Korean J Asthma Allergy Clin Immunol.  2012 Mar;32(1):56-60.

A Case of Churg-Strauss Syndrome with Hemorrhagic Cystitis after Prolonged Oral Cyclophosphamide Therapy

Abstract

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.

MeSH Terms

Churg-Strauss Syndrome
Cyclophosphamide
Cystitis
Foot
Hematuria
Humans
Neurologic Manifestations
Peripheral Nervous System Diseases
Urinalysis
Wrist
Cyclophosphamide