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New type of encephalomyelitis responsive to trimethoprim/sulfamethoxazole treatment in Japan
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 Japanes...
Autores principales: | , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
Lippincott Williams & Wilkins
2015
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Materias: | |
Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4537311/ https://www.ncbi.nlm.nih.gov/pubmed/26309903 http://dx.doi.org/10.1212/NXI.0000000000000143 |
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author | 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 |
author_facet | 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 |
author_sort | Sakiyama, Yusuke |
collection | PubMed |
description | 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. |
format | Online Article Text |
id | pubmed-4537311 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2015 |
publisher | Lippincott Williams & Wilkins |
record_format | MEDLINE/PubMed |
spelling | pubmed-45373112015-08-25 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 Neurol Neuroimmunol Neuroinflamm Article 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. Lippincott Williams & Wilkins 2015-08-13 /pmc/articles/PMC4537311/ /pubmed/26309903 http://dx.doi.org/10.1212/NXI.0000000000000143 Text en © 2015 American Academy of Neurology This is an open access article distributed under the terms of the Creative Commons Attribution-NonCommercial-NoDerivatives License 4.0 (CC BY-NC-ND) (http://creativecommons.org/licenses/by-nc-nd/4.0/) , which permits downloading and sharing the work provided it is properly cited. The work cannot be changed in any way or used commercially. |
spellingShingle | Article 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 New type of encephalomyelitis responsive to trimethoprim/sulfamethoxazole treatment in Japan |
title | New type of encephalomyelitis responsive to trimethoprim/sulfamethoxazole treatment in Japan |
title_full | New type of encephalomyelitis responsive to trimethoprim/sulfamethoxazole treatment in Japan |
title_fullStr | New type of encephalomyelitis responsive to trimethoprim/sulfamethoxazole treatment in Japan |
title_full_unstemmed | New type of encephalomyelitis responsive to trimethoprim/sulfamethoxazole treatment in Japan |
title_short | New type of encephalomyelitis responsive to trimethoprim/sulfamethoxazole treatment in Japan |
title_sort | new type of encephalomyelitis responsive to trimethoprim/sulfamethoxazole treatment in japan |
topic | Article |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4537311/ https://www.ncbi.nlm.nih.gov/pubmed/26309903 http://dx.doi.org/10.1212/NXI.0000000000000143 |
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