Evidence map›Paper›PMID 37582615›Full record

ArticleNeurology(R) neuroimmunology & neuroinflammation2023

Dramatic Response to Anti-IL-6 Receptor Therapy in Children With Life-Threatening Myelin Oligodendrocyte Glycoprotein-Associated Disease.

Loren A McLendon, Claudia Gambrah-Lyles, Angela Viaene, Nina A Fainberg, Elizabeth I Landzberg, Alexander M Tucker, Peter J Madsen, Jimmy Huh, Maya R Silver, John D Arena and 2 more

Open access · goldAbstract readCase Reports
In one paragraph

Article in Neurology(R) neuroimmunology & neuroinflammation, 2023. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 14 papers.

0numbers the graph read from it
0cells of the map it votes in
14citing papers in PubMed
7.7field-weighted citation impact, top 2% 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

14 citing papers in PubMed, 30 citations in OpenAlex.

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  12. Life-Threatening MOG Antibody-Associated Hemorrhagic ADEM With Elevated CSF IL-6.Neurology(R) neuroimmunology & neuroinflammation · 2024
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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

12 authors at 1 institution in 1 country.

Loren A McLendonFrom the Division of Neurology (L.A.M., C.G., M.R.S., B.B.), Department of Pediatrics, Children's Hospital of Philadelphia; Department of Neurology, Perelman School of Medicine, University of Pennsylvania; Department of Pathology (A.V.), Children's Hospital of Philadelphia, Perelman School of Medicine, University of Pennsylvania; Division of Critical Care Medicine (N.A.F., E.I.L., J.H., M.F.K.), Department of Anesthesiology and Critical Care Medicine, Children's Hospital of Philadelphia, Perelman School of Medicine, University of Pennsylvania; and Division of Neurosurgery (A.M.T., P.J.M., J.D.A.), Children's Hospital of Philadelphia; Department of Neurosurgery, Perelman School of Medicine, University of Pennsylvania.ORCID 0000-0002-9040-6420
Claudia Gambrah-LylesFrom the Division of Neurology (L.A.M., C.G., M.R.S., B.B.), Department of Pediatrics, Children's Hospital of Philadelphia; Department of Neurology, Perelman School of Medicine, University of Pennsylvania; Department of Pathology (A.V.), Children's Hospital of Philadelphia, Perelman School of Medicine, University of Pennsylvania; Division of Critical Care Medicine (N.A.F., E.I.L., J.H., M.F.K.), Department of Anesthesiology and Critical Care Medicine, Children's Hospital of Philadelphia, Perelman School of Medicine, University of Pennsylvania; and Division of Neurosurgery (A.M.T., P.J.M., J.D.A.), Children's Hospital of Philadelphia; Department of Neurosurgery, Perelman School of Medicine, University of Pennsylvania.
Angela ViaeneFrom the Division of Neurology (L.A.M., C.G., M.R.S., B.B.), Department of Pediatrics, Children's Hospital of Philadelphia; Department of Neurology, Perelman School of Medicine, University of Pennsylvania; Department of Pathology (A.V.), Children's Hospital of Philadelphia, Perelman School of Medicine, University of Pennsylvania; Division of Critical Care Medicine (N.A.F., E.I.L., J.H., M.F.K.), Department of Anesthesiology and Critical Care Medicine, Children's Hospital of Philadelphia, Perelman School of Medicine, University of Pennsylvania; and Division of Neurosurgery (A.M.T., P.J.M., J.D.A.), Children's Hospital of Philadelphia; Department of Neurosurgery, Perelman School of Medicine, University of Pennsylvania.ORCID 0000-0001-6430-8360
Nina A FainbergFrom the Division of Neurology (L.A.M., C.G., M.R.S., B.B.), Department of Pediatrics, Children's Hospital of Philadelphia; Department of Neurology, Perelman School of Medicine, University of Pennsylvania; Department of Pathology (A.V.), Children's Hospital of Philadelphia, Perelman School of Medicine, University of Pennsylvania; Division of Critical Care Medicine (N.A.F., E.I.L., J.H., M.F.K.), Department of Anesthesiology and Critical Care Medicine, Children's Hospital of Philadelphia, Perelman School of Medicine, University of Pennsylvania; and Division of Neurosurgery (A.M.T., P.J.M., J.D.A.), Children's Hospital of Philadelphia; Department of Neurosurgery, Perelman School of Medicine, University of Pennsylvania.
Elizabeth I LandzbergFrom the Division of Neurology (L.A.M., C.G., M.R.S., B.B.), Department of Pediatrics, Children's Hospital of Philadelphia; Department of Neurology, Perelman School of Medicine, University of Pennsylvania; Department of Pathology (A.V.), Children's Hospital of Philadelphia, Perelman School of Medicine, University of Pennsylvania; Division of Critical Care Medicine (N.A.F., E.I.L., J.H., M.F.K.), Department of Anesthesiology and Critical Care Medicine, Children's Hospital of Philadelphia, Perelman School of Medicine, University of Pennsylvania; and Division of Neurosurgery (A.M.T., P.J.M., J.D.A.), Children's Hospital of Philadelphia; Department of Neurosurgery, Perelman School of Medicine, University of Pennsylvania.
Alexander M TuckerFrom the Division of Neurology (L.A.M., C.G., M.R.S., B.B.), Department of Pediatrics, Children's Hospital of Philadelphia; Department of Neurology, Perelman School of Medicine, University of Pennsylvania; Department of Pathology (A.V.), Children's Hospital of Philadelphia, Perelman School of Medicine, University of Pennsylvania; Division of Critical Care Medicine (N.A.F., E.I.L., J.H., M.F.K.), Department of Anesthesiology and Critical Care Medicine, Children's Hospital of Philadelphia, Perelman School of Medicine, University of Pennsylvania; and Division of Neurosurgery (A.M.T., P.J.M., J.D.A.), Children's Hospital of Philadelphia; Department of Neurosurgery, Perelman School of Medicine, University of Pennsylvania.
Peter J MadsenFrom the Division of Neurology (L.A.M., C.G., M.R.S., B.B.), Department of Pediatrics, Children's Hospital of Philadelphia; Department of Neurology, Perelman School of Medicine, University of Pennsylvania; Department of Pathology (A.V.), Children's Hospital of Philadelphia, Perelman School of Medicine, University of Pennsylvania; Division of Critical Care Medicine (N.A.F., E.I.L., J.H., M.F.K.), Department of Anesthesiology and Critical Care Medicine, Children's Hospital of Philadelphia, Perelman School of Medicine, University of Pennsylvania; and Division of Neurosurgery (A.M.T., P.J.M., J.D.A.), Children's Hospital of Philadelphia; Department of Neurosurgery, Perelman School of Medicine, University of Pennsylvania.
Jimmy HuhFrom the Division of Neurology (L.A.M., C.G., M.R.S., B.B.), Department of Pediatrics, Children's Hospital of Philadelphia; Department of Neurology, Perelman School of Medicine, University of Pennsylvania; Department of Pathology (A.V.), Children's Hospital of Philadelphia, Perelman School of Medicine, University of Pennsylvania; Division of Critical Care Medicine (N.A.F., E.I.L., J.H., M.F.K.), Department of Anesthesiology and Critical Care Medicine, Children's Hospital of Philadelphia, Perelman School of Medicine, University of Pennsylvania; and Division of Neurosurgery (A.M.T., P.J.M., J.D.A.), Children's Hospital of Philadelphia; Department of Neurosurgery, Perelman School of Medicine, University of Pennsylvania.
Maya R SilverFrom the Division of Neurology (L.A.M., C.G., M.R.S., B.B.), Department of Pediatrics, Children's Hospital of Philadelphia; Department of Neurology, Perelman School of Medicine, University of Pennsylvania; Department of Pathology (A.V.), Children's Hospital of Philadelphia, Perelman School of Medicine, University of Pennsylvania; Division of Critical Care Medicine (N.A.F., E.I.L., J.H., M.F.K.), Department of Anesthesiology and Critical Care Medicine, Children's Hospital of Philadelphia, Perelman School of Medicine, University of Pennsylvania; and Division of Neurosurgery (A.M.T., P.J.M., J.D.A.), Children's Hospital of Philadelphia; Department of Neurosurgery, Perelman School of Medicine, University of Pennsylvania.
John D ArenaFrom the Division of Neurology (L.A.M., C.G., M.R.S., B.B.), Department of Pediatrics, Children's Hospital of Philadelphia; Department of Neurology, Perelman School of Medicine, University of Pennsylvania; Department of Pathology (A.V.), Children's Hospital of Philadelphia, Perelman School of Medicine, University of Pennsylvania; Division of Critical Care Medicine (N.A.F., E.I.L., J.H., M.F.K.), Department of Anesthesiology and Critical Care Medicine, Children's Hospital of Philadelphia, Perelman School of Medicine, University of Pennsylvania; and Division of Neurosurgery (A.M.T., P.J.M., J.D.A.), Children's Hospital of Philadelphia; Department of Neurosurgery, Perelman School of Medicine, University of Pennsylvania.
Martha F KienzleFrom the Division of Neurology (L.A.M., C.G., M.R.S., B.B.), Department of Pediatrics, Children's Hospital of Philadelphia; Department of Neurology, Perelman School of Medicine, University of Pennsylvania; Department of Pathology (A.V.), Children's Hospital of Philadelphia, Perelman School of Medicine, University of Pennsylvania; Division of Critical Care Medicine (N.A.F., E.I.L., J.H., M.F.K.), Department of Anesthesiology and Critical Care Medicine, Children's Hospital of Philadelphia, Perelman School of Medicine, University of Pennsylvania; and Division of Neurosurgery (A.M.T., P.J.M., J.D.A.), Children's Hospital of Philadelphia; Department of Neurosurgery, Perelman School of Medicine, University of Pennsylvania.
Brenda BanwellFrom the Division of Neurology (L.A.M., C.G., M.R.S., B.B.), Department of Pediatrics, Children's Hospital of Philadelphia; Department of Neurology, Perelman School of Medicine, University of Pennsylvania; Department of Pathology (A.V.), Children's Hospital of Philadelphia, Perelman School of Medicine, University of Pennsylvania; Division of Critical Care Medicine (N.A.F., E.I.L., J.H., M.F.K.), Department of Anesthesiology and Critical Care Medicine, Children's Hospital of Philadelphia, Perelman School of Medicine, University of Pennsylvania; and Division of Neurosurgery (A.M.T., P.J.M., J.D.A.), Children's Hospital of Philadelphia; Department of Neurosurgery, Perelman School of Medicine, University of Pennsylvania. banwellb@email.chop.edu.
Children's Hospital of Philadelphia · US

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

objectivesMyelin oligodendrocyte glycoprotein antibody-associated disease (MOGAD) is an immune-mediated neuroinflammatory disorder leading to demyelination of the CNS. Interleukin (IL)-6 receptor blockade is under study in relapsing MOGAD as a preventative strategy, but little is known about the role of such treatment for acute MOGAD attacks.

methodsWe discuss the cases of a 7-year-old boy and a 15-year-old adolescent boy with severe acute CNS demyelination and malignant cerebral edema with early brain herniation associated with clearly positive serum titers of MOG-IgG, whose symptoms were incompletely responsive to standard acute therapies (high-dose steroids, IV immunoglobulins (IVIGs), and therapeutic plasma exchange).

resultsBoth boys improved quickly with IL-6 receptor inhibition, administered as tocilizumab. Both patients have experienced remarkable neurologic recovery. DISCUSSION: We propose that IL-6 receptor therapies might also be considered in acute severe life-threatening presentations of MOGAD.

Indexed as

Demyelinating DiseasesAdolescentChildHumansImmunoglobulins, IntravenousMaleMyelin-Oligodendrocyte GlycoproteinPlasma ExchangePlasmapheresisReceptors, Interleukin-6IL6R protein, humanImmunoglobulins, IntravenousMyelin-Oligodendrocyte GlycoproteinReceptors, Interleukin-6

Identifiers

PMID37582615
PMCPMC10427143
OpenAlexW4385830190

What OpenQuestion holds

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Read underepoch 390

Registered trials

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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.