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Chronic enteroviral meningoencephalitis in agammaglobulinemia: case report and literature review.

Abstract：

:Chronic enteroviral meningoencephalitis is a well-recognized complication in patients with X-linked agammaglobulinemia (XLA). The majority of published cases refers to its occurrence in patients on no replacement therapy or on only intramuscular immunoglobulin. The advent of intravenous immunoglobulin (IVIg) in the early 1980s and its widespread use in XLA was thought to have virtually eradicated enteroviral meningoencephalitis in these patients. We describe the development of echovirus meningoencephalitis in an 11-year-old boy on regular IVIg replacement whose serum IgG levels were maintained at between 6 and 8 g/L (NR 6-13 g/L). Treatment with daily high-dose IVIg was commenced, with significant clinical improvement being noted within a few weeks in association with a reduction in blood-brain barrier permeability. The persistence of live virus, however, necessitated the use of intraventricular immunoglobulin. The virus proved resistant to two courses of specific intraventricular immunoglobulin and a 6-week course of oral ribavirin and eventually proved fatal 5 months after presentation. In view of the therapeutic uncertainties we have reviewed the use of immunoglobulin in the treatment of enteroviral meningoencephalitis over the past 6 years.

journal_name

J Clin Immunol

journal_title

Journal of clinical immunology

authors

Misbah SA,Spickett GP,Ryba PC,Hockaday JM,Kroll JS,Sherwood C,Kurtz JB,Moxon ER,Chapel HM

doi

10.1007/BF00918150

subject

Has Abstract

pub_date

1992-07-01 00:00:00

pages

266-70

issue

4

eissn

0271-9142

issn

1573-2592

journal_volume

12

pub_type

杂志文章,评审

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