Tocilizumab in refractory aortitis: study on 16 patients and literature review.
OBJECTIVE
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.
Department of Rheumatology, Hospital Universitario Marqués de Valdecilla, IFIMAV, Santander, Spain. jmedtor@hotmail.com.
Javier
J
Loricera
Ricardo
R
Blanco
Santos
S
Castañeda
Alicia
A
Humbría
Norberto
N
Ortego-Centeno
Javier
J
Narváez
Cristina
C
Mata
Sheila
S
Melchor
Elena
E
Aurrecoechea
Jaime
J
Calvo-Alén
Pau
P
Lluch
Concepción
C
Moll
Mauricio
M
Mínguez
Gabriel
G
Herrero-Beaumont
Beatriz
B
Bravo
Esteban
E
Rubio
Mercedes
M
Freire
Enriqueta
E
Peiró
Carmen
C
González-Vela
Javier
J
Rueda-Gotor
Trinitario
T
Pina
Natalia
N
Palmou-Fontana
Vanesa
V
Calvo-Río
Francisco
F
Ortiz-Sanjuán
Miguel Ángel
MÁ
González-Gay
D000328:Adult; D000368:Aged; D061067:Antibodies, Monoclonal, Humanized / Q000008:administration & dosage / Q000009:adverse effects; D001025:Aortitis / Q000145:classification / Q000175:diagnosis / Q000188:drug therapy / Q000276:immunology; D016903:Drug Monitoring; D004351:Drug Resistance; D005260:Female; D005938:Glucocorticoids / Q000008:administration & dosage / Q000009:adverse effects; D006801:Humans; D007166:Immunosuppressive Agents / Q000008:administration & dosage / Q000009:adverse effects; D015850:Interleukin-6 / Q000097:blood*; D018810:Magnetic Resonance Angiography / Q000379:methods; D008297:Male; D008875:Middle Aged; D017063:Outcome Assessment, Health Care; D049268:Positron-Emission Tomography / Q000379:methods; D011241:Prednisone / Q000008:administration & dosage / Q000009:adverse effects; D019947:Receptors, Interleukin-6 / Q000037:antagonists & inhibitors*; D012074:Remission Induction / Q000379:methods; D013030:Spain
D061067:Antibodies, Monoclonal, Humanized; D005938:Glucocorticoids; D007166:Immunosuppressive Agents; D015850:Interleukin-6; D019947:Receptors, Interleukin-6; C502936:tocilizumab; D011241:Prednisone