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      Histology of a Marfan aorta 4.5 years after personalized external aortic root support

      case-report

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          Abstract

          In 2008, a 26-year old man had personalized external aortic root support (PEARS) with a macroporous mesh. He was the 16th of 46 patients to have this operation. He had a typical Marfan habitus. His mother died of this disease as did his brother, with an aortic dissection. The patient himself died suddenly 4.5 years after his PEARS operation. At autopsy, there was no blood in the pericardium. The coronary orifices and proximal arteries were normal. His bicuspid aortic valve was minimally regurgitant as it was prior to operation and remained throughout follow-up. Macroscopically the implanted mesh was embedded in the adventitia and not separable from the aortic wall. Microscopically it was fully incorporated with collagen fibres as has been seen in our animal studies. The unsupported aortic arch showed some focal fragmentation of elastic fibres and a mild increase in mucopolysaccharides consistent with Marfan syndrome. These appearances were not present in the supported aortic root, which had the histological appearance of a normal aorta. He was the first patient to die with an implant. The histological appearances suggest the possibility that the incorporated support of the aortic root allowed recovery of the microstructure of the media.

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          Evidence for Marfan cardiomyopathy.

          Joyce Wong, Sanjay Prasad, Anne Child … (2010)
          Marfan syndrome (MFS) is an inherited connective tissue disease which frequently involves the cardiovascular system. The heart can be affected since valvular regurgitation is a common complication. However, there is still debate whether a primary cardiomyopathy exists. Our aim was to evaluate the existence of a Marfan-related cardiomyopathy using cardiovascular magnetic resonance. We retrospectively evaluated 68 consecutive adult patients with no cardiovascular surgery or significant valvular regurgitation. Left ventricular and right ventricular volumes, ejection fraction, and mass were estimated and compared with published data on a healthy control population. Patients were also assessed for heart failure, aortic dimensions, and valve disease. One quarter (25.0%) of Marfan patients had reduced left ventricular ejection fraction (LVEF), with 25.0% having increased left ventricular end-diastolic and 30.8% having increased end-systolic volumes. The right ventricular ejection fraction was reduced in 10.3%, with increased right ventricular end-diastolic volumes in 11.8% and increased end-systolic volumes in 13.2%. On univariate analysis, no association was found between reduced LVEF and age, gender, indexed aortic dimensions, presence of mitral valve prolapse, or valve regurgitation. This study supports the existence of a primary cardiomyopathy in a subgroup of Marfan patients. The biventricular enlargement and dysfunction is usually mild, asymptomatic, and independent from other cardiovascular manifestations. Further studies are needed to assess underlying causes and natural history of this condition. Routine monitoring and treatment in MFS may need to be tailored not only to prevent aortic root expansion but also to support myocardial function.
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            Long-term experience of girdling the ascending aorta with Dacron mesh as definitive treatment for aneurysmal dilation.

            Oved Cohen, Jonah Odim, David De La Zerda … (2007)
            The management of the mildly to moderately dilated ascending aorta (3.5 to 4.9 cm) in cardiac surgery remains controversial. Therapeutic options have included radical aortic resection with synthetic graft substitution, external aortic reinforcement or wrap, with or without partial aortic wall excision, and a watch-and-wait approach. We reviewed our institutional experience with Dacron (DuPont, Wilmington, DE) mesh wrap support of dilated ascending aortas. During the last 20 years, 102 patients with aneurysmal dilatation of the ascending aorta underwent wrapping of the ascending aorta with a fine Dacron mesh from the ventricular-aortic junction to the origin of the innominate artery. For the last 10 years, the wrap was anchored to the aortic annulus with pledgeted sutures. Aortic diameters up to 6 cm, without focal areas of thinning, were wrapped. Aortic diameters exceeding 6 cm, or with focal thinning, underwent tailored aortic wall resection and wrapping. Primary end points of the study included mortality, aortic diameter growth, dissection or rupture, or both. The mean age of the group was 54.7 +/- 19 years (range, 12 to 90 years). A single patient underwent aortic wrapping without cardiopulmonary bypass. Sixty-six patients (65%) required additional aortic valve surgery. Five patients (5%) had reinforcement of dilated sinuses with glutaraldehyde-treated pericardial patches combined with wrapping. Twenty-seven patients (26%) had combined coronary and valve surgery, and 2 patients had coronary revascularization alone. There was neither early nor hospital mortality. Among the 81 patients (79%) we were able to contact, 7 (7%) late deaths had occurred at 0.5, 1, 3, and 9 years after operation that were unrelated to aortic pathology. Various levels of follow-up were obtained in the 88 patients (86.2%). In 78 patients, echocardiograms, computed tomography angiograms, or magnetic resonance angiograms were obtained. In 2 of these patients, aneurysmal dilatation of the sinuses developed below the wrap and reoperation was required. No patient in whom the mesh wrap was anchored to the aortic annulus required reoperation. All 81 patients that were contacted by us and monitored by referring physicians were asymptomatic and free of problems related to the aorta. The mean (+/- SD) preoperative diameter of the ascending aorta was 49.2 +/- 7.8 mm (range, 35 to 87 mm), the postwrap intraoperative diameter was 32.9 +/- 3.4 mm (range, 20 to 40 mm), and the follow-up postoperative aortic diameter was 35.6 +/- 12.7 mm (range, 27 to 52 mm). The mean average change in the aortic diameter during the follow-up period was 2.6 +/- 14.8 mm (range, -7 to 22 mm), a mean of 8%. The mean follow-up period was 5.7 years (median, 4.77 years; range, 9 days to 21 years). There were no infections or other early complications related to the wrap. Dacron mesh support of the moderately dilated aneurysmal ascending aorta, alone or in conjunction with coronary revascularization, aortic root surgery, or valvular operations, or both, is safe and durable. Dacron mesh is transparent and stretchable, permitting tight girdling of the aorta. These properties prevent hematoma formation, facilitate proximal vein graft anastomoses, and provide visualization and access to aortic suture lines. Finally, this technique retards further aortic dilation, altering the natural history of aortic aneurysms.
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              Surgical management of aortic root disease in Marfan syndrome and other congenital disorders associated with aortic root aneurysms

              Tom Treasure, J Takkenberg, John Pepper (2014)
              Elective root replacement in Marfan syndrome has improved life expectancy in affected patients. Three forms of surgery are now available: total root replacement (TRR) with a valved conduit, valve sparing root replacement (VSRR) and personalised external aortic root support (PEARS) with a macroporous mesh sleeve. TRR can be performed irrespective of aortic dimensions and a mechanical replacement valve is a secure and near certain means of correcting aortic valve regurgitation but has thromboembolic and bleeding risks. VSRR offers freedom from anticoagulation and attendant risks of bleeding but reoperation for aortic regurgitation runs at 1.3% per annum. A prospective multi-institutional study has found this to be an underestimate of the true rate of valve-related adverse events. PEARS conserves the aortic root anatomy and optimises the chance of maintaining valve function but average follow-up is under 5 years and so the long-term results are yet to be determined. Patients are on average in their 30s and so the cumulative lifetime need for reoperation, and of any valve-related complications, are consequently substantial. With lowering surgical risk of prophylactic root replacement, the threshold for intervention has reduced progressively over 30 years to 4.5 cm and so an increasing number of patients who are not destined to have a dissection are now having root replacement. In evaluation of these three forms of surgery, the number needed to treat to prevent dissection and the balance of net benefit and harm in future patients must be considered.
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                Author and article information

                Journal
                Journal ID (nlm-ta): Eur J Cardiothorac Surg
                Journal ID (iso-abbrev): Eur J Cardiothorac Surg
                Journal ID (publisher-id): ejcts
                Journal ID (hwp): ejctsurg
                Title: European Journal of Cardio-Thoracic Surgery : Official Journal of the European Association for Cardio-thoracic Surgery
                Publisher: Oxford University Press
                ISSN (Print): 1010-7940
                ISSN (Electronic): 1873-734X
                Publication date (Print): September 2015
                Publication date (Electronic): 18 November 2014
                Publication date PMC-release: 18 November 2014
                Volume: 48
                Issue: 3
                Pages: 502-505
                Affiliations
                [a ]National Institute of Health Research (NIHR) Cardiovascular Biomedical Research Unit (BRU), Royal Brompton Hospital , London, UK
                [b ]Department of Pathology, Papworth Hospital and University of Cambridge , Cambridge, UK
                [c ]Cardiovascular MR Unit, Royal Brompton Hospital and Imperial College London , London, UK
                [d ]Clinical Operational Research Unit, Department of Mathematics, University College London , London, UK
                Author notes
                [* ]Corresponding author. Clinical Operational Research Unit, Department of Mathematics, University College London, London, UK. Tel: +44-7957-168754; fax: +44-01233-740378; e-mail: tom.treasure@ 123456gmail.com (T. Treasure).
                Article
                Publisher ID: ezu415
                DOI: 10.1093/ejcts/ezu415
                PMC ID: 4535558
                PubMed ID: 25406424
                SO-VID: 10342617-6af8-4fb2-8d3a-a1c540eeb795
                Copyright © © The Author 2014. Published by Oxford University Press on behalf of the European Association for Cardio-Thoracic Surgery.
                License:

                This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License ( http://creativecommons.org/licenses/by-nc/4.0/), which permits non-commercial re-use, distribution, and reproduction in any medium, provided the original work is properly cited. For commercial re-use, please contact journals.permissions@oup.com

                History
                Date received : 12 July 2014
                Date revision received : 1 October 2014
                Date accepted : 6 October 2014
                Categories
                Subject: 26
                Subject: Case Reports

                ScienceOpen disciplines: Surgery
                Keywords: marfan syndrome,aortic root aneurysm,sudden death
                Data availability:
                ScienceOpen disciplines: Surgery
                Keywords: marfan syndrome, aortic root aneurysm, sudden death

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