Abstract

IntroductionHughes-Stovin syndrome is a rare condition characterized by peripheral deep venous thrombosis accompanied by single or multiple pulmonary arterial aneurysms. The limited number of cases has precluded controlled studies of the management of pulmonary artery aneurysms, which usually cause massive hemoptysis leading to death. This is the first report of a new endovascular treatment of a single large pulmonary arterial aneurysm.Case presentationAn 18-year-old Caucasian man was referred to our department with recurrent severe hemoptysis. His medical history included Hughes-Stovin syndrome diagnosed during a recent hospital admission. The patient was initially treated with corticosteroids. Because of his recurrent hemoptysis, we decided to embolize a 3.5 cm pulmonary arterial aneurysm using an Amplatzer Vascular Plug. The procedure was not complicated, and the patient's post-intervention course was uneventful. The patient has remained free from any complications of the embolization 36 months after the procedure.ConclusionPercutaneous embolization of a single large pulmonary artery aneurysm with an Amplatzer Vascular Plug in a patient with Hughes-Stovin syndrome is a less invasive procedure that represents the best multidisciplinary approach in treating these patients.

Highlights

  • Hughes-Stovin syndrome is a rare condition characterized by peripheral deep venous thrombosis accompanied by single or multiple pulmonary arterial aneurysms

  • Hughes-Stovin syndrome is a rare condition first described in 1959 that is characterized by peripheral deep venous thrombosis (DVT) often involving the vena cava and accompanied by single or multiple pulmonary arterial aneurysms [1]

  • We describe a new endovascular treatment of a single large pulmonary arterial aneurysm in a patient with Hughes-Stovin

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Summary

Introduction

Hughes-Stovin syndrome is a rare condition first described in 1959 that is characterized by peripheral deep venous thrombosis (DVT) often involving the vena cava and accompanied by single or multiple pulmonary arterial aneurysms [1]. He had had some incidents of spontaneous non-productive cough several days prior to his admission During his previous admission three months before his current presentation, his physical examination was normal apart from mild, tender swelling of his left thigh and calf caused by DVT (left external iliac and femoral vein) detected by color duplex examination. The rare diagnosis of systemic vasculitis Hughes-Stovin syndrome was made on the basis of the pulmonary artery aneurysm and peripheral venous thromboses in such a young patient. His hemoptysis resolved with intravenous methylprednisolone treatment after about 25 days Upon his second admission, because of his recurrent hemoptysis, we decided to perform percutaneous embolization of the pulmonary artery aneurysm. The patient developed a new DVT in his lower extremities during follow-up (in the 30th month) after temporary discontinuation of anti-coagulant treatment He has not had any complications from the aneurysm 36 months after. Undergoing embolization, and follow-up CT showed the completely thrombosed pulmonary artery aneurysm

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