A Rare Case of Laryngeal Crohnʼs Treated with Laser Debridement, Infliximab and Azathioprine: 2011 ACG Presidential Poster

Purpose: A 48-year-old man presented to Otolaryngology with daytime somnolence, stridor and difficulty breathing. The patient had a past history severe obstructive sleep apnea and a remote history of Crohn's, requiring an ileocecal resection in the early 1980's. The patient denied having fevers, night sweats, weight loss or dermatologic changes. There was no perianal pathology and the abdomen was benign. On examining the oropharynx, the floor of the mouth appeared thickened but no ulcers were seen. Flexible laryngoscopy showed dramatic supraglottic edema involving the aryepiglottic folds, cricoarytenoid joints and epiglottis (Figure 1). Following an unsuccessful trial of prednisone, tissue surrounding the supraglottic area was excised via carbon dioxide laser therapy. Pathology from biopsies taken from the left epiglottic fold and arytenoids revealed extensive non-necrotizing granulomatous inflammation with fibrosis, edema and lymphangiectasia, favoring laryngeal Crohn's disease. Given the rarity of laryngeal Crohn's, other granulomatous processes were considered. Tuberculin skin testing was negative and the patient's chest x-ray was unremarkable. Serology was negative for ANCA. Mycobacterial tissue culture from the surgical specimen and tissue stains for fungi and acid-fast bacilli were negative. A small bowel follow through confirmed a prior ileocecal resection. A colonoscopy performed thereafter noted an area of ulceration suggesting mild disease. The patient was diagnosed with Crohn's involving the supraglottic larynx and was started on azathioprine. The patient had ongoing airway obstruction requiring three additional courses of laser debridement. Infliximab was started in an attempt to obviate the need for further surgery. Despite persistent obstruction, follow-up laryngoscopy suggested that the disease process in the larynx had stabilized. The manifestation of oropharyngeal Crohn's is a well documented phenomenon; however, true laryngeal manifestation is exceedingly rare and has been reported sporadically. Given this patient's severe airway obstruction, we would advocate for first-line treatment with infliximab and azathioprine in patients presenting with laryngeal manifestations of Crohn's unresponsive to steroid therapy.Figure: No Caption available.

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