Hughes-Stovin Syndrome: a case report and review of the literature

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Abstract

Background

Hughes-Stovin syndrome is a rare entity. The aetiology of Hughes-Stovin syndrome is still unknown and the natural course of the illness is usually fatal; however it is supposed to be a clinical variant manifestation of Behçet disease.

Case presentation

We report the case of an 18 years old, greek male patient with Hughes-Stovin syndrome, who initially presented with deep vein thrombosis. There were no findings consistent with Behçet disease and the haemoptysis was treated successfully with methylprednisolone. Pathogenesis, imaging investigation and treatment of this syndrome are also briefly discussed.

Conclusion

In young men presenting with venous thrombosis as revealed on imaging examination, with platelet count and coagulation tests within normal and hemoptysis the eventuality of Hughes-Stovin syndrome is to be considered.

Related collections

Most cited references 17

Record: found
Abstract: not found
Article: not found

Behçet's disease.

T Sakane, M Takeno, N. Suzuki … (1999)

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Behçet's disease.

J B Michelson, F V Chisari (1982)

Behçet's disease is characterized by three primary components: iridocyclitis (historically with hypopyon), aphthous lesions in the mouth, and ulceration of the genitalia. Erythema nodosum, arthropathy and thrombophlebitis often accompany these manifestations, but the ocular symptoms may be the most important and serious manifestations of the disease. Central nervous system involvement, most often due to necrotizing vasculitis, may be the most protean manifestation of the disease, leading to death. The frequency of ocular manifestations is 70-85% in patients with the disease; the underlying disease mechanism in all organ systems is an occlusive vasculitis. Although the most common ocular symptom is that of anterior uveitis, often with hypopyon as a very late sign, the presence of necrotizing retinal vascular lesions is well known and often obscured by the severity of the anterior reaction. Definitions, incidence, clinical characteristics, differential diagnosis, and management of Behçet's ocular disease are discussed, as are the interrelationships of the different organ manifestations. The ophthalmologist should be familiar with the full spectrum of disease presentation since he or she may be the first physician to encounter the Behçet's patient.

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Bronchial and nonbronchial systemic arteries at multi-detector row CT angiography: comparison with conventional angiography.

Martine Remy-Jardin, Philippe Dumont, N Bouaziz … (2004)

To retrospectively evaluate bronchial and nonbronchial systemic arteries at multi-detector row helical computed tomography (CT) compared with conventional angiography in patients undergoing endovascular treatment of hemoptysis. Neither institutional board approval nor informed consent was required. Forty-eight consecutive patients (39 men, nine women; mean age, 55.7 years; range, 20-82 years) with hemoptysis of bronchial and nonbronchial systemic artery origin underwent multi-detector row helical CT angiography of the thorax with use of a four-detector row (n = 31) or 16-detector row (n = 17) scanner prior to embolization. Findings on CT angiograms, including CT scans, maximum intensity projections, and three-dimensional volume-rendered images, were used to evaluate the depiction of bronchial and nonbronchial systemic arteries. Retrospective analysis of the ostium and the course of bronchial and/or nonbronchial systemic arteries on CT angiograms enabled evaluation of the accuracy of this technique in identification of the relevant vasculature. Among the 46 patients initially treated with bronchial artery embolization, 58 bronchial arteries were identified at CT and/or angiography. In 50 (86%) cases, concordant findings were observed with both modalities. In five (9%) cases, CT could not be used to identify the ostia of bronchial arteries. In three (5%) cases, CT depicted bronchial arteries that could not be selectively catheterized. Three-dimensional images were found to be superior to transverse CT scans in depicting the ectopic origin of the bronchial arteries, which enabled the interventional radiologists to perform successful embolization after direct catherization of the ectopic vessel in every case. In five (11%) patients, the nonbronchial systemic origin of bronchial bleeding was identified on CT angiograms. Multi-detector row helical CT angiography provides more precise depiction of bronchial and nonbronchial systemic arteries than does conventional angiography. (c) RSNA, 2004.

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Author and article information

Journal

Journal ID (nlm-ta): Cases J

Title: Cases Journal

Publisher: BioMed Central

ISSN (Electronic): 1757-1626

Publication date Collection: 2009

Publication date (Electronic): 29 January 2009

Volume: 2

Page: 98

Affiliations

[1 ]Alexandra' University Hospital, Department of Radiology, Vas Sofias ave 80, 11528, Athens, Greece

[2 ]401 Army General Hospital, Department of Radiology, Mesogeion ave 138, 115 25 Athens, Greece

Article

Publisher ID: 1757-1626-2-98

DOI: 10.1186/1757-1626-2-98

PMC ID: 2649053

PubMed ID: 19178695

SO-VID: c2ef69c4-01c0-4fd8-baee-3aee6acac036

License:

This is an Open Access article distributed under the terms of the Creative Commons Attribution License ( http://creativecommons.org/licenses/by/2.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

Hughes-Stovin Syndrome: a case report and review of the literature

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Abstract

Background

Case presentation

Conclusion

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Most cited references 17

Behçet's disease.

Behçet's disease.

Bronchial and nonbronchial systemic arteries at multi-detector row CT angiography: comparison with conventional angiography.

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