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Case Series
Therapeutic responses to interferon-alpha in HTLV-1-associated myelopathy/tropical spastic paraparesis (HAM/TSP): Differing clinical responses to leukocyte-derived human interferon-alpha versus recombinant interferon-alpha
1 MD, MPH, Clinical Research Associate, Department of Neurology, University of Miami Miller School of Medicine, Miami, Florida, USA
2 MD, Professor of Neurology, Department of Neurology, University of Miami Miller School of Medicine, Miami, Florida, USA
3 MD, Associate Professor of Neurology, Department of Neurology, University of Miami Miller School of Medicine, Miami, Florida, USA
4 MD, Neurocritical Care Fellow, Department of Neurosurgery, UT Southwestern Medical Center, Dallas, Texas, USA
5 MD, PhD, Professor of Neurology, Department of Neurology, University of Miami Miller School of Medicine, Miami, Florida, USA
6
MD, Assistant Professor of Neurology, Department of Neurology, University of Miami Miller School of Medicine, Miami, Florida, USA,
† Author is deceased as of March 29, 2017.
Address correspondence to:
Melissa R Ortega
MD, 1120 NW 14th St, Miami, Florida 33136,
USA
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Article ID: 101118Z01CZ2020
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Zheng C, Sheremata WA, Tornes L, Campos Y, McCarthy M, Ortega MR. Therapeutic responses to interferon-alpha in HTLV-1-associated myelopathy/tropical spastic paraparesis (HAM/TSP): Differing clinical responses to leukocyte-derived human interferon-alpha versus recombinant interferon-alpha. Int J Case Rep Images 2020;11:101118Z01CZ2020.ABSTRACT
Introduction: HTLV-1-associated myelopathy/tropical spastic paraparesis (HAM/TSP) is a progressive spinal cord syndrome seen in patients infected by human T-lymphotropic virus type 1 (HTLV-1). Initial infection by the virus is usually asymptomatic. As the disease progresses, a series of neurological symptoms can appear gradually in years or, in some cases, rapidly within months. Interferon-alpha (IFN-α) is still considered one possible disease modifying treatment option for this disease due to the immunological pathogenesis of HAM/TSP.
Case Series: We followed 3 HTLV-1 infected HAM/TSP patients who presented with rapidly progressive neurological impairment. Each patient underwent serial neurological examinations that were recorded according to the Expanded Disability Status Scale (EDSS). Then treatment with preparations of human leukocyte-derived natural interferon-α (IFN-αn3) was started and continued for varying intervals of time according to the availability of IFN-αn3. With this treatment, the HAM/TSP symptoms were controlled and even alleviated, with decreased EDSS scores. However, interferon-α was switched to the recombinant product IFN-α2b when the supply of IFN-αn3 was unavailable. Surprisingly, all patients experienced a significant worsening of their neurological symptoms and EDSS scores after they switched to IFN-α2b.
Conclusion: Since IFN-α is still used to treat HAM/TSP patients globally, this differential response to IFN-αn3 and IFN-α2b treatment is important to be monitored and warrants further reporting.
Keywords: Human T-lymphotropic virus type 1, Interferon-alpha, Myelopathy, Tropical spastic paraparesis
SUPPORTING INFORMATION
Acknowledgments
This paper is dedicated to the late Dr. William A Sheremata, a pioneer in multiple sclerosis clinical research and care, who was an inspiration to his colleagues and patients.
Author ContributionsChao Zheng - Analysis of data, Interpretation of data, Drafting the article, Revising it critically for important intellectual content, Final approval of the version to be published
William A Sheremata† - Substantial contributions to conception and design, Acquisition of data, Analysis of data, Interpretation of data, Drafting the article, Revising it critically for important intellectual content, Final approval of the version to be published
Leticia Tornes - Substantial contributions to conception and design, Acquisition of data, Analysis of data, Interpretation of data, Drafting the article, Revising it critically for important intellectual content, Final approval of the version to be published
Yesica Campos - Acquisition of data, Analysis of data, Interpretation of data, Drafting the article, Revising it critically for important intellectual content, Final approval of the version to be published
Micheline McCarthy - Acquisition of data, Analysis of data, Interpretation of data, Drafting the article, Revising it critically for important intellectual content, Final approval of the version to be published
Melissa R Ortega - Substantial contributions to conception and design, Acquisition of data, Analysis of data, Interpretation of data, Drafting the article, Revising it critically for important intellectual content, Final approval of the version to be published
Guarantor of SubmissionThe corresponding author is the guarantor of submission.
Source of SupportNone
Consent StatementThis case report was prepared according to the “Uniform Requirements for Manuscripts Submitted to Biomedical Journals given by the International Committee of Medical Journal Editors (ICMJE).” Identifying information for each patient has been excluded. Prior to submission, the report was reviewed and approved by the Office of HIPAA Privacy and Data Security, University of Miami Miller School of Medicine.
Data AvailabilityAll relevant data are within the paper and its Supporting Information files.
Conflict of InterestAuthors declare no conflict of interest
Copyright© 2020 Chao Zheng et al. This article is distributed under the terms of Creative Commons Attribution License which permits unrestricted use, distribution and reproduction in any medium provided the original author(s) and original publisher are properly credited. Please see the copyright policy on the journal website for more information.
