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Detection of autoantibody in pediatric encephalitis

Research Project

Project/Area Number 19K10613
Research Category

Grant-in-Aid for Scientific Research (C)

Allocation TypeMulti-year Fund
Section一般
Review Section Basic Section 58020:Hygiene and public health-related: including laboratory approach
Research InstitutionKyushu University (2021-2023)
Fukuoka Children’s Hospital (2019-2020)

Principal Investigator

Chong Pin Fee  九州大学, 大学病院, 助教 (90835167)

Co-Investigator(Kenkyū-buntansha) 酒井 康成  九州大学, 医学研究院, 准教授 (10380396)
Project Period (FY) 2019-04-01 – 2024-03-31
Project Status Completed (Fiscal Year 2023)
Budget Amount *help
¥4,420,000 (Direct Cost: ¥3,400,000、Indirect Cost: ¥1,020,000)
Fiscal Year 2022: ¥910,000 (Direct Cost: ¥700,000、Indirect Cost: ¥210,000)
Fiscal Year 2021: ¥910,000 (Direct Cost: ¥700,000、Indirect Cost: ¥210,000)
Fiscal Year 2020: ¥910,000 (Direct Cost: ¥700,000、Indirect Cost: ¥210,000)
Fiscal Year 2019: ¥1,690,000 (Direct Cost: ¥1,300,000、Indirect Cost: ¥390,000)
Keywords急性脳炎 / 小児 / 自己免疫性脳炎 / 中枢脱髄性疾患 / 脂質代謝 / 抗MOG抗体 / 急性脳症 / 中枢性脱髄性疾患 / 疫学調査 / 小児脳炎 / 自己抗体
Outline of Research at the Start

本研究の概要は、福岡県を中心とした九州内の小児医療機関の多施設、前向きコホート研究を基盤とした小児脳炎の疫学調査である。感染性脳炎、自己免疫性脳炎、中枢脱髄性疾患の割合、発症頻度を始め、異なる病態ごと脳炎の予後の相違等を明らかにするために行う研究である。

Outline of Final Research Achievements

To classify etiologically unknown pediatric encephalitis cases systemically, we developed a research procedure employing a combination of cell-based assay and FilmArray meningitis/encephalitis panel. During the research period, in collaboration with medical institutions in Fukuoka prefecture, we were able to identify causative viruses in 5 cases, and NMDA receptor antibody in 6 cases from cerebrospinal fluid of pediatric encephalitis patients with unknown cause. We were unable to detect LGI1, CASPR2, GABAB receptor, and AMPA1/2 receptor antibodies as seen in adult patients. We also detected 15 cases of MOG antibodies in blood specimens, confirming the diagnosis of MOG antibody-positive central demyelinating disease. Analysis of lipid metabolism from cerebrospinal fluid specimens of MOG antibody-positive patients revealed a metabolic profile that differed from that of controls.

Academic Significance and Societal Importance of the Research Achievements

小児期発症の脳炎は原因不明なことが多く、本研究では福岡県における感染性脳炎、特異的自己抗体による自己免疫性脳炎や中枢脱髄性疾患の疫学特徴を検討した。小児脳炎における自己抗体の検出傾向は成人と異なることを判明した。また、小児脳炎において最も頻度の多いMOG抗体関連疾患につき、非脱髄性疾患と比較し、特異的な脂質代謝を示すことを明らかにした。代謝経路が判明できれば、今後、MOG抗体関連疾患の病態解明や将来的に病態に選択性の高い創薬へとつながる可能性がある。

Report

(6 results)
  • 2023 Annual Research Report   Final Research Report ( PDF )
  • 2022 Research-status Report
  • 2021 Research-status Report
  • 2020 Research-status Report
  • 2019 Research-status Report
  • Research Products

    (34 results)

All 2024 2023 2022 2021 2020 2019

All Journal Article (21 results) (of which Int'l Joint Research: 2 results,  Peer Reviewed: 21 results,  Open Access: 4 results) Presentation (13 results) (of which Int'l Joint Research: 4 results,  Invited: 6 results)

  • [Journal Article] Persistent intracranial hyper-inflammation in ruptured cerebral aneurysm after COVID-19: case report and review of the literature2024

    • Author(s)
      Chong PF, Higashi K, Matsuoka W, Arimura K, Sangatsuda Y, Iwaki K, Sonoda Y, Ichimiya Y, Kamori A, Kawakami A, Mizuguchi S, Kaku N, Sakai Y, Ohga S.
    • Journal Title

      BMC Neurol

      Volume: 2 Issue: 1 Pages: 17-17

    • DOI

      10.1186/s12883-023-03493-z

    • Related Report
      2023 Annual Research Report
    • Peer Reviewed / Open Access
  • [Journal Article] The Etiology and Outcome of Area Postrema Syndrome in Childhood: Two Cases and a Literature Review2024

    • Author(s)
      Tomari Y, Igata Y, Chong PF, Kajiwara K, Hatai E, Sonoda Y, Oba U, Kaku N, Koga Y, Sakai Y, Ohga S.
    • Journal Title

      Pediatr Neurol

      Volume: 152 Pages: 11-15

    • DOI

      10.1016/j.pediatrneurol.2023.12.010

    • Related Report
      2023 Annual Research Report
    • Peer Reviewed
  • [Journal Article] Divergent neurodevelopmental profiles of very-low-birth-weight infants2024

    • Author(s)
      Ogata R, Watanabe K, Chong PF, Okamoto J, Sakemi Y, Nakashima T, Ohno T, Nomiyama H, Sonoda Y, Ichimiya Y, Inoue H, Ochiai M, Yamashita H, Sakai Y, Ohga S.
    • Journal Title

      Pediatr Res

      Volume: 95 Issue: 1 Pages: 233-240

    • DOI

      10.1038/s41390-023-02778-w

    • Related Report
      2023 Annual Research Report
    • Peer Reviewed
  • [Journal Article] Long-lasting pain and somatosensory disturbances in children with myelin oligodendrocyte glycoprotein antibody-associated disease2023

    • Author(s)
      Ichimiya Yuko、Chong Pin Fee、Sonoda Yuri、Tocan Vlad、Watanabe Mitsuru、Torisu Hiroyuki、Kira Ryutaro、Takahashi Toshiyuki、Kira Jun-Ichi、Isobe Noriko、Sakai Yasunari、Ohga Shouichi
    • Journal Title

      European Journal of Pediatrics

      Volume: 182 Issue: 7 Pages: 3175-3185

    • DOI

      10.1007/s00431-023-04989-z

    • Related Report
      2023 Annual Research Report
    • Peer Reviewed / Open Access
  • [Journal Article] Critical vitamin deficiencies in autism spectrum disorder: Reversible and irreversible outcomes2022

    • Author(s)
      P. F. Chong, M. Torio, F. Fujii, Y. Hirata, W. Matsuoka, Y. Sonoda, et al.
    • Journal Title

      Eur J Clin Nutr

      Volume: 76 Issue: 11 Pages: 1618-1621

    • DOI

      10.1038/s41430-022-01170-x

    • Related Report
      2022 Research-status Report
    • Peer Reviewed
  • [Journal Article] Clinical and electrophysiological features of acute flaccid myelitis: A national cohort study.2021

    • Author(s)
      Chong PF, Torisu H, Yasumoto S, Okumura A, Mori H, Sato T, Kimura J, Ohga S, Tanaka-Taya K, Kira R
    • Journal Title

      Clin Neurophysiol.

      Volume: 132 Issue: 10 Pages: 2456-2463

    • DOI

      10.1016/j.clinph.2021.07.013

    • Related Report
      2021 Research-status Report
    • Peer Reviewed / Open Access
  • [Journal Article] Acute flaccid myelitis: cause, diagnosis, and management2021

    • Author(s)
      Murphy OC, Messacar K, Benson L, et al.
    • Journal Title

      The Lancet

      Volume: 397 Issue: 10271 Pages: 334-346

    • DOI

      10.1016/s0140-6736(20)32723-9

    • Related Report
      2020 Research-status Report
    • Peer Reviewed / Int'l Joint Research
  • [Journal Article] Three-Year Longitudinal Motor Function and Disability Level of Acute Flaccid Myelitis2021

    • Author(s)
      Chong Pin Fee、Kira Ryutaro、Torisu Hiroyuki、Yasumoto Sawa、Okumura Akihisa、Mori Harushi、Tanaka-Taya Keiko.
    • Journal Title

      Pediatric Neurology

      Volume: 116 Pages: 14-19

    • DOI

      10.1016/j.pediatrneurol.2020.11.019

    • Related Report
      2020 Research-status Report
    • Peer Reviewed
  • [Journal Article] Infantile spasms and early-onset progressive polycystic renal lesions associated with TSC2/PKD1 contiguous gene deletion syndrome2021

    • Author(s)
      Matsubara Yoshie、Akamine Satoshi、Chong Pin Fee、Kawakami Saori、Maehara Kenji、Kaku Yoshitsugu、Kurokawa Mari、Morisada Naoya、Iijima Kazumoto、Kira Ryutaro
    • Journal Title

      Seizure

      Volume: 86 Pages: 82-84

    • DOI

      10.1016/j.seizure.2021.01.016

    • Related Report
      2020 Research-status Report
    • Peer Reviewed
  • [Journal Article] Disseminated cortical and subcortical lesions in neonatal enterovirus 71 encephalitis2020

    • Author(s)
      Nakamura Ryoko、Chong Pin Fee、Haraguchi Kohei、Katano Harutaka、Tanaka-Taya Keiko、Kira Ryutaro
    • Journal Title

      Journal of NeuroVirology

      Volume: 26 Issue: 5 Pages: 790-792

    • DOI

      10.1007/s13365-020-00843-2

    • Related Report
      2020 Research-status Report
    • Peer Reviewed
  • [Journal Article] Acute Flaccid Myelitis With Neuroradiological Finding of Brachial Plexus Swelling2020

    • Author(s)
      Chong Pin Fee、Yoshida Takeshi、Yuasa Shota、Mori Harushi、Tanaka-Taya Keiko、Kira Ryutaro
    • Journal Title

      Pediatric Neurology

      Volume: 109 Pages: 85-88

    • DOI

      10.1016/j.pediatrneurol.2020.04.004

    • Related Report
      2020 Research-status Report 2019 Research-status Report
    • Peer Reviewed
  • [Journal Article] Influenza-associated encephalopathy with focal late reduced diffusion circumscribing a pre-existing cortical lesion2020

    • Author(s)
      Maeda Kenichi、Chong Pin Fee、Yamashita Fumiya、Akamine Satoshi、Kawakami Saori、Lee Sooyoung、Kira Ryutaro
    • Journal Title

      Journal of Neuroradiology

      Volume: 47 Issue: 3 Pages: 241-243

    • DOI

      10.1016/j.neurad.2020.01.086

    • Related Report
      2020 Research-status Report
    • Peer Reviewed
  • [Journal Article] Isolated cranial neuritis of the oculomotor nerve: Expanding the MOG phenotype?2020

    • Author(s)
      Kawakami Saori、Akamine Satoshi、Chong Pin Fee、Yamashita Fumiya、Maeda Kenichi、Takahashi Toshiyuki、Kira Ryutaro
    • Journal Title

      Multiple Sclerosis and Related Disorders

      Volume: 41 Pages: 102040-102040

    • DOI

      10.1016/j.msard.2020.102040

    • Related Report
      2019 Research-status Report
    • Peer Reviewed
  • [Journal Article] Disseminated cortical and subcortical lesions in neonatal enterovirus 71 encephalitis2020

    • Author(s)
      Nakamura Ryoko, Chong Pin Fee, Haraguchi Kohei, Katano Harutaka, Tanaka-Taya Keiko, Kira Ryutaro
    • Journal Title

      Journal of NeuroVirology

      Volume: -

    • Related Report
      2019 Research-status Report
    • Peer Reviewed
  • [Journal Article] West Syndrome in an Infant With Vitamin B12 Deficiency Born to Autoantibodies Positive Mother2019

    • Author(s)
      Chong Pin Fee、Matsukura Masaru、Fukui Kaoru、Watanabe Yoriko、Matsumoto Naomichi、Kira Ryutaro
    • Journal Title

      Frontiers in Pediatrics

      Volume: 7

    • DOI

      10.3389/fped.2019.00531

    • Related Report
      2019 Research-status Report
    • Peer Reviewed / Open Access
  • [Journal Article] Cytotoxic lesion of the corpus callosum exclusively at the genu in a case of callosal hypogenesis2019

    • Author(s)
      Tsuji Moeri、Chong Pin Fee、Yamashita Fumiya、Maeda Kenichi、Kira Ryutaro
    • Journal Title

      Journal of Neuroradiology

      Volume: 46 Issue: 3 Pages: 222-223

    • DOI

      10.1016/j.neurad.2018.11.001

    • Related Report
      2019 Research-status Report
    • Peer Reviewed
  • [Journal Article] The recurrent postzygotic pathogenic variant p.Glu47Lys in RHOA causes a novel recognizable neuroectodermal phenotype2019

    • Author(s)
      Yigit Gokhan、Saida Ken、DeMarzo Danielle、Miyake Noriko、Fujita Atsushi、Yang Tan Tiong、White Susan M.、Wadley Alexandrea、Toliat Mohammad R.、Motameny Susanne、Franitza Marek、Stutterd Chloe A.、Chong Pin F.、Kira Ryutaro、et al.
    • Journal Title

      Human Mutation

      Volume: 41 Issue: 3 Pages: 591-599

    • DOI

      10.1002/humu.23964

    • Related Report
      2019 Research-status Report
    • Peer Reviewed / Int'l Joint Research
  • [Journal Article] Genomic backgrounds of Japanese patients with undiagnosed neurodevelopmental disorders2019

    • Author(s)
      Yamamoto Toshiyuki、Imaizumi Taichi、Yamamoto-Shimojima Keiko、Kurahashi Hirokazuら,
    • Journal Title

      Brain and Development

      Volume: 41 Issue: 9 Pages: 776-782

    • DOI

      10.1016/j.braindev.2019.05.007

    • Related Report
      2019 Research-status Report
    • Peer Reviewed
  • [Journal Article] Global Central Nervous System Atrophy in Spinal Muscular Atrophy Type 02019

    • Author(s)
      Maeda Kenichi、Chong Pin Fee、Yamashita Fumiya、Akamine Satoshi、Kawakami Saori、Saito Kayoko、Takahata Yasushi、Kira Ryutaro
    • Journal Title

      Annals of Neurology

      Volume: 86 Issue: 5 Pages: 801-802

    • DOI

      10.1002/ana.25596

    • Related Report
      2019 Research-status Report
    • Peer Reviewed
  • [Journal Article] Long surviving classical Menkes disease treated with weekly intravenous copper therapy2019

    • Author(s)
      Ogata Reina、Chong Pin Fee、Maeda Kenichi、Imagi Toru、Nakamura Ryoko、Kawamura Nobuko、Kira Ryutaro
    • Journal Title

      Journal of Trace Elements in Medicine and Biology

      Volume: 54 Pages: 172-174

    • DOI

      10.1016/j.jtemb.2019.04.020

    • Related Report
      2019 Research-status Report
    • Peer Reviewed
  • [Journal Article] Sequential radiologic findings in osteopathia striata with cranial sclerosis.2019

    • Author(s)
      Tomita Y, Chong P-F, Yamamoto T, Akaminea S, Imaizumi T, Kira R.
    • Journal Title

      Diagn Interv Imaging.

      Volume: 100 Issue: 9 Pages: 529-531

    • DOI

      10.1016/j.diii.2019.04.001

    • Related Report
      2019 Research-status Report
    • Peer Reviewed
  • [Presentation] The utility of multiplex assay in the differential diagnosis of pediatric CNS infection2023

    • Author(s)
      Chong Pin Fee
    • Organizer
      World Meningitis Day Forum 2023
    • Related Report
      2023 Annual Research Report
    • Invited
  • [Presentation] 小児の炎症性脊髄疾患2023

    • Author(s)
      チョン ピンフィー
    • Organizer
      第65回日本小児神経学会学術集会
    • Related Report
      2023 Annual Research Report
    • Invited
  • [Presentation] AFPの全国調査の概要と臨床的な視点2023

    • Author(s)
      チョン ピンフィー
    • Organizer
      第27回日本ワクチン学会・第64回日本臨床ウイルス学会合同学術集会
    • Related Report
      2023 Annual Research Report
    • Invited
  • [Presentation] COVID-19感染後に発症した脳動脈瘤破裂によるくも膜下出血の男児例2023

    • Author(s)
      チョン ピンフィー、東加奈子、松岡若利、有村公一、三月田祐平、岩城克馬、家守章子、川上晶子、園田有里、一宮優子、水口壮一、賀来典之、酒井康成、大賀正一
    • Organizer
      第65回日本小児神経学会学術集会
    • Related Report
      2023 Annual Research Report
  • [Presentation] Pediatric and transitional MOGAD: distinctive features and pathogenesis2022

    • Author(s)
      Chong Pin Fee
    • Organizer
      14th Pan-Asian Committee for Treatment and Research in Multiple Sclerosis Congress
    • Related Report
      2022 Research-status Report
    • Int'l Joint Research / Invited
  • [Presentation] A patient with West syndrome and autistic spectrum disorder with SCN2A gene deletion without SCN1A involvement2020

    • Author(s)
      Pin Fee Chong, Saori Kawakami, Satoshi Akamine, Ryutaro Kira
    • Organizer
      21st Annual Meeting of Infantile Seizure Society
    • Related Report
      2020 Research-status Report
    • Int'l Joint Research
  • [Presentation] RHOA体細胞モザイク変異認めた伊藤白斑の1症例2020

    • Author(s)
      チョン ピンフィー、川上 沙織、山下 文也、前田 謙一、赤峰 哲、才田 謙、藤田 京志、三宅 紀子、松本 直通、吉良龍太郎
    • Organizer
      第62回日本小児神経学会学術集会
    • Related Report
      2020 Research-status Report
  • [Presentation] Restrictively defined acute flaccid myelitis: stricter definition for diagnosing acute flaccid myelitis2019

    • Author(s)
      Chong Pin Fee
    • Organizer
      15th Asian & Oceanian Congress of Child Neurology
    • Related Report
      2019 Research-status Report
    • Int'l Joint Research
  • [Presentation] An infant with dietary vitamin B12 deficiency due to maternal antibodies presenting with West syndrome2019

    • Author(s)
      Pin Fee Chong, Fumiya Yamashita, Kenichi Maeda, Satoshi Akamine, Ryutaro Kira
    • Organizer
      20th Annual Meeting of Infantile Seizure Society
    • Related Report
      2019 Research-status Report
    • Int'l Joint Research
  • [Presentation] 急性弛緩性脊髄炎の臨床像:2018-2019の動向2019

    • Author(s)
      チョン ピンフィー、吉良龍太郎
    • Organizer
      第61回日本小児神経学術集会
    • Related Report
      2019 Research-status Report
    • Invited
  • [Presentation] 尿中マルベリー細胞を認めた腎機能正常な古典型ファブリー病男児例2019

    • Author(s)
      チョン ピンフィー
    • Organizer
      Conference of Fabry disease
    • Related Report
      2019 Research-status Report
    • Invited
  • [Presentation] 成人期まで経静脈的銅補充を行った古典型メンケス病の一例2019

    • Author(s)
      チョン ピンフィー、緒方怜奈、前田謙一、中村涼子、今城透、山下文也、赤峰哲、吉良龍太郎
    • Organizer
      第61回日本小児神経学会学術集会
    • Related Report
      2019 Research-status Report
  • [Presentation] 憤怒けいれんのmimickers2019

    • Author(s)
      チョン ピンフィー、川上沙織、山下文也、前田謙一、赤峰哲、吉良龍太郎
    • Organizer
      第53回日本てんかん学会学術集会
    • Related Report
      2019 Research-status Report

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Published: 2019-04-18   Modified: 2025-01-30  

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