INTRODUCTION: Tracheobronchitis caused by ulcerative colitis has been rarely documented in literature. The case reported here is unique because of similar pathologic findings observed on colonic and tracheal biopsy specimens and because of the original images observed on computed tomography (CT), magnetic resonance imaging (MRI) and endobronchial ultrasonography (EBUS).
CASE PRESENTATION: A 49-year-old woman presented with a one-year history of dyspnea on exertion and hoarseness. The patient had a total colectomy for ulcerative colitis 17 years prior to our evaluation, with a strong family history of collagen vascular disease. Her mother and sister both had rheumatoid arthritis. Physical examination revealed stridor during both inspiration and expiration but it was louder during inspiration. Flow volume loop showed flattening of both inspiratory and expiratory curves, which is consistent with fixed intrathoracic obstruction. CT and MRI showed diffuse narrowing of the trachea and thickening of the mucosal tissue. Bronchoscopy revealed edematous, hypervascular and floppy vocal cords as well as diffuse friable and hemorrhagic tracheobronchial mucosa. EBUS revealed circumferential thickening of the mucosa and intact tracheobronchial cartilaginous structures. The pathological findings of the trachea showed severe inflammatory cell concentration in the sub mucosal tissue, similar to the findings found on colonic biopsy. Symptoms improved after one week of treatment with corticosteroids and Cyclosporine.
DISCUSSIONS: There are some other differential diagnoses of diffuse tracheobronchial narrowing.•Wegener granulomatosis also causes the obstruction of the central airway. Wegener granulomatosis is characterized by the systemic granulomatous inflammation and necrotizing angiitis. Our paitent had no symptom of necrotizing angiitis such as renal or nervous system disorders. Anti-neutrophil cytoplasmic antibodies (ANCA) were negative and no parenchymal lung disease was seen. Amyloidosis can be excluded on the point of clinical view because it is also a systemic disease. EBUS images•helped because mucosa was thickened circumferentially and the cartilage was normal while in relapsing polychondritis (RP) the cartilage is destroyed and the posterior membrane is normal. Rhinoscleroma or tuberculosis can cause laryngotracheitis, but the patient had no risk factor or symptoms for these infections. In our case, the patient has a medical history of ulcerative colitis. It is rare that tracheobroncitis is involved in ulcerative colitis. To us knowledge, only 12 cases have been reported previously. All reported patients had bowel disease and at least 75% patients had some respiratory symptoms such as cough, stridor or dyspnea. After comparing the tracheal apecimen with the lesected colon we noted similarities showing inliltration of the inflammatory cells around the grandular systems from pathology. Microabscesses were seen on both tracheal and colonic biopsy tissues. We started the treatment by oral prednisolone and cyclosporine. The symptoms and MRI findings exhibited improvement.
CONCLUSION: We report a unique case of ulcerative colitis with tracheobronchitis in which we found similar pathologic findings on tracheal and colonic mucosal biopsies. EBUS images helped differentiate from other diseases that cause diffuse tracheobronchial narrowing.
DISCLOSURE: Miho Nakamura, None.