Trial of intraventricular ribavirin therapy for subacute sclerosing panencephalitis in Japan (original) (raw)
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High-Dose Intravenous Ribavirin Therapy for Subacute Sclerosing Panencephalitis
Antimicrobial Agents and Chemotherapy, 2001
Two patients with subacute sclerosing panencephalitis (SSPE) were treated safely and effectively with high doses of intravenous ribavirin combined with intraventricular alpha interferon. The ribavirin concentrations maintained in the serum and cerebrospinal fluid were higher than those which inhibit SSPE virus replication in vitro and in vivo.
Antimicrobial Agents and Chemotherapy, 2004
Ribavirin is a broad-spectrum antiviral drug with inhibitory activity against many RNA viruses, including measles virus. Five patients with subacute sclerosing panencephalitis (SSPE) were treated with ribavirin by intraventricular administration. Although there were transient side effects attributed to ribavirin, such as drowsiness, headache, lip and gingival swelling, and conjunctival hyperemia, intraventricular ribavirin therapy was generally safe and well tolerated. The cerebrospinal fluid (CSF) ribavirin concentration decreased, as described by a monoexponential function, after a single intraventricular dose. There was considerable interindividual variability, however, in the peak level and half-life.
Combined treatment with interferon-alpha and ribavirin for subacute sclerosing panencephalitis
Pediatric Neurology, 2001
Two patients with subacute sclerosing panencephalitis (SSPE) are described. They were diagnosed on admission to the hospital with SSPE, as judged on cerebrospinal fluid examination involving reverse transcription followed by polymerase chain reaction, at the second stage of Jabbour's classification. They first were treated with intraventricular interferon-alpha monotherapy; however, the combination of interferonalpha and IV ribavirin was started at 8 and 5 months after beginning the interferon-alpha monotherapy, respectively. Although slow progressive brain atrophy was observed in Patient 1 on brain magnetic resonance imaging before the ribavirin therapy, no further progression was noted 11 months after starting combination therapy with ribavirin. The event-related potential study results and audiography of the right ear improved in Patient 1 after the combination therapy was initiated. In Patient 2 the hypertonicity, neurobladder incontinence, and dysphagia improved 3 months after starting the combination treatment. Although this group of patients is small, these results suggest treatment with intrathecal high-dose interferon-alpha and IV ribavirin is effective in the treatment of SSPE. Early administration of intrathecal high-dose interferon-alpha and IV ribavirin should be considered as a possible therapy for SSPE patients, especially interferon-nonresponding ones. © 2001 by Elsevier Science Inc. All rights reserved.
Brain and Development, 1993
Eighteen patients, 16 boys and 2 girls, aged 5-14 years, with subacute sclerosing panencephalitis (SSPE) were treated with oral isoprinosine (100 mg/kg/day) and intraventricular alpha-interferon 2b (Intron A, Schering Corp.), starting at 500,000 U twice a week and later increasing to 3 million U biweekly. Minimal follow-up of living patients is 12 months; maximal 40 months. On the basis of the Neurological Disability Index (NDI) scores and staging, 8 have treatment-induced remissions (3 improved, 5 arrested), 4 are worse and 6 died. This 44% (8/18) rate of remission/improvement compares well with the 9% (1/11) remission in historical controls in the same institution (p = < 0.05) and 5% spontaneous remission in the literature. Combined oral isoprinosine-intraventricular alpha-interferon appears to be an effective treatment for SSPE.
Brain and Development, 2003
Subacute sclerosing panencephalitis (SSPE) is a degenerative disease of the central nervous system that leads to death within a few years. Recently, it has been reported that combination therapy with intraparenchymal interferon-a (INF-a) and intraventricular ribavirin is effective. An 11-year-old SSPE patient whose clinical symptoms progressed rapidly, was treated first with intraventricular INF-a and then with combined intraventricular INF-a and ribavirin therapy. To monitor viral load over the course of the therapy, measles virus RNA was quantified using a real-time polymerase chain reaction assay. Measles virus RNA decreased rapidly after the INF-a therapy was started, paralleling the decrease in the measles antibody titer in the cerebrospinal fluid and the improvement in the neurological disability. After intraventricular ribavirin was combined with INF-a therapy, no further improvement was observed. The neurological disability gradually progressed, although the amount of virus RNA remained low. q
A Multinational Survey on Actual Diagnostics and Treatment of Subacute Sclerosing Panencephalitis
Neuropediatrics, 2015
Subacute sclerosing panencephalitis (SSPE) is a chronic infection of the central nervous system caused by the measles virus (MV). Its prevalence remains high in resource poor countries and is likely to increase in the Northern Europe as vaccination rates decrease. Clinical knowledge of this devastating condition, however, is limited. We therefore conducted this multinational survey summarizing experience obtained from more than 500 patients treated by 24 physicians in seven countries. SSPE should be considered in all patients presenting with otherwise unexplained acquired neurological symptoms. In most patients, the diagnosis will be established by the combination of typical clinical symptoms (characteristic repetitive myoclonic jerks), a strong intrathecal synthesis of antibodies to MV and typical electroencephalogram findings (Radermecker complexes). Whereas the therapeutic use of different antiviral (amantadine, ribavirin) and immunomodulatory drugs (isoprinosine, interferons) an...
Subacute Sclerosing Panencephalitis (SSPE): Experience from a Tertiary-Care Pediatric Center
Journal of Neurosciences in Rural Practice, 2022
Introduction Subacute sclerosing panencephalitis (SSPE) is a devastating neurodegenerative disease occurring as a complication of measles infection that is still prevalent in low-resource countries. Clinical and electrographical variability in SSPE can lead to diagnostic delays. Methods Children diagnosed with SSPE in a tertiary care pediatric hospital in India in a period of 8 years were included in the study. The diagnosis was established on the basis of Dyken's criteria. The demographic data, clinical presentations, investigations, treatment approaches, and outcomes were reviewed and recorded. Results Thirty-four patients were included in the analysis. Average age at symptom onset was 7 years, 5 months. Majority of the children were not vaccinated for measles. Most patients (80%) presented with stage 2 of illness. Nearly 25% presented with atypical clinical features. Myoclonus was the most predominant feature seen after diagnosis. Electroencephalography (EEG) was the most use...
Options in the Treatment of Subacute Sclerosing Panencephalitis: Implications for Low Resource Areas
Current Treatment Options in Neurology, 2022
Purpose of the review Subacute sclerosing panencephalitis (SSPE) is a rare, slowly progressive, and frequently fatal neurodegenerative disorder caused by measles virus. The risk of SSPE remains significant globally, with fluctuating incidence noted in in tandem with measles vaccine uptake. This review aims to explore the current global status of SSPE, its treatment, and preventive measures. Recent findings An increase in measles cases have been reported in various parts of the world for different reasons related to the regional context of the outbreak. With reduction in measles vaccine doses since the onset of the COVID-19 pandemic, the future risk of SSPE can only accelerate. In recent years, subsequent cases of SSPE have been reported in the period following documented measles outbreaks in different settings. Concomitantly, there have been efforts to evaluate the efficacy of immunomodulatory, antiviral, and anti-seizure therapies that could ameliorate the devastating effects of th...
Combined Interferon-α and Inosiplex Treatment of Subacute Sclerosing Panencephalitis: A Case Report
The Kaohsiung Journal of Medical Sciences, 2003
Subacute sclerosing panencephalitis (SSPE) is rare in Taiwan. On admission to hospital, a 1 5 -y e a r -o l d b o y w a s d i a g n o s e d w i t h S S P E b a s e d o n t h e c l i n i c a l p i c t u r e , electroencephalogram, cerebrospinal fluid studies, and brain biopsy. The initial clinical picture was a decline in school performance and a change in personality, followed by progressive tic-like involuntary movements and mental impairment for 8 months, then a rapidly progressive course. After the patient was treated with oral inosiplex and intraventricular interferon-α (IFN-α), his condition stabilized and the neurologic disability index score improved slightly. There were no major side effects during treatment except for a transient initial elevation of body temperature that lasted for several days. Oral inosiplex and intraventricular IFN-α appear to be safe and effective. Early identification and aggressive treatment of SSPE is important.