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Long-term outcomes of refractory neurosarcoidosis treated with infliximab

Journal of Neurology volume 264, pages 891–897 (2017)Cite this article

Abstract

Central nervous system localizations of sarcoidosis may be refractory to conventional treatment such as steroids and immunosuppressive drugs. Infliximab, a TNF-α antagonist chimeric antibody, has been shown to be effective for treatment of these localizations. The aim of this study was to evaluate the efficacy and safety, in particular the long-term outcomes, of the use of infliximab for the treatment of neurosarcoidosis. We retrospectively reviewed medical records of patients with neurosarcoidosis who had been treated with infliximab between 2009 and 2015. All patients had histologically proven non-caseating granulomas. Eighteen patients with histologically proven sarcoidosis were included in this study. All had neurological involvement consisting of meningeal (n = 16), cerebral (n = 10), spinal cord (n = 6), and/or optic nerve (n = 5) involvement. Sixteen patients had previously received at least one immunosuppressive drug in addition to corticosteroids, including cyclophosphamide in 11 patients. All patients received treatment with infliximab (3–7.5 mg/kg) associated with corticosteroids (n = 18), low-dose methotrexate (n = 15), azathioprine (n = 2), or mycophenolate (n = 1). Sixteen out of 18 patients improved clinically (initial median modified Rankin scale score of 3, final median score of 1; p < 0.0001). At 6 months after initiation of infliximab, six patients obtained complete remission (33%), ten attained partial remission (56%), and two had stable disease (11%). The median follow-up time was 20 months (range 6–93). Nine patients relapsed during follow-up (50%). Eight patients developed toxic side effects and seven of these side effects were infectious events. Infliximab is an efficacious treatment of refractory neurosarcoidosis. However, relapses frequently occurred during follow-up.

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Authors and Affiliations

  1. AP-HP, Service de Médecine Interne 2, Institut e3m, Centre National de Référence Maladies Auto-immunes Systémiques Rares, Groupe Hospitalier Pitié-Salpétrière, 47-83 Boulevard de l’hôpital, 75013, Paris Cedex 13, France

    Fleur Cohen Aubart, Julien Haroche, Miguel Hie, Alexis Mathian & Zahir Amoura

  2. Université Paris VI Pierre et Marie Curie, Sorbonnes Universités, 75013, Paris, France

    Fleur Cohen Aubart, Damien Galanaud, Julien Haroche & Zahir Amoura

  3. AP-HP, Service de Pneumologie, Hôpital Avicenne, 93000, Bobigny, France

    Diane Bouvry, Hilario Nunes & Dominique Valeyre

  4. AP-HP, Service de Neuroradiologie Diagnostique et Fonctionnelle, Groupe Hospitalier Pitié-Salpêtrière, 75013, Paris, France

    Damien Galanaud

  5. AP-HP, Service de Neurologie, Groupe Hospitalier Pitié-Salpêtrière, 75013, Paris, France

    Caroline Dehais & Dimitri Psimaras

  6. Service de Neurologie, Centre hospitalier Régional de Metz-Thionville, 57530, Ars-Laquenexy, France

    Guillaume Mathey

  7. Service de Neurologie, Centre Hospitalier René Dubos, 95300, Pontoise, France

    Corinne Pottier

  8. Service de Médecine Interne, University Hospital, 21000, Dijon, France

    Hervé Devilliers

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  1. Fleur Cohen Aubart
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  2. Diane Bouvry
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  3. Damien Galanaud
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  4. Caroline Dehais
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  7. Julien Haroche
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  9. Miguel Hie
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  10. Alexis Mathian
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Correspondence to Fleur Cohen Aubart.

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Cohen Aubart, F., Bouvry, D., Galanaud, D. et al. Long-term outcomes of refractory neurosarcoidosis treated with infliximab. J Neurol 264, 891–897 (2017). https://doi.org/10.1007/s00415-017-8444-9

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  • DOI: https://doi.org/10.1007/s00415-017-8444-9

Keywords

  • Neurosarcoidosis
  • Infliximab
  • Relapses
  • Side effects