Cargando…

GABA(A) receptor encephalitis associated with human parvovirus B19 virus infection: Case report

RATIONALE: Human parvovirus B19 (B19) infection can produce a spectrum of clinical syndromes, including neurological manifestations, most notably encephalitis. Although symptoms suggestive of autoimmune disease in patients with B19 infection have been previously described, a clear association of aut...

Descripción completa

Detalles Bibliográficos
Autores principales: do Valle, Daniel Almeida, Santos, Mara Lúcia S. Ferreira, Spinosa, Mônica J., Telles, Bruno A., Prando, Carolina, Cordeiro, Mara L.
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Lippincott Williams & Wilkins 2021
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8202551/
https://www.ncbi.nlm.nih.gov/pubmed/34115048
http://dx.doi.org/10.1097/MD.0000000000026324
_version_ 1783708005750013952
author do Valle, Daniel Almeida
Santos, Mara Lúcia S. Ferreira
Spinosa, Mônica J.
Telles, Bruno A.
Prando, Carolina
Cordeiro, Mara L.
author_facet do Valle, Daniel Almeida
Santos, Mara Lúcia S. Ferreira
Spinosa, Mônica J.
Telles, Bruno A.
Prando, Carolina
Cordeiro, Mara L.
author_sort do Valle, Daniel Almeida
collection PubMed
description RATIONALE: Human parvovirus B19 (B19) infection can produce a spectrum of clinical syndromes, including neurological manifestations, most notably encephalitis. Although symptoms suggestive of autoimmune disease in patients with B19 infection have been previously described, a clear association of autoimmune encephalitis with B19 infection has yet to be established. PATIENT CONCERNS: We describe the case of a 6-year-old boy who was hospitalized due to status epilepticus, which evolved to super-refractory status epilepticus that was only mildly responsive to anticonvulsant drugs. DIAGNOSIS: A cerebrospinal fluid study identified slight pleocytosis and B19 positivity. A subsequent autoimmunity cerebrospinal fluid study revealed the presence of anti-γ-aminobutyric acid type A (GABA(A)) receptor antibodies. INTERVENTIONS: After pulse therapy with methylprednisolone and continuous therapy with prednisolone with cyclosporine, the patient experiencing seizure persistence with disordered motor function manifestations and only minor improvement in consciousness, and so, plasmapheresis was performed. With continued immunosuppressive treatments with cyclosporine and prednisolone, the patient's clinical picture showed progressive improvement, with good control of seizures. Although the patient tolerated withdrawal of the anticonvulsant drugs well, he developed seizures when corticosteroid therapy withdrawal was attempted, so was started on azathioprine. OUTCOMES: After immunosuppressive therapy, the patient evolved with complete remission of symptoms, normal neurological examination and age-appropriate neuropsychomotor development. LESSONS: The present case characteristics, together with previous findings, support the hypothesis that autoimmunity may be triggered by extensive antigen release due to degeneration of infected neurons. This case highlights the importance of early clinical suspicion and treatment.
format Online
Article
Text
id pubmed-8202551
institution National Center for Biotechnology Information
language English
publishDate 2021
publisher Lippincott Williams & Wilkins
record_format MEDLINE/PubMed
spelling pubmed-82025512021-06-15 GABA(A) receptor encephalitis associated with human parvovirus B19 virus infection: Case report do Valle, Daniel Almeida Santos, Mara Lúcia S. Ferreira Spinosa, Mônica J. Telles, Bruno A. Prando, Carolina Cordeiro, Mara L. Medicine (Baltimore) 6200 RATIONALE: Human parvovirus B19 (B19) infection can produce a spectrum of clinical syndromes, including neurological manifestations, most notably encephalitis. Although symptoms suggestive of autoimmune disease in patients with B19 infection have been previously described, a clear association of autoimmune encephalitis with B19 infection has yet to be established. PATIENT CONCERNS: We describe the case of a 6-year-old boy who was hospitalized due to status epilepticus, which evolved to super-refractory status epilepticus that was only mildly responsive to anticonvulsant drugs. DIAGNOSIS: A cerebrospinal fluid study identified slight pleocytosis and B19 positivity. A subsequent autoimmunity cerebrospinal fluid study revealed the presence of anti-γ-aminobutyric acid type A (GABA(A)) receptor antibodies. INTERVENTIONS: After pulse therapy with methylprednisolone and continuous therapy with prednisolone with cyclosporine, the patient experiencing seizure persistence with disordered motor function manifestations and only minor improvement in consciousness, and so, plasmapheresis was performed. With continued immunosuppressive treatments with cyclosporine and prednisolone, the patient's clinical picture showed progressive improvement, with good control of seizures. Although the patient tolerated withdrawal of the anticonvulsant drugs well, he developed seizures when corticosteroid therapy withdrawal was attempted, so was started on azathioprine. OUTCOMES: After immunosuppressive therapy, the patient evolved with complete remission of symptoms, normal neurological examination and age-appropriate neuropsychomotor development. LESSONS: The present case characteristics, together with previous findings, support the hypothesis that autoimmunity may be triggered by extensive antigen release due to degeneration of infected neurons. This case highlights the importance of early clinical suspicion and treatment. Lippincott Williams & Wilkins 2021-06-11 /pmc/articles/PMC8202551/ /pubmed/34115048 http://dx.doi.org/10.1097/MD.0000000000026324 Text en Copyright © 2021 the Author(s). Published by Wolters Kluwer Health, Inc. https://creativecommons.org/licenses/by/4.0/This is an open access article distributed under the Creative Commons Attribution License 4.0 (CCBY), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited. http://creativecommons.org/licenses/by/4.0 (https://creativecommons.org/licenses/by/4.0/)
spellingShingle 6200
do Valle, Daniel Almeida
Santos, Mara Lúcia S. Ferreira
Spinosa, Mônica J.
Telles, Bruno A.
Prando, Carolina
Cordeiro, Mara L.
GABA(A) receptor encephalitis associated with human parvovirus B19 virus infection: Case report
title GABA(A) receptor encephalitis associated with human parvovirus B19 virus infection: Case report
title_full GABA(A) receptor encephalitis associated with human parvovirus B19 virus infection: Case report
title_fullStr GABA(A) receptor encephalitis associated with human parvovirus B19 virus infection: Case report
title_full_unstemmed GABA(A) receptor encephalitis associated with human parvovirus B19 virus infection: Case report
title_short GABA(A) receptor encephalitis associated with human parvovirus B19 virus infection: Case report
title_sort gaba(a) receptor encephalitis associated with human parvovirus b19 virus infection: case report
topic 6200
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8202551/
https://www.ncbi.nlm.nih.gov/pubmed/34115048
http://dx.doi.org/10.1097/MD.0000000000026324
work_keys_str_mv AT dovalledanielalmeida gabaareceptorencephalitisassociatedwithhumanparvovirusb19virusinfectioncasereport
AT santosmaraluciasferreira gabaareceptorencephalitisassociatedwithhumanparvovirusb19virusinfectioncasereport
AT spinosamonicaj gabaareceptorencephalitisassociatedwithhumanparvovirusb19virusinfectioncasereport
AT tellesbrunoa gabaareceptorencephalitisassociatedwithhumanparvovirusb19virusinfectioncasereport
AT prandocarolina gabaareceptorencephalitisassociatedwithhumanparvovirusb19virusinfectioncasereport
AT cordeiromaral gabaareceptorencephalitisassociatedwithhumanparvovirusb19virusinfectioncasereport