Drug-indiced aseptic meningitis: development of subacute sclerosing panencephalitis following repeated intraventricular infusion therapy with interferon alpha/beta |
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Authors: | Imataka George Nakagawa Eiji Yamanouchi Hideo Arisaka Osamu |
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Institution: | (1) Department of Pediatrics, Dokkyo Medical University School of Medicine, Kitakobayashi 880, Mibu Tochigi, 321-0293, Japan;(2) Department of Pediatric Neurology, National Center of Neurology and Psychiatry, Musashi Hospital, Kodaira, Japan;(3) Department of Pediatrics, Saitama Medical University School of Medicine, Saitama, Japan |
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Abstract: | Interferon (IFN)-α was reported to be effective in longterm intrathecal treatment of subacute sclerosing panencephalitis (SSPE).
However, the side effects related with longterm use of IFN-α/β are unclear. We evaluated the therapeutic effects of IFN-α/β
in a 13-years-old patient with SSPE. The cerebrospinal fluid (CSF) measles antibody titer was 64 × NT/128×HI, IgG-index was
4.5, and the SSPE diagnosis was based on electroencephalography (Jabbour-stage II on admission). With Inosiplex (INP) given
orally, IFN-α (3 × 106 units) was infused intraventricularly twice-a-week for 1-year. Resultantly, CSF cell count was elevated (2502/3), total protein
and glucose levels were normal; however, DIAM occurred repeatedly. Consequently, reduced IFN-α (5 × 105 units) with hydrocorton was administered at 2-months interval for 19 months, during which, DIAM occurred four times. Therefore,
IFN-β (3 × 106 units; twice-a-week) therapy was started and continued for 3 years. Although the symptoms were improved considerably, DIAM
recurred after 15-months therapy and CSF cell counts were also elevated (2121/3). Since SSPE progressed to Jabbour-stage IV,
indicated by irreversible consciousness disorder, IFN therapy was discontinued and INP monotherapy was followed for another
3 years. We, therefore, concluded that the longterm intraventricular IFN-α/β infusion therapy of SSPE involved the potential
risk of DIAM with serious irreversible neurological sequelae and should be monitored carefully. |
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