Abstract

Introduction: Hughes-Stovin syndrome (HSS) is a rare clinical disorder characterized by thrombo-phlebitis and multiple pulmonary aneurysms.  Most patients develop peripheral venous thrombosis.

Case presentation : A 27-year male presented with massive hemoptysis in the emergency department, work up done showed bilateral pulmonary artery aneurysm with signs of leakage and thrombosis. There was no venous involvement.  The choice of treatment was challenging since during the course in the ward since there was evidence of bilateral chronic deep venous thrombosis.  Patient underwent bronchial artery embolization. In addition, there were no other systemic manifestations thus the patient was diagnosed as Hughes-Stovin syndrome.  Pulse steroids and cyclophosphamide was given.  The patient received treatment in the form of intravenous pulse methylprednisolone 1 gm for 3 days then shifted to oral prednisone. The hemoptysis improved and was scheduled for monthly CYC infusion for 1 year . However on the 3rd month of CYC infusion, patient developed covid infection and after 3 week subsequently had another episode of massive hemoptysis which led to the demise of the patient.

Conclusion: HSS require aggressive immune suppressive treatment, however referral to interventional radiology for embolization and surgery can be attempted when indicated especially when presented with massive hemoptysis