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June 2020; 10 (3) Research

Treatment and outcome of childhood cerebral sinovenous thrombosis

Ryan J. Felling, Sahar M.A. Hassanein, Jennifer Armstrong, Luis Aversa, Lori Billinghurst, Neil A. Goldenberg, Jo Ellen Lee, Emily C. Maxwell, Michael J. Noetzel, Warren Lo
First published September 12, 2019, DOI: https://doi.org/10.1212/CPJ.0000000000000720
Ryan J. Felling
Division of Child Neurology (RJF), Department of Neurology and Department of Pediatrics, Johns Hopkins University School of Medicine, Baltimore, MD; Department of Pediatrics (SMAH), Faculty of Medicine, Ain Shams University, Egypt; Section of Child Neurology and Hemophilia and Thrombosis Center (JA, ECM), Department of Pediatrics, University of Colorado, Boulder; Department of Hematology (LA), Ricardo Gutierrez Children's Hospital, Buenos Aires, Argentina; Department of Neurology (LB), Children's Hospital of Philadelphia, University of Pennsylvania School of Medicine; Divisions of Hematology (NAG), Departments of Pediatrics and Medicine, Johns Hopkins University School of Medicine, Baltimore, MD; All Children's Research Institute (NAG), Johns Hopkins All Children's Hospital, St. Petersburg, FL; Department of Neurology (JEL, WL), the Ohio State University and Nationwide Children's Hospital; College of Nursing (JEL), the Ohio State University, Columbus; Department of Neurology and Pediatrics (MJN), Washington University School of Medicine, St. Louis, MO; and Department of Pediatrics (WL), the Ohio State University and Nationwide Children's Hospital.
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Sahar M.A. Hassanein
Division of Child Neurology (RJF), Department of Neurology and Department of Pediatrics, Johns Hopkins University School of Medicine, Baltimore, MD; Department of Pediatrics (SMAH), Faculty of Medicine, Ain Shams University, Egypt; Section of Child Neurology and Hemophilia and Thrombosis Center (JA, ECM), Department of Pediatrics, University of Colorado, Boulder; Department of Hematology (LA), Ricardo Gutierrez Children's Hospital, Buenos Aires, Argentina; Department of Neurology (LB), Children's Hospital of Philadelphia, University of Pennsylvania School of Medicine; Divisions of Hematology (NAG), Departments of Pediatrics and Medicine, Johns Hopkins University School of Medicine, Baltimore, MD; All Children's Research Institute (NAG), Johns Hopkins All Children's Hospital, St. Petersburg, FL; Department of Neurology (JEL, WL), the Ohio State University and Nationwide Children's Hospital; College of Nursing (JEL), the Ohio State University, Columbus; Department of Neurology and Pediatrics (MJN), Washington University School of Medicine, St. Louis, MO; and Department of Pediatrics (WL), the Ohio State University and Nationwide Children's Hospital.
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Jennifer Armstrong
Division of Child Neurology (RJF), Department of Neurology and Department of Pediatrics, Johns Hopkins University School of Medicine, Baltimore, MD; Department of Pediatrics (SMAH), Faculty of Medicine, Ain Shams University, Egypt; Section of Child Neurology and Hemophilia and Thrombosis Center (JA, ECM), Department of Pediatrics, University of Colorado, Boulder; Department of Hematology (LA), Ricardo Gutierrez Children's Hospital, Buenos Aires, Argentina; Department of Neurology (LB), Children's Hospital of Philadelphia, University of Pennsylvania School of Medicine; Divisions of Hematology (NAG), Departments of Pediatrics and Medicine, Johns Hopkins University School of Medicine, Baltimore, MD; All Children's Research Institute (NAG), Johns Hopkins All Children's Hospital, St. Petersburg, FL; Department of Neurology (JEL, WL), the Ohio State University and Nationwide Children's Hospital; College of Nursing (JEL), the Ohio State University, Columbus; Department of Neurology and Pediatrics (MJN), Washington University School of Medicine, St. Louis, MO; and Department of Pediatrics (WL), the Ohio State University and Nationwide Children's Hospital.
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Luis Aversa
Division of Child Neurology (RJF), Department of Neurology and Department of Pediatrics, Johns Hopkins University School of Medicine, Baltimore, MD; Department of Pediatrics (SMAH), Faculty of Medicine, Ain Shams University, Egypt; Section of Child Neurology and Hemophilia and Thrombosis Center (JA, ECM), Department of Pediatrics, University of Colorado, Boulder; Department of Hematology (LA), Ricardo Gutierrez Children's Hospital, Buenos Aires, Argentina; Department of Neurology (LB), Children's Hospital of Philadelphia, University of Pennsylvania School of Medicine; Divisions of Hematology (NAG), Departments of Pediatrics and Medicine, Johns Hopkins University School of Medicine, Baltimore, MD; All Children's Research Institute (NAG), Johns Hopkins All Children's Hospital, St. Petersburg, FL; Department of Neurology (JEL, WL), the Ohio State University and Nationwide Children's Hospital; College of Nursing (JEL), the Ohio State University, Columbus; Department of Neurology and Pediatrics (MJN), Washington University School of Medicine, St. Louis, MO; and Department of Pediatrics (WL), the Ohio State University and Nationwide Children's Hospital.
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Lori Billinghurst
Division of Child Neurology (RJF), Department of Neurology and Department of Pediatrics, Johns Hopkins University School of Medicine, Baltimore, MD; Department of Pediatrics (SMAH), Faculty of Medicine, Ain Shams University, Egypt; Section of Child Neurology and Hemophilia and Thrombosis Center (JA, ECM), Department of Pediatrics, University of Colorado, Boulder; Department of Hematology (LA), Ricardo Gutierrez Children's Hospital, Buenos Aires, Argentina; Department of Neurology (LB), Children's Hospital of Philadelphia, University of Pennsylvania School of Medicine; Divisions of Hematology (NAG), Departments of Pediatrics and Medicine, Johns Hopkins University School of Medicine, Baltimore, MD; All Children's Research Institute (NAG), Johns Hopkins All Children's Hospital, St. Petersburg, FL; Department of Neurology (JEL, WL), the Ohio State University and Nationwide Children's Hospital; College of Nursing (JEL), the Ohio State University, Columbus; Department of Neurology and Pediatrics (MJN), Washington University School of Medicine, St. Louis, MO; and Department of Pediatrics (WL), the Ohio State University and Nationwide Children's Hospital.
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Neil A. Goldenberg
Division of Child Neurology (RJF), Department of Neurology and Department of Pediatrics, Johns Hopkins University School of Medicine, Baltimore, MD; Department of Pediatrics (SMAH), Faculty of Medicine, Ain Shams University, Egypt; Section of Child Neurology and Hemophilia and Thrombosis Center (JA, ECM), Department of Pediatrics, University of Colorado, Boulder; Department of Hematology (LA), Ricardo Gutierrez Children's Hospital, Buenos Aires, Argentina; Department of Neurology (LB), Children's Hospital of Philadelphia, University of Pennsylvania School of Medicine; Divisions of Hematology (NAG), Departments of Pediatrics and Medicine, Johns Hopkins University School of Medicine, Baltimore, MD; All Children's Research Institute (NAG), Johns Hopkins All Children's Hospital, St. Petersburg, FL; Department of Neurology (JEL, WL), the Ohio State University and Nationwide Children's Hospital; College of Nursing (JEL), the Ohio State University, Columbus; Department of Neurology and Pediatrics (MJN), Washington University School of Medicine, St. Louis, MO; and Department of Pediatrics (WL), the Ohio State University and Nationwide Children's Hospital.
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Jo Ellen Lee
Division of Child Neurology (RJF), Department of Neurology and Department of Pediatrics, Johns Hopkins University School of Medicine, Baltimore, MD; Department of Pediatrics (SMAH), Faculty of Medicine, Ain Shams University, Egypt; Section of Child Neurology and Hemophilia and Thrombosis Center (JA, ECM), Department of Pediatrics, University of Colorado, Boulder; Department of Hematology (LA), Ricardo Gutierrez Children's Hospital, Buenos Aires, Argentina; Department of Neurology (LB), Children's Hospital of Philadelphia, University of Pennsylvania School of Medicine; Divisions of Hematology (NAG), Departments of Pediatrics and Medicine, Johns Hopkins University School of Medicine, Baltimore, MD; All Children's Research Institute (NAG), Johns Hopkins All Children's Hospital, St. Petersburg, FL; Department of Neurology (JEL, WL), the Ohio State University and Nationwide Children's Hospital; College of Nursing (JEL), the Ohio State University, Columbus; Department of Neurology and Pediatrics (MJN), Washington University School of Medicine, St. Louis, MO; and Department of Pediatrics (WL), the Ohio State University and Nationwide Children's Hospital.
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Emily C. Maxwell
Division of Child Neurology (RJF), Department of Neurology and Department of Pediatrics, Johns Hopkins University School of Medicine, Baltimore, MD; Department of Pediatrics (SMAH), Faculty of Medicine, Ain Shams University, Egypt; Section of Child Neurology and Hemophilia and Thrombosis Center (JA, ECM), Department of Pediatrics, University of Colorado, Boulder; Department of Hematology (LA), Ricardo Gutierrez Children's Hospital, Buenos Aires, Argentina; Department of Neurology (LB), Children's Hospital of Philadelphia, University of Pennsylvania School of Medicine; Divisions of Hematology (NAG), Departments of Pediatrics and Medicine, Johns Hopkins University School of Medicine, Baltimore, MD; All Children's Research Institute (NAG), Johns Hopkins All Children's Hospital, St. Petersburg, FL; Department of Neurology (JEL, WL), the Ohio State University and Nationwide Children's Hospital; College of Nursing (JEL), the Ohio State University, Columbus; Department of Neurology and Pediatrics (MJN), Washington University School of Medicine, St. Louis, MO; and Department of Pediatrics (WL), the Ohio State University and Nationwide Children's Hospital.
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Michael J. Noetzel
Division of Child Neurology (RJF), Department of Neurology and Department of Pediatrics, Johns Hopkins University School of Medicine, Baltimore, MD; Department of Pediatrics (SMAH), Faculty of Medicine, Ain Shams University, Egypt; Section of Child Neurology and Hemophilia and Thrombosis Center (JA, ECM), Department of Pediatrics, University of Colorado, Boulder; Department of Hematology (LA), Ricardo Gutierrez Children's Hospital, Buenos Aires, Argentina; Department of Neurology (LB), Children's Hospital of Philadelphia, University of Pennsylvania School of Medicine; Divisions of Hematology (NAG), Departments of Pediatrics and Medicine, Johns Hopkins University School of Medicine, Baltimore, MD; All Children's Research Institute (NAG), Johns Hopkins All Children's Hospital, St. Petersburg, FL; Department of Neurology (JEL, WL), the Ohio State University and Nationwide Children's Hospital; College of Nursing (JEL), the Ohio State University, Columbus; Department of Neurology and Pediatrics (MJN), Washington University School of Medicine, St. Louis, MO; and Department of Pediatrics (WL), the Ohio State University and Nationwide Children's Hospital.
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Warren Lo
Division of Child Neurology (RJF), Department of Neurology and Department of Pediatrics, Johns Hopkins University School of Medicine, Baltimore, MD; Department of Pediatrics (SMAH), Faculty of Medicine, Ain Shams University, Egypt; Section of Child Neurology and Hemophilia and Thrombosis Center (JA, ECM), Department of Pediatrics, University of Colorado, Boulder; Department of Hematology (LA), Ricardo Gutierrez Children's Hospital, Buenos Aires, Argentina; Department of Neurology (LB), Children's Hospital of Philadelphia, University of Pennsylvania School of Medicine; Divisions of Hematology (NAG), Departments of Pediatrics and Medicine, Johns Hopkins University School of Medicine, Baltimore, MD; All Children's Research Institute (NAG), Johns Hopkins All Children's Hospital, St. Petersburg, FL; Department of Neurology (JEL, WL), the Ohio State University and Nationwide Children's Hospital; College of Nursing (JEL), the Ohio State University, Columbus; Department of Neurology and Pediatrics (MJN), Washington University School of Medicine, St. Louis, MO; and Department of Pediatrics (WL), the Ohio State University and Nationwide Children's Hospital.
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Citation
Treatment and outcome of childhood cerebral sinovenous thrombosis
Ryan J. Felling, Sahar M.A. Hassanein, Jennifer Armstrong, Luis Aversa, Lori Billinghurst, Neil A. Goldenberg, Jo Ellen Lee, Emily C. Maxwell, Michael J. Noetzel, Warren Lo
Neurol Clin Pract Jun 2020, 10 (3) 232-244; DOI: 10.1212/CPJ.0000000000000720

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Abstract

Objective To test our hypothesis that anticoagulation is associated with better neurologic outcomes in childhood cerebral sinovenous thrombosis (CSVT), we analyzed treatment and outcomes in a population of 410 children from the International Pediatric Stroke Study (IPSS).

Methods We included patients enrolled in the IPSS registry with a diagnosis of CSVT at age >28 days with radiologic confirmation, in isolation or with concomitant arterial ischemic stroke. The primary outcome was the neurologic status at discharge. We defined unfavorable outcome as severe neurologic impairment or death at discharge. The Pediatric Stroke Outcome Measure was used for long-term outcome in those with follow-up. Predictors of anticoagulation use and outcome were analyzed by logistic regression.

Results Most children (95%) had identifiable risk factors, and 82% received anticoagulation. Shift analysis demonstrated better outcomes at discharge in children who were anticoagulated, and this persisted with longer-term outcomes. In multivariable analysis, anticoagulation was significantly associated with favorable outcomes (adjusted odds ratio [aOR] unfavorable 0.32, p = 0.007) whereas infarct was associated with unfavorable outcome (aOR unfavorable 6.71, p < 0.001). The trauma/intracranial surgery was associated with a lower odds of anticoagulation use (aOR 0.14, p < 0.001).

Conclusions Within the IPSS registry, children with risk factors of trauma or intracranial surgery were less likely to receive anticoagulation for CSVT. Anticoagulation was associated with a lower odds of severe neurologic impairment or death at hospital discharge, but this finding is limited and needs further confirmation in randomized, controlled, prospective studies.

Footnotes

  • Funding information and disclosures are provided at the end of the article. Full disclosure form information provided by the authors is available with the full text of this article at Neurology.org/cp.

  • ↵* These authors contributed equally to the manuscript.

  • Received March 11, 2019.
  • Accepted July 26, 2019.
  • © 2019 American Academy of Neurology
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