Complex Pulmonary Arteriovenous Fistula Presented With Ventricular Tachycardia and Type 2 Acute Myocardial Infarction.

Pulmonary arteriovenous fistula (PAVF) is a rare congenital vascular malformation primarily manifested as dyspnea, migraine, ischemic stroke, hemoptysis, and nervous system complications. However, in our case, an 18-year-old male patient with PAVF presented with sudden onset of ventricular tachycardia and type 2 acute myocardial infarction as initial symptoms. A diagnosis was achieved through pulmonary artery computer tomography angiography (CTA) and three-dimensional (3D) computed tomography (CT) reconstruction, revealing a complex and giant PAVF. Following multidisciplinary team (MDT) consultation, the patient underwent thoracoscopic surgery and experienced a successful recovery during follow-up.

A massive pulmonary arteriovenous fistula complicated with coronary atherosclerotic heart disease treated by interventional therapy: a case report.

BACKGROUND: Pulmonary arteriovenous fistula (PAVF) is a rare disease, and its symptoms lack specificity. For patients with coronary heart disease(CHD), hypertension and other common cardiovascular diseases, PAVF is easy to be ignored. We presented a case of massive PAVF complicated with coronary atherosclerotic heart disease by interventional treatment to improve the understanding of this complex disease. CASE PRESENTATION: A 77-year-old female patient was admitted to the hospital due to chest tightness and shortness of breath following activities, which was diagnosed with CHD and hypoxemia in other hospitals. Coronary angiography showed that the patient had severe stenosis of coronary artery while pulmonary vascular DSA showing the patient had PAVF. After interventional therapy of both coronary artery and PAVF, the patient's symptoms were significantly improved. CONCLUSION: We presented a case of massive PAVF complicated with CHD by interventional treatment. For patients with unexplained hypoxemia and symptoms similar with CHD, the possibility of PAVF often leads to oversight, and various auxiliary examinations should be improved to avoid missed diagnosis. And intervention treatment should be carried out to improve the prognosis of patients as much as possible.

Large pulmonary arteriovenous malformation lost to follow-up with 10 years of asymptomatic interval growth: A case report.

Pulmonary arteriovenous malformations, previously considered a rare condition, have been increasingly identified in asymptomatic patients over the past 2 decades. Usually congenital and associated with hereditary hemorrhagic telangiectasia, these fistulae result in right-to-left shunting of blood by abnormal communication of pulmonary arteries and veins lacking capillary beds. Clinical findings of right-to-left shunting in the presence of feeding and draining vessels identified on imaging confirm the diagnosis, for which the first-line therapy is embolization. This report highlights the presentation and management of a large asymptomatic PAVM detected incidentally in a patient who was lost to follow-up for 10 years and represented with acute hypoxic respiratory failure secondary to a viral infection with an interval increase of PAVM size.

The chief culprit of intractable hypoxemia: a case report of rare pulmonary arteriovenous fistula complicated with giant hemangioma.

BACKGROUND: Pulmonary arteriovenous fistula (PAVF) is a rare disease, which can lead to the direct return of unoxidized venous blood to pulmonary veins and left heart, resulting in right-to-left shunt leading to hypoxia. Long term, the right-to-left shunt will cause severe pathophysiological changes in the patient's body and pulmonary circulation, and the prognosis will be poor if PAVF is not treated timely. CASE PRESENTATION: Here, we report the case of a 71-year-old man who presented with chest tightness and shortness of breath. After a series of examinations, PAVF and giant hemangioma were diagnosed, which are difficult to operate.Transcatheter interventional therapy was initiated. The patient recovered on the third day after operation and was discharged smoothly. During the long-term follow-up of nearly 4 years after discharge, the general condition and quality of life of the patient basically returned to normal. CONCLUSIONS: PAVF is rare but very important clinical problem. When the clinical manifestations of persistent unexplained hypoxia appear, it is necessary to fully consider the possibility of PAVF. Once the diagnosis of PAVF is clear, timely treatment is recommended to avoid deterioration of the disease and affecting the prognosis.

Hereditary Hemorrhagic Telangiectasia in a Patient Undergoing Hemodialysis with Anticoagulants and Antiplatelets: A Case Report.

Hereditary hemorrhagic telangiectasia (HHT) is an autosomal dominant disorder that causes abnormal blood vessel formation and bleeding. We herein report a 61-year-old woman with aggravated HHT symptoms after hemodialysis initiation. She was diagnosed with HHT based on her recurrent bleeding, abnormal blood vessel formation, and family history. Despite bleeding complications, the patient required anticoagulants and antiplatelet agents to treat cardiovascular complications. Eventually, the patient died of extensive cerebral hemorrhaging. Our experience suggests that special attention should be paid to bleeding complications in high-risk patients.

Lobectomy for Pulmonary Arteriovenous Fistula in a Patient With Rendu-Osler-Weber Disease: A Case Report.

BACKGROUND: Rendu-Osler-Weber disease (Osler's disease) is the most common cause of pulmonary arteriovenous fistula. We report a case of pulmonary arteriovenous fistula associated with Osler's disease that was treated by lobectomy. CASE REPORT: A 44-year-old man with Osler's disease presented with respiratory distress. Computed tomography showed a pulmonary arteriovenous fistula, which had a 26-mm-long diameter in S6 of the left lung. Transcatheter treatment had a high risk of recurrence, and surgery was indicated. The pulmonary arteriovenous fistula was found at the beginning of A6. A6 and the basilar artery were cut together with a stapler. The postoperative course was uneventful, and respiratory distress symptoms improved with no recurrence. CONCLUSION: We report a case of pulmonary arteriovenous fistula caused by Osler's disease treated by lobectomy. Although transcatheter treatment is the mainstream treatment for pulmonary arteriovenous fistulas, surgical resection may be effective depending on the size of the lesion.

Attention to pulmonary arteriovenous fistula in a case of transient hypoxemia and cerebral infarction during pregnancy: a case report and literature review.

BACKGROUND: Pulmonary arteriovenous fistula is rare during pregnancy. Pulmonary arteriovenous fistula presents no pulmonary symptoms in most patients but can be exacerbated by pregnancy. If not diagnosed and treated promptly, pulmonary arteriovenous fistula can lead to respiratory failure, stroke, spontaneous hemothorax, or other fatal complications. CASE PRESENTATION: A 29-year-old healthy pregnant woman presented with a transient drop in blood oxygen level of unknown cause during a routine examination at 34 weeks of gestation and during a cesarean section at 38 weeks of pregnancy. The patient's oxygen saturation quickly returned to normal and was not further investigated. On day 3 postpartum, the patient suddenly displayed slurred speech and right limb myasthenia. A head magnetic resonance imaging revealed cerebral infarction in the left basal ganglia. Subsequent computed tomography pulmonary arteriography revealed bilateral pulmonary arteriovenous fistula, which was likely the cause of cerebral infarction. The patient was transferred to the Department of Thoracic Surgery after one month of treatment and successfully underwent percutaneous embolization of pulmonary arteriovenous fistula. CONCLUSION: Pulmonary arteriovenous fistula should not be neglected if a pregnant woman presents with transient hypoxemia and cerebral infarction. A transient decrease in pulse oxygen saturation that cannot be explained by common clinical causes can be an early warning sign of the disease. Early diagnosis and multidisciplinary management could improve the prognosis.

Heart-brain axis: Association of congenital heart abnormality and brain diseases.

Brain diseases are a major burden on human health worldwide, and little is known about how most brain diseases develop. It is believed that cardiovascular diseases can affect the function of the brain, and many brain diseases are associated with heart dysfunction, which is called the heart-brain axis. Congenital heart abnormalities with anomalous hemodynamics are common treatable cardiovascular diseases. With the development of cardiovascular surgeries and interventions, the long-term survival of patients with congenital heart abnormalities continues to improve. However, physicians have reported that patients with congenital heart abnormalities have an increased risk of brain diseases in adulthood. To understand the complex association between congenital heart abnormalities and brain diseases, the paper reviews relevant clinical literature. Studies have shown that congenital heart abnormalities are associated with most brain diseases, including stroke, migraine, dementia, infection of the central nervous system, epilepsy, white matter lesions, and affective disorders. However, whether surgeries or other interventions could benefit patients with congenital heart abnormalities and brain diseases remains unclear because of limited evidence.

Transient ischemic attack induced by pulmonary arteriovenous fistula in a child: A case report.

BACKGROUND: Cerebral ischemic stroke is attributed to paradoxical cerebral embolism. Pulmonary arteriovenous fistula (PAVF) is a rare potential cause of cerebral ischemic stroke, and cerebral ischemic stroke induced by PAVF in children is rare. CASE SUMMARY: We report a case of right PAVF that presented as a transient ischemic attack (TIA) in a 13-year-old boy. The patient underwent embolization therapy and remained clinically stable for 2 years after treatment. CONCLUSION: TIA induced by PAVF in children is rare, lacks typical clinical manifestations, and should not be ignored.

A Case of Paradoxical Cerebral Embolism Due to Pulmonary Arteriovenous Fistula Mimicking Vertebral Artery Dissection With Wallenberg Syndrome.

Pulmonary arteriovenous fistula (PAVF) leads to paradoxical cerebral embolism, which can be fatal if left untreated. We report a rare case of brainstem infarction with acute severe headache and Wallenberg syndrome caused by a PAVF mimicking vertebral artery (VA) dissection. A 40-year-old man presented with a sudden occipital headache accompanied by right hemisensory disturbance. Magnetic resonance imaging revealed left lateral medullary infarction and poor depiction of the left VA. However, it was clearly recanalized on day six, and there were no findings of VA dissection. Whole-body contrast-enhanced computed tomography (CT) revealed a PAVF in the right lung and a thrombus in the feeding artery. The patient was diagnosed with hereditary hemorrhagic telangiectasia due to recurrent epistaxis and peripheral vasodilation of the tongue. An anticoagulant was administered for preventing further ischemic stroke, and a follow-up CT confirmed the disappearance of the thrombus in three months. Thoracoscopic partial lung resection was performed five months after the onset, and no recurrence of ischemic stroke was observed.

Diagnosis of descending aorta-pulmonary venous fistula combined with pulmonary arteriovenous fistula by color Doppler ultrasound combined with CT: A case report.

Aorto-pulmonary venous fistula combined with pulmonary arteriovenous fistula is a rare condition with an unknown incidence. We experienced a case of descending aorto-pulmonary venous fistula combined with a pulmonary arteriovenous fistula, which was treated with pulmonary arteriovenous fistula embolization and improved.

Transcatheter management of life-threatening pulmonary arteriovenous fistula with extracorporeal membrane oxygenation support in an infant.

Pulmonary arteriovenous malformation is a rare disease leading to cyanosis, where there is a direct relation between the pulmonary artery and pulmonary vein without a capillary structure. Arteriovenous fistulae may be single or multiple. Clinical signs emerge depending on the size of the fistulae and amount of shunt. Due to the advancements in transcatheter devices and increased experience render enable the fistula embolisation procedure as an alternative to surgical treatment. Extracorporeal membrane oxygenation is used to support the patient haemodynamically and respirationally in cases of treatment-resistant, severe and revocable cardiac or pulmonary sufficiency. This paper presents an infant patient with pulmonary arteriovenous malformation, who had haemodynamic instability due to severe hypoxia and received successful transcatheter fistula embolisation via extracorporeal membrane oxygenation under emergency conditions.

Unexpected Complications 25 Years after Coil Embolization for Pulmonary Arteriovenous Fistula.

An 87-year-old woman who had undergone coil embolization 25 years ago for pulmonary arteriovenous fistula, which was diagnosed following repeated cerebral infarction, presented with massive hemoptysis. The coils migrated and were excreted in stool following hemoptysis during long-term follow-up. Although the technical success rate of coil embolization for pulmonary arteriovenous malformations is extremely high, and coil embolization-related complications are rare, little is known about the long-term complications. We herein report the clinical course of our case, review previous reports related to coil migration as a long-term complication, and discuss the associated mechanism.

Case Report: Clinical characteristics and genetic analysis of two patients with hereditary hemorrhagic telangiectasia.

Objective: To analyze the clinical features and genetic characteristics of two patients with hereditary hemorrhagic telangiectasia (HHT) and to review the relevant literature. Methods: The clinical data of two HHT patients admitted to the author's hospital between April 2019 and February 2022 were retrospectively analyzed. Meanwhile, the genetic analysis was performed with their consent. Results: The first patient was a 62-year-old woman who had been complaining of shortness of breath and fever for 20 days. Her previous medical history included brain abscess drainage and video-assisted thoracoscopic surgery for a pulmonary hemangioma. A right heart catheterization revealed no pulmonary arterial hypertension, and an abdominal enhanced magnetic resonance imaging revealed multiple arteriovenous malformations in the liver. Her ACVRL1 heterozygous variants were discovered through whole-exon gene testing. The second case involved a 47-year-old woman who had been experiencing chest tightness for the past 2 years. Several years ago, she underwent brain abscess drainage and embolization of a pulmonary arteriovenous fistula. Ultrasound revealed generalized hepatic vascular dilation, and enhanced computed tomography revealed numerous pulmonary venous fistulas scattered in both lungs as well as multiple arteriovenous malformations in the liver. Her whole-exon gene testing revealed that she, like her son, had heterozygous ENG variants. Conclusion: HHT patients may experience infection, bleeding, dyspnea, and other symptoms. Imaging is important in disease diagnosis and management because early detection and treatment can prevent major complications and disability or even death.

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

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.

Images of the month 1: Ischaemic stroke due to pulmonary arteriovenous fistula.

A 38-year-old man was admitted to the hospital due to a "suddenly developed right hemiplegia, unconsciousness and gaze to the right". Pulmonary arteriovenous fistulas (PAVFs) are rare but an important cause of stroke in young people, which is easy to be clinically neglected. Therefore, for young patients with pulmonary diseases and cerebral infarction, the possibility of PAVF should be considered.

Transcatheter coil embolization for large pulmonary arteriovenous fistulae through an artificial tricuspid ball valve.

Pulmonary arteriovenous fistulae (PAFs) occur congenitally or are acquired. A PAF can cause hypoxemia, sudden death from rupture, abscess formation, and embolism. Treatment for PAF is transcatheter embolization or surgery. Transcatheter embolization is the first choice of treatment; however, this treatment is impossible to perform if a patient has had tricuspid or pulmonary valve replacement. In this paper, we describe a case of PAFs complicated with tricuspid valve replacement with a ball valve (which had been performed 40 years earlier) that was treated with transcatheter embolization. .

A Comparative Study of Invasive Modalities for Evaluation of Pulmonary Arteriovenous Fistula after Bidirectional Glenn Shunt.

Development of pulmonary AV fistula (PAVF) after bidirectional glenn shunt (BDG) results in significant cyanosis, impaired exercise performance, and increased morbidity and mortality. We attempted to detect and quantify PAVF in post-BDG patients by saline contrast transesophageal echocardiography (TEE) and compare with pulmonary angiography and pulmonary vein oximetry. This was a prospective study done between 2017 and 2018. Twenty-five children who underwent BDG and planned for cardiac catheterization prior to Fontan completion were included in the study. All patients underwent pulmonary angiography, oximetry, and saline contrast TEE at the time of cardiac catheterization. Twenty-two patients had undergone unilateral BDG surgery and three were palliated by bilateral BDG. The mean oxygen saturation was 80 ± 5.2%. Thirteen patients (52%) had preserved antegrade pulmonary blood flow. Eighteen patients (72%) had PAVF by angiography and oximetry, while 19 (76%) had PAVF identified by contrast echocardiography. There was moderate correlation between the degree of pulmonary venous desaturation and grading of PAVF by contrast echocardiography. PAVF was predominantly located in the lower zones of the lungs. Higher grades of PAVF were not seen in patients with preserved antegrade flow after BDG. Angiographically detected PAVF showed a steady increase with increasing delay to cardiac catheterization from BDG. Significant reduction in systemic saturation was limited to advanced grades of PAVF in patients after BDG. Saline contrast TEE, pulmonary venous oximetry, and pulmonary angiography equally identified PAVF in patients after BDG. Prognostic utility of the same needs to be assessed by long-term follow-up of these subjects.

Declarations: management of a pulmonary arteriovenous fistulae by uniportal video-assisted thoracoscopic surgery: a case report.

BACKGROUND: A pulmonary arteriovenous fistula (PAVF) is a rare condition that is associated with pulmonary arteriovenous malformation (PAVM). Few reports have described managing PAVMs using uniportal video-assisted thoracoscopic surgery (VATS). CASE PRESENTATION: A 13-year-old child with PAVF in the left inferior pulmonary artery was treated by uniportal VATS with left lower lobectomy. After surgery, hemoptysis did not recur and there were no postoperative complications. Six months after the operation, postoperative review of computerized tomography showed no recrudescence of PAVF. CONCLUSIONS: PAVF is a rare case that should be diagnosed and treated early. 3D- computerized tomography (CT) reconstruction is useful for diagnosis and preoperative assessment. The case shows that PAVF can be managed with uniportal VATS.

Pulmonary arteriovenous fistula diagnosed by transthoracic contrast echocardiography: A case report.

We report the case of a 38-year-old woman who was admitted due to right limb numbness. A right-to-left shunt (RLS) was detected by transthoracic contrast echocardiography (cTTE), and microbubbles appeared in the left heart seven cardiac cycles after opacification of the right heart, suggesting pulmonary arteriovenous fistula. This was supported by computed tomography angiography. She underwent thoracoscopic partial right lower pneumonectomy and recovered after surgery. At 1 year follow-up, she had no relapse of stroke or cerebral infarction. This case shows the typical signs of pulmonary arteriovenous fistula on contrast-enhanced trans-thoracic echocardiography, which allowed an accurate and rapid diagnosis.