title: Long-term outcome of infliximab in severe chronic and refractory systemic sarcoidosis: a report of 16 cases.
abstract: OBJECTIVE
  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.
authors.affiliation: 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.
authors.forename: Catherine
authors.initials: C
authors.lastname: Chapelon-Abric
authors.affiliation: 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.
authors.forename: David
authors.initials: D
authors.lastname: Saadoun
authors.affiliation: Department of Biostatistics and Medical Information (SBIM), Hôpital Saint Louis, Paris, France.
authors.forename: Lucie
authors.initials: L
authors.lastname: Biard
authors.affiliation: Service de Médecine Interne, Hôpital Lariboisière, Paris, France.
authors.forename: Damien
authors.initials: D
authors.lastname: Sene
authors.affiliation: Department of Biostatistics and Medical Information (SBIM), Hôpital Saint Louis, Paris, France.
authors.forename: Matthieu
authors.initials: M
authors.lastname: Resche-Rigon
authors.affiliation: 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.
authors.forename: Baptiste
authors.initials: B
authors.lastname: Hervier
authors.affiliation: Service de Médecine Interne, Hôpital Cochin, Université René Descartes, Centre de Référence Maladies Auto-immunes et Systémiques Rares, Paris, France.
authors.forename: Nathalie
authors.initials: N
authors.lastname: Costedoat-Chalumeau
authors.affiliation: Département de Neuroradiologie, Université Pierre et Marie Curie, Paris, France.
authors.forename: Aurelie
authors.initials: A
authors.lastname: Drier
authors.affiliation: Département de Neurologie, Université Pierre et Marie Curie, Paris, France.
authors.forename: Jean Marc
authors.initials: JM
authors.lastname: Leger
authors.affiliation: 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.
authors.forename: Patrice
authors.initials: P
authors.lastname: Cacoub
mesh_terms: D000328:Adult; D000911:Antibodies, Monoclonal / Q000008:administration & dosage / Q000009:adverse effects; D002493:Central Nervous System Diseases / Q000209:etiology*; D004351:Drug Resistance; D005260:Female; D005938:Glucocorticoids / Q000008:administration & dosage / Q000009:adverse effects; D006331:Heart Diseases / Q000209:etiology*; D006801:Humans; D007166:Immunosuppressive Agents / Q000008:administration & dosage / Q000009:adverse effects; D007239:Infections / Q000209:etiology*; D000069285:Infliximab; D008297:Male; D012008:Recurrence; D012074:Remission Induction / Q000379:methods; D012189:Retrospective Studies; D012507:Sarcoidosis / Q000150:complications / Q000188:drug therapy / Q000503:physiopathology; D012720:Severity of Illness Index; D012737:Sex Factors; D013997:Time Factors; D016896:Treatment Outcome
chemical_list: D000911:Antibodies, Monoclonal; D005938:Glucocorticoids; D007166:Immunosuppressive Agents; D000069285:Infliximab