Pulmonary arteriovenous fistula ruptured in an adolescent girl 1 week after her mother’s rupture: a report of a case

Mitsui, Suguru; Tauchi, Shunsuke; Tanaka, Yugo; Tobe, Satoshi

doi:10.1007/s11748-020-01410-6

Cited by 3 publications

(1 citation statement)

References 10 publications

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“…About 47%–80% of congenital PAVF also have hereditary hemorrhagic telangiectasia (HHT), and more than 20% of HHT patients develop PAVF. According to Curcao’s diagnostic criteria, a diagnosis of definite HHT is recognized if at least three of the following are present: spontaneous recurrent nosebleeds, mucocutaneous telangiectasia, visceral involvement, and an affected first-degree relative ( 6 , 7 ). Therefore, our patient cannot be diagnosed with HHT but can be diagnosed with congenital PAVF.…”

Section: Discussionmentioning

confidence: 99%

Bilateral Lung Transplantation for Congenital Pulmonary Arteriovenous Fistula with Intraoperative Venovenous ECMO Support: The First Case Report in China

et al. 2022

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Pulmonary arteriovenous fistula (PAVF) is a rare pulmonary vascular lesion, more than 80% of which is caused by congenital abnormal development of pulmonary capillaries. The incidence of PAVF ranges from 2/100,000 to 3/100,000, with no difference in the male and female ratio. Congenital PAVF is often associated with hereditary hemorrhagic telangiectasia (HHT). In this article, we report a patient with only congenital PAVF that was successfully treated by bilateral lung transplantation (BLT) with intraoperative venovenous extracorporeal membrane oxygenation (ECMO) support because both lungs have been affected by PAVF and secondary pulmonary hypertension. To the best of our knowledge, this is the first report of BLT for PAVF in China and the second report that explains the clinical course of a patient to receive BLT for congenital PAVF without HHT. Some investigators have proposed lung transplantation as a definitive treatment, but the results are controversial. On the basis of the current condition of this patient, we believe lung transplantation is a viable option for certain patients, but the long-term effect remains to be studied.

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Section: Discussionmentioning

confidence: 99%

Bilateral Lung Transplantation for Congenital Pulmonary Arteriovenous Fistula with Intraoperative Venovenous ECMO Support: The First Case Report in China

et al. 2022

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Rupture of pulmonary arteriovenous fistula in the setting of hereditary hemorrhagic teleangiectasia

Tan

Kong

2023

The American Journal of the Medical Sciences

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Massive hemothorax induced by pulmonary arteriovenous malformation rupture: a case report and literature review

Li,

Duan,

et al. 2024

J Cardiothorac Surg

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Background Pulmonary arteriovenous malformation (PAVM), also known as pulmonary arteriovenous fistula, is a rare vascular developmental anomaly. Most cases of PAVM are associated with hereditary hemorrhagic telangiectasia (HHT). Hemothorax associated with PAVM is even rarer, and management concerning this complication still challenges. Case presentation A 55-year-old man with sudden onset of dyspnea and chest pain was admitted to our hospital. He had a medical history of epistaxis, intraperitoneal germ cell tumor and PAVM. Chest unenhanced CT revealed the left-sided pleural effusion together with partial passive atelectasis and gradual increase at the interval of six days. Diagnostic thoracocentesis further revealed hemorrhagic effusion. CT angiography (CTA) showed tortuously dilated lumen of the left lower pulmonary artery and PAVM with the formation of aneurysm. Due to his family's refusal of surgery, the patient underwent transcatheter embolization therapy. However, the left pleural effusion did not significantly reduce and there was a slow drop in hemoglobin value even after interventional treatment, indicating the possibility of ongoing active bleeding. Eventually, the patient received lobectomy of the left lower lobe with a satisfactory outcome. Conclusions Massive hemothorax resulting from PAVM rupture into the pleural space can lead to fatal outcomes. CTA can accurately diagnose this pathologic condition. Transcatheter embolization is frequently used in the treatment of PAVM, but it may be challenging to achieve the desirable effect in patients with hemothorax. Combined with our case and literature review, direct radical surgery can lead to a successful outcome when PAVM complicated with hemothorax and a large diameter of the draining vein.

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Pulmonary arteriovenous fistula ruptured in an adolescent girl 1 week after her mother’s rupture: a report of a case

Cited by 3 publications

References 10 publications

Bilateral Lung Transplantation for Congenital Pulmonary Arteriovenous Fistula with Intraoperative Venovenous ECMO Support: The First Case Report in China

Bilateral Lung Transplantation for Congenital Pulmonary Arteriovenous Fistula with Intraoperative Venovenous ECMO Support: The First Case Report in China

Rupture of pulmonary arteriovenous fistula in the setting of hereditary hemorrhagic teleangiectasia

Massive hemothorax induced by pulmonary arteriovenous malformation rupture: a case report and literature review

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