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Article: Meningoencephalitis in primary antibody deficiency: Our experience from northwest India

TitleMeningoencephalitis in primary antibody deficiency: Our experience from northwest India
Authors
KeywordsCommon Variable Immunodeficiency
Enterovirus
Intravenous immunoglobulin
Meningoencephalitis
Primary antibody deficiency
X-linked agammaglobulinemia
Issue Date8-Aug-2022
PublisherElsevier
Citation
Journal of Neuroimmunology, 2022, v. 371 How to Cite?
Abstract

Background/Objectives: Patients with primary antibody deficiency (PAD) are predisposed to develop meningoencephalitis, often considered to be enteroviral. However, there is a paucity of literature on this subject, and there are no studies from developing countries. Methods: We analyzed our cohort of children with PAD who developed meningoencephalitis.Results: This complication was observed in 13/135 (10.4%) patients with PAD - 5 patients had X-linked agammaglobulinemia (XLA), 7 had common variable immunodeficiency (CVID) and 1 had suspected nuclear factor kappa B essential modulator (NEMO) defect. Mean age at onset of neurological illness was 9.3 years. Presenting features included seizures (n=8), neurodevelopmental delay (n=2), regression of milestones (n=2), and acute flaccid paralysis (n=1). Trough IgG levels were found to be low in 12/13 patients at the time of development of neurological symptoms. Herpes simplex virus (HSV), cytomegalovirus (CMV), and Streptococcus pneumoniae were isolated in 1 each. Eight (72.7%) patients had altered signal hyperintensities in gray matter and deep white matter on magnetic resonance imaging (MRI), while 4 patients showed global cerebral atrophy. All patients were treated with high-dose intravenous immunoglobulin (IVIg). Fluoxetine was given to 3 patients. Eight patients in the present series have died, 3 have recovered with varying degrees of neurological sequelae and 2 patients are showing gradual recovery.


Persistent Identifierhttp://hdl.handle.net/10722/331433
ISSN
2023 Impact Factor: 2.9
2023 SCImago Journal Rankings: 0.897
ISI Accession Number ID

 

DC FieldValueLanguage
dc.contributor.authorJindal, AK-
dc.contributor.authorChaudhary, H-
dc.contributor.authorTyagi, R-
dc.contributor.authorRawat, A-
dc.contributor.authorSuri, D-
dc.contributor.authorPatra, PK-
dc.contributor.authorArora, K-
dc.contributor.authorChawla, S-
dc.contributor.authorVyas, S-
dc.contributor.authorArora, M-
dc.contributor.authorAggarwal, R-
dc.contributor.authorBasu, S-
dc.contributor.authorBansal, R-
dc.contributor.authorSachdeva, MUS-
dc.contributor.authorGupta, A-
dc.contributor.authorPandiarajan, V-
dc.contributor.authorSankhyan, N-
dc.contributor.authorSuthar, R-
dc.contributor.authorSahu, JK-
dc.contributor.authorSingh, M-
dc.contributor.authorMani, R-
dc.contributor.authorSharma, R-
dc.contributor.authorSaka, R-
dc.contributor.authorImai, K-
dc.contributor.authorOhara, O-
dc.contributor.authorNonoyama, S-
dc.contributor.authorHammarstrom, L-
dc.contributor.authorChan, KW-
dc.contributor.authorLau, YL-
dc.contributor.authorSingh, S-
dc.date.accessioned2023-09-21T06:55:41Z-
dc.date.available2023-09-21T06:55:41Z-
dc.date.issued2022-08-08-
dc.identifier.citationJournal of Neuroimmunology, 2022, v. 371-
dc.identifier.issn0165-5728-
dc.identifier.urihttp://hdl.handle.net/10722/331433-
dc.description.abstract<p></p><p>Background/Objectives: Patients with primary antibody deficiency (PAD) are predisposed to develop meningoencephalitis, often considered to be enteroviral. However, there is a paucity of literature on this subject, and there are no studies from developing countries. Methods: We analyzed our cohort of children with PAD who developed meningoencephalitis.Results: This complication was observed in 13/135 (10.4%) patients with PAD - 5 patients had X-linked agammaglobulinemia (XLA), 7 had common variable immunodeficiency (CVID) and 1 had suspected nuclear factor kappa B essential modulator (NEMO) defect. Mean age at onset of neurological illness was 9.3 years. Presenting features included seizures (n=8), neurodevelopmental delay (n=2), regression of milestones (n=2), and acute flaccid paralysis (n=1). Trough IgG levels were found to be low in 12/13 patients at the time of development of neurological symptoms. Herpes simplex virus (HSV), cytomegalovirus (CMV), and Streptococcus pneumoniae were isolated in 1 each. Eight (72.7%) patients had altered signal hyperintensities in gray matter and deep white matter on magnetic resonance imaging (MRI), while 4 patients showed global cerebral atrophy. All patients were treated with high-dose intravenous immunoglobulin (IVIg). Fluoxetine was given to 3 patients. Eight patients in the present series have died, 3 have recovered with varying degrees of neurological sequelae and 2 patients are showing gradual recovery.<br></p>-
dc.languageeng-
dc.publisherElsevier-
dc.relation.ispartofJournal of Neuroimmunology-
dc.rightsThis work is licensed under a Creative Commons Attribution-NonCommercial-NoDerivatives 4.0 International License.-
dc.subjectCommon Variable Immunodeficiency-
dc.subjectEnterovirus-
dc.subjectIntravenous immunoglobulin-
dc.subjectMeningoencephalitis-
dc.subjectPrimary antibody deficiency-
dc.subjectX-linked agammaglobulinemia-
dc.titleMeningoencephalitis in primary antibody deficiency: Our experience from northwest India-
dc.typeArticle-
dc.identifier.doi10.1016/j.jneuroim.2022.577952-
dc.identifier.scopuseid_2-s2.0-85136463176-
dc.identifier.volume371-
dc.identifier.isiWOS:000855690900001-
dc.identifier.issnl0165-5728-

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