INTRODUCTION:Pulmonary artery (PA) stenosis is an infrequent complication of histoplasmosis induced fibrosing mediastinitis (FM). Despite various forms of pharmacologic treatment and surgical interventions most patients with PA stenosis succumb to right heart failure secondary to severe pulmonary hypertension (1). Recent evidence suggests that percutaneous endovascular stent placement can effectively alleviate PA stenosis in these patients (2). In-stent restenosis, however, appears to limit their long-term durability (1). With the advent of cutting balloon angioplasty limiting coronary restenosis and it’s proven success in treating pediatric PA stenosis, we attempted PA rehabilitation using such a device.
CASE PRESENTATION:A 41-year-old white female with previously treated histoplasmosis presented with increasing dyspnea, cough and pleuritic chest pain and was subsequently diagnosed with community acquired pneumonia. A chest roentegram revealed a large right perihilar mass. This was confirmed with chest computer tomography and revealed near complete occlusion of the right pulmonary artery. Positron emission tomography was obtained and depicted increased metabolic activity in the mediastinal region. Mediastinoscopy revealed extensive fibrosis while histological examination confirmed FM. Subsequently, cardiac catheterization was performed which revealed severe right PA stenosis, moderate pulmonary hypertension and right ventricular diastolic dysfunction. Successful right PA rehabilitation using cutting balloon angioplasty and stent insertion was performed. Currently, one year after the procedure, the right PA is still patent.
DISCUSSIONS:FM is a rare chronic complication of infection with Histoplasma capsulatum, occurring years to decades after exposure. Furthermore, there are sparse reports of FM induced PA stenosis. Traditionally, medical and surgical treatment for PA disease has been ineffective with mortality rates approaching 50% when both PA’s are involved in symptomatic patients. Percutaneous stent placement with balloon angioplasty has recently provided an effective technique for alleviating such stenoses (2). Unfortunately, in-stent restenosis limits the long term durability of this procedure. A recent randomized control study provides evidence that bare metal stenting (BMS) preceded by cutting balloon angioplasty is superior to standard angioplasty and subsequent BMS in preventing coronary restenosis. It is therefore conceivable that less PA in-stent restenosis could occur after cutting balloon PA rehabilitation. Regrettably, such data for PA stenosis in adults is currently not available. Additionally, dilation of congenital PA stenosis with cutting balloons has been used with success in the pediatric population. Due to the severe stenosis encountered in this case such a catheter was utilized with a relatively low inflation pressure of 8 atmospheres.
CONCLUSION:To our knowledge this is the first reported case of successful PA rehabilitation in an adult patient with histoplasmosis induced FM utilizing cutting balloon angioplasty with subsequent stent insertion.
DISCLOSURE:Steven Kadiev, No Financial Disclosure Information; No Product/Research Disclosure Information