Hughes–Stovin syndrome: an uncommon cause of pulmonary aneurysms

Valdés-Corona, Luis F; Kimura‐Hayama, Eric; Mendez-Cano, Victor; Hernández-Villegas, Carlos Antonio; Hernández-Molina, Gabriela

doi:10.1093/rheumatology/kez663

Cited by 4 publications

(3 citation statements)

References 1 publication

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“…A total of 40 patients were analyzed. 3 4 5 6 7 8 9 10 11 12 13 14 15 16 17 18 19 20 21 22 23 24 25 26 27 28 29 30 31 32 33 34 35 36 37 38 39 40 41 42 43 44 45 46 47 48 49 50 51 52 53 54 55 56 57 58 59 The comparison of surgical/medical or medical alone therapy on length of follow-up is reported in Fig 3 . No differences in survival rate were observed between the two groups.…”

Section: Discussionmentioning

confidence: 99%

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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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Section: Discussionmentioning

confidence: 99%

“…The advent of endovascular procedures has expanded the possibilities for treatment. At the beginning, therapeutically options were represented by pneumectomy, 16 lobectomy, 3 4 5 9 25 37 50 52 and segmentectomy. 40 Aneurysm resection with saphenous vein replacement 9 or lung transplantation 10 were also proposed.…”

Section: Discussionmentioning

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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“…Other causes of PAAs and PAPAs include vasculitis, pulmonary hypertension, lung neoplasms, and iatrogenic factors such as cardiopulmonary surgery, radiotherapy, and percutaneous procedures [15,16]. Post-traumatic PAPAs due to direct or indirect trauma and iatrogenic pseudoaneurysm formation due to Swan-Ganz catheter mispositioning or repetitive traumatic injury of permanent venous or arterial catheters can also occur [17][18][19][20][21][22][23][24][25][26][27][28][29][30][31]. The presentation of PAPAs may be asymptomatic or, often, characterized by massive hemoptysis, dyspnea, and coughing, which can quickly evolve into hypovolemic hemorrhagic shock if not treated promptly [32][33][34][35][36][37][38][39][40][41][42][43].…”

Section: Introductionmentioning

confidence: 99%

Pulmonary Artery Pseudoaneurysms Embolization: Bicentric Experience and Review of the Literature

Fontana

Piacentino

Curti

et al. 2023

JCM

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The purpose of this bicentric case series was to report the safety, efficacy, and clinical outcome of transcatheter embolization in pulmonary artery pseudoaneurysms (PAPAs). Between January 2016 and June 2021, eight patients with PAPA were subjected to transcatheter embolization. The total number of patients was eight, of which five were female, with a mean age of 62 ± 14 years (average ± standard deviation). Etiology was traumatic in 2/8 cases and iatrogenic in 6/8 cases (after positioning a Swan-Ganz catheter in 5/6 cases and a temporary pacemaker in the latter case). In a single case, the PAPA was incidentally discovered during a routine X-ray, in the remaining 7 cases, the procedure was performed in emergency settings. PAPA embolization was performed using detachable coils alone in 3 cases; coils and glue in 1 case; coils, glue, and vascular plug in 1 case; coils and non-adhesive liquid embolic agents (Onyx and Squid respectively) in 2 cases; and non-adhesive liquid embolic agent alone (Onyx) in 1 case. No peri-procedural or post-procedural complications were recorded. Both the technical and clinical success rates were 100.0%. In conclusion, endovascular embolization is a technically feasible and safe therapeutic option for patients with PAPAs.

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Case Report: HUGHES-STOVIN SYNDROME: A RARE ENTITY AND CHALLENGING THERAPEUTIC DECISION: CASE SERIES AND REVIEW OF THE LITERATURE

Mahjoub,

Kallala,

Hammami

et al. 2023

F1000Res

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Hughes-Stovin Syndrome is a systematic disorder generally revealed by deep venous thrombosis (DVT) and pulmonary artery (PA) aneurysms. Its pathogenesis is still unclear and there are no previously established diagnostic criteria or treatment guidelines. It is usually associated with Behçet disease. The aim of this study was to further enlighten this entity as it is often misdiagnosed. We report a case series of five patients with this rare pathology with a systematic literature review of all the previously reported cases.

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Hughes–Stovin syndrome: an uncommon cause of pulmonary aneurysms

Cited by 4 publications

References 1 publication

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

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

Pulmonary Artery Pseudoaneurysms Embolization: Bicentric Experience and Review of the Literature

Case Report: HUGHES-STOVIN SYNDROME: A RARE ENTITY AND CHALLENGING THERAPEUTIC DECISION: CASE SERIES AND REVIEW OF THE LITERATURE

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