Tocilizumab treatment in childhood takayasu arteritis: Case series of four patients and systematic review of the literature

• Published in: Seminars in arthritis and rheumatism Vol. 46; no. 4; pp. 529 - 535
• Main Authors: Batu, Ezgi Deniz, Sönmez, Hafize Emine, Hazırolan, Tuncay, Özaltın, Fatih, Bilginer, Yelda, Özen, Seza
• Format: Journal Article
• Language: English
• Published: United States Elsevier Inc 01.02.2017
• Subjects: Adolescent; Antibodies, Monoclonal, Humanized - therapeutic use; Child; Child, Preschool; Female; Humans; Immunosuppressive Agents - therapeutic use; Male; Remission Induction; Rheumatology; Takayasu arteritis; Takayasu Arteritis - drug therapy; Tocilizumab; Treatment; Treatment Outcome; CRP; Regulated on Activation, Normal T Cell Expressed and Secreted; RANTES; Tocilizumab; PET-CT; National Institutes of Health; APRs; Indian Takayasu Clinical Activity Score 2010; interleukin 6; tumor necrosis factor alpha; computed tomographic angiography; TNF α; IL 6; C reactive protein; Child; erythrocyte sedimentation rate; ESR; NIH; acute phase reactants; ITAS2010; CTA; Treatment; positron emission tomography-computed tomography; Takayasu arteritis; MRA; magnetic resonance angiography; TAK
• ISSN: 0049-0172; 1532-866X
• DOI: 10.1016/j.semarthrit.2016.07.012

Abstract Objective Our aim was to describe our experience with tocilizumab (interleukin 6 receptor antagonist) treatment in children with Takayasu arteritis and to review previous studies regarding tocilizumab use in Takayasu arteritis patients. Patients and methods We reviewed the charts of all pediatric Takayasu arteritis patients followed between 2000 and 2015 in Department of Pediatric Rheumatology in Hacettepe University, Ankara, Turkey and we present the patients who were treated with tocilizumab. We screened PubMed and MEDLINE for articles involving Takayasu arteritis patients treated with tocilizumab. Results We have followed four pediatric Takayasu arteritis patients who received tocilizumab. The median duration of immunosuppressive treatment before tocilizumab onset was 16 (1–60) months. The median duration of tocilizumab treatment was 9.5 (7–13) months. One of our patients received tocilizumab as a first line immunosuppressive treatment directly after methylprednisolone. Others were resistant to their initial immunosuppressive treatment (cyclophosphamide, methotrexate, or azathioprine). All achieved complete response to tocilizumab at the third month of treatment. None of the patients reported any adverse events during follow-up. In literature review, we identified 19 articles describing 75 Takayasu arteritis patients treated with tocilizumab. Eight of these received tocilizumab before the age of 18 years. Tocilizumab was the first line immunosuppressive treatment in six patients (five adults and one children). Conclusion Our small series suggests that tocilizumab may be a promising alternative for Takayasu arteritis treatment. Long-term controlled studies are warranted to provide better evidence for tocilizumab treatment in childhood Takayasu arteritis.