Hughes-Stovin syndrome: a case report and literature review

Authors

  • Imran Nazir Department of Internal Medicine Security Forces Hospital Makkah, KSA
  • Elham Hamed Alrubai Pulmonologist Security Forces Hospital Makkah, KSA
  • Muhammad Zafar Iqbal Department of Radiology, University Medical and Dental College, Faisalabad, Pakistan
  • Amani Abdulmoniem Ahmad Elimam Saudi Board-IM, Security Forces Hospital Makkah (SFHM), KSA
  • Amal Abdulmoniem Ahmad Elimam Saudi Board-IM, Security Forces Hospital Makkah (SFHM), KSA
  • Sadeen Essam Ezzat Saudi Board-IM, Security Forces Hospital Makkah (SFHM), KSA
  • Thekra A. Aseeri Saudi Board-IM, Security Forces Hospital Makkah (SFHM), KSA
  • Sara Salem Alqethami Saudi Board-IM, Security Forces Hospital Makkah (SFHM), KSA
  • Jumanah Faisal Algoufi Saudi Board-IM, Security Forces Hospital Makkah (SFHM), KSA
  • Sayed S. Rahman Nephrologist Security Forces Hospital Makkah (SFHM), KSA

DOI:

https://doi.org/10.18203/2320-6012.ijrms20250266

Keywords:

Hemoptysis, Pulmonary artery aneurysms, Huges-Stovin syndrome

Abstract

Hughes-Stovin syndrome (HSS) is a rare clinical entity comprised of thrombophlebitis and multiple pulmonary and bronchial aneurysms. Medical literature has documented fewer than 40 cases of HSS. Its pathogenesis and etiology are still unknown. It is considered a variant of Behcet’s disease (BD). HSS is being managed either medically or surgically. We report the case of a 32-year-old Saudi male patient with HSS who initially presented with a pulmonary embolism (PE). Later, he developed hemoptysis that was treated successfully with methylprednisolone. The genetic, etiologic, and pathologic basis of HSS should be explored more in the future to elucidate the disease. Exploration of better and optimal medical and surgical therapeutic regimens is a need for time to treat and prevent morbidity in BD and HSS. There is a dilemma of anticoagulation in these patients that needs to be addressed and investigated properly.

Metrics

Metrics Loading ...

References

Gupta M, Agrawal A, Iakovou A, Cohen S, Shah R, Talwar A. Pulmonary artery aneurysm: a review. Pulmonary Circulation. 2020;10(1):2045894020908780. DOI: https://doi.org/10.1177/2045894020908780

Kandathil A, Chamarthy M. Pulmonary vascular anatomy and anatomical variants. Cardiovasc Diagn Ther. 2018;8(3):201-07. DOI: https://doi.org/10.21037/cdt.2018.01.04

Samreen I, Darji P, Genobaga S, Doosetty S, Mohta T, Maity G, et al. Pulmonary Artery Aneurysm in Behcet Disease: Medical, Endovascular or Surgical Intervention. Cureus. 2023;15(11):e49368. DOI: https://doi.org/10.7759/cureus.49368

Bu C, Zhang M, Sun Q, Zhang H, Luo J, Liu Q, et al. Pulmonary artery aneurysm: computed tomography (CT) imaging findings and diagnosis. Quant Imaging Med Surg. 20241;14(8):6147-60. DOI: https://doi.org/10.21037/qims-24-462

Giannessi C, Smorchkova O, Cozzi D, Zantonelli G, Bertelli E, Moroni C, et al. Behçet’s Disease: A Radiological Review of Vascular and Parenchymal Pulmonary Involvement. Diagnostics. 2022;12(11):2868. DOI: https://doi.org/10.3390/diagnostics12112868

Park JH, Han MC, Bettmann MA. Arterial manifestations of Behcet disease. AJR. 1984;143:821-25. DOI: https://doi.org/10.2214/ajr.143.4.821

Chalazonitis AN, Lachanis SB, Mitseas P, Argyriou P, Tzovara J, Porfyrides P, et al. Hughes-Stovin Syndrome: a case report and review of literature. Cases J. 2009;(2):98. DOI: https://doi.org/10.1186/1757-1626-2-98

Sakane T, Takeno M, Suzuki N, Inaba G. Behcet's disease. N Engl J Med. 1999;341:1284. DOI: https://doi.org/10.1056/NEJM199910213411707

Tsai CL, Lu TS, Tsai KC, Chen WJ. Hemoptysis caused by Hughes-Stovin syndrome. Am J Emerg Med. 2005;23:209-11. DOI: https://doi.org/10.1016/j.ajem.2004.05.009

Balci NC, Semelka RC, Noone TC. Multiple pulmonary aneurysms secondary to Hughes-Stovin syndrome: demonstration by MR angiography. J Magn Reson Imaging. 1998;8(6):1323-5. DOI: https://doi.org/10.1002/jmri.1880080620

Ketchum E, Zamanian R, Fleischmann D. CT Angiography of Pulmonary Artery Aneurysms in Hughes-Stovin Syndrome. AJR. 2005;185:330-32. DOI: https://doi.org/10.2214/ajr.185.2.01850330

Remy-Jardin M, Bouaziz N, Dumont P, Brillet PY, Bruzzi J, Remy J. Bronchial and Nonbronchial Systemic Arteries at Multi-Detector Row CT Angiography: Comparison with Conventional Angiography. Radiology. 2004;233:741-9. DOI: https://doi.org/10.1148/radiol.2333040031

Mahlo HR, Elsner K, Rieber A. New approach in the diagnosis of and therapy for Hughes-Stovin syndrome. Am J Roentgenol. 1996;167:817-8. DOI: https://doi.org/10.2214/ajr.167.3.8751710

Lee J, Noh JW, Hwang JW, Kim H, Ahnn JK, Koh EM, et al. Successful cyclophosphamide therapy with complete resolution of pulmonary artery aneurysm in Hughes Stovin syndrome patient. Clin Rheumatol. 2008;27(11):1455-58. DOI: https://doi.org/10.1007/s10067-008-0951-8

Downloads

Published

2025-01-30

How to Cite

Nazir, I., Alrubai, E. H., Iqbal, M. Z., Ahmad Elimam, A. A., Ahmad Elimam, A. A., Ezzat, S. E., Aseeri, T. A., Alqethami, S. S., Algoufi, J. F., & Rahman , S. S. (2025). Hughes-Stovin syndrome: a case report and literature review. International Journal of Research in Medical Sciences, 13(2), 850–852. https://doi.org/10.18203/2320-6012.ijrms20250266

Issue

Section

Case Reports