Remission of chronic type ATL in a patient with rheumatoid arthritis after withdrawing methotrexate and infliximab combination therapy: a case report

Abstract

A 70-year-old Japanese female was diagnosed with seropositive rheumatoid arthritis (RA) in January 2008. She had high disease activity and was treated with prednisolone, methotrexate (MTX) and infliximab (IFX). She was found to be positive for human T-lymphotropic virus type 1 (HTLV-1) antibody in 2012, and her HTLV-1 proviral DNA load (PVL) was as high as 5.82 copies per 100 white blood cells (WBCs). In 2014, she complained of fever and showed elevated soluble IL-2 receptors (sIL-2R) and WBCs. Abnormal lymphocytes with convoluted nuclei were found on blood smear analysis (13% of WBCs). She was negative for lymphadenopathy, skin lesion and organomegaly. She was diagnosed with chronic type adult T-cell leukaemia-lymphoma (ATL), and treatment with IFX and MTX was discontinued. Abnormal lymphocytes disappeared and sIL-2R level decreased a few months later. ATL did not relapse for more than 2 years. This case emphasises the need for careful observation in HTLV-1 positive patients with RA, especially in patients with high PVL. In addition, there is a possibility that prompt withdraw of biologics and MTX may contribute to the spontaneous remission of ATL.

Keywords:

• Rheumatoid arthritis
• human T-lymphotropic virus type 1
• adult T-cell leukaemia/lymphoma
• disease modifying anti-rheumatic drugs

Acknowledgements

We thank Dr. Hajime Nomura, Dr. Yuki Hashikura, Dr. Ikuo Yamamoto, Dr. Kazumi Umeki and Ms. Yuki Kaseda of University of Miyazaki for their technical support in this work. We also thank Ms. Michiyo Koufuku of the Institute of Rheumatology, Zenjin-kai Shimin-no-mori Hospital for data management.

Patient consent

Written informed consent for publication of this report was obtained from the patient by the author.

Conflict of interest

None.

Additional information

Funding

This work was supported by Research on rare and intractable diseases, Health, Labour and Welfare Sciences Research Grants and the Japan Agency for Medical Research and Development.