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Long-term outcome of infliximab in severe chronic and refractory systemic sarcoidosis: a report of 16 cases

1, 2, 3, 4, 5, 6, 7, 8, 9, 10

 

  1. 1.Sorbonne Universités, UPMC Univ Paris, UMR7211 & Inflammation-Immunopathology-Biotherapy Dept. (DHU i2B); 2.INSERM, UMR_S959, Paris; 3.CNRS, FRE3632, Paris; 4.APHP, Groupe Hosp. Pitié Salpétrière, Dept. Internal Medicine & Clin.Immunol, Paris, France.
  2. 1.Sorbonne Universités, UPMC Univ Paris, UMR7211 & Inflammation-Immunopathology-Biotherapy Dept. (DHU i2B); 2.INSERM, UMR_S959, Paris; 3.CNRS, FRE3632, Paris; 4.APHP, Groupe Hosp. Pitié Salpétrière, Dept. Internal Medicine & Clin.Immunol, Paris, France.
  3. Department of Biostatistics and Medical Information (SBIM), Hôpital Saint Louis, Paris, France.
  4. Service de Médecine Interne, Hôpital Lariboisière, Paris, France.
  5. Department of Biostatistics and Medical Information (SBIM), Hôpital Saint Louis, Paris, France.
  6. 1.Sorbonne Universités, UPMC Univ Paris, UMR7211 & Inflammation-Immunopathology-Biotherapy Dept. (DHU i2B); 2.INSERM, UMR_S959, Paris; 3.CNRS, FRE3632, Paris; 4.APHP, Groupe Hosp. Pitié Salpétrière, Dept. Internal Medicine & Clin.Immunol, Paris, France.
  7. Service de Médecine Interne, Hôpital Cochin, Université René Descartes, Centre de Référence Maladies Auto-immunes et Systémiques Rares, Paris, France.
  8. Département de Neuroradiologie, Université Pierre et Marie Curie, Paris, France.
  9. Département de Neurologie, Université Pierre et Marie Curie, Paris, France.
  10. 1.Sorbonne Universités, UPMC Univ Paris, UMR7211 & Inflammation-Immunopathology-Biotherapy Dept. (DHU i2B); 2.INSERM, UMR_S959, Paris; 3.CNRS, FRE3632, Paris; 4.APHP, Groupe Hosp. Pitié Salpétrière, Dept. Internal Medicine & Clin.Immunol, Paris, France.

CER8047
2015 Vol.33, N°4
PI 0509, PF 0515
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PMID: 26120779 [PubMed]

Received: 21/10/2014
Accepted : 02/03/2015
In Press: 29/06/2015
Published: 20/07/2015

Abstract

OBJECTIVES:
Infliximab (IFX) appears to be effective in refractory sarcoidosis. However, data are lacking regarding its efficacy in severe sarcoidosis (i.e. with cardiac and/or neurological involvement).
METHODS:
Retrospective single-centre study including 16 unselected consecutive patients with biopsy proven, severe, and resistant sarcoidosis, who were treated by infliximab (3 or 5 mg /kg at 0, 2 and 6 weeks, then every 8 weeks) between 2005 and 2013.
RESULTS:
Following IFX therapy we observed an improvement in 92% of cases, with a marked decrease of the severity score [median score 6 (3–12) vs. 2 (1–8), p<0.0001] and trend toward steroid sparing effect [12.5 (0–40) vs. 8.5 mg/d (0–30), p=0.11] between baseline and the end of follow-up, respectively. Regarding the index organ response, we observed a remission of cardiac and central nervous system involvements in 4 out of 4 and 11 out 12 cases, respectively. Thirty-eight percent of patients experienced a relapse. After a median follow-up of 57 months (2 to 91), we observed 7 (44%) infectious complications, 1 paradoxical cutaneous granuloma and 1 leucoencephalopathy. Infectious complications were mostly observed in male [6/7 (86%), p=0.06], with a longer duration of steroids (108 vs. 39 months, p=0.11) and immunosuppressant use prior IFX (42 vs. 24 months, p=0.08) compared to their negative counterpart, respectively.
CONCLUSIONS:
IFX was efficient in severe and refractory sarcoidosis. Infectious complications were frequent and occurred mainly in male patients with longer duration of steroids and immunosuppressant use prior to IFX.

Rheumatology Article