Sustained response to tocilizumab in a patient with relapsing polychondritis with aortic involvement: a case based review.
This paper presents a case with refractory relapsing polychondritis (RPC), complicated with severe aortic involvement, which is successfully treated with tocilizumab. Previous treatments consisted of methotrexate, corticosteroids, cyclosporine, cyclophosphamide, infliximab, and etanercept. With these treatments, the patient had recurrent episodes of fever, polyarthritis, tenosynovitis, subcutaneous nodules, and progressive cardiac disease. One year after the start of treatment with tocilizumab, there is resolution of all symptoms, normalization of C-reactive protein (CRP) and erythrocyte sedimentation rate (ESR), and the dose of prednisolone is tapered down to 2 mg/day. We have reviewed the English literature for reports of patients with refractory RPC, successfully treated with tocilizumab. We found five additional case reports. In one case report, a patient with refractory RPC complicated with aortitis was successfully treated with tocilizumab. In three case reports, patients with refractory RPC complicated with laryngotracheal involvement were successfully treated with tocilizumab. All cases had, like our patient, failed conventional treatment. We also reviewed the literature for reports of the effect of biologicals on cardiac involvement in RPC. Current literature is presented and discussed.
Department of Rheumatology, University Hospital of Ghent, De Pintelaan 185, 9000, Ghent, Belgium, rebecca_stael@hotmail.com.
Rebecca
R
Stael
Vanessa
V
Smith
Ruth
R
Wittoek
David
D
Creytens
Herman
H
Mielants
D000328:Adult; D000893:Anti-Inflammatory Agents / Q000627:therapeutic use*; D061067:Antibodies, Monoclonal, Humanized / Q000627:therapeutic use*; D001025:Aortitis / Q000150:complications / Q000188:drug therapy*; D006801:Humans; D008297:Male; D011081:Polychondritis, Relapsing / Q000150:complications / Q000188:drug therapy*; D016896:Treatment Outcome
D000893:Anti-Inflammatory Agents; D061067:Antibodies, Monoclonal, Humanized; C502936:tocilizumab