Dataset Information

New type of encephalomyelitis responsive to trimethoprim/sulfamethoxazole treatment in Japan.

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 ?m 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.

SUBMITTER: Sakiyama Y

PROVIDER: S-EPMC4537311 | biostudies-literature | 2015 Oct

REPOSITORIES: biostudies-literature

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Publications

New type of encephalomyelitis responsive to trimethoprim/sulfamethoxazole treatment in Japan.

Sakiyama Yusuke Y Kanda Naoaki N Higuchi Yujiro Y Yoshimura Michiyoshi M Wakaguri Hiroyuki H Takata Yoshiharu Y Watanabe Osamu O Yuan Junhui J Tashiro Yuichi Y Saigo Ryuji R Nozuma Satoshi S Yoshimura Akiko A Arishima Shiho S Ikeda Kenichi K Shinohara Kazuya K Arata Hitoshi H Michizono Kumiko K Higashi Keiko K Hashiguchi Akihiro A Okamoto Yuji Y Hirano Ryuki R Shiraishi Tadafumi T Matsuura Eiji E Okubo Ryuichi R Higuchi Itsuro I Goto Masamichi M Hirano Hirofumi H Sano Akira A Iwasaki Takuya T Matsuda Fumihiko F Izumo Shuji S Takashima Hiroshi H

Neurology(R) neuroimmunology & neuroinflammation 20150813 5

<h4>Objective</h4>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.<h4>Methods</h4>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 ...[more]

PMID: 26309903

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Dataset Information

New type of encephalomyelitis responsive to trimethoprim/sulfamethoxazole treatment in Japan.

Objective

Methods

Results

Conclusions

Publications

New type of encephalomyelitis responsive to trimethoprim/sulfamethoxazole treatment in Japan.

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