Evidence map›Paper›PMID 37550789›Full record

ArticleAllergy, asthma, and clinical immunology : official journal of the Canadian Society of Allergy and Clinical Immunology2023

Myelin oligodendrocyte glycoprotein antibody-associated disease as a novel presentation of central nervous system autoimmunity in a pediatric patient with Wiskott-Aldrich syndrome.

Vivien X Xie, Wilson File, Christina Wiedl, Brant R Ward, Blachy Dávila Saldaña, Michael D Keller, Alexandra B Kornbluh

Open access · goldAbstract read
PubMed Full text at PMC Publisher
In one paragraph

Article in Allergy, asthma, and clinical immunology : official journal of the Canadian Society of Allergy and Clinical Immunology, 2023. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 1 paper, 1 of them a synthesis that pooled it.

0numbers the graph read from it
0cells of the map it votes in
1citing papers in PubMed, 1 pooled it
0.4field-weighted citation impact, top 36% of its field
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

1 citing paper in PubMed, 1 synthesis or guideline pooled it, 2 citations in OpenAlex.

  1. Pooled it
4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

7 authors at 3 institutions in 1 country.

Vivien X XieDepartment of Neurology, District of Columbia, Children's National Hospital, 111 Michigan Ave NW, Washington, 20010, USA. vxie@childrensnational.org.ORCID http://orcid.org/0000-0003-0426-4268
Wilson FileDivision of Hematology and Oncology, Eastern Virginia Medical School and Children's Hospital of The King's Daughters, Norfolk, VG, USA.
Christina WiedlDivision of Hematology and Oncology, District of Columbia, Children's National Hospital, Washington, USA.
Brant R WardDivision of Allergy and Immunology, Children's Hospital of Richmond, Virginia Commonwealth University, Richmond, VA, USA.
Blachy Dávila SaldañaDivision of Blood and Marrow Transplantation, Children's National Hospital, Washington, DC, USA.
Michael D KellerCenter for Cancer and Immunology Research, Division of Allergy and Immunology, Children's National Hospital, Washington, DC, USA.
Alexandra B KornbluhDepartment of Neurology, District of Columbia, Children's National Hospital, 111 Michigan Ave NW, Washington, 20010, USA.
Children's National · USEastern Virginia Medical School · USVirginia Commonwealth University · US

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

backgroundWiskott-Aldrich syndrome (WAS) is an X-linked primary immunodeficiency caused by mutations in the WAS gene that leads to increased susceptibility to infections, thrombocytopenia, eczema, malignancies, and autoimmunity. Central nervous system (CNS) autoimmune manifestations are uncommon. CASE PRESENTATION: We describe the case of a five-year-old boy with refractory thrombocytopenia and iron deficiency anemia who developed relapsing bilateral optic neuritis. Myelin oligodendrocyte glycoprotein antibody (MOG-IgG) via serum fluorescence-activated cell sorting assay was positive (titer 1:100), confirming a diagnosis of myelin oligodendrocyte glycoprotein antibody-associated disease (MOGAD). At age six, molecular panel testing for genes associated with primary immunodeficiency identified a missense WAS gene variant. He was subsequently found to have decreased WAS protein expression, consistent with a diagnosis of WAS.

conclusionsThis case expands the reported spectrum of CNS autoimmunity associated with WAS and may help to inform long-term therapeutic options.

Indexed as

DemyelinationMyelin oligodendrocyte glycoprotein antibody associated diseaseOptic neuritisWiskott-Aldrich syndrome

Identifiers

PMID37550789
PMCPMC10408201
OpenAlexW4385638872

What OpenQuestion holds

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LicenceCC BY
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Registered trials

None linked

Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.