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BMC Musculoskeletal Disorders
Published in: BMC Musculoskeletal Disorders 1/2023

Open Access 01-12-2023 | Spastic Paraplegia | Case report

HTLV-1-associated myelopathy/tropical spastic paraplegia with sporadic late-onset nemaline myopathy: a case report

Authors: Eiji Matsuura, Satoshi Nozuma, Ayano Shigehisa, Mika Dozono, Tomonori Nakamura, Masakazu Tanaka, Ryuji Kubota, Akihiro Hashiguchi, Hiroshi Takashima

Published in: BMC Musculoskeletal Disorders | Issue 1/2023

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Abstract

Background

Sporadic late onset nemaline myopathy (SLONM) is a muscle disorder characterized by the presence of nemaline rods in muscle fibers. SLONM has no known genetic cause but has been associated with monoclonal gammopathy of undetermined significance and with human immunodeficiency virus (HIV) infection. Human T-cell leukemia virus-1 (HTLV-1) is a known causative agent of adult T-cell leukemia/lymphoma and HTLV-1 associated myelopathy/tropical spastic paraplegia (HAM/TSP), a chronic inflammatory neurological disease. HTLV-1 has been reported to be implicated in inflammatory myopathies, as well as in HIV infection.; however, there have been no reports of an association between HTLV-1 infection and SLONM to date.

Case presentation

A 70-year-old Japanese woman presented with gait disturbance, lumbar kyphosis, and respiratory dysfunction. The diagnosis of HAM/TSP with SLONM was made based on characteristic clinical symptoms of HAM/TSP, such as spasticity in the lower extremities, and cerebrospinal fluid test results; and of SLONM, such as generalized head drooping, respiratory failure, and muscle biopsy results. Steroid treatment was initiated and improvement in her stooped posture was observed after 3 days of treatment.

Conclusion

This is the first case report of SLONM combined with HTLV-1 infection. Further studies are needed to elucidate the relationship between retroviruses and muscle diseases.
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Metadata
Title
HTLV-1-associated myelopathy/tropical spastic paraplegia with sporadic late-onset nemaline myopathy: a case report
Authors
Eiji Matsuura
Satoshi Nozuma
Ayano Shigehisa
Mika Dozono
Tomonori Nakamura
Masakazu Tanaka
Ryuji Kubota
Akihiro Hashiguchi
Hiroshi Takashima
Publication date
01-12-2023
Publisher
BioMed Central
Published in
BMC Musculoskeletal Disorders / Issue 1/2023
Electronic ISSN: 1471-2474
DOI
https://doi.org/10.1186/s12891-023-06461-3

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