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Unilateral pulmonary edema due to pulmonary venous obstruction from fibrosing mediastinitis

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Abstract

An unusual case of fibrosing mediastinitis with obstruction of the inferior and superior left pulmonary veins and severe narrowing of the right pulmonary artery, disclosed after unilateral pulmonary edema, is described. The 18-year-old male patient had a long history of cough, progressive dyspnea and recurrent hemoptysis and the possible diagnosis of “interstitial fibrosis” from a previous lung biopsy. The diagnosis and the pulmonary vessels involvement were suspected after right heart catheterization combined with transesophageal echocardiography and confirmed during urgent thoracotomy and at postmortem examination.

Introduction

Fibrosing mediastinitis is rarely met; moreover, vascular obstruction due to this process is an uncommon condition with involvement of the superior vena cava usually resulting in clinical manifestations. A few cases of pulmonary artery involvement have also been reported [1], [2], [3]; narrowing or even occlusion of the major pulmonary veins is one of the less commonly known and most insidious vascular complication of this disease, always followed by diagnostic difficulties [2], [4], [5], [6], [7], [8], [9]. We report on an 18-year-old male with both pulmonary artery and pulmonary veins obstruction, causing unilateral pulmonary edema, due to fibrosing mediastinitis surgically confirmed after a long history of symptoms and diagnostic procedures.

Section snippets

Case report

An 18-year-old white male was admitted to the Intensive Care Unit because of severe dyspnea, hypoxemia and hemoptysis. His past medical history started at 4 years of age when he was found to have a positive tuberculin skin test with a normal chest X-ray. He was given isoniazide and rifampicine for 1 year. From the age of 9 onwards he experienced recurrent episodes of upper respiratory track “infections” with non-productive cough and wheezing which was felt to be consistent with “asthma”. Two

Discussion

Fibrosing mediastinitis is a rare clinical entity, the manifestations of which depend upon the structures of the mediastinum involved. Vascular obstruction due to fibrosing mediastinitis is an uncommon condition; superior vena cava obstruction is the most commonly reported as it is easily diagnosed. Pulmonary artery compression has also been reported in a few patients since the first report [3]; pulmonary vein occlusion is rare and the most insidious manifestation. Clinical manifestation is

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