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Neurology
Inflammatory progressive multifocal leukoencephalopathy with human T-cell lymphotropic virus-1 coinfection
  1. Sachiko Hasebe1,
  2. Kota Maekawa2,
  3. Yukiko Shishido-Hara3,
  4. Kazuo Nakamichi4,
  5. Nobuaki Funata5 and
  6. Makio Takahashi6
  1. 1 Department of Neurology, Kyoto University Graduate School of Medicine Faculty of Medicine, Kyoto, Japan
  2. 2 Department of Neurology, Kobe City Medical Center General Hospital, Osaka, Japan
  3. 3 Pathology and Applied Neurobiology, Kyoto Prefectural University of Medicine, Kyoto, Japan
  4. 4 Department of Virology 1, National Institute of Infectious Diseases, Osaka, Japan
  5. 5 Department of Pathology, Tokyo Metropolitan Cancer and Infectious Diseases Center Komagome Hospital, Osaka, Japan
  6. 6 Neurodegenerative Disorders, Kansai Medical University, Hirakata, Osaka, Japan
  1. Correspondence to Dr Sachiko Hasebe; sachiko_h{at}kuhp.kyoto-u.ac.jp

Abstract

A middle-aged man with progressive multifocal leukoencephalopathy (PML) in a human T-cell lymphotropic virus type-1 (HTLV-1) carrier on haemodialysis presented with mild dysarthria and ataxia. Brain MRI revealed asymmetric T2-hyperintense lesions in the cerebral white matter, cerebellum and brainstem. A small amount of JC virus (JCV) genome in cerebrospinal fluid was detected by PCR and cerebellar biopsy demonstrated JCV-DNA presence. Pathological findings showed demyelinating lesions and glial cells with mildly enlarged nuclei, accompanied by T-lymphocytes, neutrophils and plasma cell infiltration. The CD4+/CD8+ratio was 0.83. High-dose corticosteroid therapy was effective for inflammatory PML lesions, and the administration of mefloquine combined with mirtazapine led to favourable outcome. The encephalitis in this case is considered to have occurred secondarily to JCV infection in the presence of HTLV-1 infection. Therefore, it is crucial to investigate the presence of HTLV-1 in order to understand the aetiology of this brain inflammation.

  • Infection (neurology)
  • Brain stem / cerebellum

https://doi.org/10.1136/bcr-2023-257805

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    Footnotes

    • Contributors The following authors were responsible for drafting of the text, sourcing and editing of clinical images, investigation results, drawing original diagrams and algorithms, and critical revision for important intellectual content: SH, KM, YS-H, KN, NF and MT. The following authors gave final approval of the manuscript: SH, KM, YS-H, KN, NF and MT.

    • Funding This work was supported by the Research Committee of Prion Disease and Slow Virus Infection, Research on Policy Planning and Evaluation for Rare and Intractable Diseases, Health and Labour Sciences Research Grants, The Ministry of Health, Labour and Welfare, Japan, and by JSPS KAKENHI (grant number 21K07450).

    • Case reports provide a valuable learning resource for the scientific community and can indicate areas of interest for future research. They should not be used in isolation to guide treatment choices or public health policy.

    • Competing interests None declared.

    • Provenance and peer review Not commissioned; externally peer-reviewed.