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  1. Article ; Online: A mycobacterium fortuitum infection in chronic mucocutaneous candidiasis.

    Hoshina, Yuta / Okai, Masashi / Shoji, Kensuke / Ishikawa, Takashi / Kawai, Toshinao

    Pediatrics international : official journal of the Japan Pediatric Society

    2022  Volume 64, Issue 1, Page(s) e15166

    MeSH term(s) Candidiasis, Chronic Mucocutaneous/microbiology ; Humans ; Mutation ; Mycobacterium Infections ; Mycobacterium fortuitum
    Language English
    Publishing date 2022-04-22
    Publishing country Australia
    Document type Journal Article
    ZDB-ID 1470376-2
    ISSN 1442-200X ; 1328-8067
    ISSN (online) 1442-200X
    ISSN 1328-8067
    DOI 10.1111/ped.15166
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  2. Article ; Online: Bronchial stenosis associated with non-tuberculous mycobacterial infection.

    Ishikawa, Takashi / Okai, Masashi / Funata, Keiko / Miyazaki, Osamu / Kawai, Toshinao

    Pediatrics international : official journal of the Japan Pediatric Society

    2022  Volume 64, Issue 1, Page(s) e14947

    MeSH term(s) Constriction, Pathologic ; Humans ; Nontuberculous Mycobacteria
    Language English
    Publishing date 2022-04-18
    Publishing country Australia
    Document type Journal Article
    ZDB-ID 1470376-2
    ISSN 1442-200X ; 1328-8067
    ISSN (online) 1442-200X
    ISSN 1328-8067
    DOI 10.1111/ped.14947
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  3. Article ; Online: Granulicatella adiacens Bacteremia in Chronic Granulomatous Disease.

    Okai, Masashi / Ishikawa, Takashi / Tamura, Eiichiro / Matsui, Toshihiro / Kawai, Toshinao

    Journal of clinical immunology

    2022  Volume 43, Issue 1, Page(s) 85–87

    MeSH term(s) Humans ; Granulomatous Disease, Chronic/complications ; Granulomatous Disease, Chronic/diagnosis ; Carnobacteriaceae ; Anti-Bacterial Agents/therapeutic use ; Bacteremia/diagnosis ; Bacteremia/drug therapy
    Chemical Substances Anti-Bacterial Agents
    Language English
    Publishing date 2022-10-03
    Publishing country Netherlands
    Document type Letter ; Research Support, Non-U.S. Gov't
    ZDB-ID 779361-3
    ISSN 1573-2592 ; 0271-9142
    ISSN (online) 1573-2592
    ISSN 0271-9142
    DOI 10.1007/s10875-022-01371-w
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  4. Article ; Online: Clinical and molecular significance of flow cytometric analysis for reactive oxygen species production and residual p67

    Miyazawa, Hanae / Muraoka, Masahiro / Matsuda, Yusuke / Toma, Tomoko / Morio, Tomohiro / Shigemura, Tomonari / Haraguchi, Kohei / Matsubayashi, Tadashi / Kawai, Toshinao / Shirai, Yuya / Wada, Taizo

    Scandinavian journal of immunology

    2024  , Page(s) e13372

    Abstract: Chronic granulomatous disease (CGD) is a primary immunodeficiency disease caused by molecular defects in nicotinamide adenine dinucleotide phosphate (NADPH) oxidase. ... ...

    Abstract Chronic granulomatous disease (CGD) is a primary immunodeficiency disease caused by molecular defects in nicotinamide adenine dinucleotide phosphate (NADPH) oxidase. p67
    Language English
    Publishing date 2024-04-23
    Publishing country England
    Document type Journal Article
    ZDB-ID 120476-2
    ISSN 1365-3083 ; 0300-9475
    ISSN (online) 1365-3083
    ISSN 0300-9475
    DOI 10.1111/sji.13372
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  5. Article ; Online: Autoimmune hemolytic anemia associated with Takenouchi-Kosaki syndrome.

    Ishikawa, Kotaro / Uchiyama, Toru / Kaname, Tadashi / Kawai, Toshinao / Ishiguro, Akira

    Pediatrics international : official journal of the Japan Pediatric Society

    2021  Volume 63, Issue 12, Page(s) 1528–1530

    MeSH term(s) Anemia, Hemolytic ; Anemia, Hemolytic, Autoimmune/complications ; Anemia, Hemolytic, Autoimmune/diagnosis ; Humans ; Mutation ; Syndrome
    Language English
    Publishing date 2021-08-17
    Publishing country Australia
    Document type Journal Article
    ZDB-ID 1470376-2
    ISSN 1442-200X ; 1328-8067
    ISSN (online) 1442-200X
    ISSN 1328-8067
    DOI 10.1111/ped.14651
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  6. Article ; Online: Hepcidin in Kawasaki disease: upregulation by acute inflammation in patients having resistance to intravenous immunoglobulin therapy.

    Ishikawa, Takashi / Wada, Yasuyuki / Namba, Hiroyuki / Kawai, Toshinao

    Clinical rheumatology

    2021  Volume 40, Issue 12, Page(s) 5019–5024

    Abstract: Hepcidin is an iron metabolism inhibitor that increases with chronic inflammation. However, it is unclear whether hepcidin indicates acute inflammatory response in Kawasaki disease (KD), which is an acute systemic vasculitis. In this study, we examined ... ...

    Abstract Hepcidin is an iron metabolism inhibitor that increases with chronic inflammation. However, it is unclear whether hepcidin indicates acute inflammatory response in Kawasaki disease (KD), which is an acute systemic vasculitis. In this study, we examined the serum hepcidin levels before and after intravenous immunoglobulin (IVIG) therapy in responders and non-responders to IVIG. This was a pilot prospective observational study at a university hospital. All KD patients were initially administered 2 g/kg of IVIG as the first IVIG therapy (IVIG
    MeSH term(s) Hepcidins ; Humans ; Immunoglobulins, Intravenous/therapeutic use ; Infant ; Inflammation ; Mucocutaneous Lymph Node Syndrome/drug therapy ; Up-Regulation
    Chemical Substances Hepcidins ; Immunoglobulins, Intravenous
    Language English
    Publishing date 2021-06-19
    Publishing country Germany
    Document type Journal Article ; Observational Study
    ZDB-ID 604755-5
    ISSN 1434-9949 ; 0770-3198
    ISSN (online) 1434-9949
    ISSN 0770-3198
    DOI 10.1007/s10067-021-05822-4
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  7. Article ; Online: [A therapeutic approach towards chronic granulomatous disease].

    Kawai, Toshinao

    Nihon Rinsho Men'eki Gakkai kaishi = Japanese journal of clinical immunology

    2014  Volume 37, Issue 6, Page(s) 437–446

    Abstract: Chronic granulomatous disease (CGD) is a primary immunodeficiency (PID) characterized by the inability of phagocytes to produce reactive oxygen intermediates (ROIs) due to a defect in the NADPH oxidase complex. Recent studies have revealed that ROIs are ... ...

    Abstract Chronic granulomatous disease (CGD) is a primary immunodeficiency (PID) characterized by the inability of phagocytes to produce reactive oxygen intermediates (ROIs) due to a defect in the NADPH oxidase complex. Recent studies have revealed that ROIs are involved in inflammatory signaling in phagocytes, illuminating the underlying mechanisms of hyper-inflammation in CGD. CGD patients frequently suffer from CGD-associated bowel inflammation, granuloma, and life-threatening infections. Based on the discovery of the regulatory function of ROIs in the immune response, therapeutic methods for excessive inflammation focusing on inflammatory cytokines are being developed for CGD. Although hematopoietic stem cell (HSC) transplantation (HSCT) is a curative therapy for CGD, successful transplants greatly depend on HSC source selection and the degree of matching of potential donors. Gene therapy trials for PID have been performed on over 120 patients with no HLA identical donor for HSCT, and have demonstrated clinical benefits. Genotoxicity in HSC gene therapy trials has expanded our knowledge on the mechanisms of vector-associated clonal expansion of gene-modified cells, which will advance gene therapy development using self-inactivating retrovirus and lentivirus vectors. We will discuss the complications of HSCT for CGD. We will then outline the status of gene therapy approaches in the treatment of CGD.
    MeSH term(s) Cytokines/metabolism ; Genetic Therapy/methods ; Genetic Vectors ; Granulomatous Disease, Chronic/etiology ; Granulomatous Disease, Chronic/therapy ; Hematopoietic Stem Cell Transplantation/adverse effects ; Humans ; Inflammation Mediators/metabolism ; Molecular Targeted Therapy ; NADPH Oxidases/deficiency ; Phagocytes/metabolism ; Reactive Oxygen Species/metabolism ; Thalidomide/therapeutic use
    Chemical Substances Cytokines ; Inflammation Mediators ; Reactive Oxygen Species ; Thalidomide (4Z8R6ORS6L) ; NADPH Oxidases (EC 1.6.3.-)
    Language Japanese
    Publishing date 2014
    Publishing country Japan
    Document type Journal Article ; Review
    ZDB-ID 2019767-6
    ISSN 1349-7413 ; 0911-4300
    ISSN (online) 1349-7413
    ISSN 0911-4300
    DOI 10.2177/jsci.37.437
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    Zs.A 5254: Show issues Location:
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  8. Article ; Online: Patient-reported outcomes in patients with primary immunodeficiency diseases in Japan: baseline results from a prospective observational study.

    Kanegane, Hirokazu / Ishimura, Masataka / Kawai, Toshinao / Okada, Satoshi / Okamatsu, Nobuaki / Go, Madoka / Noto, Shinichi

    Frontiers in immunology

    2023  Volume 14, Page(s) 1244250

    Abstract: Introduction: Primary immunodeficiency diseases (PIDs) are rare inherited diseases resulting in impaired immunity. People with PID experience lower health-related quality of life (HR-QOL) and disease-related burdens in daily activities. This ongoing, ... ...

    Abstract Introduction: Primary immunodeficiency diseases (PIDs) are rare inherited diseases resulting in impaired immunity. People with PID experience lower health-related quality of life (HR-QOL) and disease-related burdens in daily activities. This ongoing, prospective observational study aims to evaluate disease activity, treatment status, treatment-related burden, daily activities, and HR-QOL in patients with PID in Japan over a 1-year period. In this interim report (database lock: July 29, 2022), we present baseline results.
    Methods: Participants were enrolled between November 2021 and May 2022; data were collected four times/year per participant until May 2023 using an online electronic patient-reported outcomes system. Patients with PID and healthy volunteers aged ≥12 years, residing in Japan, and with access to a smartphone were eligible. HR-QOL (primary endpoint) was assessed by the EuroQol-5 Dimensions-5 Levels (EQ-5D-5L) and the Medical Outcomes Study 36-Item Short Form Health Survey (SF-36). Work productivity was assessed by the Work Productivity and Activity Impairment (WPAI) Questionnaire. Other aspects of PID and burden were assessed with a new questionnaire developed in-house. The study is registered at the University hospital Medical Information Network clinical trials registry (UMIN000045622).
    Results: The full interim analysis set comprised 71 patients with PID and 47 healthy volunteers. The most common International Union of Immunological Societies PID category was primary antibody deficiency (56.3% of patients). Complications were common, especially recurrent respiratory tract infections (63.4%). Most patients with PID were treated with immunoglobulin replacement therapy (73.2%); 22.4% of these patients had serum immunoglobulin levels <700 mg/dL. Among patients who did not undergo hematopoietic cell transplantation, EQ-5D-5L (n=67) and SF-36 (n=59) Physical and Mental Component Summary scores were significantly lower than in healthy volunteers (p < 0.001). WPAI absenteeism, work productivity loss, and activity impairment scores were significantly lower in 42 working patients with PID than in 37 working healthy volunteers (p < 0.05). Other results indicated that patients with PID experience substantial burdens related to medical visits, expenses, work, and daily activities.
    Discussion: This interim analysis confirms that patients with PID in Japan have lower HR-QOL and work productivity compared with healthy individuals and experience substantial limitations and burdens in their daily lives.
    MeSH term(s) Humans ; Immunoglobulins ; Japan/epidemiology ; Patient Reported Outcome Measures ; Primary Immunodeficiency Diseases/therapy ; Quality of Life ; Prospective Studies
    Chemical Substances Immunoglobulins
    Language English
    Publishing date 2023-09-27
    Publishing country Switzerland
    Document type Observational Study ; Journal Article ; Research Support, Non-U.S. Gov't
    ZDB-ID 2606827-8
    ISSN 1664-3224 ; 1664-3224
    ISSN (online) 1664-3224
    ISSN 1664-3224
    DOI 10.3389/fimmu.2023.1244250
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  9. Article ; Online: Elevated Soluble PD-L1 in Pregnant Women's Serum Suppresses the Immune Reaction.

    Okuyama, Mai / Mezawa, Hidetoshi / Kawai, Toshinao / Urashima, Mitsuyoshi

    Frontiers in immunology

    2019  Volume 10, Page(s) 86

    Abstract: Background: ...

    Abstract Background:
    MeSH term(s) Adult ; Antibodies, Blocking/metabolism ; B7-H1 Antigen/blood ; B7-H1 Antigen/immunology ; Cells, Cultured ; Cohort Studies ; Cytokines/metabolism ; Enzyme-Linked Immunosorbent Assay ; Female ; Fetal Blood/chemistry ; Humans ; Immune Tolerance ; Lymphocyte Culture Test, Mixed ; Male ; Pregnancy/immunology ; Programmed Cell Death 1 Receptor/metabolism ; T-Lymphocytes/immunology ; Trophoblasts/metabolism ; Up-Regulation
    Chemical Substances Antibodies, Blocking ; B7-H1 Antigen ; CD274 protein, human ; Cytokines ; PDCD1 protein, human ; Programmed Cell Death 1 Receptor
    Language English
    Publishing date 2019-02-18
    Publishing country Switzerland
    Document type Journal Article ; Research Support, Non-U.S. Gov't
    ZDB-ID 2606827-8
    ISSN 1664-3224 ; 1664-3224
    ISSN (online) 1664-3224
    ISSN 1664-3224
    DOI 10.3389/fimmu.2019.00086
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  10. Article ; Online: Thymitis in chronic granulomatous disease.

    Kanamori, Keita / Tamura, Eiichiro / Onodera, Masafumi / Ishiguro, Akira / Kawai, Toshinao

    Pediatrics international : official journal of the Japan Pediatric Society

    2019  Volume 61, Issue 4, Page(s) 429–431

    MeSH term(s) Granulomatous Disease, Chronic/complications ; Humans ; Infant ; Lymphatic Diseases/complications ; Magnetic Resonance Imaging ; Male ; Thymus Gland/pathology ; Tomography, X-Ray Computed
    Language English
    Publishing date 2019-04-14
    Publishing country Australia
    Document type Case Reports ; Journal Article
    ZDB-ID 1470376-2
    ISSN 1442-200X ; 1328-8067
    ISSN (online) 1442-200X
    ISSN 1328-8067
    DOI 10.1111/ped.13810
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