Tocilizumab in refractory aortitis: study on 16 patients and literature review

Clin Exp Rheumatol. 2014 May-Jun;32(3 Suppl 82):S79-89. Epub 2014 May 15.

Abstract

Objectives: Non-infectious aortitis is often refractory to standard immunosuppressive therapy. Since IL-6 has been implicated in the pathogenesis of aortitis, we assessed the efficacy of the anti-IL6 receptor monoconal antibody tocilizumab (TCZ) in a series of patients with refractory non-infectious aortitis.

Methods: Review of 16 patients (14 women/2 men) with refractory aortitis diagnosed by imaging (CT angiography, MR angiography, and/or PET) that were treated with TCZ.

Results: The mean age±SD was 51.4±20.1 years. The underlying conditions were: Takayasu arteritis (TakA) (n=7 cases), giant cell arteritis (GCA) (n=7), relapsing polychondritis (RP) (n=1), and aortitis associated with retroperitoneal fibrosis (n=1). TCZ was the first biologic drug used in all patients with GCA and in the patient with aortitis associated with retroperitoneal fibrosis but in only 2 of 7 TakA patients. In the remaining cases anti-TNF inhibitors were prescribed before TCZ (standard dose was 8 mg/kg/iv/4 weeks). After a mean±SD follow-up of 11.8±6.6 months most patients experienced clinical improvement, showing reduction of erythrocyte sedimentation rate from 43±36 mm/1st h to 5±4 mm/1st h at last visit. At TCZ onset, 25% of patients had fever and 19% polymyalgia rheumatica. These manifestations disappeared after 3 months of TCZ therapy. A corticosteroid sparing effect was also achieved (from 27.3±17.6 mg/day of prednisone at TCZ onset to 4.2±3.8 mg/day at last visit). TCZ had to be discontinued in a patient because of severe neutropenia.

Conclusions: TCZ appears to be effective and relatively safe in patients with inflammatory aortitis refractory to corticosteroids or to other biologic immunosuppressive drugs.

Publication types

  • Multicenter Study
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adult
  • Aged
  • Antibodies, Monoclonal, Humanized* / administration & dosage
  • Antibodies, Monoclonal, Humanized* / adverse effects
  • Aortitis* / classification
  • Aortitis* / diagnosis
  • Aortitis* / drug therapy
  • Aortitis* / immunology
  • Drug Monitoring
  • Drug Resistance
  • Female
  • Glucocorticoids / administration & dosage
  • Glucocorticoids / adverse effects
  • Humans
  • Immunosuppressive Agents / administration & dosage
  • Immunosuppressive Agents / adverse effects
  • Interleukin-6 / blood*
  • Magnetic Resonance Angiography / methods
  • Male
  • Middle Aged
  • Outcome Assessment, Health Care
  • Positron-Emission Tomography / methods
  • Prednisone* / administration & dosage
  • Prednisone* / adverse effects
  • Receptors, Interleukin-6 / antagonists & inhibitors*
  • Remission Induction / methods
  • Spain

Substances

  • Antibodies, Monoclonal, Humanized
  • Glucocorticoids
  • Immunosuppressive Agents
  • Interleukin-6
  • Receptors, Interleukin-6
  • tocilizumab
  • Prednisone