Literature DB >> 19888559

[Aggressive immunotherapy in Susac's syndrome].

M Klein1, T Illies, S Georgi, T Rosenkranz, C Terborg.   

Abstract

Susac's syndrome is a rare but important differential diagnosis of aseptic encephalitis of young women with focal neurological deficits and white matter lesions on cerebral MRI. We report on a previously healthy 36-year-old woman who presented with encephalopathy, central weakness of her right leg and multiple white matter lesions on MRI. Shortly thereafter, inner ear deafness developed and funduscopy revealed occlusions of branch retinal arteries. A diagnosis of retino-cochlear-cerebral vasculopathy or Susac's syndrome was established and steroid-based immunotherapy with high-dose corticosteroids was initiated. Steroid reduction led to repeated clinical worsening, so that immunotherapy was sequentially escalated. Finally, high-dose cyclophosphamide every 4 weeks led to sufficient control of disease activity. Recent publications have argued for an early and aggressive immunosuppression in Susac's syndrome based on clinical and histological similarities with juvenile dermatomyositis, where such a regimen has already been established. We report on these treatment guidelines with respect to the current literature and the case presented.

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Year:  2009        PMID: 19888559     DOI: 10.1007/s00115-009-2866-2

Source DB:  PubMed          Journal:  Nervenarzt        ISSN: 0028-2804            Impact factor:   1.214


  12 in total

Review 1.  Treatment of Susac's syndrome.

Authors:  Robert M Rennebohm; John O Susac
Journal:  J Neurol Sci       Date:  2007-02-26       Impact factor: 3.181

Review 2.  Retinocochleocerebral vasculopathy.

Authors:  G W Petty; A G Engel; B R Younge; J Duffy; T Yanagihara; C F Lucchinetti; J D Bartleson; J E Parisi; J L Kasperbauer; M Rodriguez
Journal:  Medicine (Baltimore)       Date:  1998-01       Impact factor: 1.889

3.  [The Susac syndrome--a retinocochleocerebral angiopathy].

Authors:  M Winterholler; C Lang; U Schonherr; J G Heckmann; B Neundorfer
Journal:  Fortschr Neurol Psychiatr       Date:  2000-10       Impact factor: 0.752

4.  Treatment of Susac syndrome with gamma globulin and corticosteroids.

Authors:  Robert J Fox; Fiona Costello; Alexander R Judkins; Steven L Galetta; Albert M Maguire; Brian Leonard; Clyde E Markowitz
Journal:  J Neurol Sci       Date:  2006-10-17       Impact factor: 3.181

5.  Long-term outcome in Susac syndrome.

Authors:  Fleur Aubart-Cohen; Isabelle Klein; Jean-François Alexandra; Bahram Bodaghi; Serge Doan; Christine Fardeau; Philippa Lavallée; Jean-Charles Piette; Phuc Le Hoang; Thomas Papo
Journal:  Medicine (Baltimore)       Date:  2007-03       Impact factor: 1.889

Review 6.  Susac's syndrome: 1975-2005 microangiopathy/autoimmune endotheliopathy.

Authors:  John O Susac; Robert A Egan; Robert M Rennebohm; Martin Lubow
Journal:  J Neurol Sci       Date:  2007-02-28       Impact factor: 3.181

7.  Microangiopathy of the brain and retina.

Authors:  J O Susac; J M Hardman; J B Selhorst
Journal:  Neurology       Date:  1979-03       Impact factor: 9.910

8.  Aggressive management of juvenile dermatomyositis results in improved outcome and decreased incidence of calcinosis.

Authors:  Rita E Fisler; Marilyn G Liang; Robert C Fuhlbrigge; Ali Yalcindag; Robert P Sundel
Journal:  J Am Acad Dermatol       Date:  2002-10       Impact factor: 11.527

9.  Treatment of Susac's Syndrome.

Authors:  Robert M Rennebohm; Robert A Egan; John O Susac
Journal:  Curr Treat Options Neurol       Date:  2008-01       Impact factor: 3.598

10.  Aggressive immunosuppressive treatment of Susac's syndrome in an adolescent: using treatment of dermatomyositis as a model.

Authors:  Robert M Rennebohm; Martin Lubow; Jerome Rusin; Lisa Martin; Deborah M Grzybowski; John O Susac
Journal:  Pediatr Rheumatol Online J       Date:  2008-01-29       Impact factor: 3.054

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  2 in total

Review 1.  Characteristics of Susac syndrome: a review of all reported cases.

Authors:  Jan Dörr; Sarah Krautwald; Brigitte Wildemann; Sven Jarius; Marius Ringelstein; Thomas Duning; Orhan Aktas; Erich Bernd Ringelstein; Friedemann Paul; Ilka Kleffner
Journal:  Nat Rev Neurol       Date:  2013-04-30       Impact factor: 42.937

2.  [Retinal arteritis in pregnancy].

Authors:  D Finis; J Stammen; J Gonnermann
Journal:  Ophthalmologe       Date:  2011-07       Impact factor: 1.059

  2 in total

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