INTRODUCTION:Tracheobronchitis related to Crohn's disease (CD) can rarely cause severe upper airway stenosis1,2. We present a case of severe isolated segmental bronchial stenosis related to CD. CASE PRESENTATION: A 52-year-old male presented to our clinic with three months of exertional dyspnea and productive cough. Diagnosed with CD 20 years ago he was initially managed with infliximab, prednisone and mesalamine. He later transitioned to adalimumab in 2017. He was a non-smoker followed by our clinic for chronic bronchitis secondary to CD. Pulmonary function testing in 2013 was normal with a FEV1/FVC of 78%, FVC of 92% and DLCO of 95%. His bronchitis was managed with inhaled fluticasone and as needed albuterol.Computed tomography (CT) in February 2019 for surveillance of pulmonary nodules showed stable nodules with interval appearance of concentric narrowing in the proximal segmental bronchus to the superior segment of the left lower lobe (LLL). In May the patient reported increased sputum production, dyspnea and chest tightness worsened in the left decubitus position. Repeat imaging showed worsening bronchial wall thickening with tree in bud opacities in the anterior LLL.Bronchoscopy showed severe narrowing of the LLL bronchus with a fibrotic band like stricture. Knife assisted balloon dilation was performed along with local injection of methylprednisolone. Following this the patient reported significant improvement of dyspnea, sputum production and resolution of positional chest tightness.Seven months later he reported recurrence of productive cough and chest tightness. Chest CT showed circumferential narrowing of the proximal LLL bronchus with worsened wall thickening and complete occlusion of the medial segment of the LLL. Bronchoscopy revealed cobblestoning in the left mainstem bronchus and fibrotic bronchial stenosis at LLL with 90% occlusion. Knife assisted balloon dilation was again performed with local injection of methylprednisolone. Endobronchial biopsy showed bronchial mucosa with mixed acute and chronic inflammatory infiltrate. The patient was treated with a course of prednisone and levofloxacin and was started inhaled budesonide and formoterol. Following this he had significant improvement in his symptoms.DISCUSSION: Previously reported cases of airway stenosis related to CD involve the trachea1,2. Our patient developed severe isolated segmental bronchial stenosis related to CD. Case reports have shown that corticosteroids reduce mucosal inflammation, but have limited efficacy on underlying stenosis2. Cases of tracheal stenosis resistant to corticosteroids were later successfully treated with dilation via bronchoscopy1.CONCLUSIONS: This case highlights the role of interventional pulmonology in management of a rare case of isolated segmental bronchial stenosis related to CD.
Stridor can be associated with a variety of etiologies as a sign of upper airway obstruction. From malignancy of the airway to trauma secondary to intubation, stridor is a cause for concern of impending obstruction. Obstructive fibrinous tracheal pseudomembrane (OFTP) is an uncommon cause of airway obstruction. OFTP is a condition with unclear pathophysiology which causes a subacute presentation of stridor usually in the post-extubation period. In the case report our patient developed a pseudomembrane post-traumatic intubation that grew Hemophilus parainfluenza. This is the first case reported of OFTP associated with Hemophilus parainfluenza growth.
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