Recurrent Pulmonary Aneurysms

Bennji, Sami; Preez, Leonard du; Griffith‐Richards, Stephanie; Smit, Derrick; Rigby, Jonathan; Koegelenberg, Coenraad F.N.; Irusen, Elvis; Allwood, Brian

doi:10.1016/j.chest.2017.07.015

Cited by 21 publications

(12 citation statements)

References 4 publications

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“…14 - 16 Also, extrapulmonary arterial involvement in HSS has been previously reported in the splenic artery, 17 internal iliac artery aneurysm, 18 , 19 and femoral arteries branches. 12 Optic neuritis, 20 as seen, 2 of our patients, and pan-uveitis, 21 have been previously reported in HSS. Overlapping features between HSS and BD have been previously reported in the literature.…”

Section: Discussionsupporting

confidence: 61%

Hughes-Stovin Syndrome: An Experience of Management of 3 Cases From a Tertiary Health Care Centre

Sachdev

Yadav

Balakrishnan

et al. 2023

Clin Med�Insights�Arthritis�Musculoskelet Disord

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Section: Discussionsupporting

confidence: 61%

Hughes-Stovin Syndrome: An Experience of Management of 3 Cases From a Tertiary Health Care Centre

Sachdev

Yadav

Balakrishnan

et al. 2023

Clin Med�Insights�Arthritis�Musculoskelet Disord

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“…We summarize the current management options of HSS. Table 1 Summary of the Hughes-Stovin syndrome (HSS) case reports sharing some features of the classic triad of Behçet's Disease (BD), e. g., mouth and/or genital ulcers and/or eye inflammation and other cases with HSS lacking any of the classical features of BD Summary of HSS case reports revealing some of the classical features of BD Kechida et al [14] Case 1: aortic aneurysm in a 55-year-old man who initially presented with deep venous thrombosis; the diagnosis of HSS revealing BD was made given the history of recurrent oral and genital ulcers Bennji et al [3] Case 2: multiple pulmonary aneurysms with life-threatening hemoptysis; pulmonary artery coil embolization and right lower lobectomy were performed; extensive bilateral femoral deep vein thrombosis extending into the inferior vena cava, massive hemoptysis. A final diagnosis of BD was made after extensive investigations El Jammal et al [15] Case 3: 19-year-old man with hemoptysis; tongue ulcers; CT angiography revealed femoral vein thrombosis, large threatening aneurysm of a left pulmonary artery segment.…”

Section: Discussionmentioning

confidence: 99%

“…The current consensus is that HSS results from a vasculitis similar to that in Behçet's disease (BD). Several investigators have suggested that HSS is actually a variant of BD rather than a discrete clinical entity [2][3][4]. However, the link between HSS and BD remains speculative because the etiologies of both disorders are still uncertain.…”

Section: Introductionmentioning

confidence: 99%

A case of Hughes–Stovin syndrome (incomplete Behçet’s disease) with extensive arterial involvement

et al. 2019

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A case of Hughes-Stovin syndrome (incomplete Behçet's disease) with extensive arterial involvement Unmasking the true face of a rare syndrome Robinson et al. [16] Case 4: A 21-year-old male; recurrent oral ulcers; no genital ulceration; superficial thrombophlebitis; pulmonary CT angiography a 35 mm right lateral segmental PAA with multiple pulmonary artery in situ thromboses Demirkan and Gültekin [17] Case 5, 6: two patients with HSS who presented with pulmonary artery aneurysm, thrombophlebitis, hemoptysis, and oral ulcers Al-Jahdali [18] Case 7: 23-year-old Saudi woman; recurrent oral ulceration; right-lower lobe PAA; papilledema; DVT Yagi et al. [19] Case 8: A 32-year-old male; multiple PAA; DVT of the right leg and the right femoral vein; thrombosis of the vena cava; aphthous ulcer in the oral cavity, an ulcer in the genital region Madiha and Sami [20] Case 9: DVT; oral ulcers; giant aneurysm in the left lower lobe pulmonary artery Summary of HSS case reports without features of the classic triad of BD Hughes and Stovin [1] Case 1, 2: two male patients; segmental PPA with peripheral venous thrombosis Kopp and Green [21] Case 3, 4: two male patients; PAA and recurrent thrombophlebitis Fabi et al. [22] Case 5: A 12-year-old boy; right atrium endocardial mass; jugular vein and cerebral venous thrombosis; deep venous thromboses; PAA Abdelbary et al. [23] Case 6: A 35-year-old Egyptian female lower limb deep vein thrombosis; pulmonary aneurysm Ribeiro et al. [24] Case 7: A 43-year-old male; superficial thrombophlebitis and DVT of the lower limbs; PAA Pankl et al. [25] Case 8: A 41-year-old man; deep venous thrombosis of the right leg, and PAA Al-Zeedy et al. [26] Case 9: A 53-year-old man; DVT and PAA Kably and Reveron [27] Case 10: A 41-year-old male; massive hemoptysis; ruptured PAA; DVT; cardio-venous thromboembolism; pulmonary infarction El Aoud et al. [28] Case 11: A 42-year-old woman; DVT; PAA Jaramillo et al. [29] Case 12: A 47-year-old male; dilated main pulmonary arteries, multiple right bronchial artery aneurysms and a splenic artery aneurysm Silva et al. [30] Case 13: A 25-year-old male; DVT; PAA Grembiale et al. [31] Case 14: DVT; PAA; Budd-Chiari syndrome; a thrombotic occlusion of inferior vena cava Amezyane et al. [32] Case 15: A 28-year-old female; right ventricular thrombus, PAA, iliac vein thrombosis; caval thrombosis Kim et al. [7] Case 16: A 45-year-old man; massive hemoptysis; DVT; bilateral PAA and inferior vena caval thrombosis Chalazonitis et al. [9] Case 17: A 18-year-old, Greek male patient; DVT; PAA; superior sagittal and transverse sinuses Emad et al. [2] Case 18, 19: two male patients; DVT, PAA, superior sagittal sinus thrombosis in one case Balci et al. [33] Case 20: A 41-year-old patient; multiple PAA; thrombus in both the inferior and superior vena cavae Herb et al. [34] Case 21: A 25-year-old man; PAA; multiple aneurysms of the bronchial arteries; severe hemoptysis; aneurysm of the left hepatic artery Margolesky et al. [35] Case 22: A 38-year-old woman; DVT, PAA; right ventricle th...

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“…More recently, embolization, even with repeated procedures, have been performed, with good immediate and mid-term results (See ►Supplementary Materials 1-3). [13][14][15]22,28,33,37,42 The simultaneous presence of phlebitis and aneurysms makes the subsequent therapeutic path difficult because of the hemorrhagic risk associated with oral anticoagulation. In our case, the association between deep vein thrombosis and the simultaneous presence of a contralateral pulmonary aneurysm was addressed using a cava filter, which thus reduces the risk of DVT embolism and at the same time protects the patient from complications of anticoagulant therapy and aneurysm bleeding.…”

Section: Therapeutically Optionsmentioning

confidence: 99%

A Rare but Fatal Behçet Variant: The Hughes–Stovin Syndrome—Successful Case Report and New Evidence from Literature Review

Ascoli Marchetti,

Belvivere,

Argirò

et al. 2023

Aorta (Stamford)

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Hughes–Stovin syndrome (HSS) is a rare potentially fatal vasculitis supposedly belonging to the spectrum of Behçet disease without ocular involvement. HSS tends to play by a temporal pattern, starting with thrombosis and followed by formation of pulmonary aneurysms. Since its mortality can reach 25% of cases, early recognition and appropriate therapy represent the major clinical challenges. We describe a rare case of HSS successfully treated via multidisciplinary management by an endovascular approach and immunosuppressive therapy.

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Recurrent Pulmonary Aneurysms

Cited by 21 publications

References 4 publications

Hughes-Stovin Syndrome: An Experience of Management of 3 Cases From a Tertiary Health Care Centre

Hughes-Stovin Syndrome: An Experience of Management of 3 Cases From a Tertiary Health Care Centre

A case of Hughes–Stovin syndrome (incomplete Behçet’s disease) with extensive arterial involvement

A Rare but Fatal Behçet Variant: The Hughes–Stovin Syndrome—Successful Case Report and New Evidence from Literature Review

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