Publications by authors named "Hiroko Sato"

114 Publications

Multi-targeted therapy for refractory eosinophilic granulomatosis with polyangiitis characterized by intracerebral hemorrhage and cardiomyopathy: a case-based review.

Rheumatol Int 2021 Jul 21. Epub 2021 Jul 21.

Department of Hematology and Rheumatology, Tohoku University Graduate School of Medicine, Sendai, Miyagi, Japan.

Eosinophilic granulomatosis with polyangiitis (EGPA) is a systemic autoimmune disorder classified under anti-neutrophil cytoplasmic antibody (ANCA)-associated vasculitis, predominantly affecting small- to medium-sized vessels, characterized by asthma, eosinophilia, and necrotizing granulomatous inflammation. Most patients with EGPA experience peripheral neuropathy, whereas intracerebral hemorrhage is rare as EGPA-related presentation in central nervous system involvement, causing severe morbidity and mortality. Here, we present a 45-year-old man with refractory EGPA who developed intracerebral hemorrhage as the first manifestation, followed by cardiac involvement. This patient with a history of bronchial asthma developed a right putaminal hemorrhage caused by EGPA. Although intravenous cyclophosphamide (IVCY) and mepolizumab (MPZ) induced remission, relapse was frequently observed. Subsequently, he developed cardiomyopathy despite administration of rituximab (RTX) substituted from IVCY and MPZ. Combined immunosuppressive therapy, including IVCY, MPZ, and RTX was required to inhibit vascular inflammation, leading to sustained remission. We review previously published literature while focusing on the clinical features of patients with intracerebral hemorrhage caused by EGPA and describe clinical characteristics for detecting EGPA in patients with intracerebral hemorrhage, emphasizing rapid evaluation and recognition of EGPA and adequate intervention in the early vasculitic phase of this disease. We also refer to the immunological aspects of this case. It is important to consider "multi-targeted therapy" through interleukin-5 suppression and B cell depletion in the management of refractory EGPA.
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http://dx.doi.org/10.1007/s00296-021-04950-zDOI Listing
July 2021

Long-term outcome of partial resection in venous adventitial cystic disease.

J Vasc Surg Cases Innov Tech 2021 Sep 20;7(3):382-385. Epub 2021 May 20.

Department of Molecular Diagnostic Pathology, Iwate Medical University School of Medicine, Morioka, Iwate, Japan.

Venous adventitial cystic disease is extremely rare. Therefore, standard treatment methods have not been clearly defined. Some reports suggest that complete cyst removal is an effective treatment. However, considering the relatively high recurrence rate, follow-up periods were short. Herein, we report the case of a 75-year-old man with venous adventitial cystic disease successfully treated with partial cyst wall excision. No recurrence was observed for 10 years postoperatively. This case suggests that complete cyst wall excision might not be necessary for the treatment of venous adventitial cystic disease.
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http://dx.doi.org/10.1016/j.jvscit.2021.04.022DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC8261535PMC
September 2021

Raccoon eyes in an infant with neuroblastoma.

Pediatr Int 2021 Jul;63(7):863-864

Department of Pediatrics, Yamagata University Faculty of Medicine, Yamagata, Japan.

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http://dx.doi.org/10.1111/ped.14618DOI Listing
July 2021

Cyclophosphamide-associated enteritis presenting with severe protein-losing enteropathy in granulomatosis with polyangiitis: A case report.

World J Gastroenterol 2021 May;27(20):2657-2663

Department of Hematology and Rheumatology, Tohoku University Graduate School of Medicine, Sendai 9808574, Japan.

Background: Although cyclophosphamide (CPA) is the key drug for the treatment of autoimmune diseases including vasculitides, it has some well-known adverse effects, such as myelosuppression, hemorrhagic cystitis, infertility, and infection. However, CPA-associated severe enteritis is a rare adverse effect, and only one case with a lethal clinical course has been reported. Therefore, the appropriate management of patients with CPA-associated severe enteritis is unclear.

Case Summary: We present the case of a 61-year-old woman diagnosed with granulomatosis with polyangiitis based on the presence of symptoms in ear, lung, and, kidney with positive myeloperoxidase-antineutrophil cytoplasmic antibody. She received pulsed methylprednisolone followed by prednisolone 55 mg/d and intravenous CPA at a dose of 500 mg/mo. Ten days after the second course of intravenous CPA, she developed nausea, vomiting, and diarrhea, and was admitted to the hospital. Laboratory testing revealed hypoalbuminemia, suggesting protein-losing enteropathy. Computed tomography revealed wall thickening of the stomach, small intestine, and colon with contrast enhancement on the lumen side. Antibiotics and immunosuppressive therapy were not effective, and the patient's enteritis did not improve for > 4 mo. Because her condition became seriously exhausted, corticosteroids were tapered and supportive therapies including intravenous hyperalimentation, replenishment of albumin and gamma globulin, plasma exchange, and infection control were continued. These supportive therapies improved her condition, and her enteritis gradually regressed. She was finally discharged 7 mo later.

Conclusion: Immediate discontinuation of CPA and intensive supportive therapy are crucial for the survival of patients with CPA-associated severe enteritis.
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http://dx.doi.org/10.3748/wjg.v27.i20.2657DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC8160622PMC
May 2021

Necrotizing lymphadenitis may be induced by overexpression of Toll-like receptor7 (TLR7) caused by reduced TLR9 transport in plasmacytoid dendritic cells (PDCs).

J Clin Exp Hematop 2021 Jun 14;61(2):85-92. Epub 2021 May 14.

Department of Pediatrics, Iwaki City Medical Center, Iwaki, Japan.

Necrotizing lymphadenitis (NEL) is a self-limited systemic disease exhibiting characteristic clinical features. The pathogenesis of the disease remains unclear, but it may be associated with viral infection. In lymph nodes affected by this disease, innumerable plasmacytoid dendritic cells produce interferon-α when triggered by certain viral stimuli. IFN-α presents antigens causing the transformation of CD8 cells into immunoblasts and apoptosis of CD4 cells. From the perspective of innate immunity, UNC93B1, an endoplasmic reticulum (ER)-resident protein, associates more strongly with TLR9 than TLR7. Homeostasis is maintained under normal conditions. However, in NEL, TLR 7 was observed more than TLR 9, possibly because mutant type UNC93B1 associates more tightly with TLR7. The inhibitory effects against TLR7 by TLR9 were reported to disappear. It is likely that more TLR7 than TLR9 is transported from the ER to endolysosomes. In conclusion, overexpression of TLR7, an innate immune sensor of microbial single-stranded RNA, is inferred. Consequently, NEL may be induced.
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http://dx.doi.org/10.3960/jslrt.20060DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC8265496PMC
June 2021

Association of various myositis-specific autoantibodies with dermatomyositis and polymyositis triggered by pregnancy.

Rheumatol Int 2021 Apr 10. Epub 2021 Apr 10.

Department of Hematology and Rheumatology, Tohoku University Graduate School of Medicine, 1-1 Seiryo-machi, Aoba-ku, Sendai, Miyagi, 980-8574, Japan.

Although pregnancy is an important risk factor for autoimmune rheumatic diseases, little is known regarding the association between pregnancy and dermatomyositis (DM) or polymyositis (PM). Herein, we present two patients with DM that developed during the perinatal period. The first patient was positive for anti-aminoacyl synthetase (ARS) antibody and developed DM in the 14th week of pregnancy. Despite treatment, her foetus died of intrauterine growth restriction in the 27th week. The second patient was positive for anti-melanoma differentiation-associated gene 5 (MDA-5) antibody and developed DM 1 week after miscarriage at 9 weeks of gestation. The patient developed severe interstitial pneumonia, and intensive therapy including tofacitinib and rituximab administration was required. Our cases and a literature review revealed that various myositis-specific autoantibodies, including anti-ARS, anti-Mi-2, anti-TIF-1γ, and anti-MDA-5, are associated with DM and PM triggered by pregnancy. We also found that delay in commencing treatment in case of active disease including myositis and interstitial pneumonia, and poor response to corticosteroids were related to poor foetal outcomes in DM and PM. Although rare in pregnant women, it is critical to consider the possibility of DM and PM in patients presenting with rash, fever, weakness, and cough, and testing for myositis-specific autoantibodies is recommended.
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http://dx.doi.org/10.1007/s00296-021-04851-1DOI Listing
April 2021

Spontaneous ovarian choriocarcinoma in a young ICR mouse.

J Toxicol Pathol 2021 Jan 15;34(1):123-125. Epub 2020 Nov 15.

Safety Research Laboratories, Sohyaku, Innovative Research Division, Mitsubishi Tanabe Pharma Corporation, 2-26-1 Muraoka-Higashi, Fujisawa, Kanagawa 251-8555, Japan.

This paper describes the spontaneous ovarian choriocarcinoma observed in a young female Crl:CD1 (ICR) mouse. The mouse was sacrificed at 8 weeks of age after oral administration of a compound for 2 weeks. The left ovary was found to be cystically enlarged with dark red hemorrhaging. The cystic mass contained abundant blood plasma and erythrocytes. At the peripheral regions of the mass, large pleomorphic tumor cells with bizarre shaped nuclei were detected. Tumor cells contained a single large nucleus and abundant eosinophilic to amphophilic cytoplasm. Histopathology of the tumor cells resembled that of trophoblastic giant cells. Therefore, the observed ovarian lesion was diagnosed as a choriocarcinoma. No microscopic lesions were observed in the right ovary or other reproductive organs. Ovarian choriocarcinoma was considered to be of non-gestational origin. This is the first report of ovarian choriocarcinoma in a young ICR mouse.
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http://dx.doi.org/10.1293/tox.2020-0062DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7890175PMC
January 2021

ALOX12 mutation in a family with dominantly inherited bleeding diathesis.

J Hum Genet 2021 Aug 10;66(8):753-759. Epub 2021 Feb 10.

Department of Pediatrics, Yamagata University School of Medicine, Yamagata, Japan.

The arachidonic acid (AA) cascade plays a significant role in platelet aggregation. AA released from membrane phospholipids is metabolized by cyclooxygenase (COX) pathway to thromboxane A (TXA) or by 12S-lipoxygenase (ALOX12) to 12-hydroperoxyeicosatetraenoic acid (12-HPETE). In contrast to a well-known role of the COX pathway in platelet aggregation, the role of ALOX12 is not well understood. Platelets of ALOX12-deficient mice exhibit increased sensitivity for ADP-induced aggregation. However, recent evidence strongly suggests a significant role of ALOX12 in platelet aggregation and calcium signaling. 12-HPETE potentiates thrombin- and thromboxane-induced platelet aggregation, and calcium signaling. Inhibition experiments of ALOX12 demonstrated decreased platelet aggregation and calcium signaling in stimulated platelets. We studied a family with a dominantly inherited bleeding diathesis using next-generation sequencing analysis. Platelet aggregation studies revealed that the proband's platelets had defective aggregation responses to ADP, TXA mimetic U46619, collagen, and AA, normal affinity of TXA receptor for U46619, and normal induction of GTPase activity upon stimulation with U46619. However, the production of inositol 1,4,5-triphosphate (IP) was only increased up to 30% of the control upon U46619 stimulation, suggesting a defect in phospholipase C-β2 (PLCB2) activation downstream from TXA receptors. Affected family members had no mutation of PLCB2, but had a heterozygous c.1946A > G (p.Tyr649Cys) mutation of ALOX12. ALOX12 activity in platelets from the affected members was decreased to 25-35% of the control. Our data strongly suggested that a heterozygous c.1946A > G ALOX12 mutation was a disease-causing mutation; however, further experiments are required to confirm the pathogenesis of ALOX12 mutation in platelet aggregation.
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http://dx.doi.org/10.1038/s10038-020-00887-6DOI Listing
August 2021

Spontaneous hyaline glomerulopathy in a young Slc:ICR mouse.

J Toxicol Pathol 2020 Oct 25;33(4):303-307. Epub 2020 Aug 25.

Safety Research Laboratories, Sohyaku. Innovative Research Division, Mitsubishi Tanabe Pharma Corporation, 2-26-1 Muraoka-Higashi, Fujisawa, Kanagawa 251-8555, Japan.

Hyaline glomerulopathy is a type of glomerular lesion that occurs in aging mice. Spontaneous hyaline glomerulopathy is rare in young mice. Here, we report spontaneous hyaline glomerulopathy in a young adult (15-week-old) ICR mouse. Necropsy revealed discoloration and roughness of the kidney surface. Microscopically, diffuse glomerular lesions were prominent. Amorphous, eosinophilic materials were deposited globally in the glomeruli. The mesangial region was expanded; however, the mesangial cells showed no proliferation. Thickening of the Bowman's capsule with proliferation of parietal epithelial cells was observed. Glomerular deposits were strongly positive for anti-IgM, anti-IgG, and periodic acid-Schiff stain and were stained red by Masson's trichrome stain. The deposits were negative for anti C3 and stained negatively with Congo red stain. Periodic acid methenamine silver and electron microscopy revealed glomerular deposits limited to intraglomerular capillaries. Based on the histological features, we diagnosed this lesion as hyaline glomerulopathy. This case could improve our understanding of spontaneous lesions in toxicological and pharmacological studies.
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http://dx.doi.org/10.1293/tox.2020-0027DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7677629PMC
October 2020

Development of a Pediatric Dermatology Screening Tool Based on Two Parent-Reported Skin Symptoms: Comparison of Parental Recognition and Physician Diagnosis of Skin Symptoms of Infants and Toddlers.

J Prim Care Community Health 2020 Jan-Dec;11:2150132720974883

Kawabata Dermatology Clinic, Tokyo, Japan.

Aim: The objective of the present study was to develop a tool for mothers to assess their children's skin condition with the ultimate goal of its wider use in general health checkups for infants and toddlers.

Methods: This was a single-institution, cross-sectional study in Japan, targeting parents of 200 infants and toddlers who visited a dermatology clinic between December 2018 and March 2019. The parents completed a self-administered survey of the mother's perception of her child's dermatological symptoms, itchiness, and sleep status, and a quality-oflife assessment, using a nine-question version of the Quality of life in Primary Caregivers of children with Atopic Dermatitis questionnaire (QP9). The attending physician was asked to complete a form about the child's dermatological condition and the treatment prescribed. The severity of the dermatological condition noted by the physician was compared with the combined response to the 3 perception items (dermatological condition, itchiness, and sleep) reported by the parents, in order to identify the optimal cutoff score.

Results: Of 200 parent questionnaires distributed, 198 (99% response rate) were returned and analyzed, along with the responses from 198 physician records (100% response rate). The optimal cutoff score was 2/3 for the total score (range 0-6) for 2 items, itchiness and sleep status (sensitivity 73%, specificity 64%). There was a significant difference in QP9 scores between the 2 groups categorized by the cutoff score.

Conclusions: A pediatric dermatological screening tool based on 2 symptoms reported by the parents of children with atopic dermatitis was developed, and its precision and criterion-related validity were confirmed. This simple tool could help parents become better aware of their children's skin condition and allow healthcare workers to provide adequate skin care advice. This practical tool could be widely applicable in primary child care and public health service settings.
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http://dx.doi.org/10.1177/2150132720974883DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7682225PMC
June 2021

Protocol for -mediated transformation of tall fescue and future perspective on the application of genome editing.

Plant Biotechnol (Tokyo) 2020 Jun;37(2):157-161

Hokkaido Agricultural Research Center, National Agriculture and Food Research Organization, Sapporo, Hokkaido 062-8555, Japan.

Tall fescue ( Schreb.) is a major cool-season perennial grass grown for forage and turf. We have obtained transgenic tall fescue by -mediated transformation to improve agronomically important traits. In our protocol, we use embryogenic calli derived from not only mature seeds but also shoot tips. Although tall fescue cultivars consist of various genotypes with different genetic variation, we can produce transgenic plants at any time with calli induced from shoot tips of in vitro-maintained responsive genotypes. When the hygromycin phosphotransferase gene is used as a selectable marker, transformants are selected by incubation with 100 mg l hygromycin in both selection and regeneration media. Since tall fescue is an anemophilous species, the cultivation of transgenic plants poses the risk of transgenic pollen flow. Recently, it has been reported that genome-edited plants without the integration of foreign DNA fragments can be produced by an -mediated transient gene expression system. We hope that our protocol will contribute to production of transgene-free genome-edited tall fescue.
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http://dx.doi.org/10.5511/plantbiotechnology.20.0309aDOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7434684PMC
June 2020

Effects of water immersion on shear bond strength reduction after current application of resin-modified glass-ionomer-cements with and without an ionic liquid.

Dent Mater J 2021 Jan 7;40(1):35-43. Epub 2020 Aug 7.

Department of Biomaterials and Bioengineering, Institute of Biomedical Sciences, Tokushima University Graduate School.

The enhancement in the bonding strength of advanced dental cements has enabled long-lasting dental restorations. However, the high bonding strength can cause difficulty in removing these restorations. Therefore, "smart" dental cements with simultaneous strong bonding and easy on-demand debonding ability are required. A resin-modified glass-ionomer-cement (RMGIC) with an ionic liquid (IL) has demonstrated significant reduction in the bonding strength with current application (CA). This research investigates the effect of immersion in distilled water on the electric conductivity and bonding strength of RMGIC with and without an IL and CA. The RMGIC without the IL exhibited significant electric conductivity after immersion, and a significant decrease in bonding strength with CA. In comparison, the electric conductivity after immersion and the decrease in bonding strength with CA were greater for RMGIC with the IL. Thus, the feasibility of smart dental cements capable of electrically debonding-on-demand is indicated.
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http://dx.doi.org/10.4012/dmj.2019-371DOI Listing
January 2021

Migratory Aortitis Associated with Granulocyte-colony-stimulating Factor.

Intern Med 2020 Jun 19;59(12):1559-1563. Epub 2020 Mar 19.

Department of Hematology and Rheumatology, Tohoku University Graduate School of Medicine, Japan.

We herein report a case of migratory aortitis after the administration of granulocyte-colony-stimulating factor (G-CSF) to a 65-year-old woman with a history of pancreatic cancer. She was being administered pegfilgrastim and developed aortitis around the aortic arch. Although it resolved within two weeks, she again developed aortitis around the descending aorta, presenting as migratory aortitis, after pegfilgrastim was resumed. We further experienced three additional cases of G-CSF-induced aortitis that also showed spontaneous resolution, suggesting no or short-term use of immunosuppression. Aortitis due to G-CSF can present as migratory aortitis, since aortitis can quickly resolve and inflammation can recur at a different location.
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http://dx.doi.org/10.2169/internalmedicine.4331-19DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7364257PMC
June 2020

Three Consecutive Cases of Familial Hemophagocytic Lymphohistiocytosis, Including a Case Due to Maternal Uniparental Disomy.

J Pediatr Hematol Oncol 2020 11;42(8):e819-e821

Department of Pediatrics, Yamagata University School of Medicine, Yamagata.

We have experienced 3 consecutive cases of familial hemophagocytic lymphohistiocytosis (FHL). All affected infants had mutations in exon 3 of the perforin gene. The first had a homozygous mutation, c.1168C>T (p.R390*), caused by maternal uniparental isodisomy. The second and third had compound heterozygous mutations: c.781G>A (p.E261K) and c.1491T>A (p.C497*); c.1724G>T (p.C242G) and p.R390*, respectively. FHL is very rare in Northern Japan but should be suspected if infants exhibit prolonged fever. This is the first report of a relationship of p.R390* with FHL caused by uniparental isodisomy, and the second reported case of FHL type 2 with this form of inheritance.
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http://dx.doi.org/10.1097/MPH.0000000000001681DOI Listing
November 2020

Refractory Takayasu arteritis successfully treated with rituximab: case-based review.

Rheumatol Int 2019 Nov 6;39(11):1989-1994. Epub 2019 Aug 6.

Department of Hematology and Rheumatology, Tohoku University Graduate School of Medicine, 1-1 Seiryo-machi, Aoba-ku, Sendai, Miyagi, 980-8574, Japan.

Takayasu arteritis (TAK) is a subtype of the large-vessel vasculitis, affecting the aorta and its major branches. Although T cell-mediated autoimmunity is mainly involved in vascular inflammation, in recent years, accumulating evidence suggests the important role of B cells in the pathogenesis and effectiveness of B-cell-targeted therapy with rituximab (RTX), a chimeric anti-CD20 monoclonal antibody in refractory TAK. Herein, we report for the first time a case involving a 34-year-old man with TAK who was refractory to four different biologic agents, such as one selective T-cell co-stimulation modulator (abatacept), one anti-interleukin-6 receptor monoclonal antibody (tocilizumab), and two tumor necrosis factor-α inhibitors (infliximab and etanercept), but eventually achieved remission with RTX. He received a total of six courses of RTX, and doses of prednisolone and methotrexate were tapered without relapse. The current case provided further evidence to the potential role of RTX therapy in patients with refractory TAK, and its efficacy needs to be validated in a controlled trial.
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http://dx.doi.org/10.1007/s00296-019-04390-wDOI Listing
November 2019

Scrub typhus caused by Shimokoshi type Orientia tsutsugamushi showing variant 56-kDa type-specific antigen gene sequence in Tohoku region, Japan.

Microbiol Immunol 2019 Jul 21;63(7):280-284. Epub 2019 Jun 21.

Department of Microbiology, Yamagata Prefectural Institute of Public Health, Yamagata City, Yamagata, Japan.

In 2018, a patient was diagnosed with Shimokoshi type scrub typhus in Yamagata Prefecture, Japan. The causative pathogen was likely a variant type because 43 (8.3%) of 521 deduced amino acid sequences of the 56-kDa type-specific antigen (TSA) were different from those of the Shimokoshi prototype strain. The patient's paired sera showed low antibody titers against the Shimokoshi prototype strain. Two cases of scrub typhus reported in the Tohoku region during 2011-2012 also involved the same 56-kDa TSA gene sequence. These findings suggest the presence of diversity in Shimokoshi type Orientia tsutsugamushi, which may impede the laboratory diagnosis of scrub typhus.
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http://dx.doi.org/10.1111/1348-0421.12690DOI Listing
July 2019

Adult hypophosphatasia with compound heterozygous p.Phe327Leu missense and c.1559delT frameshift mutations in tissue-nonspecific alkaline phosphatase gene: a case report.

J Med Case Rep 2019 Apr 24;13(1):101. Epub 2019 Apr 24.

Department of General Medicine, Gunma University Graduate School of Medicine, 3-39-15 Showa-machi, Maebashi, Gunma, 371-8511, Japan.

Background: Hypophosphatasia is an inherited bone disease characterized by low alkaline phosphatase activity encoded by ALPL. Clinically, hypophosphatasia can be categorized as perinatal, infantile, childhood, and adult forms, as well as odonto-hypophosphatasia, according to the age at first sign or dental manifestations. Adult hypophosphatasia typically presents in middle-aged patients who appear to be in good health in early adulthood and manifests as painful feet caused by recurrent, slow-healing stress fractures of the lower limb. Because the symptoms of adult hypophosphatasia vary and are common, many patients with hypophosphatasia might be not diagnosed accurately and thus may receive inappropriate treatment.

Case Presentation: We report a case of a 35-year-old Japanese woman with low serum alkaline phosphatase detected at a routine medical checkup. She had mild muscle/bone pain but no history of rickets, fractures, or dental problems. Measurement of bone mineral density of the lumbar spine and the femoral neck revealed osteopenia below the expected range for age in a young adult. Abdominal ultrasonography revealed numerous microcalcifications in both kidneys. Analysis of amino acids in urine revealed that phosphoethanolamine was elevated. Low serum alkaline phosphatase activity, elevation of phosphoethanolamine, and low bone mineral density supported the diagnosis of hypophosphatasia. ALPL mutation analysis revealed two mutations: p.Phe327Leu and c.1559delT. These genetic abnormalities were previously reported in perinatal, infantile, and childhood but not adult hypophosphatasia. On the basis of the clinical presentation, laboratory and imaging findings, and genetic analyses, the patient was definitively diagnosed with adult hypophosphatasia. To the best of our knowledge, this is the first case report of adult hypophosphatasia with the compound heterozygous mutations p.Phe327Leu and c.1559delT.

Conclusions: Although the risk of bone fracture was high in this case, treatment approaches differ between osteoporosis and hypophosphatasia. Because adult hypophosphatasia diagnosis is often difficult because of their varied symptoms, hypophosphatasia should be considered in the differential diagnosis of low serum alkaline phosphatase. Early diagnosis is important so that appropriate treatment can be initiated.
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http://dx.doi.org/10.1186/s13256-019-2045-4DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6480864PMC
April 2019

Isolation and molecular detection of Ehrlichia species from ticks in western, central, and eastern Japan.

Ticks Tick Borne Dis 2019 02 13;10(2):344-351. Epub 2018 Nov 13.

United Graduate School of Agricultural Science and Veterinary Science, Gifu University, Gifu, Japan; Department of Virology 1, National Institute of Infectious Diseases, Tokyo, Japan.

Ehrlichiosis is a tick-borne bacterial disease caused by pathogens of the Ehrlichia genus. Although human ehrlichiosis has not been reported in Japan, Ehrlichia spp., which are closely related to Ehrlichia chaffeensis, were detected in several species of ixodid ticks. In this study, the presence of Ehrlichia spp. in ticks in Japan was studied by using isolation and molecular detection methods. In total, 1237 ticks were collected from vegetation in western, central, and eastern parts of Japan. The ticks were tested for detection of ehrlichial DNA with a nested polymerase chain reaction and/or isolation by inoculation of mice with the homogenate. Ehrlichial DNA was detected in 29 of these ticks. The ehrlichial DNAs, groEL and 16S rRNA genes, detected in Ixodes turdus showed a high similarity to those of E. chaffeensis with 94.7% and 99.2% identity, respectively. Ehrlichia sp. HF and Candidatus Neoehrlichia mikurensis were also detected in I. ovatus. Furthermore, Ehrlichia sp. HF was isolated from laboratory mice that were intraperitoneal inoculated with I. ovatus tick homogenate. Some ehrlichial agents detected in Ixodes ticks might be a previously unknown Ehrlichia species. In this study, Candidatus N. mikurensis was detected in I. ovatus ticks. Because I. ovatus is distributed widely and cases of its tick bite in humans are ubiquitously reported in Japan, there is a potential for ehrlichiosis to be endemic to Japan, necessitating intensive surveillance of this infectious disease.
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http://dx.doi.org/10.1016/j.ttbdis.2018.11.010DOI Listing
February 2019

Medical services at the 2017 Sapporo Asian winter games: injury and illness epidemiology at a 34-nation multisport event.

Br J Sports Med 2019 Jan 12;53(1):32-36. Epub 2018 Oct 12.

Medical Committee and Anti-Doping Commission of the Olympic Council of Asia, Kuwait City, Kuwait.

Objectives: We describe the medical services provided and report the injuries and illnesses that occurred at the eighth Asian Winter Games 2017.

Methods: A total of 2010 athletes and team officials from 32 National Olympic Committees and 2 guest countries attended this event; medical services were provided for 16 days. Medical data (medical care and physiotherapy) were collected for the same period by the organising committee for athletes and non-athletes (team officials, workforce, media and spectators) and recorded on the electronic medical record system at the medical rooms in the venues and the team residences.

Results: We recorded 745 medical encounters (medical care, 443; physiotherapy, 302), of which 549 (74%) were among athletes. There were 214 injuries as well as 144 illnesses and other medical conditions. Of the 1164 athletes, 549 (47%) utilised the services. Ice hockey, snowboarding and alpine skiing had high rate of medical encounters. More than half of the delegations were not accompanied by team doctor, and rate of medical encounters was high in these teams. The vast majority of patients transferred to hospital for further care were mostly athletes (n=36 out of 41), mostly alpine skiers and ice hockey players.

Conclusion: Injuries and illnesses varied depending on NOC medical staffing and sport events. These data will serve organisers of medical and physiotherapy services in the Asian Winter Games and similar large events.
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http://dx.doi.org/10.1136/bjsports-2018-099061DOI Listing
January 2019

Imaging mass spectrometry for toxicity assessment: a useful technique to confirm drug distribution in histologically confirmed lesions.

J Toxicol Pathol 2018 Jul 3;31(3):221-227. Epub 2018 May 3.

Safety Research Laboratories, Sohyaku. Innovative Research Division, Mitsubishi Tanabe Pharma Corporation, 2-2-50 Kawagishi, Toda-shi, Saitama 335-8505, Japan.

To evaluate the usefulness of imaging mass spectrometry (IMS) technology for assessing drug toxicity, we analyzed animal tissues in an amiodarone (AMD)-induced phospholipidosis model by IMS and confirmed the relationship between the distribution of AMD, its metabolites, and representative phospholipids (phosphatidylcholine, PC) and histological changes. AMD was administered to rats for 7 days at 150 mg/kg/day. The lung, spleen, and mesenteric lymph node were histologically examined and analyzed using IMS. The detection intensities of AMD, its metabolites, and typical PCs were higher in regions infiltrated by foamy macrophages compared with normal areas. This tendency was common in all three organs analyzed in this study. For the spleen, signals for AMD, its metabolites, and typical PCs were significantly more intense in the marginal zone, where foamy macrophages and vacuolated lymphocytes are abundant, than in the other areas. These results indicate that AMD, its metabolites, and PCs accumulate together in foamy or vacuolated cells, which is consistent with the mechanism of AMD-induced phospholipidosis. They also indicate that IMS is a useful technique for evaluating the distribution of drugs and biological components in the elucidation of toxicity mechanisms.
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http://dx.doi.org/10.1293/tox.2018-0006DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6077156PMC
July 2018

[Achievement of a stringent complete response with low-dose pomalidomide monotherapy in a multiple myeloma patient].

Rinsho Ketsueki 2018;59(4):395-400

Division of Hematology and Rheumatology, Nihon University School of Medicine.

An 80-year-old man presented to our hospital with a thoracic vertebrae compression fracture. He was diagnosed with IgG-λ myeloma (International Staging System stage II, Durie-Salmon stage IIIA). Since melphalan-prednisolone (MP) was not effective, we treated him with lenalidomide and low-dose dexamethasone (DEX) (Ld), achieving a partial response. As DEX provoked edema and psychiatric symptoms, the patient disagreed with its use, and pomalidomide (POM) monotherapy was initiated. Although the POM dosage was reduced to 1-2 mg/day due to somnolence, which was reported as an adverse event, stringent complete response (sCR) was achieved and sustained for 10 months following 11 cycles of low-dose POM monotherapy. It is assumed that sCR was achieved with low-dose POM monotherapy due to its early introduction as well as there being no high-risk chromosomal abnormalities. Even though adverse events develop with a standard dose, a continuation of low-dose POM is considered more important than discontinuation.
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http://dx.doi.org/10.11406/rinketsu.59.395DOI Listing
July 2019

Evaluation of Diagnostic Assay for Rickettsioses Using Duplex Real-Time PCR in Multiple Laboratories in Japan.

Jpn J Infect Dis 2018 Jul 27;71(4):267-273. Epub 2018 Apr 27.

Department of Food and Nutritional Sciences, Graduate School of Integrated Pharmaceutical and Nutritional Sciences, University of Shizuoka.

Tsutsugamushi disease and Japanese spotted fever are representative rickettsioses in Japan, and are caused by infection with Orientia tsutsugamushi and Rickettsia japonica, respectively. For molecular-based diagnosis, conventional PCR assays, which independently amplify respective rickettsial DNA, are usually used; however, this approach is time-consuming. Here, we describe a new duplex real-time PCR assay for the simultaneous detection of O. tsutsugamushi and spotted fever group rickettsiae, and its evaluation using several PCR conditions in 6 public health laboratories. The detection limit of the assay was estimated to be 10 copies and the sensitivity was almost identical to that of 3 conventional PCR methods. A total of 317 febrile patients were selected as clinically suspected or confirmed cases of rickettsioses. The detection efficiency of this assay for O. tsutsugamushi from blood or skin (eschar) specimens appeared to be almost the same as that of the conventional PCR method, even when performed in different laboratories, whereas the efficiency for spotted fever group rickettsiae tended to be higher than that of the 2 traditional double PCR assays. Our duplex real-time PCR is thus a powerful tool for the rapid diagnosis of rickettsioses, especially at the acute stage of infection.
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http://dx.doi.org/10.7883/yoken.JJID.2017.447DOI Listing
July 2018

Isolation Rate of Neisseria meningitidis in Japanese Children with Respiratory Tract Infections.

Jpn J Infect Dis 2018 05 28;71(3):244-246. Epub 2018 Feb 28.

Department of Pediatrics, Chiba University Graduate School of Medicine.

Although invasive meningococcal disease is rare in Japan (0.028 cases per 100,000 population), its incidence is 10 times greater in many other countries. Colonization is a prerequisite for invasive meningococcal disease. However, no study in Japan has involved specifically analyzing the carriage rate of Neisseria meningitidis in children. During 5 months in 2015, the respiratory tract specimens of patients who presented to 3 hospitals with respiratory symptoms were cultured. The bacteria were identified in selective media using a meningococcal detection kit and the serogroup was identified using polymerase chain reaction analysis. In 389 patients aged ≤15 years with respiratory symptoms, the N. meningitidis isolation rate was 0.26% (1/389). The serogroup of the only child who tested positive was Y. In this study, we detected a low meningococcal isolation rate in pediatric patients. Due to increasing globalization, the risk of invasive meningococcal disease is likely increasing in Japan. Accordingly, invasive meningococcal diseases should be continuously monitored in Japan. Future large-scale studies should assess meningococcal isolation rates and corresponding serogroups.
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http://dx.doi.org/10.7883/yoken.JJID.2017.244DOI Listing
May 2018

Quality assessment of long-term stored formalin-fixed paraffin embedded tissues for histopathological evaluation.

J Toxicol Pathol 2018 Jan 4;31(1):61-64. Epub 2017 Sep 4.

Safety Research Laboratories, Sohyaku. Innovative Research Division, Mitsubishi Tanabe Pharma Corporation, 2-2-50 Kawagishi, Toda-shi, Saitama 335-8505, Japan.

Histopathological examination of formalin-fixed paraffin-embedded (FFPE) tissues that had been stored for 30 years was conducted, and reconstructivity of the results was verified. These FFPE tissues, which were from all organs of male and female rats, were re-sectioned and histopathologically examined using hematoxylin and eosin (HE) staining. In particular, the stainability and morphology of HE sections and reproducibility of microscopic findings in the liver and kidney demonstrated in the original final reports were evaluated. Although the stainability of hematoxylin was slightly weaker and some morphological artifacts were observed in tissues in re-prepared slides, these deteriorations in the quality of HE sections were considered to be permissible for histopathological examination so long as control sections were also prepared. Most microscopic findings recorded in the original final reports were confirmed using re-prepared HE sections in the present study. While some focal findings, which were judged to be either incidental or spontaneous in nature, were not observed in the sections as expected, this was not considered to be a problem in reconstructing the results of the original histopathological examination because most findings related to the test articles were generally observed diffusely or multifocally in each organ. We concluded that results of the original histopathological examinations could be reconstructed using paraffin blocks that had been stored for up to 30 years.
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http://dx.doi.org/10.1293/tox.2017-0046DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC5820105PMC
January 2018

Eltrombopag with i.v. immunoglobulin for safe splenectomy in refractory immune thrombocytopenia.

Pediatr Int 2018 Feb 22;60(2):191-192. Epub 2017 Dec 22.

Department of Pediatrics, Yamagata University School of Medicine, Yamagata, Japan.

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http://dx.doi.org/10.1111/ped.13446DOI Listing
February 2018

Analysis of Streptococcus pneumoniae and Haemophilus influenzae isolated from middle ear fluid before and after the introduction of government subsidies for pneumococcal and H. influenzae type b vaccines in Japan.

J Infect Chemother 2017 Feb 23;23(2):85-89. Epub 2016 Nov 23.

Department of Infectious Diseases, Medical Mycology Research Center, Chiba University, Chiba, Japan.

This study aimed to identify trends in frequency, serotype, and antimicrobial susceptibility of Streptococcus pneumoniae and Haemophilus influenzae isolated from middle ear fluid specimens of children aged≤15 years (mean, 2 years), before and after the introduction of the 7-valent pneumococcal conjugate vaccine (PCV7) and the H. influenzae type b vaccine, at a pediatric facility in Japan. Sixty-six S. pneumoniae and 88 H. influenzae strains were isolated from 820 middle ear fluid samples. Serotyping and antimicrobial susceptibility testing were performed. The study time-frame was divided into period 1 (2007-2010) and period 2 (2011-2014), according to the availability of vaccine public funding. The S. pneumoniae detection rate decreased from 9.6% in period 1-6.1% in period 2 (p = 0.042). PCV7 serotypes decreased from 56.8% to 9.1% (p = 0.0002). No significant change was observed for the 13-valent pneumococcal conjugate vaccine (PCV13) serotypes: 72.7% in period 1 and 59.1% in period 2. Penicillin-resistant strains (penicillin G-MIC ≥2 μg/mL) decreased from 25% to 4.5% (p = 0.038). Detection rates for H. influenzae did not change significantly: 10.3% in period 1 and 11.3% in period 2. Serotypes were mostly non-typeable: 97.9% in period 1 and 90.2% in period 2, and only one serotype b strain was isolated in each period. The frequency of ampicillin-resistant strains (MIC ≥4 μg/mL) did not change. These results show a preventative effect of PCV7 on otitis media due to S. pneumoniae. PCV7 was replaced with PCV13 in 2013 in Japan; therefore, a further decrease in pneumococcal otitis media is anticipated in the future.
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http://dx.doi.org/10.1016/j.jiac.2016.10.008DOI Listing
February 2017

A case of intraductal papillary neoplasm of the bile duct preoperatively diagnosed using contrast-enhanced endoscopic ultrasonography.

Nihon Shokakibyo Gakkai Zasshi 2016 07;113(7):1236-43

Department of Gastroenterology, Saitama Medical University International Medical Center.

A 71-year-old man was referred to our hospital and was diagnosed with jaundice and a liver function disorder. Although we suspected an intraductal papillary neoplasm of the bile duct (IPNB)-derived caudate branches on the basis of contrast-enhanced CT, MRI, and endoscopic retrograde cholangiopancreatography, we could not clearly identify the tumor. Therefore, we examined the lesion using endoscopic ultrasonography (EUS). We could visualize an iso-hyperechoic elevated tumor in the caudate branches. The tumor was observed as a hypervascular lesion using contrast-enhanced EUS, which is useful in preoperatively diagnosing IPNB and detecting the presence of lesions.
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http://dx.doi.org/10.11405/nisshoshi.113.1236DOI Listing
July 2016

[Examination of Measures for Preventing Exposure in Nurses Who Handle Cyclophosphamide].

Gan To Kagaku Ryoho 2015 Dec;42(13):2457-9

Dept. of Hospital Pharmacy, Yamagata Prefectural Central Hospital.

Health hazards due to long-term exposure to anticancer drugs have been reported among health care professionals. In Yamagata Prefectural Central Hospital, constant use of personal protective equipment(gloves and mask with face shield)is mandatory, but there is no clear description of the protective gown. To verify the exposure status of nurses while handling cyclophosphamide and the usefulness of a protective gown as a protective measure, urinary concentration of cyclophosphamide was measured for nurses who handled cyclophosphamide. No cyclophosphamide was detected in the urine samples collected from nurses who handled cyclophosphamide while wearing protective gowns or in the samples collected from nurses who handled cyclophosphamide without protective gowns. This finding suggests that gloves and a mask with a face shield are sufficient for preventing exposure to cyclophosphamide. However, considering that only experienced nurses were included as subjects in this study, we cannot conclude that a protective gown is unnecessary, because inexperienced nurses may be exposed to cyclophosphamide. Our study's findings may be one reference to examine measures for preventing exposure in nurses.
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December 2015

Serum uric acid levels and mortality in the Japanese population: the Yamagata (Takahata) study.

Clin Exp Nephrol 2016 Dec 16;20(6):904-909. Epub 2016 Jan 16.

Department of Cardiology, Pulmonology, and Nephrology, Yamagata University School of Medicine, 2-2-2, Iida-Nishi, Yamagata, 990-9585, Japan.

Background: Serum uric acid level is regulated by gender, dietary habit, genetic predisposition, and renal function, and is associated with the development of renal and cardiovascular diseases. This study prospectively investigated the association between serum uric acid levels and mortality in a community-based population.

Methods: Three thousand four hundred and eighty-seven subjects regardless of the antihyperuricemic medication (45 % male; mean age 62 years old) from the Takahata town in Japan participated in this study and were followed up for 8 years (median 7.5 years). We examined the association between serum uric acid levels at baseline and the all-cause and cardiovascular mortality, respectively, in this population.

Results: One hundred seventy-nine subjects died during the follow-up period, with 49 deaths attributed to cardiovascular causes. Kaplan-Meier analysis revealed that the all-cause mortality was significantly higher along with the increase in serum uric acid levels at baseline among female (Log-rank P < 0.01), but not male subjects (P = 0.97). Cox-proportional hazard model analysis with adjustment for possible confounders including age, renal function, and comorbidities revealed that hyperuricemia (uric acid ≥7.0 mg/dL) was an independent risk factor for all-cause and cardiovascular mortality, respectively, in female [hazard ratio (HR) 5.92, 95 % confidence interval (CI) 2.10-14.6 for all-cause mortality, and HR 10.7, 95 % CI 1.76-50.2 for cardiovascular mortality], but not male subjects.

Conclusion: Hyperuricemia was an independent risk for all-cause and cardiovascular mortality in female, but not among the male subjects in a community-based population.
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http://dx.doi.org/10.1007/s10157-016-1228-1DOI Listing
December 2016
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