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Published on: March 19, 2018
Treatment of subacute sclerosing panencephalitis with interferon-alpha, ribavirin, and inosiplex
Tom Solomon1, C Anthony Hart, Sobhan Vinjamuri
1Department of Neurological Science, University of Liverpool, United Kingdom. tsolomon@liv.ac.uk
Abstract:
Subacute sclerosing panencephalitis is an almost universally fatal late complication of measles infection for which there is no established treatment. We report a patient with subacute sclerosing panencephalitis who was bed-bound and ataxic and had a left hemiparesis and frequent myoclonus. He was started on a new regimen consisting of intraventricular interferon-alpha (starting at 100,000 U/m2/day, building up to 1 million U/m2/day), ribavirin (60 mg/kg/day intravenously), and inosiplex (3 g/day) and improved markedly. At 10 weeks, the intraventricular reservoir was removed because of bacterial infection, and he was discharged home on oral ribavirin (1200 mg/kg/day) and inosiplex. He continued to improve as judged by neurologic examination, functional independence measurement, neuropsychometry and single photon emission computed tomography (SPECT) imaging. However, after 10 months, he deteriorated suddenly and died before further intraventricular treatment could be instituted. Further trials are needed to evaluate long-term combination therapy in subacute sclerosing panencephalitis.
Insights
Subacute sclerosing panencephalitis (SSPE), a fatal measles complication, showed marked improvement with a novel combination therapy including interferon-alpha, ribavirin, and inosiplex. Though temporary, this treatment highlights potential therapeutic avenues for SSPE.
Area of Science:
- Neurology
- Infectious Diseases
- Virology
Background:
- Subacute sclerosing panencephalitis (SSPE) is a rare, progressive, and fatal neurological complication of measles virus infection.
- Currently, no established effective treatment exists for SSPE, leading to a universally fatal outcome.
Observation:
- A patient with severe SSPE, presenting with significant neurological deficits including ataxia, hemiparesis, and myoclonus, was treated with a novel regimen.
- The treatment involved intraventricular interferon-alpha, intravenous ribavirin, and oral inosiplex.
Findings:
- The patient demonstrated marked neurological and functional improvement during the initial 10 weeks of combination therapy.
- Despite initial success and continued improvement on oral therapy, the patient experienced sudden deterioration and expired after 10 months.
Implications:
- This case suggests that combination therapy, particularly with intraventricular interferon-alpha, may offer temporary benefits in managing severe SSPE.
- Further research and clinical trials are warranted to explore the long-term efficacy and optimal strategies for combination therapy in SSPE.
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