A case report : Hughes-Stovin Syndrome

Authors

  • สุภัค ชุติไพจิตร Suratthani Hospital

Keywords:

Hughes-Stovin syndrome, Behcet’s disease, massive hemop tysis, thrombophlebitis, multiple pulmonary and/or bronchial aneu rysms, computed tomography of the chest

Abstract

              Hughes-Stovin Syndrome (HSS) is a very rare clinical disorder characterized by thrombophlebitis and multiple pulmonary and/or bronchial aneurysms. The exact etiology and pathogenesis of HSS is unknown; possible causes include infections and angiodysplasia. HSS has also been considered as a variant of Behcet’s disease.
              A case report of young male patient, initially presented with cerebral venous sinus thrombosis. Later, he presented with massive hemoptysis. The computed tomography of the chest demonstrated multiple pulmonary aneurysms. Pathogenesis, computed tomography, surgical pathology and treatment of this syndrome are also briefly discussed.
              In young men presenting with venous thrombosis as revealed on imaging examination, with platelet count and coagulation tests within normal and hemoptysis the eventuality of HSS is to be considered.

References

1. Umair Khalid, Taimur Saleem. Hughes-Stovin Syndrome. Orphanet Journal of Rare Diseases 2011; 15 : 1-11.

2. Hughes JP, Stovin PG: Segmental pulmonary artery aneurysms with peripheral venous thrombosis. Br J Dis Chest 1959 ; 53 : 19-27.

3. Kopp WL, Green RA: Pulmonary artery aneurysms with recurrent thrombo phlebitis: the “Hughes-Stovin syndrome”. Ann Intern Med 1962; 56 :105-14.

4. Reimold WV, Emmrich J, Harmjanz D, Kochsiek K: Multiple aneurysms of the pulmonary artery following recurrent septic pulmonary embolism (Hughes-Stovin syndrome): report of 1 case. Arch Klin Med 1968; 215 : 1-18.

5. Khalil A, Parrot A, Fartoukh M, Marsault C, Carette MF: Images in cardiovascular medicine. Large pulmonary artery aneurysm rupture in Hughes-Stovin syndrome: multidetector computed tomography pattern and endovascular treatment. Circulation 2006; 114 :e380-1.

6. Al-Jahdali H: Massive hemoptysis and deep venous thrombosis presenting in a woman with Hughes-Stovin syndrome: a case report. J Med Case Reports 2010; 4 : 109.

7. Mahlo HR, Elsner K, Rieber A, Brambs HJ: New approach in the diagnosis of and therapy for Hughes-Stovin syndrome. AJR Am J Roentgenol 1996 ;167 : 817-8.

8. Athanasios N Chalazonitis, Stefanos B Lachanis, Panagiotis Mitseas, Panagiotis Argyriou, Joannie Tzovara, Petros Porfyrides, Evangelia Sotiropoulou, Nikolaos Ptohis. Hughes-Stovin Syndrome: a case report and review of the literature. Cases Journal 2009; 2 : 98.

9. Weintraub JL, DeMayo R, Haskal ZJ, Susman J. SCVIR Annual Meeting Film Panel Session: Diagnosis and Discussion of Case 1. J Vasc Interv Radiol 2001; 12 : 531-534.

10. 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 :
736-41.

11. Eric S. Ketchum, Roham T. Zamanian, Dominik Fleischmann. CT Angiography of Pulmonary Artery An eurysms in Hughes-Stovin Syndrome. AJR 2005 ; 185 : 330–332.

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Published

2019-03-01

How to Cite

ชุติไพจิตร ส. (2019). A case report : Hughes-Stovin Syndrome. Region 11 Medical Journal, 33(1), 35–44. Retrieved from https://he02.tci-thaijo.org/index.php/Reg11MedJ/article/view/188131

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Section

Case Report