Abstract

HTLV-1-associated myelopathy/tropical spastic paraparesis (HAM/TSP) is an immune mediated myelopathy caused by the human T-lymphotropic virus type 1 (HTLV-1). The efficacy of treatments used for patients with HAM/TSP is uncertain. The aim of this study is to document the efficacy of pulsed methylprednisolone in patients with HAM/TSP. Data from an open cohort of 26 patients with HAM/TSP was retrospectively analysed. 1g IV methylprednisolone was infused on three consecutive days. The outcomes were pain, gait, urinary frequency and nocturia, a range of inflammatory markers and HTLV-1 proviral load. Treatment was well tolerated in all but one patient. Significant improvements in pain were: observed immediately, unrelated to duration of disease and maintained for three months. Improvement in gait was only seen on Day 3 of treatment. Baseline cytokine concentrations did not correlate to baseline pain or gait impairment but a decrease in tumour necrosis factor-alpha (TNF-α) concentration after pulsed methylprednisolone was associated with improvements in both. Until compared with placebo, treatment with pulsed methylprednisolone should be offered to patients with HAM/TSP for the treatment of pain present despite regular analgesia.

Highlights

  • The human T lymphotropic virus type 1 (HTLV-1) was recognised as the first human retrovirus, with oncogenic potential, following its discovery by independent groups in the USA[1] and Japan[2]

  • Of the 25 patients treated with 1g IV Methylprednisolone on three consecutive days, 24 completed the treatment without any side effects

  • The prevalence of the improvement in pain gradually decreased over time

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Summary

Introduction

The human T lymphotropic virus type 1 (HTLV-1) was recognised as the first human retrovirus, with oncogenic potential, following its discovery by independent groups in the USA[1] and Japan[2]. The prevalence of HTLV-1, estimated at 5–10 million worldwide, is variable between and even within countries[3]. High prevalence, defined as greater than 1:10,000 of the general population or greater than 1:100 first time blood donors, is reported in Japan, the Caribbean, Western and Southern Africa, South America and Melanesia[4]. European countries have a low prevalence with the exception of Romania[4]. Gessain et al first reported the PLOS ONE | DOI:10.1371/journal.pone.0152557. Gessain et al first reported the PLOS ONE | DOI:10.1371/journal.pone.0152557 April 14, 2016

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