Giant Neonatal Pulmonary Arteriovenous Malformation: An Imaging and Management Challenge.
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ABSTRACT:
SUBMITTER: Lin Y
PROVIDER: S-EPMC7756164 | biostudies-literature | 2020 Dec
REPOSITORIES: biostudies-literature
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Giant Neonatal Pulmonary Arteriovenous Malformation: An Imaging and Management Challenge.
Lin Yalin Y Hogan Whitnee W Stillwell Kristina K Moore Phillip P Peyvandi Shabnam S Amin Elena E Quezada Emilio E
CASE (Philadelphia, Pa.) 20200916 6
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Project description:BACKGROUND:Pulmonary arteriovenous malformations are abnormal direct connections between the pulmonary artery and pulmonary vein which result in a right-to-left shunt. They are associated with substantial morbidity and mortality mainly from the effects of paradoxical emboli. Potential complications include stroke, cerebral abscess, pulmonary haemorrhage and hypoxaemia. Embolisation is an endovascular intervention based on the occlusion of the feeding arteries the pulmonary arteriovenous malformations thus eliminating the abnormal right-to-left-shunting. This is an update of a previously published review. OBJECTIVES:To determine the efficacy and safety of embolisation in patients with pulmonary arteriovenous malformations including a comparison with surgical resection and different embolisation devices. SEARCH METHODS:We searched the Cystic Fibrosis and Genetic Disorders Group's Trials Register; date of last search: 10 April 2017.We also searched the following databases: the Australian New Zealand Clinical Trials Registry; ClinicalTrials.gov; International Standard Randomised Controlled Trial Number Register; International Clinical Trials Registry Platform Search Portal (last searched 27 August 2017). to be updatedWe checked cross-references and searched references from review articles. SELECTION CRITERIA:Trials in which individuals with pulmonary arteriovenous malformations were randomly allocated to embolisation compared to no treatment, surgical resection or embolisation using a different embolisation device. DATA COLLECTION AND ANALYSIS:Studies identified for potential inclusion were independently assessed for eligibility by two authors, with excluded studies further checked by a third author. No trials were identified for inclusion in the review and hence no analysis was performed. MAIN RESULTS:There were no randomised controlled trials included in the review; one ongoing trial has been identified which may be eligible for inclusion in the future. AUTHORS' CONCLUSIONS:There is no evidence from randomised controlled trials for embolisation of pulmonary arteriovenous malformations. However, randomised controlled trials are not always feasible on ethical grounds. Accumulated data from observational studies suggest that embolisation is a safe procedure which reduces morbidity and mortality. A standardised approach to reporting with long-term follow-up through registry studies can help to strengthen the evidence for embolisation in the absence of randomised controlled trials.
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Project description:Genital arteriovenous malformations are rare and present unique surgical challenges in preserving urogenital function, abdominal wall integrity, and lower limb perfusion. A 32-year-old man with a giant abdominoscrotal arteriovenous malformation presented with recurrent heavy bleeding. Due to the high risk of rebleeding and fatal hemorrhage, surgery with curative intent was proposed and the patient was counseled on the risks of ischemia to the lower limb, testes, and penis. Preoperative embolization of the feeding vessels was performed. Three days later, surgical excision of the mass with the affected scrotum, left rectus muscle, sheath, and overlying abdominal skin followed. The testes were dissected from the malformation and preserved along with the right internal pudendal artery. The left thigh skin was advanced to the scrotal remnants and a neoscrotum created. The resulting large abdominal wall defect was reconstructed in layers with a pedicled anterolateral thigh flap, including innervated vastus lateralis muscle, to prevent herniation. Recovery was uneventful, and a 4-year follow-up revealed no significant clinical or radiological recurrence with recovery of flap sensation, retained erectile function, and no herniation. We report this case due to rarity of giant abdominoscrotal arteriovenous malformations and present preoperative embolization, surgical resection, and functional anterolateral thigh flap reconstruction as a valuable treatment option of this life-threatening illness.
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Project description:Pulmonary arteriovenous malformation (AVM) is a congenital vascular disease in which interventional radiologists can play both diagnostic and therapeutic roles in patient management. The diagnosis of pulmonary AVM is simple and can usually be made based on CT images. Endovascular treatment, that is, selective embolization of the pulmonary artery feeding the nidus of the pulmonary AVM, and/or selectively either the nidus or draining vein, has become a first-line treatment with advances in interventional devices. However, some vascular diseases can simulate pulmonary AVMs on CT and pulmonary angiography. This subset can confuse interventional radiologists and referring physicians. Vascular mimickers of pulmonary AVM have not been widely known and described in detail in the literature, although some of these require surgical correction, while others require regular follow-up. This article reviews the clinical and radiologic features of pulmonary AVMs and their mimickers.
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Project description:We present a case of recurrent strokes in a patient with absent left internal carotid artery (ICA) and pulmonary arteriovenous malformation. Pulmonary arteriovenous malformations (PAVMs) are abnormal communications between pulmonary artery and pulmonary vein, cause extracardiac right to left shunting of blood and are known to significantly increase the risk of stroke primarily due to paradoxical embolization. They are often hereditary and are commonly associated with hereditary hemorrhagic telangiectasias (HHT). Delayed bubbles seen in the left ventricle (after 3 cardiac cycles) on transthoracic echocardiogram with bubble study is often the first clue to the presence of PAVMs. CT scan of the chest can confirm the diagnosis. Percutaneous embolotherapy is the treatment of choice with reduction in stroke risk post embolization.
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Project description:Arteriovenous malformations (AVMs) are rare congenital vascular anomalies where rupture can be fatal. This report describes a case of a 69-year-old woman who presented with sudden severe lower back pain radiating to her left buttock and thigh and an inability to move her left leg. Computed tomography angiography (CTA) showed an extensive ruptured retroperitoneal AVM on the left paravertebral side with active bleeding and a large hematoma. Cardiopulmonary resuscitation with considerable blood transfusion was required. The AVM was effectively treated with coil embolization of the feeding lumbar arteries. Shortly after the intervention, the patient developed abdominal compartment syndrome. Urgent laparotomy with evacuation of the retroperitoneal hematoma was required. The patient was discharged 16 days after admission in a good physical condition and has been free of symptoms for more than a year after intervention. However, follow-up CTA showed residual contrast enhancement in the AVM and further surveillance is required.
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Project description:A pulmonary arteriovenous malformation (PAVM) is caused by abnormal communications between the pulmonary arteries and veins. In this study, a 64-year old woman presented with a large PAVM in the central upper lobe of the right lung. As feeding vessels were large and short, the patient was scheduled for resection therapy. By clamping the right main pulmonary artery, the blood flow into the PAVM was controlled and lobectomy was performed safely. Although advances in interventional radiology have led to the introduction of obliterative techniques, surgical resection is still an effective first option for patients with a large, centrally located PAVM.
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Project description:Pulmonary arteriovenous malformations (PAVMs) may manifest with bleeding or embolic events necessitating intervention. Transcatheter coil embolization through the pulmonary artery (PA) is an established approach. We present a case of recurrent PAVMs despite numerous PA coil embolizations. PAVM occlusion was achieved through plug placement by a transseptal and pulmonary venous approach. (Level of Difficulty: Advanced.).
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Project description:BackgroundConcurrent patent foramen ovale (PFO) and pulmonary arteriovenous malformation (PAVM) are rare but can cause paradoxical embolism and platypnoea-orthodeoxia syndrome (POS).Case summaryA 72-year-old female with embolic stroke history developed positional dyspnoea. Evaluation revealed right-to-left shunting through PFO and PAVM in the right middle lobe. Orthodeoxia was confirmed by 6% SpO2 decrease upon position change. A staged approach was adopted: PFO closure with Amplatzer™ Occluder, followed by PAVM embolization 1 month later. Symptoms improved significantly post-procedure. No residual shunting or symptoms have been observed during the 2-year follow-up.DiscussionThis case emphasizes thorough evaluation in patients with cryptogenic stroke and POS, especially when symptoms persist. It demonstrates the effectiveness of staged treatment for concurrent PFO and PAVM, highlighting the importance of individualized strategies and long-term follow-up.
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Project description:Brain arteriovenous malformations (AVMs) are complex and heterogeneous lesions that can rupture, causing significant morbidity and mortality. While ruptured lesions are usually treated, the management of unruptured AVMs remains unclear. A Randomized trial of Unruptured Brain Arteriovenous Malformations (ARUBA) was the first trial conducted to compare the effects of medical and interventional therapy. Although it concluded that medical therapy was superior in preventing stroke and death over a follow-up period of 33 months, the findings were met with intense criticism regarding several aspects of study design, progression, and analysis/conclusion. Namely, the increased use of stand-alone embolisation relative to microsurgery in a cohort with predominantly low-grade lesions combined with a short follow-up period amplified treatment risk. Subsequently, several observational studies were conducted on ARUBA-eligible patients to investigate the safety and efficacy of microsurgery, radiosurgery, and endovascular embolisation over longer follow-up periods. These reports showed that favourable safety profiles and cure rates can be achieved with appropriate patient selection and judicious use of different treatment modalities in multidisciplinary centres. Since large prospective randomised trials on AVMs may not be feasible, it is important to make use of practice-based data beyond the flawed ARUBA study to optimise patients' lifetime outcomes.
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