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New type of encephalomyelitis responsive to trimethoprim/sulfamethoxazole treatment in Japan.
Sakiyama, Yusuke; Kanda, Naoaki; Higuchi, Yujiro; Yoshimura, Michiyoshi; Wakaguri, Hiroyuki; Takata, Yoshiharu; Watanabe, Osamu; Yuan, Junhui; Tashiro, Yuichi; Saigo, Ryuji; Nozuma, Satoshi; Yoshimura, Akiko; Arishima, Shiho; Ikeda, Kenichi; Shinohara, Kazuya; Arata, Hitoshi; Michizono, Kumiko; Higashi, Keiko; Hashiguchi, Akihiro; Okamoto, Yuji; Hirano, Ryuki; Shiraishi, Tadafumi; Matsuura, Eiji; Okubo, Ryuichi; Higuchi, Itsuro; Goto, Masamichi; Hirano, Hirofumi; Sano, Akira; Iwasaki, Takuya; Matsuda, Fumihiko; Izumo, Shuji; Takashima, Hiroshi.
Afiliação
  • Sakiyama Y; Department of Neurology and Geriatrics (Y.S., N.K., Y.H., M.Y., Y. Takata, O.W., J.Y., Y. Tashiro, R.S., S.N., A.Y., K.I., K.S., H.A., K.M., K.H., A.H., Y.O., R.H., T.S., E.M., R.O., I.H., T.I., H.T.), Center for Chronic Viral Diseases (S.A., S.I.), Department of Neurosurgery (H.H.), and Department
  • Kanda N; Department of Neurology and Geriatrics (Y.S., N.K., Y.H., M.Y., Y. Takata, O.W., J.Y., Y. Tashiro, R.S., S.N., A.Y., K.I., K.S., H.A., K.M., K.H., A.H., Y.O., R.H., T.S., E.M., R.O., I.H., T.I., H.T.), Center for Chronic Viral Diseases (S.A., S.I.), Department of Neurosurgery (H.H.), and Department
  • Higuchi Y; Department of Neurology and Geriatrics (Y.S., N.K., Y.H., M.Y., Y. Takata, O.W., J.Y., Y. Tashiro, R.S., S.N., A.Y., K.I., K.S., H.A., K.M., K.H., A.H., Y.O., R.H., T.S., E.M., R.O., I.H., T.I., H.T.), Center for Chronic Viral Diseases (S.A., S.I.), Department of Neurosurgery (H.H.), and Department
  • Yoshimura M; Department of Neurology and Geriatrics (Y.S., N.K., Y.H., M.Y., Y. Takata, O.W., J.Y., Y. Tashiro, R.S., S.N., A.Y., K.I., K.S., H.A., K.M., K.H., A.H., Y.O., R.H., T.S., E.M., R.O., I.H., T.I., H.T.), Center for Chronic Viral Diseases (S.A., S.I.), Department of Neurosurgery (H.H.), and Department
  • Wakaguri H; Department of Neurology and Geriatrics (Y.S., N.K., Y.H., M.Y., Y. Takata, O.W., J.Y., Y. Tashiro, R.S., S.N., A.Y., K.I., K.S., H.A., K.M., K.H., A.H., Y.O., R.H., T.S., E.M., R.O., I.H., T.I., H.T.), Center for Chronic Viral Diseases (S.A., S.I.), Department of Neurosurgery (H.H.), and Department
  • Takata Y; Department of Neurology and Geriatrics (Y.S., N.K., Y.H., M.Y., Y. Takata, O.W., J.Y., Y. Tashiro, R.S., S.N., A.Y., K.I., K.S., H.A., K.M., K.H., A.H., Y.O., R.H., T.S., E.M., R.O., I.H., T.I., H.T.), Center for Chronic Viral Diseases (S.A., S.I.), Department of Neurosurgery (H.H.), and Department
  • Watanabe O; Department of Neurology and Geriatrics (Y.S., N.K., Y.H., M.Y., Y. Takata, O.W., J.Y., Y. Tashiro, R.S., S.N., A.Y., K.I., K.S., H.A., K.M., K.H., A.H., Y.O., R.H., T.S., E.M., R.O., I.H., T.I., H.T.), Center for Chronic Viral Diseases (S.A., S.I.), Department of Neurosurgery (H.H.), and Department
  • Yuan J; Department of Neurology and Geriatrics (Y.S., N.K., Y.H., M.Y., Y. Takata, O.W., J.Y., Y. Tashiro, R.S., S.N., A.Y., K.I., K.S., H.A., K.M., K.H., A.H., Y.O., R.H., T.S., E.M., R.O., I.H., T.I., H.T.), Center for Chronic Viral Diseases (S.A., S.I.), Department of Neurosurgery (H.H.), and Department
  • Tashiro Y; Department of Neurology and Geriatrics (Y.S., N.K., Y.H., M.Y., Y. Takata, O.W., J.Y., Y. Tashiro, R.S., S.N., A.Y., K.I., K.S., H.A., K.M., K.H., A.H., Y.O., R.H., T.S., E.M., R.O., I.H., T.I., H.T.), Center for Chronic Viral Diseases (S.A., S.I.), Department of Neurosurgery (H.H.), and Department
  • Saigo R; Department of Neurology and Geriatrics (Y.S., N.K., Y.H., M.Y., Y. Takata, O.W., J.Y., Y. Tashiro, R.S., S.N., A.Y., K.I., K.S., H.A., K.M., K.H., A.H., Y.O., R.H., T.S., E.M., R.O., I.H., T.I., H.T.), Center for Chronic Viral Diseases (S.A., S.I.), Department of Neurosurgery (H.H.), and Department
  • Nozuma S; Department of Neurology and Geriatrics (Y.S., N.K., Y.H., M.Y., Y. Takata, O.W., J.Y., Y. Tashiro, R.S., S.N., A.Y., K.I., K.S., H.A., K.M., K.H., A.H., Y.O., R.H., T.S., E.M., R.O., I.H., T.I., H.T.), Center for Chronic Viral Diseases (S.A., S.I.), Department of Neurosurgery (H.H.), and Department
  • Yoshimura A; Department of Neurology and Geriatrics (Y.S., N.K., Y.H., M.Y., Y. Takata, O.W., J.Y., Y. Tashiro, R.S., S.N., A.Y., K.I., K.S., H.A., K.M., K.H., A.H., Y.O., R.H., T.S., E.M., R.O., I.H., T.I., H.T.), Center for Chronic Viral Diseases (S.A., S.I.), Department of Neurosurgery (H.H.), and Department
  • Arishima S; Department of Neurology and Geriatrics (Y.S., N.K., Y.H., M.Y., Y. Takata, O.W., J.Y., Y. Tashiro, R.S., S.N., A.Y., K.I., K.S., H.A., K.M., K.H., A.H., Y.O., R.H., T.S., E.M., R.O., I.H., T.I., H.T.), Center for Chronic Viral Diseases (S.A., S.I.), Department of Neurosurgery (H.H.), and Department
  • Ikeda K; Department of Neurology and Geriatrics (Y.S., N.K., Y.H., M.Y., Y. Takata, O.W., J.Y., Y. Tashiro, R.S., S.N., A.Y., K.I., K.S., H.A., K.M., K.H., A.H., Y.O., R.H., T.S., E.M., R.O., I.H., T.I., H.T.), Center for Chronic Viral Diseases (S.A., S.I.), Department of Neurosurgery (H.H.), and Department
  • Shinohara K; Department of Neurology and Geriatrics (Y.S., N.K., Y.H., M.Y., Y. Takata, O.W., J.Y., Y. Tashiro, R.S., S.N., A.Y., K.I., K.S., H.A., K.M., K.H., A.H., Y.O., R.H., T.S., E.M., R.O., I.H., T.I., H.T.), Center for Chronic Viral Diseases (S.A., S.I.), Department of Neurosurgery (H.H.), and Department
  • Arata H; Department of Neurology and Geriatrics (Y.S., N.K., Y.H., M.Y., Y. Takata, O.W., J.Y., Y. Tashiro, R.S., S.N., A.Y., K.I., K.S., H.A., K.M., K.H., A.H., Y.O., R.H., T.S., E.M., R.O., I.H., T.I., H.T.), Center for Chronic Viral Diseases (S.A., S.I.), Department of Neurosurgery (H.H.), and Department
  • Michizono K; Department of Neurology and Geriatrics (Y.S., N.K., Y.H., M.Y., Y. Takata, O.W., J.Y., Y. Tashiro, R.S., S.N., A.Y., K.I., K.S., H.A., K.M., K.H., A.H., Y.O., R.H., T.S., E.M., R.O., I.H., T.I., H.T.), Center for Chronic Viral Diseases (S.A., S.I.), Department of Neurosurgery (H.H.), and Department
  • Higashi K; Department of Neurology and Geriatrics (Y.S., N.K., Y.H., M.Y., Y. Takata, O.W., J.Y., Y. Tashiro, R.S., S.N., A.Y., K.I., K.S., H.A., K.M., K.H., A.H., Y.O., R.H., T.S., E.M., R.O., I.H., T.I., H.T.), Center for Chronic Viral Diseases (S.A., S.I.), Department of Neurosurgery (H.H.), and Department
  • Hashiguchi A; Department of Neurology and Geriatrics (Y.S., N.K., Y.H., M.Y., Y. Takata, O.W., J.Y., Y. Tashiro, R.S., S.N., A.Y., K.I., K.S., H.A., K.M., K.H., A.H., Y.O., R.H., T.S., E.M., R.O., I.H., T.I., H.T.), Center for Chronic Viral Diseases (S.A., S.I.), Department of Neurosurgery (H.H.), and Department
  • Okamoto Y; Department of Neurology and Geriatrics (Y.S., N.K., Y.H., M.Y., Y. Takata, O.W., J.Y., Y. Tashiro, R.S., S.N., A.Y., K.I., K.S., H.A., K.M., K.H., A.H., Y.O., R.H., T.S., E.M., R.O., I.H., T.I., H.T.), Center for Chronic Viral Diseases (S.A., S.I.), Department of Neurosurgery (H.H.), and Department
  • Hirano R; Department of Neurology and Geriatrics (Y.S., N.K., Y.H., M.Y., Y. Takata, O.W., J.Y., Y. Tashiro, R.S., S.N., A.Y., K.I., K.S., H.A., K.M., K.H., A.H., Y.O., R.H., T.S., E.M., R.O., I.H., T.I., H.T.), Center for Chronic Viral Diseases (S.A., S.I.), Department of Neurosurgery (H.H.), and Department
  • Shiraishi T; Department of Neurology and Geriatrics (Y.S., N.K., Y.H., M.Y., Y. Takata, O.W., J.Y., Y. Tashiro, R.S., S.N., A.Y., K.I., K.S., H.A., K.M., K.H., A.H., Y.O., R.H., T.S., E.M., R.O., I.H., T.I., H.T.), Center for Chronic Viral Diseases (S.A., S.I.), Department of Neurosurgery (H.H.), and Department
  • Matsuura E; Department of Neurology and Geriatrics (Y.S., N.K., Y.H., M.Y., Y. Takata, O.W., J.Y., Y. Tashiro, R.S., S.N., A.Y., K.I., K.S., H.A., K.M., K.H., A.H., Y.O., R.H., T.S., E.M., R.O., I.H., T.I., H.T.), Center for Chronic Viral Diseases (S.A., S.I.), Department of Neurosurgery (H.H.), and Department
  • Okubo R; Department of Neurology and Geriatrics (Y.S., N.K., Y.H., M.Y., Y. Takata, O.W., J.Y., Y. Tashiro, R.S., S.N., A.Y., K.I., K.S., H.A., K.M., K.H., A.H., Y.O., R.H., T.S., E.M., R.O., I.H., T.I., H.T.), Center for Chronic Viral Diseases (S.A., S.I.), Department of Neurosurgery (H.H.), and Department
  • Higuchi I; Department of Neurology and Geriatrics (Y.S., N.K., Y.H., M.Y., Y. Takata, O.W., J.Y., Y. Tashiro, R.S., S.N., A.Y., K.I., K.S., H.A., K.M., K.H., A.H., Y.O., R.H., T.S., E.M., R.O., I.H., T.I., H.T.), Center for Chronic Viral Diseases (S.A., S.I.), Department of Neurosurgery (H.H.), and Department
  • Goto M; Department of Neurology and Geriatrics (Y.S., N.K., Y.H., M.Y., Y. Takata, O.W., J.Y., Y. Tashiro, R.S., S.N., A.Y., K.I., K.S., H.A., K.M., K.H., A.H., Y.O., R.H., T.S., E.M., R.O., I.H., T.I., H.T.), Center for Chronic Viral Diseases (S.A., S.I.), Department of Neurosurgery (H.H.), and Department
  • Hirano H; Department of Neurology and Geriatrics (Y.S., N.K., Y.H., M.Y., Y. Takata, O.W., J.Y., Y. Tashiro, R.S., S.N., A.Y., K.I., K.S., H.A., K.M., K.H., A.H., Y.O., R.H., T.S., E.M., R.O., I.H., T.I., H.T.), Center for Chronic Viral Diseases (S.A., S.I.), Department of Neurosurgery (H.H.), and Department
  • Sano A; Department of Neurology and Geriatrics (Y.S., N.K., Y.H., M.Y., Y. Takata, O.W., J.Y., Y. Tashiro, R.S., S.N., A.Y., K.I., K.S., H.A., K.M., K.H., A.H., Y.O., R.H., T.S., E.M., R.O., I.H., T.I., H.T.), Center for Chronic Viral Diseases (S.A., S.I.), Department of Neurosurgery (H.H.), and Department
  • Iwasaki T; Department of Neurology and Geriatrics (Y.S., N.K., Y.H., M.Y., Y. Takata, O.W., J.Y., Y. Tashiro, R.S., S.N., A.Y., K.I., K.S., H.A., K.M., K.H., A.H., Y.O., R.H., T.S., E.M., R.O., I.H., T.I., H.T.), Center for Chronic Viral Diseases (S.A., S.I.), Department of Neurosurgery (H.H.), and Department
  • Matsuda F; Department of Neurology and Geriatrics (Y.S., N.K., Y.H., M.Y., Y. Takata, O.W., J.Y., Y. Tashiro, R.S., S.N., A.Y., K.I., K.S., H.A., K.M., K.H., A.H., Y.O., R.H., T.S., E.M., R.O., I.H., T.I., H.T.), Center for Chronic Viral Diseases (S.A., S.I.), Department of Neurosurgery (H.H.), and Department
  • Izumo S; Department of Neurology and Geriatrics (Y.S., N.K., Y.H., M.Y., Y. Takata, O.W., J.Y., Y. Tashiro, R.S., S.N., A.Y., K.I., K.S., H.A., K.M., K.H., A.H., Y.O., R.H., T.S., E.M., R.O., I.H., T.I., H.T.), Center for Chronic Viral Diseases (S.A., S.I.), Department of Neurosurgery (H.H.), and Department
  • Takashima H; Department of Neurology and Geriatrics (Y.S., N.K., Y.H., M.Y., Y. Takata, O.W., J.Y., Y. Tashiro, R.S., S.N., A.Y., K.I., K.S., H.A., K.M., K.H., A.H., Y.O., R.H., T.S., E.M., R.O., I.H., T.I., H.T.), Center for Chronic Viral Diseases (S.A., S.I.), Department of Neurosurgery (H.H.), and Department
Neurol Neuroimmunol Neuroinflamm ; 2(5): e143, 2015 Oct.
Article em En | MEDLINE | ID: mdl-26309903
ABSTRACT

OBJECTIVE:

To determine the causative pathogen and investigate the effective treatment of a new type of encephalomyelitis with an unknown pathogen in Japan and report the preliminary ultrastructural and genomic characterization of the causative agent.

METHODS:

From 2005 to 2012, we treated 4 Japanese patients with geographic clustering and comparable clinical features, serum/CSF cytology, and radiologic findings. Brain biopsy was conducted in all patients to analyze neuropathologic changes by histology, and electron microscopy was applied to reveal the features of the putative pathogen. Genomic DNA was obtained from the affected brain tissues and CSF, and an unbiased high-throughput sequencing approach was used to screen for specific genomic sequences indicative of the pathogen origin.

RESULTS:

All patients exhibited progressive dementia with involuntary tongue movements. Cytologic examination of CSF revealed elevated mononuclear cells. Abnormal MRI signals were observed in temporal lobes, subcortical white matter, and spinal cord. Biopsied brain tissue exhibited aggregated periodic acid-Schiff-positive macrophages and 2-7 µm diameter round/oval bodies without nuclei or cell walls scattered around the vessels. Unbiased high-throughput sequencing identified more than 100 archaea-specific DNA fragments. All patients were responsive to trimethoprim/sulfamethoxazole (TMP-SMX) plus corticosteroid therapy.

CONCLUSIONS:

We report 4 cases of encephalomyelitis due to an unknown pathogen. On the basis of ultrastructural and genomic studies, we propose a new disease entity resulting from a causative pathogen having archaeal features. TMP-SMX therapy was effective against this new type of encephalomyelitis.
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Texto completo: 1 Coleções: 01-internacional Base de dados: MEDLINE Tipo de estudo: Prognostic_studies Idioma: En Revista: Neurol Neuroimmunol Neuroinflamm Ano de publicação: 2015 Tipo de documento: Article
Texto completo
Imprimir
XML
PubMed Links
Buscar no Google

Texto completo: 1 Coleções: 01-internacional Base de dados: MEDLINE Tipo de estudo: Prognostic_studies Idioma: En Revista: Neurol Neuroimmunol Neuroinflamm Ano de publicação: 2015 Tipo de documento: Article