Several cases of Churg-Strauss syndrome (CSS) have been reported in asthmatic patients treated with leukotriene receptor antagonists (LTRAs). It is not clear whether LTRA is a causative factor in the development of vasculitis. We present the case of a 26-year-old patient, who developed severe central and peripheral neuropathy after a short-term treatment with LTRA, followed by gastrointestinal perforation and bleeding. The patient was successfully treated with high-dose glucocorticoids, immunoglobulins, and cyclophosphamide. His monozygotic twin brother treated for asthma does not meet classification criteria for Churg-Strauss syndrome at the moment, but his condition is being monitored. Both asthma and rheumatology specialists should consider the possibility of CSS development in patients treated with LTRAs.
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