Evidence map›Paper›PMID 34475249›Full record

ArticleNeurology(R) neuroimmunology & neuroinflammation2021

Increased Prevalence of Familial Autoimmune Disease in Children With Opsoclonus-Myoclonus Syndrome.

Jonathan D Santoro, Lauren M Kerr, Rachel Codden, Theron Charles Casper, Benjamin M Greenberg, Emmanuelle Waubant, Sek Won Kong, Kenneth D Mandl, Mark P Gorman

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

Article in Neurology(R) neuroimmunology & neuroinflammation, 2021. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 3 papers.

0numbers the graph read from it
0cells of the map it votes in
3citing papers in PubMed
0.3field-weighted citation impact, top 38% 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

3 citing papers in PubMed, 6 citations in OpenAlex.

  1. Article
  2. Article
  3. Review
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

9 authors at 2 institutions in 1 country.

Jonathan D SantoroFrom the Department of Neurology (J.D.S.), Massachusetts General Hospital, Boston; Department of Neurology (J.D.S., J.M.K., M.P.G.), Boston Children's Hospital, MA; Harvard Medical School (J.D.S., K.D.M.), Boston, MA; Division of Neurology (J.D.S.), Department of Pediatrics, Children's Hospital of Los Angeles, CA; Department of Neurology (J.D.S.), Keck School of Medicine at the University of Southern California, Los Angeles; Department of Pediatrics (R.C., T.C.C.), University of Utah School of Medicine, Salt Lake City; Department of Neurology and Neurotherapeutics (B.M.G.), The University of Texas Southwestern Medical Center at Dallas, TX; UCSF Weill Institute for Neurosciences (E.W.), Department of Neurology, University of California San Francisco, CA; Computational Health Informatics Program (S.W.K., K.D.M.), Boston Children's Hospital, MA; and Department of Pediatrics (S.W.K., K.D.M.), Boston Children's Hospital, MA. jdsantoro@chla.usc.edu.ORCID 0000-0002-8350-8234
Lauren M KerrFrom the Department of Neurology (J.D.S.), Massachusetts General Hospital, Boston; Department of Neurology (J.D.S., J.M.K., M.P.G.), Boston Children's Hospital, MA; Harvard Medical School (J.D.S., K.D.M.), Boston, MA; Division of Neurology (J.D.S.), Department of Pediatrics, Children's Hospital of Los Angeles, CA; Department of Neurology (J.D.S.), Keck School of Medicine at the University of Southern California, Los Angeles; Department of Pediatrics (R.C., T.C.C.), University of Utah School of Medicine, Salt Lake City; Department of Neurology and Neurotherapeutics (B.M.G.), The University of Texas Southwestern Medical Center at Dallas, TX; UCSF Weill Institute for Neurosciences (E.W.), Department of Neurology, University of California San Francisco, CA; Computational Health Informatics Program (S.W.K., K.D.M.), Boston Children's Hospital, MA; and Department of Pediatrics (S.W.K., K.D.M.), Boston Children's Hospital, MA.
Rachel CoddenFrom the Department of Neurology (J.D.S.), Massachusetts General Hospital, Boston; Department of Neurology (J.D.S., J.M.K., M.P.G.), Boston Children's Hospital, MA; Harvard Medical School (J.D.S., K.D.M.), Boston, MA; Division of Neurology (J.D.S.), Department of Pediatrics, Children's Hospital of Los Angeles, CA; Department of Neurology (J.D.S.), Keck School of Medicine at the University of Southern California, Los Angeles; Department of Pediatrics (R.C., T.C.C.), University of Utah School of Medicine, Salt Lake City; Department of Neurology and Neurotherapeutics (B.M.G.), The University of Texas Southwestern Medical Center at Dallas, TX; UCSF Weill Institute for Neurosciences (E.W.), Department of Neurology, University of California San Francisco, CA; Computational Health Informatics Program (S.W.K., K.D.M.), Boston Children's Hospital, MA; and Department of Pediatrics (S.W.K., K.D.M.), Boston Children's Hospital, MA.
Theron Charles CasperFrom the Department of Neurology (J.D.S.), Massachusetts General Hospital, Boston; Department of Neurology (J.D.S., J.M.K., M.P.G.), Boston Children's Hospital, MA; Harvard Medical School (J.D.S., K.D.M.), Boston, MA; Division of Neurology (J.D.S.), Department of Pediatrics, Children's Hospital of Los Angeles, CA; Department of Neurology (J.D.S.), Keck School of Medicine at the University of Southern California, Los Angeles; Department of Pediatrics (R.C., T.C.C.), University of Utah School of Medicine, Salt Lake City; Department of Neurology and Neurotherapeutics (B.M.G.), The University of Texas Southwestern Medical Center at Dallas, TX; UCSF Weill Institute for Neurosciences (E.W.), Department of Neurology, University of California San Francisco, CA; Computational Health Informatics Program (S.W.K., K.D.M.), Boston Children's Hospital, MA; and Department of Pediatrics (S.W.K., K.D.M.), Boston Children's Hospital, MA.
Benjamin M GreenbergFrom the Department of Neurology (J.D.S.), Massachusetts General Hospital, Boston; Department of Neurology (J.D.S., J.M.K., M.P.G.), Boston Children's Hospital, MA; Harvard Medical School (J.D.S., K.D.M.), Boston, MA; Division of Neurology (J.D.S.), Department of Pediatrics, Children's Hospital of Los Angeles, CA; Department of Neurology (J.D.S.), Keck School of Medicine at the University of Southern California, Los Angeles; Department of Pediatrics (R.C., T.C.C.), University of Utah School of Medicine, Salt Lake City; Department of Neurology and Neurotherapeutics (B.M.G.), The University of Texas Southwestern Medical Center at Dallas, TX; UCSF Weill Institute for Neurosciences (E.W.), Department of Neurology, University of California San Francisco, CA; Computational Health Informatics Program (S.W.K., K.D.M.), Boston Children's Hospital, MA; and Department of Pediatrics (S.W.K., K.D.M.), Boston Children's Hospital, MA.
Emmanuelle WaubantFrom the Department of Neurology (J.D.S.), Massachusetts General Hospital, Boston; Department of Neurology (J.D.S., J.M.K., M.P.G.), Boston Children's Hospital, MA; Harvard Medical School (J.D.S., K.D.M.), Boston, MA; Division of Neurology (J.D.S.), Department of Pediatrics, Children's Hospital of Los Angeles, CA; Department of Neurology (J.D.S.), Keck School of Medicine at the University of Southern California, Los Angeles; Department of Pediatrics (R.C., T.C.C.), University of Utah School of Medicine, Salt Lake City; Department of Neurology and Neurotherapeutics (B.M.G.), The University of Texas Southwestern Medical Center at Dallas, TX; UCSF Weill Institute for Neurosciences (E.W.), Department of Neurology, University of California San Francisco, CA; Computational Health Informatics Program (S.W.K., K.D.M.), Boston Children's Hospital, MA; and Department of Pediatrics (S.W.K., K.D.M.), Boston Children's Hospital, MA.
Sek Won KongFrom the Department of Neurology (J.D.S.), Massachusetts General Hospital, Boston; Department of Neurology (J.D.S., J.M.K., M.P.G.), Boston Children's Hospital, MA; Harvard Medical School (J.D.S., K.D.M.), Boston, MA; Division of Neurology (J.D.S.), Department of Pediatrics, Children's Hospital of Los Angeles, CA; Department of Neurology (J.D.S.), Keck School of Medicine at the University of Southern California, Los Angeles; Department of Pediatrics (R.C., T.C.C.), University of Utah School of Medicine, Salt Lake City; Department of Neurology and Neurotherapeutics (B.M.G.), The University of Texas Southwestern Medical Center at Dallas, TX; UCSF Weill Institute for Neurosciences (E.W.), Department of Neurology, University of California San Francisco, CA; Computational Health Informatics Program (S.W.K., K.D.M.), Boston Children's Hospital, MA; and Department of Pediatrics (S.W.K., K.D.M.), Boston Children's Hospital, MA.ORCID 0000-0003-4877-7567
Kenneth D MandlFrom the Department of Neurology (J.D.S.), Massachusetts General Hospital, Boston; Department of Neurology (J.D.S., J.M.K., M.P.G.), Boston Children's Hospital, MA; Harvard Medical School (J.D.S., K.D.M.), Boston, MA; Division of Neurology (J.D.S.), Department of Pediatrics, Children's Hospital of Los Angeles, CA; Department of Neurology (J.D.S.), Keck School of Medicine at the University of Southern California, Los Angeles; Department of Pediatrics (R.C., T.C.C.), University of Utah School of Medicine, Salt Lake City; Department of Neurology and Neurotherapeutics (B.M.G.), The University of Texas Southwestern Medical Center at Dallas, TX; UCSF Weill Institute for Neurosciences (E.W.), Department of Neurology, University of California San Francisco, CA; Computational Health Informatics Program (S.W.K., K.D.M.), Boston Children's Hospital, MA; and Department of Pediatrics (S.W.K., K.D.M.), Boston Children's Hospital, MA.ORCID 0000-0002-9781-0477
Mark P GormanFrom the Department of Neurology (J.D.S.), Massachusetts General Hospital, Boston; Department of Neurology (J.D.S., J.M.K., M.P.G.), Boston Children's Hospital, MA; Harvard Medical School (J.D.S., K.D.M.), Boston, MA; Division of Neurology (J.D.S.), Department of Pediatrics, Children's Hospital of Los Angeles, CA; Department of Neurology (J.D.S.), Keck School of Medicine at the University of Southern California, Los Angeles; Department of Pediatrics (R.C., T.C.C.), University of Utah School of Medicine, Salt Lake City; Department of Neurology and Neurotherapeutics (B.M.G.), The University of Texas Southwestern Medical Center at Dallas, TX; UCSF Weill Institute for Neurosciences (E.W.), Department of Neurology, University of California San Francisco, CA; Computational Health Informatics Program (S.W.K., K.D.M.), Boston Children's Hospital, MA; and Department of Pediatrics (S.W.K., K.D.M.), Boston Children's Hospital, MA.
University of Southern California · USBoston Children's Hospital · US

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

BACKGROUND AND

objectivesOpsoclonus-myoclonus syndrome (OMS) is a rare autoimmune disorder associated with neuroblastoma in children, although idiopathic and postinfectious etiologies are present in children and adults. Small cohort studies in homogenous populations have revealed elevated rates of autoimmunity in family members of patients with OMS, although no differentiation between paraneoplastic and nonparaneoplastic forms has been performed. The objective of this study was to investigate the prevalence of autoimmune disease in first-degree relatives of pediatric patients with paraneoplastic and nonparaneoplastic OMS.

methodsA single-center cohort study of consecutively evaluated children with OMS was performed. Parents of patients were prospectively administered surveys on familial autoimmune disease. Rates of autoimmune disease in first-degree relatives of pediatric patients with OMS were compared using Fisher exact

resultsThirty-five patients (18 paraneoplastic, median age at onset 19.0 months; 17 idiopathic, median age at onset 25.0 months) and 68 first-degree relatives (median age 41.9 years) were enrolled. One patient developed systemic lupus erythematosus 7 years after OMS onset. Paraneoplastic OMS was associated with a 50% rate of autoimmune disease in a first-degree relative compared with 29% in idiopathic OMS ( DISCUSSION: In a cohort of pediatric patients with OMS, there were elevated rates of first-degree relative autoimmune disease, with no difference in rates observed between paraneoplastic and idiopathic etiologies, suggesting an autoimmune genetic contribution to the development of OMS in children.

Indexed as

AdultAutoimmune DiseasesChild, PreschoolCohort StudiesFamilyFemaleGenetic Predisposition to DiseaseHumansInfantMaleMiddle AgedOpsoclonus-Myoclonus SyndromePrevalence

Identifiers

PMID34475249
PMCPMC8422990
OpenAlexW3198315322

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