title: Treatment of subacute sclerosing panencephalitis with interferon-alpha, ribavirin, and inosiplex.
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.
authors.affiliation: Department of Neurological Science, University of Liverpool, United Kingdom. tsolomon@liv.ac.uk
authors.forename: Tom
authors.initials: T
authors.lastname: Solomon
authors.forename: C Anthony
authors.initials: CA
authors.lastname: Hart
authors.forename: Sobhan
authors.initials: S
authors.lastname: Vinjamuri
authors.forename: Nicholas J
authors.initials: NJ
authors.lastname: Beeching
authors.forename: Conor
authors.initials: C
authors.lastname: Malucci
authors.forename: Peter
authors.initials: P
authors.lastname: Humphrey
mesh_terms: D000284:Administration, Oral; D000293:Adolescent; D000998:Antiviral Agents / Q000008:administration & dosage / Q000627:therapeutic use*; D004359:Drug Therapy, Combination; D017809:Fatal Outcome; D006801:Humans; D007276:Injections, Intraventricular; D007542:Inosine Pranobex / Q000008:administration & dosage / Q000627:therapeutic use*; D016898:Interferon-alpha / Q000008:administration & dosage / Q000627:therapeutic use*; D008297:Male; D012254:Ribavirin / Q000008:administration & dosage / Q000627:therapeutic use*; D013344:Subacute Sclerosing Panencephalitis / Q000175:diagnosis / Q000188:drug therapy*
chemical_list: D000998:Antiviral Agents; D016898:Interferon-alpha; D012254:Ribavirin; D007542:Inosine Pranobex