Churg-Strauss 증후군 환자에서 저용량 경구 Cyclophosphamide 사용 중 발생한 출혈성 방광염 1예 (A Case of Churg-Strauss Syndrome with Hemorrhagic Cystitis after Prolonged Oral Cyclophosphamide Therapy)

서지정보

· 발행기관 : 대한천식알레르기학회
· 수록지 정보 : 천식 및 알레르기 / 32권 / 1호 / 56 ~ 60페이지
· 저자명 : 박은정, 이길선, 박하나, 이은영, 이병재, 최동철

초록

Background: Cyclophosphamide (CYC) has been used in patients with Churg-Strauss syndrome with a major organ involvement or peripheral neuropathy in order to reduce mortality and disability. We report a patient who developed hemorrhagic cystitis after receiving low-dose oral CYC over a 6-year period (a total cumulative dose of 137 g), to treat Churg-Strauss syndrome with peripheral neuropathy.
Case History: The patient developed left foot drop 18 months after he was diagnosed with Churg-Strauss syndrome. Despite 2 cycles of CYC pulse therapy, he complained of newly developed drop of the wrist and right foot. The neurologic manifestations had gradually improved with oral CYC therapy, which was given 1.5 mg/kg/day initially and was maintained at 0.3∼1.0 mg/kg/day in consideration of CYC pulse therapy-refractory peripheral neuropathy. Gross hematuria developed suddenly after 6 years.
Results: The patient was diagnosed with hemorrhagic cystitis related to oral CYC after ruling out other causes of hematuria. Hemorrhagic cystitis improved 1 week after the discontinuation of oral CYC. Conclusion: The maintenance of oral CYC can be efficacious when puls therapies have failed. Hemorrhagic cystitis might occur at low dose of oral CYC. Routine urinalysis and history-taking are crucial for patients receiving oral CYC for a long period even in the absence of side effects. The use of uroprotective measure should be considered during oral CYC therapy.

영어초록

Background: Cyclophosphamide (CYC) has been used in patients with Churg-Strauss syndrome with a major organ involvement or peripheral neuropathy in order to reduce mortality and disability. We report a patient who developed hemorrhagic cystitis after receiving low-dose oral CYC over a 6-year period (a total cumulative dose of 137 g), to treat Churg-Strauss syndrome with peripheral neuropathy.
Case History: The patient developed left foot drop 18 months after he was diagnosed with Churg-Strauss syndrome. Despite 2 cycles of CYC pulse therapy, he complained of newly developed drop of the wrist and right foot. The neurologic manifestations had gradually improved with oral CYC therapy, which was given 1.5 mg/kg/day initially and was maintained at 0.3∼1.0 mg/kg/day in consideration of CYC pulse therapy-refractory peripheral neuropathy. Gross hematuria developed suddenly after 6 years.
Results: The patient was diagnosed with hemorrhagic cystitis related to oral CYC after ruling out other causes of hematuria. Hemorrhagic cystitis improved 1 week after the discontinuation of oral CYC. Conclusion: The maintenance of oral CYC can be efficacious when puls therapies have failed. Hemorrhagic cystitis might occur at low dose of oral CYC. Routine urinalysis and history-taking are crucial for patients receiving oral CYC for a long period even in the absence of side effects. The use of uroprotective measure should be considered during oral CYC therapy.

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