama, Shiga, Japan.

An autopsy case of sporadic amyotrophic lateral sclerosis (ALS) associated with pleuroparenchymal fibroelastosis (PPFE), a rare form of idiopathic interstitial pneumonia, is reported. The patient, a 76-year-old man, presented with shortness of breath and dyspnea and died of progressive respiratory failure after a clinical course of 9 months. Muscle weakness and motor disturbance were mild, and the diagnosis of ALS was not established until one month prior to death. He had serum IgM-kappa monoclonal gammopathy (IgM: 1,232 mg/dL). Autopsy demonstrated ALS of the lower neuron-predominant type. The density of motor neurons in the spinal anterior horn showed a moderate decline, and many remaining neurons contained round inclusions that were immunoreactive for pTDP-43 (phosphorylated transactivation responsive DNA-binding protein of 43 kD) and ubiquitin. Betz cells and the pyramidal tracts were well preserved. The lung showed typical features of PPFE predominantly affecting the upper lobe: fibro-hyalinous thickening of the visceral pleura, a marked increase and aggregation of elastic fibers in the subpleural zone, and intra-alveolar collagenous fibrosis with an increase of elastic fibers in the alveolar septa. Although the complications from interstitial lung diseases including PPFE in ALS patients are usually regarded as incidental, PPFE is clinically important because of its markedly adverse influence on the clinical course. IgM-monoclonal gammopathy is another notable finding in the present case, which is occasionally seen in ALS patients, and the pathogenesis of PPFE is also considered to be associated with immunological derangements.
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http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6949758PMC
October 2019

Chronic Chagastic cardiomyopathy associated with membranoproliferative glomerulonephritis: Report of an autopsy case.

Pathol Int 2020 Jan 16;70(1):47-52. Epub 2019 Dec 16.

Pathos Tsutsumi, Aichi, Japan.

An autopsy case of chronic Chagas disease, a debilitating disorder caused by persistent infection by protozoa, Trypanosoma cruzi (Tr. cruzi), is reported. The patient was a 73-year-old Brazilian woman of Japanese descent, who had emigrated to Japan at the age of about 40 years. She died of chronic cardiac insufficiency about 8 years after the onset of cardiac symptoms. At autopsy, the heart showed typical features of chronic Chagastic cardiomyopathy: chronic lymphocytic myocarditis with extensive fibrosis and the formation of an apical aneurysm. The pathogenic protozoa were not detected in the cardiac tissue. The kidney showed typical features of membranoproliferative glomerulonephritis (MPGN). On the basis of experimental data which suggested that chronic infection of Tr. cruzi could elicit immune complex-mediated glomerulonephritis, we considered that the chronic persistent infection by Tr. cruzi contributed to the pathogenesis of MPGN in this patient.
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http://dx.doi.org/10.1111/pin.12883DOI Listing
January 2020

Localized atrophy of the pontine base as a sequela of prolonged ischemia: Report of an autopsy case.

Neuropathology 2019 Apr 4;39(2):156-161. Epub 2019 Mar 4.

Department of Neurology, Osaka City Kosaiin Hospital, Osaka, Japan.

An 80-year-old man with a history of diabetes mellitus and hypertension died of a progressive neurological disorder characterized by truncal ataxia, extraocular movement disturbance, and muscular rigidity. Neuroradiological examination showed progressive atrophy restricted to the pontine base. Autopsy revealed localized atrophy of the pontine base, in which both neurons and nerve fibers were lost, especially in the central region. Medium-sized and small arteries in the parenchyma of the pontine base showed marked fibro-hyalinous thickening of the walls with luminal stenosis, but no distinct tissue defect as seen in lacunar infarct was observed. Perivascular lymphocytic infiltration was mostly absent, and reactive astrocytic proliferation was weak. The pontine tegmentum, midbrain, and medulla oblongata were well preserved. Localized atrophy of the pontine base is a rare pathological condition, and its pathogenesis in the present case can be best explained by a prolonged ischemic state (hypoperfusion) due to marked sclerotic changes of perforating arteries. It is unique that the lesions were restricted to the pontine base and the formation of lacunas was not observed. Localized metabolic derangement resembling that seen in central pontine myelinolysis might have also contributed to the pathogenesis of this peculiar localized atrophy.
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http://dx.doi.org/10.1111/neup.12545DOI Listing
April 2019

Hormone signaling via androgen receptor affects breast cancer and prostate cancer in a male patient: A case report.

BMC Cancer 2018 Dec 22;18(1):1282. Epub 2018 Dec 22.

Department of Breast Surgery, Shiga General Hospital, 5-4-30, Moriyama, Moriyama-City, Shiga, 524-8524, Japan.

Background: Male breast cancer (MBC) is rare, accounting for only around 1% of all breast cancers. Most MBCs are hormone-driven. Not only the estrogen receptor (ER), but also other steroid hormone receptors, including the androgen receptor (AR) and progesterone receptor (PgR) are expressed in MBC. AR activation in breast cancer cells facilitates downstream gene expression that drives tumorigenesis in a similar manner to ER. AR-mediated signalling works paradoxically in breast cancer and prostate cancer, and cancer cells expressing the AR are endocrine-sensitive.

Case Presentation: We describe a case of double cancer of MBC and prostate cancer. A 69-year-old man was referred to our hospital with a lump in his left breast in the 1990s. The patient had cT3N3M0, stage IIIC breast cancer, and underwent a mastectomy and axillary lymph node dissection. Though adjuvant chemotherapy was administered, he experienced pleural metastasis 2 months after the surgery. Two years after the recurrence during endocrine therapy with oral 5-fluorouracil, he complained of frequent urination. Radiological and histological examinations revealed that the patient had cT3N0M0, stage III primary prostate cancer with a prostate-specific antigen (PSA) level of 40.5 ng/mL. Germline mutations in the BRCA1 and BRCA2 genes were not tested. He received multidisciplinary, continuous therapy for both breast and prostate cancer; however, 5 and 3 years after each diagnosis, respectively, he experienced a deep vein thrombosis in his right leg related to the endocrine therapy. Liver metastasis progressed after he stopped breast cancer therapy. However, long-term disease control had been achieved with anti-estrogen therapy for breast cancer and estrogen replacement therapy for prostate cancer.

Conclusions: Several studies have shown that estrogen exposure after estrogen depletion likely causes apoptosis of ER-positive breast cancer cells. Our findings indicate that this also applies to the environment in male body. AR dominant signaling prevents breast cancer recurrence and metastasis, especially in MBC patients.
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http://dx.doi.org/10.1186/s12885-018-5216-6DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6303965PMC
December 2018

Large polypoid angiomyofibroblastoma of the vulva: report of a case.

Int J Clin Exp Pathol 2018 1;11(11):5509-5513. Epub 2018 Nov 1.

Uehonmachi Hiromi Women's Clinic Osaka, Japan.

We report a case of angiomyofibroblastoma which arose in the vulva of a 46-year-old woman. The tumor formed a large pedunculated polypoid mass, measuring 14 cm in maximal dimension, which hung down from the right labium majus. It consisted of a dense or loose proliferation of fibroblastic and myofibroblastic cells on an edematous background, and the tumor cells occasionally exhibited an increased cellularity around well-developed, medium-sized or small blood vessels. In small areas, conglomerates of capillaries exhibited an appearance resembling that of capillary hemangioma. Tumor cells were immunoreactive for vimentin, desmin, estrogen receptor, and progesterone receptor, but not for α-smooth muscle actin, CD34, CD10, S-100 protein, calretinin, podoplanin, or cytokeratin. Angiomyofibroblastoma usually appears as a small subcutaneous nodule, and the formation of a large pedunculated polypoid mass is rare. The differential diagnosis from aggressive angiomyxoma and other mesenchymal tumors which preferentially involve the vulvo-vaginal region was briefly discussed.
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http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6963015PMC
November 2018

Detection of tumor cells of serous tubal intraepithelial carcinoma (STIC) in cervical smears and rapid development of the ovarian involvement: A case report.

Diagn Cytopathol 2018 Nov 30;46(11):945-949. Epub 2018 Oct 30.

Department of Gynecology, Shiga General Hospital, Moriyama, Shiga, Japan.

Serous tubal intraepithelial carcinoma (STIC) has attracted attention as a precursor lesion of high-grade serous carcinoma (HGSC) of the ovary. We report the rare case of a woman in whom adenocarcinoma cells were detected in cervical smears and demonstrated to be derived from STIC in the fimbria. The patient was a 48-year-old woman, in whom cervical smears contained adenocarcinoma cells, but cervical conization did not reveal adenocarcinoma. Because the post-conization smears again demonstrated adenocarcinoma cells, hysterectomy with bilateral salpingectomy was performed 16 months after the first detection of adenocarcinoma cells in cervical smears. Histopathological examination demonstrated STIC in the fimbria of the left fallopian tube. Bilateral ovaries appeared grossly normal at that time, but oophorectomy, which was performed 3 months later, disclosed HGSC involving the surface of bilateral ovaries. Detection of carcinoma cells from STIC in cervical smears is of marked significance for the management of patients, and we should keep in mind the possibility that adenocarcinoma cells in cervical smears are derived from STIC. The postoperative outcome of patients with STIC is considered generally favorable, and the clinical course of the present patient, in whom HGSC involving the bilateral ovaries was found shortly after salpingectomy, is exceptional.
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http://dx.doi.org/10.1002/dc.23957DOI Listing
November 2018

Müllerian carcinosarcoma with neuroendocrine differentiation arising in hydrocele of the canal of Nuck.

Pathol Int 2018 Nov 9;68(11):630-632. Epub 2018 Sep 9.

Department of Gynecology, Shiga General Hospital, Moriyama, Shiga, Japan.

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http://dx.doi.org/10.1111/pin.12717DOI Listing
November 2018

Dedifferentiated liposarcoma of the spermatic cord with features of epithelioid rhabdomyosarcoma and a rapidly fatal outcome.

Pathol Int 2018 10 9;68(10):579-581. Epub 2018 Aug 9.

Department of Pathology for Regional Communication, Kobe University Graduate School of Medicine, Kobe, Hyogo, Japan.

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http://dx.doi.org/10.1111/pin.12712DOI Listing
October 2018

Low neutrophil-lymphocyte ratio correlates with extended survival in patients with metastatic breast cancer who achieved clinically complete response following multidisciplinary therapy: A retrospective study.

Oncol Lett 2018 May 1;15(5):6681-6687. Epub 2018 Mar 1.

Department of Breast Surgery, Shiga Medical Center for Adults, Moriyama-shi, Shiga 524-8524, Japan.

The prognosis of patients with metastatic or recurrent breast cancer (MBC) is improving as novel treatments are developed. The present study compared the clinical characteristics of patients with MBC with or without a complete clinical response (cCR) and identified the survival-associated factors. This was a retrospective study, which included 171 patients treated for MBC between 2011 and 2017 at the Shiga Medical Center for Adults. Neutrophil to lymphocytes ratios (NLRs) were determined in blood samples. The median follow-up period following diagnosis of MBC was 44 months (range, 0-217 months). A total of 32 patients (18.7%) achieved a cCR. Compared with the non-cCR group, the cCR group had significantly fewer metastases or recurrences (P<0.001), significantly fewer visceral metastases (P<0.001), a significantly lower NLR (P<0.001) and were diagnosed with primary breast cancer at a significantly earlier stage (P=0.003). Prognosis was significantly improved in the cCR group compared with the non-cCR group (P<0.001) and a high NLR (≥19) independently predicted worse survival in a multivariate analysis (P=0.0218; hazard ratio, 1.75; 95% confidence interval, 1.09-2.85). In conclusion, the present study determined that achieving a cCR and having a low NLR are important for the long-term survival of patients with MBC.
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http://dx.doi.org/10.3892/ol.2018.8145DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC5920503PMC
May 2018

Adenoid basal carcinoma of the uterine cervix: a case report with an immunohistochemical and ultrastructural study.

Int J Clin Exp Pathol 2018 1;11(3):1758-1762. Epub 2018 Mar 1.

Department of Laboratory Medicine, Shiga University of Medical Science Japan.

A case of adenoid basal carcinoma (ABC) of the uterine cervix is reported. The patient was an 80-year-old woman who underwent hysterectomy for uterine prolapse, and ABC was incidentally found in the cervix. The tumor consisted of many cell nests of various sizes, which were composed of small basaloid cells and showed extensive invasion in the cervical wall. Both squamous and glandular differentiations were found in the center of some cell nests. Tumor cells showed the nuclear immunoreactivity for p40 and p63, and the diffuse cytoplasmic immunoreactivity for cytokeratin and p16. Although the deposition of hyaline material was not apparent around cell nests, the cell nests were surrounded by thin, membranous material immunoreactive for laminin, and the ultrastructural study demonstrated an accumulation of electron-dense lamellar material, which formed undulating or loop-forming, "seaweed-like" protrusions on the cell surface. These findings indicate that ABC produces redundant basal lamina material, although its amount is far less than that seen in adenoid cystic carcinoma, another neoplasm of the uterine cervix showing basaloid features.
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http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6958136PMC
March 2018

Atypical lower motor neuron disease with enlargement of Nissl substance: Report of an autopsy case.

Clin Neuropathol 2018 Mar/Apr;37(2):74-81

The patient was an 81-year-old woman diagnosed with atypical motor neuron disease who died after a long clinical course (7.5 years without mechanical assistance of ventilation) characterized by lower motor neuron signs and symptoms. Upper motor neuron signs and cognitive impairment were not apparent. Autopsy demonstrated severe neuronal loss in the anterior horn of the spinal cord, and some of the remaining neurons showed enlargement of Nissl substance and apparent thickening of the nuclear envelopes. No Bunina bodies, skein-like inclusions, or structures immunoreactive for phosphorylated transactivation response DNA-binding protein 43 were found. Immunoreactivity for superoxide dismutase-1 was focally seen in the enlarged Nissl substance. Ultrastructural examination demonstrated an increase of rough-surfaced endoplasmic reticulum (rough ER) and free ribosomes, disaggregation of polyribosomes, and dispersion of free ribosomes. Cisterns of rough ER were slightly dilated, and some of them were closely attached to the nuclear envelopes. Enlargement of Nissl substance may be related to "ER stress", and the abnormal findings of rough ER and free ribosomes may represent a degenerative process. However, another possibility, that they represent a compensatory hyperplastic change, cannot be excluded. The close attachment of cisterns of rough ER to the nuclear envelopes may be a mechanism to support or compensate for the abnormally-fragile nuclear envelopes.
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http://dx.doi.org/10.5414/NP301065DOI Listing
August 2018

Chronic recurrent pulmonary thromboembolism in a patient with essential thrombocythemia, complicating acute right ventricular infarct.

Pathol Int 2018 03 10;68(3):202-204. Epub 2018 Jan 10.

Department of Pathology, Amagasaki Central Hospital, Amagasaki, Hyogo, Japan.

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http://dx.doi.org/10.1111/pin.12622DOI Listing
March 2018

Chronic myocarditis with a long clinical course: Report of an autopsy case of probable autoimmune myocarditis.

Pathol Int 2017 Oct 6;67(10):521-525. Epub 2017 Sep 6.

Department of Cardiology, Osaka Red Cross Hospital, Osaka, Japan.

The patient was a 54-year-old woman, who died of chronic cardiac insufficiency after a clinical course of 2 years and 4 months. She had complained of myalgia, muscle weakness, and blepharoptosis before the onset of cardiac symptoms, but there was no evidence of myasthenia gravis or collagen-vascular diseases. At autopsy, the heart (280 g) showed marked dilatation of the four chambers and thinning of the ventricular walls. Diffuse and intense lymphocytic infiltration and extensive fibrosis were noted, with the latter being accentuated in the subendocardial and subepicardial zones. Small foci of myocardial necrosis were scattered and a small number of multinucleated giant cells were found, but epithelioid cell granulomas, Aschoff's nodules, and viral inclusion bodies were not observed. Some cardiomyocytes showed the aberrant expression of the HLA-DR antigen. No viral genomes were detected in myocardial tissue using a multivirus real-time polymerase chain reaction. The protracted clinical course, presence of active inflammatory changes at autopsy, expression of the HLA-DR antigen on cardiomyocytes, and absence of viral genomes in myocardial tissue suggest that autoimmune mechanisms played an important role in the pathogenesis and persistence of myocarditis in this patient.
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http://dx.doi.org/10.1111/pin.12568DOI Listing
October 2017

Is prolongation of corrected QT interval associated with seizures induced by electroconvulsive therapy reduced by atropine sulfate?

Pacing Clin Electrophysiol 2017 Nov 6;40(11):1246-1253. Epub 2017 Oct 6.

Liaison Psychiatry and Palliative Medicine, Graduate School of Medical and Dental Sciences, Tokyo Medical and Dental University, Tokyo, Japan.

Background: Electrocardiogram abnormalities have been reported during electroconvulsive therapy (ECT). A corrected QT interval (QTc) prolongation indicates delayed ventricular repolarization, which can trigger ventricular arrhythmias such as torsade de pointes (TdP). We examined the QTc changes during generalized tonic-clonic seizures induced by ECT, and the effects of atropine sulfate on these QTc changes.

Methods: We analyzed heart rate, QT interval, and QTc in 32 patients with depression who underwent ECT (25 women, 67.4 ± 8.7 years of age). The QTc from -30 to 0 seconds prestimulation was used as baseline, which was compared with QTc at 20-30 seconds and 140-150 seconds poststimulus onset.

Results: QTc was significantly prolonged at 20-30 seconds poststimulus, then significantly decreased at 140-150 seconds poststimulus, compared with baseline. QTc prolongation induced by ECT was significantly decreased by atropine sulfate.

Conclusions: These data suggest that the risk of TdP may be enhanced by ECT. Further, the risk of cardiac ventricular arrhythmias, including TdP, may be reduced by administration of atropine sulfate.
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http://dx.doi.org/10.1111/pace.13188DOI Listing
November 2017
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