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Tumour necrosis factor (TNF) inhibitor therapy in Susac's syndrome.

Abstract
Susac's syndrome is the clinical triad of encephalopathy, branch retinal artery occlusions and sensorineural hearing loss (Susac 1994) [1]. It occurs predominantly in young females and is believed to be an immune-mediated endotheliopathy of small vessels of the brain, retina and cochlea (Neumayer et al. 2009) [2]. Early, aggressive, and sustained immunosuppressive therapy has been recommended for Susac's syndrome and anecdotal evidence has suggested a therapeutic role for monoclonal antibodies (Rennebohm et al. 2008, Lee and Amezcua 2009) [3,4]. We report a case of Susac's syndrome in which the patient improved immediately after tumour necrosis factor (TNF) inhibition with the monoclonal antibody, infliximab.
AuthorsT A Hardy, R J Garsia, G M Halmagyi, S J G Lewis, B Harrisberg, M J Fulham, M H Barnett
JournalJournal of the neurological sciences (J Neurol Sci) Vol. 302 Issue 1-2 Pg. 126-8 (Mar 15 2011) ISSN: 1878-5883 [Electronic] Netherlands
PMID21167503 (Publication Type: Case Reports, Journal Article)
CopyrightCopyright © 2010 Elsevier B.V. All rights reserved.
Chemical References
  • Anti-Inflammatory Agents
  • Antibodies, Monoclonal
  • Immunoglobulins, Intravenous
  • Immunosuppressive Agents
  • Tumor Necrosis Factor-alpha
  • Cyclophosphamide
  • Infliximab
  • Azathioprine
  • Prednisone
Topics
  • Anti-Inflammatory Agents (therapeutic use)
  • Antibodies, Monoclonal (therapeutic use)
  • Azathioprine (therapeutic use)
  • Brain (pathology)
  • Cognition Disorders (etiology, psychology)
  • Cyclophosphamide (therapeutic use)
  • Epilepsy, Generalized (etiology)
  • Female
  • Humans
  • Immunoglobulins, Intravenous (therapeutic use)
  • Immunosuppressive Agents (therapeutic use)
  • Infliximab
  • Magnetic Resonance Imaging
  • Neuropsychological Tests
  • Prednisone (therapeutic use)
  • Reverse Transcriptase Polymerase Chain Reaction
  • Seizures (etiology)
  • Susac Syndrome (drug therapy, psychology)
  • Tumor Necrosis Factor-alpha (antagonists & inhibitors)
  • Young Adult

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