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HTLV-I virological and histopathological analysis in two cases of anti-centromere-antibody-seropositive Sjögren’s syndrome

  • Original Article
  • Published:
Modern Rheumatology

Abstract

Introduction

The aim of this study was to show the clinical and pathological characteristics of anti-centromere-antibody (ACA)-seropositive Sjögren’s syndrome (SS) in two anti-human T-cell leukemia virus type I (HTLV-I)-seropositive patients.

Methods

One patient was an HTLV-I carrier whereas the other was diagnosed with HTLV-I-associated myelopathy (HAM). Background data including serum HTLV-I titers, viral loads, and cytokine profiles were recorded. Azocarmine with aniline blue (Azan)–Mallory staining and immunohistochemistry of the labial salivary glands (LSGs) and a muscle biopsy specimen from the HAM patient were performed.

Results

Serum transforming growth factor beta (TGF-β), tumor necrosis factor alpha (TNF-α), and HTLV-I viral load were high in the HAM-SS patient compared with the HTLV-I carrier. Fibrous change in LSG was prominent in the HAM-SS patient. Although TGF-β expression was similar in the two patients, expression of HTLV-I-related proteins including p12, p28, group-specific antigen (GAG), and nuclear factor kappa-B (NF-κB) in the LSG were dominantly detected in the HAM-SS patient. Frequency of TGF-β staining in HTLV-I-seropositive SS patients without ACA, HTLV-I-seronegative SS patients with ACA, and HTLV-I-seronegative SS patients without ACA was lower than that of the previous two patients.

Conclusion

A high HTLV-I viral load in situ is supposed to promote the production of cytokines, especially TGF-β, resulting in the fibrous change of LSG in ACA-seropositive SS patients.

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Abbreviations

ACA:

Anti-centromere antibody

ANA:

Anti-nuclear antibody

CSF:

Cerebrospinal fluid

HAM:

HTLV-I-associated myelopathy

HTLV-I:

Human T-cell leukemia virus type I

IFN-γ:

Interferon gamma

MNC:

Mononuclear cell

LSG:

Labial salivary gland

SS:

Sjögren’s syndrome

TGF-β:

Transforming growth factor beta

TNF-α:

Tumor necrosis factor alpha

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Authors and Affiliations

  1. Unit of Translational Medicine, Department of Immunology and Rheumatology, Nagasaki University Graduate School of Biomedical Sciences, 1-7-1 Sakamoto, Nagasaki, Nagasaki, 852-8501, Japan

    Hideki Nakamura, Yoshiro Horai, Ayuko Tokuyama, Kunihiro Ichinose, Satoshi Yamasaki & Atsushi Kawakami

  2. Unit of Translational Medicine, Department of Neurology, Nagasaki University Graduate School of Biomedical Sciences, Nagasaki, Japan

    Shunsuke Yoshimura & Hideki Nakajima

  3. Department of Molecular Microbiology and Immunology, Nagasaki University Graduate School of Biomedical Sciences, Nagasaki, Japan

    Tatsufumi Nakamura

  4. Department of Pathology, Nagasaki University Hospital, Nagasaki, Japan

    Tomayoshi Hayashi

Authors
  1. Hideki Nakamura
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  2. Yoshiro Horai
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  3. Ayuko Tokuyama
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  4. Shunsuke Yoshimura
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  5. Hideki Nakajima
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  6. Kunihiro Ichinose
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  7. Satoshi Yamasaki
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  8. Tatsufumi Nakamura
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  9. Tomayoshi Hayashi
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  10. Atsushi Kawakami
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Corresponding author

Correspondence to Hideki Nakamura.

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Nakamura, H., Horai, Y., Tokuyama, A. et al. HTLV-I virological and histopathological analysis in two cases of anti-centromere-antibody-seropositive Sjögren’s syndrome. Mod Rheumatol 23, 133–139 (2013). https://doi.org/10.1007/s10165-012-0641-x

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  • DOI: https://doi.org/10.1007/s10165-012-0641-x

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