Pulmonary Manifestations of Systemic Disease: Fellow Case Report Poster - Pulmonary Manifestations of Lung Disease |

Constrictive Bronchiolitis With Pulmonary Thrombus Associated With Ulcerative Colitis FREE TO VIEW

Russell Buhr, MD; Corinne Sheth, MD
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David Geffen School of Medicine at the University of California, Los Angeles, CA

Copyright 2016, American College of Chest Physicians. All Rights Reserved.

Chest. 2016;150(4_S):1057A. doi:10.1016/j.chest.2016.08.1164
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SESSION TITLE: Fellow Case Report Poster - Pulmonary Manifestations of Lung Disease

SESSION TYPE: Affiliate Case Report Poster

PRESENTED ON: Tuesday, October 25, 2016 at 01:30 PM - 02:30 PM

INTRODUCTION: Pulmonary complications of inflammatory bowel disease (IBD) are well-described, including airway & interstitial lung disease, serositis, organizing, and eosinophilic pneumonia and venous thromboembolic disease (VTE). Abnormal pulmonary function tests (PFTs) without symptoms are also reported. Treatment with 5-ASA & TNF-α inhibitors can cause lung disease. We present a case of IBD-associated obliterative bronchiolitis (OB) with associated in situ pulmonary thrombosis.

CASE PRESENTATION: A 52-year-old female ex-smoker was diagnosed with ulcerative colitis (UC) in 2013. Dyspnea began in 2014 and was presumed due to emphysema. She did not respond to inhalers. UC was poorly controlled with adalimumab (previously 5-ASA and 6-MP). Exam was notable for hypoxia. PFTs showed severe obstruction and air trapping (FEV1 27% predicted, FVC 53%, ratio 51%, DLCO 61%, RV 167%). CTA chest did not show emphysema or VTE but suggested OB. Surgical lung biopsy confirmed this and showed thick-walled pulmonary arteries with thrombus in two lobes. V/Q scan and venous Doppler ultrasound were negative. Echocardiogram was negative for pulmonary hypertension. Oral steroids were started. Symptoms improved, diffusing capacity normalized, and oxygen requirements resolved. Steroid taper initially failed. Infliximab was started with improvement to both UC and pulmonary symptoms, now nearly off steroids with continued improvement.

DISCUSSION: Obliterative bronchiolitis is a rare complication of IBD with limited treatments. Our patient was steroid responsive and improved rapidly on infliximab. Animal models link TNF-α activity and OB, but there are limited studies showing efficacy of TNF-α inhibitors to treat lung disease, while others show infliximab can cause pneumonitis. In our case, improvement correlated with UC symptom control with infliximab. The pulmonary thrombosis on surgical biopsy was unexpected. There are no guidelines to direct therapy of in situ pulmonary thrombosis without other evidence of VTE. In our case, correction of the hypoxic low flow state caused by OB resulted in improvement to diffusing capacity and symptoms without anticoagulation.

CONCLUSIONS: Recognition of UC-associated pulmonary complications can greatly affect patient management. Controlling UC inflammatory symptoms is essential to treating UC-associated OB. If pulmonary symptoms persist despite control of UC, lung transplantation may be required.

Reference #1: Alho H et al. Transplantation 2003;76(3):516

Reference #2: Weatherhead M et al. Inflam bowel diseases 2006;12(5):427

DISCLOSURE: The following authors have nothing to disclose: Russell Buhr, Corinne Sheth

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