BACKGROUND: Churg-Strauss syndrome (CSS) is a systemic vasculitis that occurs in the setting of asthma or allergic rhinitis with eosinophilia. The development of systemic manifestations in these allergic patients needs to be recognized as a likely sign of CSS. OBJECTIVE: To describe a patient with limb paresthesia and abdominal complaints related to CSS. METHODS: Blood leukocyte count, nerve conduction study, ultrasound and computed tomography of the abdomen, laparoscopic cholecystectomy and ileum resection, and histopathologic examination of ileum and gallbladder samples. RESULTS: A 55-year-old man with chronic asthma and rhinosinusitis had acute acalculous cholecystitis after he experienced lower limb paresthesia subsequently recognized as being due to mononeuritis multiplex. His eosinophil count was 1,860/microL. Three days after laparoscopic cholecystectomy the patient developed sudden severe diffuse abdominal pain with hypotension due to perforation of the ileum. The peripheral eosinophil count increased to 14,000/microL. Ileal resection was performed. Histopathologic examination showed necrotizing vasculitis with eosinophilic infiltration of the ileum and granulomatous vasculitis with eosinophilic infiltration of the gallbladder. He was treated with pulse intravenous methylprednisolone, 1 g for 3 consecutive days, followed by pulse intravenous cyclophosphamide, 750 mg/m(2), and recovered uneventfully. He received 6 additional monthly infusions of cyclophosphamide, and oral prednisone was tapered. When last seen, 2 years later, the patient was in good clinical condition, continuing alternate-day use of oral prednisone (10 mg). CONCLUSIONS: Nonrespiratory symptoms, such as paresthesia and acalculous cholecystitis, in a patient with asthma should alert the physician to consider CSS. If the neuropathic complaints had prompted the consideration of vasculitis, medical management might have avoided one or both surgical procedures.
Warning nonrespiratory symptoms in asthma: catastrophic abdominal involvement in a case of Churg-Strauss syndrome
ROLLA, Giovanni;GUIDA, Giuseppe;HEFFLER, Enrico Marco
2007-01-01
Abstract
BACKGROUND: Churg-Strauss syndrome (CSS) is a systemic vasculitis that occurs in the setting of asthma or allergic rhinitis with eosinophilia. The development of systemic manifestations in these allergic patients needs to be recognized as a likely sign of CSS. OBJECTIVE: To describe a patient with limb paresthesia and abdominal complaints related to CSS. METHODS: Blood leukocyte count, nerve conduction study, ultrasound and computed tomography of the abdomen, laparoscopic cholecystectomy and ileum resection, and histopathologic examination of ileum and gallbladder samples. RESULTS: A 55-year-old man with chronic asthma and rhinosinusitis had acute acalculous cholecystitis after he experienced lower limb paresthesia subsequently recognized as being due to mononeuritis multiplex. His eosinophil count was 1,860/microL. Three days after laparoscopic cholecystectomy the patient developed sudden severe diffuse abdominal pain with hypotension due to perforation of the ileum. The peripheral eosinophil count increased to 14,000/microL. Ileal resection was performed. Histopathologic examination showed necrotizing vasculitis with eosinophilic infiltration of the ileum and granulomatous vasculitis with eosinophilic infiltration of the gallbladder. He was treated with pulse intravenous methylprednisolone, 1 g for 3 consecutive days, followed by pulse intravenous cyclophosphamide, 750 mg/m(2), and recovered uneventfully. He received 6 additional monthly infusions of cyclophosphamide, and oral prednisone was tapered. When last seen, 2 years later, the patient was in good clinical condition, continuing alternate-day use of oral prednisone (10 mg). CONCLUSIONS: Nonrespiratory symptoms, such as paresthesia and acalculous cholecystitis, in a patient with asthma should alert the physician to consider CSS. If the neuropathic complaints had prompted the consideration of vasculitis, medical management might have avoided one or both surgical procedures.
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