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. 2023 Sep 18;77(6):851-856.
doi: 10.1093/cid/ciad281.

Mogamulizumab for Treatment of Human T-lymphotropic Virus Type 1-Associated Myelopathy/Tropical Spastic Paraparesis: A Single-Center US-based Series

Eric A Meyerowitz  1   2 , Shibani S Mukerji  3   4 , G Kyle Harrold  3   4 , Rachel M Erdil  5 , Steven T Chen  4   6 , Emily A Rudmann  3 , Athe Tsibris  4   7   8 , Nagagopal Venna  3   4 , Gregory K Robbins  4   8
Affiliations

Mogamulizumab for Treatment of Human T-lymphotropic Virus Type 1-Associated Myelopathy/Tropical Spastic Paraparesis: A Single-Center US-based Series

Eric A Meyerowitz et al. Clin Infect Dis. .

Abstract

Background: Human T-lymphotropic virus type 1 (HTLV-1)-associated myelopathy/tropical spastic paraparesis (HAM/TSP) is a chronic neurological condition characterized by progressive myelopathic symptoms including spasticity, pain, weakness, and urinary symptoms, without proven treatments. Mogamulizumab (MOG) is a monoclonal antibody that binds CCR4 and leads to the clearance of HTLV-1-infected CCR4+ cells. A phase 1-2a study in Japan evaluated MOG for the treatment of HAM/TSP and reported decreases in HTLV-1 proviral load and neuroinflammatory markers, with clinical improvement in some participants.

Methods: We administered MOG 0.1 mg/kg every 8 weeks to individuals with HAM/TSP as a compassionate and palliative treatment. Patients who received MOG had (1) a positive peripheral HTLV-1 antibody, (2) progressive myelopathic symptoms, and (3) a diagnosis of HAM/TSP.

Results: Four female patients, ages 45-68, received MOG (range, 2-6 infusions) between 1 November 2019 and 30 November 2022. Two patients with <3 years of symptoms had milder disease, with Osame scores <4. The other 2, with >7 years of symptoms, had Osame scores >5. One patient, with 6 total treatments, received dose-reduced MOG after she developed a rash at the initial dose. The 2 patients with milder baseline disease reported symptomatic improvement and saw reductions in Osame and/or modified Ashworth scale scores during follow-up. The other 2 patients showed no improvement. All 4 developed rashes after receiving MOG-a treatment-limiting event in some cases.

Conclusions: Clinical trials are needed including diverse patient populations to assess the potential role of MOG for HAM/TSP. Our findings may help inform the development of these trials.

Keywords: HAM/TSP; HTLV-1; mogamulizumab; myelopathy; rash.

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Conflict of interest statement

Potential conflicts of interest. A. T. reports unrelated consulting fees from Gilead (Research Scholars Program grant reviewer) and Dynamed (HIV Section Editor, EBSCO) and serves on a DSMB for Dalcor Pharmaceuticals. S. M. reports grants unrelated to this work (grant numbers K23MH115812 and R01MH131194), as well as Gilead stock in a retirement fund. G. K. R. reports clinical trial support paid to the institution from Leonard Meron Inc unrelated to this manuscript. R. E. reports an honorarium for her contribution to Pocket Medicine (Pocket Medicine 8th Edition and Pocket Medicine High-Yield Board Review) from Wolters Kluwer, unrelated to this work. All other authors report no potential conflicts. All authors have submitted the ICMJE Form for Disclosure of Potential Conflicts of Interest. Conflicts that the editors consider relevant to the content of the manuscript have been disclosed.

Figures

None
This graphical abstract is also available at Tidbit: https://tidbitapp.io/tidbits/challenges-in-the-long-term-management-of-patients-with-coccidioidal-meningitis-a-retrospective-analysis-of-treatment-and-outcomes
Figure 1.
Figure 1.
Cutaneous adverse events observed in patients treated with MOG for HAM/TSP. A, Left arm of patient 1 shows erythematous and hyperpigmented patches and plaques with scale. This photo was taken 6 wks after first infusion B, Area around left elbow of patient 2 showing a morbilliform rash. This photo was taken 4 wks after second infusion, prior to development of DRESS-like rash. C, The scalp shows erythematous patches and papules with scale of patient 4. This photo was taken 6 wks after third infusion. Abbreviations: HAM/TSP, HTLV-1-associated myelopathy/tropical spastic paraparesis; MOG, mogamulizumab.
See this image and copyright information in PMC

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