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+A 33-year-old Moroccan man with a 5-month history of recurring sinusitis presented with temperature, myalgia, cough, and hemoptysis.
+Thoracic physical examination revealed reduced vesicular murmur, and chest X-ray showed bilateral diffuse alveolar infiltrates.
+Laboratory analyses showed elevation of acute-phase proteins (CRP 26 mg/dL) and creatinine (1.2 mg/dL) with nonnephrotic proteinuria and microscopic glomerular hematuria.
+An antibiotic therapy for community acquired and atypical pneumoniae with levofloxacin 500 mg daily iv was established, but on the 3rd day from admission the patient developed respiratory failure unresponsive to noninvasive ventilation and had to be intubated.
+White lung with consolidations and ground glass areas at computed tomography scan (Fig.1), diffuse airways bleeding at fiberoptic bronchoscopy, mild normocytic anemia (9 g/dL), and ANCA-PR3 positivity (18.9 U/mL) were consistent with the diagnosis of GPA.
+Despite maximal ventilatory support, gas exchange did not improve (pH 7.33, PaO2 71 mm Hg, PaCO2 51 mm Hg, HCO3– 25 mmol/L, SaO2 94%) requiring extracorporeal membrane oxygenation (ECMO).
+Treatment with high-dose methylprednisolone (1 g daily for 3 days and 1 mg/kg daily thereafter), cyclophosphamide (1.2 g/pulse every 2 weeks for the 1st 3 pulses, followed by infusions every 3 weeks for the next 2 pulses), and plasma exchange (PE), according to European vasculitis study group recommendations,[4] was immediately started with respiratory improvement that allowed ECMO and orotracheal tube withdrawal and subsequent discharge from intensive care unit.
+After 9 days from the beginning of treatment ANCA-PR3 levels normalized (3 U/mL).
+Two months later, after the 5th bolus of cyclophosphamide and with prednisone 0.3 mg/kg/day, the patient complained arthromyalgia and testicular pain with edema, cough, and shortness of breath: laboratory data showed worsening of renal function, mild anemia, and hypoxemia (PaO2 61 mm Hg).
+Epididymitis was confirmed by ultrasound.
+Because of rapidly progressive renal failure (estimated glomerular filtration rate 35 mL/min) renal biopsy was performed and showed prominent intracapillary and extracapillary proliferation, with the formation of some fibrocellular crescents and focal capillary necrosis (Fig.2).
+Lung computed tomography-scan and fiberoptic bronchoscopy also confirmed alveolar hemorrhage relapse.
+Treatment with high-dose methylprednisolone (1 g daily for 3 consecutive days) and rituximab (375 mg/m2 weekly for 4 weeks) was started with rapid resolution of urologic and pulmonary involvement but delayed and partial improvement of renal function.
+At a 1-year follow-up, the patient is alive and under treatment with azathioprine (150 mg daily) and prednisone (15 mg daily).
+Renal function has markedly improved (estimated glomerular filtration rate 94 mL/min) same as gas-exchange (pH 7.42, PaO2 86 mm Hg, PaCO2 36 mm Hg, HCO3– 23 mmol/L, SaO2 95%).
+The patient signed institutional informed consent form at the time of admission to hospitalization.
+An approval by ethics committee was not necessary because of the routine health care.