Sustained clinical remission under infliximab/rituximab combination therapy in a patient with granulomatosis with polyangiitis
Tóm tắt
Granulomatosis with polyangiitis (GPA) is a systemic autoimmune disease characterized by small and medium vessel vasculitis. The use of biological therapies such as rituximab and infliximab has improved the treatment of ocular manifestations in GPA. We report a case of a 45-year-old Caucasian male suffering with rhinitis, sinubronchitis and exophthalmos. These clinical findings, subsequent biopsy and MRI were consistent with positive anti-neutrophil cytoplasm antibody (ANCA)/proteinase-3 and he was diagnosed with GPA with orbital involvement. He was refractory to cyclophosphamide at stable doses of methotrexate and a therapy with rituximab was started. Eventually and because of family planning methotrexate was replaced by azathioprine. Symptoms worsened and MRI revealed an increase in the granulomatous lesion in the orbit. Therefore, we decided to add infliximab to the combination of azathioprine and rituximab, our patient achieved then a long-term response. During the 10 years of the combined treatment, no adverse effects or systemic involvement occurred. This case suggests that the individual use of a combination of rituximab and infliximab may be a promising strategy for the treatment in the long term of refractory orbital GPA.
Tài liệu tham khảo
Muller K, Lin JH. Orbital granulomatosis with polyangiitis (Wegener granulomatosis): clinical and pathologic findings. Arch Pathol Lab Med. 2014;138(8):1110–4.
Suhler EB, Lim LL, Beardsley RM, Giles TR, Pasadhika S, Lee ST, et al. Rituximab therapy for refractory orbital inflammation: results of a phase 1/2, dose-ranging, randomized clinical trial. JAMA Ophthalmol. 2014;132(5):572–8.
Leavitt RY, Fauci AS, Bloch DA, Michel BA, Hunder GG, Arend WP, et al. The American College of Rheumatology 1990 criteria for the classification of Wegener’s granulomatosis. Arthritis Rheum. 1990;33(8):1101–7.
Guillevin L, Pagnoux C, Karras A, Khouatra C, Aumaître O, Cohen P, et al. Rituximab versus azathioprine for maintenance in ANCA-associated vasculitis. N Engl J Med. 2014;371(19):1771–80.
Guillevin L, Mukhtyar C, Pagnoux C, Yates M. Conventional and biological immunosuppressants in vasculitis. Best Pract Res Clin Rheumatol. 2018;32(1):94–111.
Taylor SR, Salama AD, Joshi L, Pusey CD, Lightman SL. Rituximab is effective in the treatment of refractory ophthalmic Wegener’s granulomatosis. Arthritis Rheum. 2009;60(5):1540–7.
Donmez S, Pamuk ON, Gedik M, Bulut AKR. A case of granulomatosis with polyangiitis and pyoderma gangrenosum successfully treated with infliximab and rituximab. Int J Rheum Dis. 2014;17(4):471–5.
Moen BH, Nystad TW, Barrett TM, Sandvik LF. A boy in his teens with large ulcerations of the head and neck. Tidsskr Nor Laegeforen. 2019. https://doi.org/10.4045/tidsskr.18.0647.
Silva F, Cisternas M, Specks U. TNF-α blocker therapy and solid malignancy risk in ANCA-associated vasculitis. Curr Rheumatol Rep. 2012;14(6):501–8.
