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Anomalous Unilateral Single Pulmonary Vein Masquerading as a Pulmonary Arteriovenous Malformation

Guy's and St. Thomas' Hospital Trust London SE1 9RT, United Kingdom

A 38-year-old man was referred to our center after episodes of dyspnea on exertion. The initial chest radiograph (Fig. 4A) and chest CT showed a vascular malformation in the right lower lobe (Fig. 4B), suggestive of a pulmonary arteriovenous malformation, which can have a similar looplike appearance. However, the arterial blood gases on room air and hemoglobin level were normal, and the patient had no history of bleeding, systemic embolization, or clinical signs of Osler-Weber-Rendu disease. Selective pulmonary angiography, performed with a view to embolization of pulmonary arteriovenous malformation showed normal arterial anatomy. The venous phase displayed a single dilated tortuous vein forming a loop in the right lower lobe, draining the entire right lung into the left atrium (Fig. 4C). The diagnosis of an anomalous unilateral single pulmonary vein (AUSPV) was made. No associated arterial or bronchial abnormalities were present. The contralateral lung was normal. The subsequent transthoracic echocardiogram failed to reveal any associated cardiac abnormality. The dyspnea remained unexplained. No intervention or surgical correction is indicated in AUSPV.

View larger version (107K): [in this window] [in a new window] [as a PowerPoint slide]   Fig. 4A. —38-year-old man with dyspnea and anomalous unilateral single pulmonary vein (AUSPV). Chest radiograph shows vascular malformation at right heart border.

View larger version (137K): [in this window] [in a new window] [as a PowerPoint slide]   Fig. 4B. —38-year-old man with dyspnea and anomalous unilateral single pulmonary vein (AUSPV). Chest CT scan shows vascular malformation with ectasia and extreme tortuosity, suggesting pulmonary arteriovenous malformation.

View larger version (144K): [in this window] [in a new window] [as a PowerPoint slide]   Fig. 4C. —38-year-old man with dyspnea and anomalous unilateral single pulmonary vein (AUSPV). Pulmonary digital subtraction angiography (venous phase) shows that single dilated tortuous vein drains entire right lung into left atrium (AUSPV).

Anomalous single pulmonary veins with a normal connection to the left atrium are exceedingly rare. Confusion with other conditions such as scimitar syndrome, partial anomalous pulmonary venous return, pulmonary varices or venous aneurysms, and parenchymal pulmonary masses has been reported [1,2,3,4]. This is important because isolated AUSPV does not require treatment and thoracic surgeons should be aware of this anomaly before performing lung resection.

Single pulmonary veins can occur in association with other vascular or pulmonary malformations such as a contralateral partial anomalous venous connection, a bilateral AUSPV, or malformations of the bronchopulmonary airways and the related arterial blood supply. However, most commonly AUSPV is isolated, asymptomatic, and diagnosed accidentally. To our knowledge, this is the first case masquerading as a pulmonary arteriovenous malformation on CT.

Selective pulmonary angiography—the modality of choice for revealing the exact fistula site in pulmonary arteriovenous malformation—is diagnostic of this unusual abnormality, thereby avoiding any further interventional or surgical treatment. We recommend selective pulmonary angiography in the differential diagnosis of any pulmonary vascular malformation if cross-sectional findings indicate vascular intervention or surgery.

As seen in our patient, AUSPV can have a nonspecific clinical appearance. Therefore, an abnormal pathologic vascular shadow on a chest radiograph, even in the absence of arterial blood gas and hemoglobin abnormalities, should raise the suspicion of a congenital pulmonary vascular abnormality and initiate further imaging if indicated.

References

1. Benfield JR, Gots RE, Mills D. Anomalous single left pulmonary vein mimicking a parenchymal nodule. Chest 1971;59:101 -103[Abstract/Free Full Text]
2. Rey C, Vaksmann G, Francart C. Anomalous unilateral single pulmonary vein mimicking partial anomalous pulmonary venous return. Cathet Cardiovasc Diagn 1986;12:330 -333[Medline]
3. Hasuo K, Numaguchi Y, Kishikawa T, Ikeda J, Matsuura K. Anomalous unilateral single pulmonary vein mimicking pulmonary varices. Chest 1981;79:602 -604[Abstract/Free Full Text]
4. Kanemoto N, Sugiyama T, Hirose S, Goto Y. A case with pseudo-scimitar syndrome: "scimitar sign" with normal pulmonary venous drainage. Jpn Circ J 1987;51:642 -646[Medline]

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This Article Figures Only Full Text (PDF) Alert me when this article is cited Alert me if a correction is posted Services Email this article to a friend Similar articles in this journal Similar articles in PubMed Alert me to new issues of the journal Download to citation manager Citing Articles Citing Articles via Google Scholar Google Scholar Articles by Engelke, C. Articles by Reidy, J. F. Search for Related Content PubMed PubMed Citation Articles by Engelke, C. Articles by Reidy, J. F. Social Bookmarking                      What's this?