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Azathioprine/eculizumab Hypersensitivity reactions, bacterial pneumonia and Pneumocystis jirovecii pneumonia: case report

An adolescent girl [exact age at the time of reaction onset not stated] developed hypersensitivity reactions following treatment with azathioprine for granulomatosis with polyangiitis (GPA). Additionally, she developed bacterial pneumonia and Pneumocystis jirovecii pneumonia following treatment with eculizumab for GPA [routes, dosages, time to reaction onsets and outcomes not stated]. The girl presented in 2007, at the age of 9 years, with progressive fatigue, dyspnoea, stridor, rhinitis, sinusitis and conjunctivitis. Following extensive investigation, she was diagnosed with GPA. Subsequently, she was treated with prednisone, cyclophosphamide, mycophenolate mofetil, rituximab, methotrexate and immune globulin. She exhibited inadequate response and experienced multiple relapses during these treatments. In late 2013, she received a single dose of eculizumab leading to transient improvement. Thereafter, she started receiving azathioprine. However, she developed hypersensitivity reactions secondary to azathioprine. Azathioprine was discontinued. The girl was treated with leflunomide. She experienced complete collapse of her left lung due to underlying GPA. Lung transplantation was deferred due to unsatisfactory control of her disease. She experienced multiple events including osteoporosis, avascular necrosis of hips and vertebrae along with recurrent infections consisting of bacterial pneumonia, Pneumocystis jirovecii pneumonia, cytomegalovirus-related cytopenia, septic arthritis and osteomyelitis of the sinuses. Recurrent bacterial pneumonia and Pneumocystis jirovecii pneumonia were suspected to have developed secondary to eculizumab. In January 2017, she started receiving off-label treatment with oral avacopan 30mg two times per day. Thereafter, significant improvement was observed. Subsequently, her immunosuppressant therapy was gradually discontinued. Rituximab doses were spaced out. She did not require new tracheal or bronchial dilations. However, her left lung collapse and left main bronchus stenosis persisted. Ennis D, et al. Long-term use and remission of granulomatosis with polyangiitis with the oral C5a receptor inhibitor avacopan. BMJ Case Reports 13: No. 10, 29 Oct 2020. 803520637 Available from: URL: http://doi.org/10.1136/bcr-2020-236236

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Reactions 12 Dec 2020 No. 1834