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

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. Mog...

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Published in:Clinical infectious diseases 2023-09, Vol.77 (6), p.851-856
Main Authors: Meyerowitz, Eric A, Mukerji, Shibani S, Kyle Harrold, G, Erdil, Rachel M, Chen, Steven T, Rudmann, Emily A, Tsibris, Athe, Venna, Nagagopal, Robbins, Gregory K
Format: Article
Language:English
Subjects:
Antibodies, Monoclonal, Humanized - adverse effects
Exanthema
Female
Human T-lymphotropic virus 1
Humans
Major
Paraparesis, Tropical Spastic - drug therapy
Viral Load
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Summary: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 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. Four women received mogamulizumab for human T-lymphotropic virus type 1 (HTLV-1) associated myelopathy/tropical spastic paraparesis (HAM/TSP) at a single US center. Two individuals with milder baseline disease showed clinical improvement. All 4 developed rashes, which were treatment limiting. Graphical abstract 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
ISSN:1058-4838
1537-6591
DOI:10.1093/cid/ciad281