English Polski
Vol 23, No 1 (2017)
Case report
Published online: 2017-05-12

open access

Page views 1026
Article views/downloads 1526
Get Citation

Connect on Social Media

Connect on Social Media

Successful closure of pulmonary artery aneurysm in a patient with Hughes-Stovin syndrome

Shokoufeh Hajsadeghi, Roozbeh Naghshin, Mehrdad Nikfam, Ali Pourmojarab, Sayfollah Abdi
Acta Angiologica 2017;23(1):20-24.

Abstract

Hughes-Stovin syndrome is a syndrome of unknown etiology characterized by thrombophlebitis and pulmonary aneurysms. Less than 40 cases have ever been reported in the English medical literature. We are reporting a 38-year-old man with a history of right ventricular thrombus and also deep vein thrombosis who presented with massive hemoptysis. Initial work-up revealed a round opacity on chest roentgenogram which later was diagnosed as a pulmonary artery aneurysm on subsequent imaging studies. Based on the history of thrombophlebitis with this newly diagnosed pulmonary artery aneurysm a diagnosis of Hughes-Stovin syndrome was made. The patient was treated with endovascular coiling of the aneurysm. A sixteen-month follow up was uneventful. To our knowledge, this is the first case of HSS ever reported from Iran and the fourth to have a cardiac chambers involvement in the course of the disease.

References

  1. Huhhes JP, Stovin PG. Segmental pulmonary artery aneurysms with peripheral venous thrombosis. Br J Dis Chest. 1959; 53(1): 19–27.
  2. Khalid U, Saleem T. Hughes-Stovin syndrome. Orphanet J Rare Dis. 2011; 6: 15.
  3. Reimold WV, Emmrich J, Harmjanz D, et al. [Multiple aneurysms of the pulmonary artery following recurrent septic pulmonary embolism (Hughes-Stovin syndrome). Report of 1 case]. Arch Klin Med. 1968; 215(1): 1–18.
  4. Khalil A, Parrot A, Fartoukh M, et al. Images in cardiovascular medicine. Large pulmonary artery aneurysm rupture in Hughes-Stovin syndrome: multidetector computed tomography pattern and endovascular treatment. Circulation. 2006; 114(10): e380–e381.
  5. Grembiale DR, Calderazzo M, Pelaia G, et al. A case of Hughes-Stovin syndrome associated with Budd-Chiari syndrome. Case Rep Clin Pract Rev. 2002; 3: 92–94.
  6. Curci N, Bilfinger T. Successful multimodality treatment of a young woman with HughesStovin syndrome. Am Surg. 2012; 78(5): 304–305.
  7. Huzjan Korunic R, Vukelic Markovic M, Tekavec Trkanjec J, et al. Hughes-Stovin Syndrome: a case report of man with hemoptysis due to pulmonary and bronchial arterial aneurysms. ESTI. 2014: P-0019.
  8. El Aoud S, Frikha F, Snoussi M, et al. Moderate hemoptysis caused by hughes-stovin syndrome. Clin Pract. 2014; 4(3): 647.
  9. Ammann ME, Karnel F, Olbert F, et al. Radiologic findings in the diagnosis of Hughes-Stovin syndrome. AJR Am J Roentgenol. 1991; 157(6): 1353–1354.
  10. Roberts DH, Jimenez JF, Golladay ES. Multiple pulmonary artery aneurysms and peripheral venous thromboses--the Hughes Stovin syndrome. Report of a case in a 12-year-old boy and a review of the literature. Pediatr Radiol. 1982; 12(4): 214–216.
  11. Charlton RW, Du Plessis LA. Multiple pulmonary artery aneurysms. Thorax. 1961; 16: 364–371.
  12. Choh NA, Jehangir M, Mir KM, et al. Hughes-Stovin syndrome: A rare cause of hemoptysis. Lung India. 2011; 28(4): 285–286.
  13. KOPP W. Pulmonary Artery Aneurysms with Recurrent Thrombophlebitis. Annals of Internal Medicine. 1962; 56(1): 105.
  14. François MF. Is Hughes-Stovin syndrome a particular expression of Behçet's disease? Chest. 1983; 83(2): 288.
  15. Chalazonitis AN, Lachanis SB, Mitseas P, et al. Hughes-Stovin syndrome: a case report and review of the literature. Cases J. 2009; 2: 98.
  16. Al-Jahdali H. Massive hemoptysis and deep venous thrombosis presenting in a woman with Hughes-Stovin syndrome: a case report. J Med Case Rep. 2010; 4: 109.
  17. Mahlo HR, Elsner K, Rieber A, et al. New approach in the diagnosis of and therapy for Hughes-Stovin syndrome. AJR Am J Roentgenol. 1996; 167(3): 817–818.
  18. Weintraub JL, DeMayo R, Haskal ZJ, et al. SCVIR annual meeting film panel session: diagnosis and discussion of case 1: Hughes-Stovin syndrome. J Vasc Interv Radiol. 2001; 12(4): 531–534.
  19. Erkan F. Rare diseases bullet 12: Pulmonary manifestations of Behcet's disease. Thorax. 2001; 56(7): 572–578.
  20. Gortani G, Starc M, Tubaro M. Successful treatment of refractory hughes stovin syndrome with infliximab. Pediatric Rheumatology. 2014; 12(Suppl 1): P352.
  21. Durieux P, Bletry O, Huchon G, et al. Multiple pulmonary arterial aneurysms in Behcet's disease and Hughes-Stovin syndrome. Am J Med. 1981; 71(4): 736–741.