A young girl with severe polyarteritis nodosa successfully treated with tocilizumab: a case report

被引:5
作者
Boistault, Margaux [1 ]
Corbeto, Mireia Lopez [2 ]
Quartier, Pierre [1 ,3 ]
Arcobe, Laura Berbel [4 ]
Durall, Ariadna Carsi [5 ]
Aeschlimann, Florence A. [1 ]
机构
[1] Necker Univ Hosp, Assitance Publ Hopitaux Paris, Enfants Malad, Dept Pediat Immunol Hematol & Rheumatol, 149 Rue Sevres, F-75015 Paris, France
[2] Vall dHebron Hosp Campus, Pediat Rheumatol Unit, Barcelona, Spain
[3] Univ Paris, Imagine Inst, Paris, France
[4] Mutua Terrassa Univ Hosp, Rheumatol Dept, Barcelona, Spain
[5] Vall dHebron Hosp Campus, Pediat Hospitalisat Unit, Barcelona, Spain
关键词
Polyarteritis nodosa; Child; Tocilizumab; INTERLEUKIN-6; PATIENT;
D O I
10.1186/s12969-021-00654-7
中图分类号
R72 [儿科学];
学科分类号
100202 ;
摘要
Background Childhood Polyarteritis nodosa (PAN) is a systemic vasculitis with necrotizing inflammation of medium- and small-sized arteries. Disease evolution may be severe and refractory to standard treatment including prednisone, azathioprine and cyclophosphamide. Case presentation We present the case of a young girl with severe PAN resulting in progressive ischemia and necrosis of fingers and toes. Biological work-up revealed increased acute phase reactants and interleukin-6 levels. She was only partially controlled despite high-dose corticosteroids and cyclophosphamide infusions, and eventually achieved rapid improvement and sustained remission on tocilizumab. Further, we review the current evidence of the interleukin-6-inhibitor tocilizumab for the treatment of PAN. Conclusion Tocilizumab may be an efficient therapeutic option in a subset of treatment-refractory children with PAN.
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页数:6
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