An Unusual Case of Chronic Cough Revealing Ulcerative Colitis

Abstract

Question: A 22-year-old nonsmoking woman with no personal or familial medical history presented a daily productive cough over several months. She was treated with multiple empirical antibiotic therapy with transient improvement. Subsequently, she developed bloody diarrhea, diffuse abdominal pain, and a 5-kg weight loss. A colonoscopy revealed a nonulcerated pancolitis in a continuous fashion. Biopsy specimens revealed distorted crypt architecture with crypt branching and a diffuse inflammatory infiltrate composed of lymphocytes, plasma cells predominantly observed between the base of the crypts, and the muscularis mucosae and neutrophils, causing cryptitis. Perinuclear antineutrophil cytoplasmic antibodies were positive with a titer of 1/320 and anti-Saccharomyces cerevisiae antibodies were negative. Stool specimens excluded common pathogens included Clostridium difficile. A diagnosis of ulcerative colitis (UC) with E3 involvement according to Montreal classification was retained. She went into digestive remission after a steroid course and maintenance treatment with azathioprine was started. Compliance with medical therapy was poor and the patient was lost to follow-up. Four months after she stopped azathioprine, both respiratory and digestive symptoms worsened. She further presented a painful ulcerated skin lesion of the left leg. She was then hospitalized in our pneumology unit. Usual blood tests were unremarkable except for a C-reactive protein of 10 mg/L. The fecal calprotectin level was of 2221 mg/g. Stool specimens were tested negative. Endoscopic lesions were similar to the previous examination and colonic biopsies did not reveal a superinfection by cytomegalovirus. The skin biopsy was compatible with pyoderma gangrenosum. Bronchoscopy revealed a diffuse inflammatory mucosa with purulent secretions. Pathologic examination of bronchial biopsies revealed a dense infiltrate of inflammatory cells comprising mainly lymphocytes and plasma cells. Microbiological analysis of bronchial aspiration was negative. Pulmonary function testing (PFT) showed a severe obstructive ventilatory defect with a forced expiratory volume in 1 second (FEV 1) of 32% of predicted associated with lung distension. Chest imaging (chest radiographs and high-resolution computed tomography scan are presented. What is your diagnosis? Look on page 1225 for the answer and see the Gastroenterology web site (www.gastrojournal.org) for more information on submitting your favorite image to Clinical Challenges and Images in GI.