Multiple pulmonary arterial aneurysms in Behcet's disease and Hughes-Stovin syndrome

Durieux, Pierre; Blétry, O; Huchon, G; Wechsler, B; Chrétien, J; Godeau, P

doi:10.1016/0002-9343(81)90245-x

Cited by 144 publications

(80 citation statements)

References 17 publications

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“…For the cases of massive hemoptysis due to large pulmonary aneurysms or those with lesions confined to one segment or one lung (as of our case), surgical removal of the aneurysm, interventional closure, lobectomy or pneumonectomy can be carried out based on the data from published case reports [2]. Durieux et al [21] used surgical intervention for three cases of HSS who had isolated pulmonary aneurysms and none of them showed any signs of recurrence on subsequent follow-ups.…”

Section: Discussionmentioning

confidence: 99%

Successful closure of pulmonary artery aneurysm in a patient with Hughes-Stovin syndrome

Hajsadeghi¹,

Naghshin²,

Nikfam³

et al. 2017

Acta Angiologica

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Section: Discussionmentioning

confidence: 99%

Successful closure of pulmonary artery aneurysm in a patient with Hughes-Stovin syndrome

Hajsadeghi¹,

Naghshin²,

Nikfam³

et al. 2017

Acta Angiologica

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“…Respecto el uso de anticoagulantes, están contraindicados por alto riesgo de hemorragia fatal, especialmente hemoptisis masiva, por rotura de los aneurismas pulmonares 3 .…”

Section: Discussionunclassified

“…Se caracteriza por presentar aneurismas de las arterias pulmonares y trombosis venosa profunda. Esta asociación también se ha descrito en la enfermedad de Behçet (EB), sugiriendo algunas publicaciones 2,3 que el SHS puede ser una forma de presentación de EB. El cuadro clínico habitualmente presenta fiebre recurrente, tos y hemoptisis 3 , junto con hipertensión endocraneana 4 .…”

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Síndrome Hughes-Stovin: Caso clínico

Escobar

Vega

et al. 2013

Rev. méd. Chile

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“…Histologically the aneurysms may resemble mycotic aneurysms, but no infectious agent has been identified in any case to date. It has been suggested that Hughes-Stovin syndrome may be a variant of Behçet's disease (3). Their clinical presentations can overlap significantly; both are more common in young men, the pulmonary manifestations of the 2 can be identical, and the histology of the aneurysms in both entities can be similar (1,4).…”

Section: Discussionmentioning

confidence: 99%

Radiologic vignette: Hughes‐Stovin syndrome

Fischer¹,

Korman²,

West³

2005

Arthritis & Rheumatism

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A 49-year-old African American man with a medical history notable for long-standing systemic hypertension and deep vein thrombus of the lower extremities 1 year earlier presented with recurrent hemoptysis. There was no history of antecedent fever, weight loss, fatigue, headache, chest pain, dyspnea, leg edema, arthralgias, rash, mucosal ulcerations, or ocular disease. He was not receiving antiplatelet or anticoagulant therapy. Physical examination revealed stable vital signs and coarse breath sounds over bilateral lung fields. Coagulation study findings were normal, there was no leukocytosis, and the erythrocyte sedimentation rate was normal. Radiologic findingsPosteroanterior and lateral radiographs of the chest showed an ill-defined 4.5-cm mass in the basilar portion of the left lower lobe and a 3.2-cm mass lateral to the upper portion of the right hilum. Contrast-enhanced computed tomography of the thorax was notable for multiple nodular densities throughout both lung fields, the largest of which was located in the left upper lobe (see arrow in Figure 1). Gadolinium-enhanced magnetic resonance imaging with magnetic resonance angiography of the chest revealed a saccular aneurysm in the left upper lobe (see thin arrow in Figure 2). A fusiform aneurysm was identified in the left lower lobe (see thick arrow in Figure 2). A tiny saccular aneurysm was seen in the right lower lobe. In addition, multiple venous occlusions were noted in the superior vena cava and the left brachiocephalic vein.A diagnosis of Hughes-Stovin syndrome was made. Clinical courseInitial treatment consisted of intravenous methylprednisolone (1 gm/day for 3 days) and oral cyclophosphamide (1.5 mg/kg/day). The patient's hemoptysis resolved and he was discharged on a tapering dose of prednisone and daily oral cyclophosphamide. Seven months later, the patient had another episode of hemoptysis. Pulmonary angiography was performed for consideration of embolization therapy. Angiography failed to reveal any of the previously identified pulmonary aneurysms. Instead, several segmental defects were present peripherally in both lungs, consistent with healing of multiple aneurysms. Two years later, gadolinium-enhanced magnetic resonance angiography revealed complete resolution of the multiple pulmonary aneurysms (see Figure 3). In addition, no new areas of aneurysmal dilatation were noted. DiscussionAneurysms of the pulmonary arteries are rare (1). Underlying risk factors for pulmonary artery aneurysms (PAAs) include infection, structural cardiac abnormalities, structural vascular abnormalities, and pulmonary hypertension (Table 1). In many cases, PAAs appear to have resulted from the interplay among several of these factors (1). There are 2 idiopathic syndromes associated with PAAs: Behçet's disease and Hughes-Stovin syndrome (1-3). Behçet's disease is a multisystem inflammatory disease of unknown etiology (2,4,5). Patients with Behçet's disease often present with vascular, cutaneous, pulmonary, neurologic, rheumatologic, gastrointestinal, and genitourina...

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Multiple pulmonary arterial aneurysms in Behcet's disease and Hughes-Stovin syndrome

Cited by 144 publications

References 17 publications

Successful closure of pulmonary artery aneurysm in a patient with Hughes-Stovin syndrome

Successful closure of pulmonary artery aneurysm in a patient with Hughes-Stovin syndrome

Síndrome Hughes-Stovin: Caso clínico

Radiologic vignette: Hughes‐Stovin syndrome

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