Lethal Pediatric Cerebral Vasculitis Triggered by Severe Acute Respiratory Syndrome Coronavirus 2.


Journal

Pediatric neurology
ISSN: 1873-5150
Titre abrégé: Pediatr Neurol
Pays: United States
ID NLM: 8508183

Informations de publication

Date de publication:
02 2022
Historique:
received: 25 10 2021
revised: 02 11 2021
accepted: 06 11 2021
pubmed: 6 12 2021
medline: 5 2 2022
entrez: 5 12 2021
Statut: ppublish

Résumé

We report the clinical, radiological, laboratory, and neuropathological findings in support of the first diagnosis of lethal, small-vessel cerebral vasculitis triggered by severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2) in a pediatric patient. A previously healthy, eight-year-old Hispanic girl presented with subacute left-sided weakness two weeks after a mild febrile illness. SARS-CoV-2 nasopharyngeal swab was positive. Magnetic resonance imaging revealed an enhancing right frontal lobe lesion with significant vasogenic edema. Two brain biopsies of the lesion showed perivascular and intraluminal lymphohistiocytic inflammatory infiltrate consistent with vasculitis. Despite extensive treatment with immunomodulatory therapies targeting primary angiitis of the central nervous system, she experienced neurological decline and died 93 days after presentation. SARS-CoV-2 testing revealed positive serum IgG and positive cerebrospinal fluid IgM. Comprehensive infectious, rheumatologic, hematologic/oncologic, and genetic evaluation did not identify an alternative etiology. Postmortem brain autopsy remained consistent with vasculitis. This is the first pediatric presentation to suggest that SARS-CoV-2 can lead to a fatal, postinfectious, inflammatory small-vessel cerebral vasculitis. Our patient uniquely included supportive cerebrospinal fluid and postmortem tissue analysis. While most children recover from the neurological complications of SARS-CoV-2, we emphasize the potential mortality in a child with no risk factors for severe disease.

Sections du résumé

BACKGROUND
We report the clinical, radiological, laboratory, and neuropathological findings in support of the first diagnosis of lethal, small-vessel cerebral vasculitis triggered by severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2) in a pediatric patient.
PATIENT DESCRIPTION
A previously healthy, eight-year-old Hispanic girl presented with subacute left-sided weakness two weeks after a mild febrile illness. SARS-CoV-2 nasopharyngeal swab was positive. Magnetic resonance imaging revealed an enhancing right frontal lobe lesion with significant vasogenic edema. Two brain biopsies of the lesion showed perivascular and intraluminal lymphohistiocytic inflammatory infiltrate consistent with vasculitis. Despite extensive treatment with immunomodulatory therapies targeting primary angiitis of the central nervous system, she experienced neurological decline and died 93 days after presentation. SARS-CoV-2 testing revealed positive serum IgG and positive cerebrospinal fluid IgM. Comprehensive infectious, rheumatologic, hematologic/oncologic, and genetic evaluation did not identify an alternative etiology. Postmortem brain autopsy remained consistent with vasculitis.
CONCLUSION
This is the first pediatric presentation to suggest that SARS-CoV-2 can lead to a fatal, postinfectious, inflammatory small-vessel cerebral vasculitis. Our patient uniquely included supportive cerebrospinal fluid and postmortem tissue analysis. While most children recover from the neurological complications of SARS-CoV-2, we emphasize the potential mortality in a child with no risk factors for severe disease.

Identifiants

pubmed: 34864371
pii: S0887-8994(21)00248-4
doi: 10.1016/j.pediatrneurol.2021.11.003
pmc: PMC8585961
pii:
doi:

Types de publication

Case Reports

Langues

eng

Sous-ensembles de citation

IM

Pagination

1-5

Informations de copyright

Copyright © 2021 Elsevier Inc. All rights reserved.

Auteurs

Kelsey E Poisson (KE)

Department of Pediatrics, University of Cincinnati College of Medicine, Cincinnati, Ohio; Division of Child Neurology, Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio.

Alexander Zygmunt (A)

Department of Pediatrics, University of Cincinnati College of Medicine, Cincinnati, Ohio; Division of Child Neurology, Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio.

Daniel Leino (D)

Department of Pediatrics, University of Cincinnati College of Medicine, Cincinnati, Ohio; Division of Pathology, Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio.

Christine E Fuller (CE)

Department of Pediatrics, University of Cincinnati College of Medicine, Cincinnati, Ohio; Division of Pathology, Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio; Department of Pathology, State University of New York, Upstate Medical University, Syracuse, New York.

Blaise V Jones (BV)

Department of Pediatrics, University of Cincinnati College of Medicine, Cincinnati, Ohio; Division of Radiology, Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio.

David Haslam (D)

Department of Pediatrics, University of Cincinnati College of Medicine, Cincinnati, Ohio; Division of Infectious Disease, Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio.

Mary Allen Staat (MA)

Department of Pediatrics, University of Cincinnati College of Medicine, Cincinnati, Ohio; Division of Infectious Disease, Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio.

Gwendolyn Clay (G)

Department of Pediatrics, University of Cincinnati College of Medicine, Cincinnati, Ohio; Division of Rheumatology, Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio.

Tracy V Ting (TV)

Department of Pediatrics, University of Cincinnati College of Medicine, Cincinnati, Ohio; Division of Rheumatology, Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio.

Kristen Wesselkamper (K)

Department of Pediatrics, University of Cincinnati College of Medicine, Cincinnati, Ohio; Division of Child Neurology, Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio.

Barbara Hallinan (B)

Department of Pediatrics, University of Cincinnati College of Medicine, Cincinnati, Ohio; Division of Child Neurology, Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio.

Shannon Standridge (S)

Department of Pediatrics, University of Cincinnati College of Medicine, Cincinnati, Ohio; Division of Child Neurology, Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio.

Melissa E Day (ME)

Department of Pediatrics, University of Cincinnati College of Medicine, Cincinnati, Ohio; Department of Pediatrics, Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio.

Monica McNeal (M)

Department of Pediatrics, University of Cincinnati College of Medicine, Cincinnati, Ohio; Division of Infectious Disease, Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio.

Charles B Stevenson (CB)

Department of Pediatrics, University of Cincinnati College of Medicine, Cincinnati, Ohio; Division of Neurosurgery, Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio.

Marissa Vawter-Lee (M)

Department of Pediatrics, University of Cincinnati College of Medicine, Cincinnati, Ohio; Division of Child Neurology, Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio. Electronic address: Marissa.Vawter@cchmc.org.

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Classifications MeSH