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A Rare Cause of Hemoptysis in Patients with Behcet's Disease: Hughes-stovin Syndrome: About One Case

Received: 18 February 2020     Accepted: 28 February 2020     Published: 17 March 2020
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Abstract

Hughes-Stovin Syndrome (HSS) is a very rare condition characterized by pulmonary aneurysms and peripheral thrombophlebitis of lower limbs. The pathogenesis of Hughes Stovin Syndrome is unknown. It’s considered as a clinical variant of Behçet’s disease. The main clinical manifestation is haemoptysis and dyspnea. The management of HSS can either be medical or surgical. We report the case of a young patient of 25 years old followed for Bechet's disease, who presents for a medium abundance hemoptysis with a swelling of the lower limbs evolving for 20 days, the radiological investigations made it possible to pose the diagnosis of the Syndrome of Huges-Stovin showing thrombosed aneurysms of the pulmonary arteries with thrombophlebitis of the lower limbs, the patient benefited from an immunosuppressive treatment, the evolution was marked by a good clinical evolution.

Published in International Journal of Medical Imaging (Volume 8, Issue 1)
DOI 10.11648/j.ijmi.20200801.13
Page(s) 16-19
Creative Commons

This is an Open Access article, distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution and reproduction in any medium or format, provided the original work is properly cited.

Copyright

Copyright © The Author(s), 2020. Published by Science Publishing Group

Keywords

Behcet, Pulmonary Aneurysms, Thrombophlebitis, Hugues-stovin

References
[1] Hughes JP, Stovin PG: Segmental pulmonary artery aneurysms with peripheral venous thrombosis. Br J Dis Chest 1959, 53: 19-27.
[2] Hughes-Stovin Syndrome Umair Khalid and Taimur Saleem Orphanet Journal of Rare Diseases 2011, 6: 15
[3] 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: 531-4.
[4] 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.
[5] Kim JT, Oh TY, Chang WH: Rare case of multiple pulmonary artery aneurysms with caval thrombosis–Hughes-Stovin syndrome. Eur J Cardiothorac Surg 2007, 31: 561-2.
[6] Chalazonitis AN, Lachanis SB, Mitseas P, Argyriou P, Tzovara J, Porfyrides P, Sotiropoulou E, Ptohis N: Hughes-Stovin Syndrome: a case report and review of the literature. Cases J 2009, 2: 98.
[7] Farber HW, McDermott S et all Year-Old Woman with Recurrent Venous Thromboembolism and Pulmonary Artery Aneurysm. N Engl J Med. 2018 Apr 12; 378 (15): 1430-1438
[8] 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.
[9] 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.
[10] Kapsimali VD, Kanakis MA, Vaiopoulos GA, Kaklamanis PG: Etiopathogenesis of Behçet’s disease with emphasis on the role of immunological aberrations. Clin Rheumatol 2010, 29: 1211-6.
[11] Cruz VA, Muniz YA, Silva Torres PPT, et al. Síndrome de Hughes-Stovin. Rev Bras Reumatol. 2009; 49: 747–52.
[12] Khalid U, Saleem T. Hughes-Stovin syndrome. Orphanet J Rare Dis. 2011; 6: 15.
[13] Fernandes GL, Teixeira AA, Antón AGS, et al. Churg-Strauss syndrome: a case report. Radiol Bras. 2014; 47: 259–61.
[14] M.-P. Revel-Dubois, J. de Laveaucoupet. Echo-Doppler des thromboses veineuses Journal de Radiologie Volume 85, Issue 9, September 2004, Page 1208
[15] Erkan F, Kiyan E, Tunaci A: Pulmonary complications of Behçet’s disease. Clin Chest Med 2002, 23: 493-503.
[16] Balci NC, Semelka RC, Noone TC: Multiple pulmonary aneurysms secondary to Hughes-Stovin syndrome: demonstration by MRangiography. J Magn Reson Imaging 1998, 8: 1323-5.
[17] Ketchum ES, Zamanian RT, Fleischmann D: CT angiography of pulmonary artery aneurysms in Hughes-Stovin syndrome. AJR Am J Roentgenol 2005, 185: 330-2
[18] Herb S, Hetzel M, Hetzel J, Friedrich J, Weber J: An unusual case of Hughes-Stovin syndrome. Eur Respir J 1998, 11: 1191-3.
[19] Tsai CL, Lu TS, Tsai KC, Chen WJ: Hemoptysis ca used by Hughes-Stovin syndrome. Am J Emerg Med 2005, 23: 209-11.
[20] Voiriot G, Parrot A, Antoine M et All. Transcatheter embolotherapy of pulmonary artery aneurysms as emergency treatment of hemoptysis in Behcet patients: experience of a referral center and a review of the literature. Internal and Emergency Medecine. 2018 Jun; 13 (4): 491-500.
[21] Pelage JP, El Hajjam M et All. Pulmonary artery interventions: an overview Radiographics. 2005 Nov-Dec.
Cite This Article
  • APA Style

    Amine El Masloumi, Jean Frederic Adjimabou, Anass Chehboun, Badr Boutakioute, Meriem Ouali Idrissi, et al. (2020). A Rare Cause of Hemoptysis in Patients with Behcet's Disease: Hughes-stovin Syndrome: About One Case. International Journal of Medical Imaging, 8(1), 16-19. https://doi.org/10.11648/j.ijmi.20200801.13

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    ACS Style

    Amine El Masloumi; Jean Frederic Adjimabou; Anass Chehboun; Badr Boutakioute; Meriem Ouali Idrissi, et al. A Rare Cause of Hemoptysis in Patients with Behcet's Disease: Hughes-stovin Syndrome: About One Case. Int. J. Med. Imaging 2020, 8(1), 16-19. doi: 10.11648/j.ijmi.20200801.13

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    AMA Style

    Amine El Masloumi, Jean Frederic Adjimabou, Anass Chehboun, Badr Boutakioute, Meriem Ouali Idrissi, et al. A Rare Cause of Hemoptysis in Patients with Behcet's Disease: Hughes-stovin Syndrome: About One Case. Int J Med Imaging. 2020;8(1):16-19. doi: 10.11648/j.ijmi.20200801.13

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  • @article{10.11648/j.ijmi.20200801.13,
      author = {Amine El Masloumi and Jean Frederic Adjimabou and Anass Chehboun and Badr Boutakioute and Meriem Ouali Idrissi and Najat Cherif Idrissi El Ganouni},
      title = {A Rare Cause of Hemoptysis in Patients with Behcet's Disease: Hughes-stovin Syndrome: About One Case},
      journal = {International Journal of Medical Imaging},
      volume = {8},
      number = {1},
      pages = {16-19},
      doi = {10.11648/j.ijmi.20200801.13},
      url = {https://doi.org/10.11648/j.ijmi.20200801.13},
      eprint = {https://article.sciencepublishinggroup.com/pdf/10.11648.j.ijmi.20200801.13},
      abstract = {Hughes-Stovin Syndrome (HSS) is a very rare condition characterized by pulmonary aneurysms and peripheral thrombophlebitis of lower limbs. The pathogenesis of Hughes Stovin Syndrome is unknown. It’s considered as a clinical variant of Behçet’s disease. The main clinical manifestation is haemoptysis and dyspnea. The management of HSS can either be medical or surgical. We report the case of a young patient of 25 years old followed for Bechet's disease, who presents for a medium abundance hemoptysis with a swelling of the lower limbs evolving for 20 days, the radiological investigations made it possible to pose the diagnosis of the Syndrome of Huges-Stovin showing thrombosed aneurysms of the pulmonary arteries with thrombophlebitis of the lower limbs, the patient benefited from an immunosuppressive treatment, the evolution was marked by a good clinical evolution.},
     year = {2020}
    }
    

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    AU  - Amine El Masloumi
    AU  - Jean Frederic Adjimabou
    AU  - Anass Chehboun
    AU  - Badr Boutakioute
    AU  - Meriem Ouali Idrissi
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    T2  - International Journal of Medical Imaging
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    JO  - International Journal of Medical Imaging
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    PB  - Science Publishing Group
    SN  - 2330-832X
    UR  - https://doi.org/10.11648/j.ijmi.20200801.13
    AB  - Hughes-Stovin Syndrome (HSS) is a very rare condition characterized by pulmonary aneurysms and peripheral thrombophlebitis of lower limbs. The pathogenesis of Hughes Stovin Syndrome is unknown. It’s considered as a clinical variant of Behçet’s disease. The main clinical manifestation is haemoptysis and dyspnea. The management of HSS can either be medical or surgical. We report the case of a young patient of 25 years old followed for Bechet's disease, who presents for a medium abundance hemoptysis with a swelling of the lower limbs evolving for 20 days, the radiological investigations made it possible to pose the diagnosis of the Syndrome of Huges-Stovin showing thrombosed aneurysms of the pulmonary arteries with thrombophlebitis of the lower limbs, the patient benefited from an immunosuppressive treatment, the evolution was marked by a good clinical evolution.
    VL  - 8
    IS  - 1
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Author Information
  • Radiology Department, Arrazy Hospital, Mohammed VIth Teaching Center, Cadi Ayad University, Marrakesh, Morocco

  • Radiology Department, Arrazy Hospital, Mohammed VIth Teaching Center, Cadi Ayad University, Marrakesh, Morocco

  • Radiology Department, Arrazy Hospital, Mohammed VIth Teaching Center, Cadi Ayad University, Marrakesh, Morocco

  • Radiology Department, Arrazy Hospital, Mohammed VIth Teaching Center, Cadi Ayad University, Marrakesh, Morocco

  • Radiology Department, Arrazy Hospital, Mohammed VIth Teaching Center, Cadi Ayad University, Marrakesh, Morocco

  • Radiology Department, Arrazy Hospital, Mohammed VIth Teaching Center, Cadi Ayad University, Marrakesh, Morocco

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