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Digestive diseases and sciences, Oct 4, 2018
Granulomatosis with polyangiitis (GPA-Wegener’s) is a small vessel vasculitis associated with the... more Granulomatosis with polyangiitis (GPA-Wegener’s) is a small vessel vasculitis associated with the presence of antineutrophil cytoplasm autoantibodies. Gastrointestinal involvement ranges from 5 to 20% of cases. We describe a case in whom relapsing disease with widespread gastrointestinal manifestations including pharyngeal, esophageal, small and large intestine, and anus was present for the first time. A 43-years-old male, with a 10-year history of proteinase-3 (PR3-ANCA) positive GPA which presented initially with general (fever, malaise), articular, otorhinolaryngological, ocular (scleritis), and pulmonary manifestations (nodular disease, biopsy-proven), was treated with glucocorticoids (GC) and methotrexate (MTX) as remission induction therapy, followed by MTX as maintenance treatment for 5 years. After remission was achieved at month 6, gradual tapering of MTX during the following 4.5 years allowed for its withdrawal in 2010. He already had a history of a hemorrhoidal disease, leading in 2013 to ferropenic anemia, which was surgically treated with complete symptoms resolution. On March 2016, he presented with hematochezia, 10-kg weight loss over 1 month and high-grade fever (39 °C). After proctologic evaluation, he was subject to a new hemorrhoidectomy in another hospital, but his course was complicated with persistent anal pain and bleeding, plus continuous fever. He denied abdominal pain. He was treated with IV antibiotics, but due to persistent illness, he came to our center. On arrival, he was found emaciated, pale, polypneic, with oxygen saturation of 93%, febrile, with bilateral knee and left wrist arthritis and exquisite anorectal pain on physical examination. He had an indurated, ulcerated, necrotic mass on the posterior anal region, which corresponded on MRI imaging to an area of thickening of the rectal and anal walls. No abscess was present. Debridement of one of the necrotic ulcers ensued and biopsies were taken. Upper and lower endoscopies disclosed large oropharyngeal and esophageal ulcers, fibrin-covered, with multiple duodenal, ileal, and— in all segments—colonic ulcers (Fig. 1a–c). Biopsies were taken from the esophagus, stomach, duodenum, ascending colon, and rectum. Laboratory tests revealed microcytic anemia, with Hb 8.8 g/dL (ferropenic), normal serum creatinine, an active urinary sediment, CRP 185 mg/dL, ESR 104 mm/ hr, PR3-ANCA over 200 U/mL, and C-ANCA 1:640. Chest X-ray showed bilateral diffuse interstitial infiltrates. All cultures (blood, urine, stool) and C. difficile toxins A and B were negative. He received several antibiotics, but the lung disease progressed to suspected lung hemorrhage with * Luis Felipe Flores-Suárez felipe98@prodigy.net.mx
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Papers by Andrea Hernández