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Case Report

Transesophageal echocardiography in surgical management of pseudoaneurysm of mitral‑aortic intervalvular fibrosa with aneurysms of right sinus of Valsalva and left main coronary artery Shreedhar S. Joshi, Arkalgud Marigowda Jagadeesh, Arul Furtado1, Seetharam Bhat1 Departments of Cardiac Anaesthesiology and 1Cardiac Surgery, Sri Jayadeva Institute of Cardiovascular Sciences and Research, Bangalore, Karnataka, India

ABSTRACT

Received : 30-06-12 Accepted : 08-09-12

Pseudoaneurysm of mitral‑aortic intervalvular fibrosa (MAIVF) is a rare complication associated with aortic and/or mitral valve surgery complicated by infective endocarditis. We report pseudoaneurysm of MAIVF in a young adult without overt cardiac disease or previous cardiac surgery. The patient had a rare combination of pseudoaneurysm of MAIVF impinging on anterior mitral leaflet causing moderate mitral regurgitation, right sinus of Valsalva aneurysm extending into interventricular septum, and left main coronary artery aneurysm. Transesophageal echocardiography helped in confirming the lesions, delineating the anatomy of all the lesions, and assessing the adequacy of surgical repair. Key words: Aorto‑mitral curtain, Mitral aortic intervalvular fibrosa, Subaortic pseudoaneurysm

INTRODUCTION

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Website: www.annals.in PMID: 23287084 DOI: 10.4103/0971-9784.105368 Quick Response Code:

Pseudoaneurysm of mitral‑aortic intervalvular fibrosa (MAIVF) is a rare clinical entity. It involves the region of aorto‑mitral curtain. The area, being relatively avascular, is prone to infection and trauma during aortic and mitral valve surgeries. There are 89 reported cases in literature. Most common age at presentation is 3 rd to 5 th decade. Endocarditis and aortic valve surgeries are the most common etiologies. Clinical presentation is commonly as endocarditis followed by congestive heart failure (CHF) or dyspnea and chest pain due to compression of coronary vessels.[1] Among the 89 cases reported, 10% had compression of coronary arteries. All these cases had involvement of left circumflex artery (LCx), and a few had associated left main coronary artery (LMCA) and left anterior descending artery

(LAD) involvement.[2] The pseudoaneurysm of MAIVF are better detected and delineated by transoesophageal echocardiography (TEE) compared to transthoracic echocardiography (TTE) or aortography techniques.[3,4] We present a case of pseudoaneurysm of MAIVF associated with right sinus of Valsalva (RtSOV) aneurysm extending into interventricular septum (IVS) and LMCA aneurysm. There was no history of aortic valve surgery or infective endocarditis. This is a rare combination of lesions without any known etiological factors. We discuss the utility of TEE in assessing the lesions and adequacy of the surgical repair. CASE REPORT A 29‑year‑old young adult male presented with chest pain and dyspnea. TTE revealed dilated left heart chambers, moderate mitral regurgitation (MR), and multiple cystic lesions

Address for correspondence: Dr. Shreedhar Shubhash Joshi, Department of Cardiac Anaesthesia, Sri Jayadeva Institute of Cardiovascular Sciences and Research, Bannerghatta Road, Bangalore ‑ 560 069, Karnataka, India. E‑mail: [email protected]

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adjacent to the IVS. In view of chest pain, patient was subjected to computed tomography (CT) angiogram. It revealed multiple aneurysms in the aorto‑mitral region, LMCA aneurysm compressing LCx and RtSOV aneurysm. Clinical diagnosis of pseudoaneurysm of MAIVF was considered and patient was referred for surgery.

The cause of pulsatility is direct transmission of blood from LV cavity during systole and emptying of aneurysm during diastole.[1,5] Cardiopulmonary bypass (CPB) was instituted by high aortic and bicaval cannulation after adequate heparinization. Cold blood cardioplegia was given through aortic root to achieve cardiac arrest. At aortotomy, RtSOV was aneurysmal and extending into the IVS. It was obliterated and excluded with a Dacron® patch. An aneurysm of the MAIVF was confirmed, which had displaced the AML inferiorly. The opening was closed following which the MAIVF was reinforced with a Dacron patch. This procedure had an inherent risk of mitral annulus being tethered leading to mitral regurgitation post CPB. The surgical team decided to rely upon TEE assessment of MR post CPB to assess for this complication. The LMCA was excised to expose an aneurysm on its supero‑lateral aspect, containing a thrombus. The aneurysm was obliterated and excluded with a patch. The laid open LMCA and the coronary ostium were reconstituted with an untanned pericardial patch to the level of the transected aorta. As planned

After induction of general anesthesia and invasive central venous catheter placement, TEE (Philips multiplane) probe was introduced. Philips EnVisor HD ultrasound machine was used to store and analyze images. Comprehensive TEE assessment revealed RtSOV aneurysm extending into the IVS [Figures 1a, b and 2a]; Pseudoaneurysms in the mitral‑aortic intervalvular fibrosa (MAIVF) region, which was impinging on anterior mitral leaflet (AML) causing moderate MR [Figure 1c, d and Video 1]; and severely dilated LMCA ostium (7.5 mm); however, the course of the aneurysm could not be determined [Figure 2a]. The characteristic pulsatility of pseudoaneurysm, expansion during systole, and collapse during diastole was demonstrated in RtSOV and MAIVF aneurysms.

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d

Figure 1: TEE ME 4C modified view (aortic valve seen) (a, b) depicting the right sinus of Valsalva (RtSOV) aneurysm extending into interventricular septum (arrows) with expansion in systole (a) and collapse during diastole (b) TEE ME LAX (130°) view shows the pseudoaneurysm of mitral aortic intervalvular fibrosa (MAIVF) impinging on anterior mitral leaflet (AML) with expansion in systole (c, arrow heads) and collapse in diastole (d)

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before surgery, left internal mammary artery (LIMA) was grafted to LAD and venous graft to major obtuse marginal (OM) was performed. After aortic cross‑clamp time of 250 min and CPB time of 262 min, weaning from CPB was facilitated with dopamine 5 µg/kg/min. Post CPB, TEE assessment revealed obliteration of the RtSOV aneurysm [Figures 3a, b and 2b]; obliteration of the pseudoaneurysm of MAIVF [Figure 3a and Video 2]; and mild MR with a vena contracta measuring 2 mm and an MR jet area 1.8 cm2, both of which were considered acceptable [Figure 3b]. After reversal of heparin and achieving hemostasis, chest was closed. Patient was weaned off ventilator successfully after 7 h. Postoperative TTE confirmed post CPB TEE  findings. DISCUSSION This case report is presented to highlight the role of perioperative TEE in assessing the MAIVF lesions, diagnosing associated lesions, adequacy of surgical repair, and surgical complications if any. This is a rare case involving MAIVF region, LMCA aneurysm, and RtSOV

a

aneurysm. This is a rare combination of lesions involving aortic root structures – anteroseptal IVS (subaortic membrane), sinus of Valsalva, and proximal coronaries. The MAIVF is a triangular area bounded by the right and left fibrosa. It forms the continuity of aortic and mitral valves, and developmentally belongs to the fibrous skeleton of the heart. Being a relatively avascular area, it is prone to weakening and abscess collection after aortic and less commonly mitral valve surgeries. The pseudoaneurysm of MAIVF is commonly due of infective endocarditis or aortic valve surgeries. In the present case, there was no aortic or mitral valve surgery in the past. Absence of spikes of fever, negative blood cultures, negative C‑reactive protein, and anti‑streptolysin O antibody tests make infective endocarditis an unlikely etiologic factor. Culture of the excised tissue did not yield any organism growth. Though some cases of pseudoaneurysm of MAIVF have been reported with Takayasu’s arteritis,[6] the present case had normal aortic dimensions and the histopathology did not reveal any features suggestive of inflammatory involvement. The pseudoaneurysm of MAIVF is asymptomatic till it compresses or ruptures into adjacent structures. Rupture into atria causing fistula and left ventricle outflow tract causing CHF are common complications, followed by coronary compression and myocardial ischemia. TTE is useful in diagnosis but less sensitive in comparison to TEE. In a case series, TTE had a sensitivity of 36% whereas TEE had a sensitivity of 87-100% and specificity of 94.6%.[4,7,8] The posterior location of the MAIVF and frequent association with prosthetic valve make TEE a more sensitive diagnostic modality. TEE also has better localization of fistula, subaortic abscess, and compression of LCx. The pulsatility of the aneurysm helps to differentiate a pseudoaneurysm from an abscess on echocardiography, which was well documented in the present case. Coronary angiogram

b

Figure 2: TEE ME AV SAX view shows RtSOV aneurysm and dilatation of left coronary ostium (a, arrow) and the post surgical view with obliteration of the aneurysm (*) (b)

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a

b

Figure 3: TEE ME LAX (112°) reveals post‑repair status with obliterated RtSOV (*) (a), IVS (arrows), and mild MR (b)

Annals of Cardiac Anaesthesia    Vol. 16:1    Jan-Mar-2013

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is useful in delineating the coronary compression and any associated coronary atherosclerotic lesions. CT and magnetic resonance imaging (MRI) are also efficacious in delineating the aneurysm and complications; both give a better spatial orientation to surrounding structures and help in surgical planning. Conservative management has been a modality in a few reports which involved smaller aneurysms without any complications. Patients with multiple previous surgeries, where the risk of surgery is higher than the benefit, are likely candidates for conservative management.[9,10] But more often surgery is the choice of treatment, as complications are the common presentations. Rupture into the atria or pericardium may be an indication of emergency surgical intervention. Device closures have been reported in a few low‑risk cases.[11,12] In summary, we present a case of pseudoaneurysm of MAIVF without any overt cardiac disease, or cardiac surgery in the past, or connective tissue disease. The pseudoaneurysm of MAIVF was associated with a rare combination of RtSOV aneurysm extending into IVS and LMCA aneurysm. TEE helped in delineating the pseudoaneurysm and assessing the adequacy of the surgical repair. In patients with pseudoaneurysm of MAIVF, TEE evaluation of structures related to aortic root is necessary before surgical intervention. ACKNOWLEDGMENT We acknowledge the conributions made by Dr. Deepak Borde, Dr. Ashwini T, and Dr. Navaneetha in editing the manuscript.

REFERENCES 1.

Sudhakar S, Sewani A, Aggarwal M, Uretsky BF. Pseudoaneurysm of the Mitral‑Aortic Intervalvular Fibrosa (MAIVF): A comprehensive review.

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J Am Soc Echocardiogr 2010;23:1009‑18. 2. Almeida J, Pinho P, Torres JP, Garcia JM, Maciel MJ, Lima CA, et al. Pseudoaneurysm of the Mitral‑Aortic Fibrosa: Myocardial ischemia secondary to left coronary compression. J Am Soc Echocardiogr 2002;15:96‑8. 3. Afridi I, Apostolidou MA, Saad RM, Zoghbi WA. Pseudoaneurysm of the mitral‑aortic intervalvular fibrosa: Dynamic characterisation using transoesophageal echocardiography and Doppler techniques. J Am Coll Cardiol 1995;25:137‑45. 4. Karalis DG, Bansal RC, Hauck AJ, Ross JJ Jr, Applegate PM, Jutzy KR, et al. Transesophageal echocardiographic recognition of subaortic complications in aortic valve endocarditis. Clinical and surgical implications. Circulation 1992;86:353‑62. 5. Bansal RC, Moloney PM, Marsa RJ, Jacobson JG. Echocardiographic features of a mycotic aneurysm of the left ventricular outflow tract caused by perforation of mitral‑aortic intervalvular fibrosa. Circulation 1983;67:930‑4. 6. Tufekcioglu O, Ozlu MF, Cay S, Tuna F, Basar N, Gurel OM, et al. Pseudoaneurysm of the mitral‑aortic intervalvular fibrosa in a patient with Takayasu’s arteritis. Can J Cardiol 2008;24:718. 7. Leung DY, Cranney GB, Hopkins AP, Walsh WF. Role of transoesophageal echocardiography in the diagnosis and management of aortic root abscess. Br Heart J 1994;72:175‑81. 8. Daniel WG, Mügge A, Martin RP, Lindert O, Hausmann D, Nonnast‑Daniel B, et al. Improvement in the diagnosis of abscesses associated with endocarditis by transoesophageal echocardiography. N Engl J Med 1991;324:795‑80. 9. Hasin T, Reisner SA, Agmon Y. Large pseudoaneurysms of the mitral‑aortic intervalvular fibrosa: Long‑term natural history without surgery in two patients. Eur J Echocardiogr 2011;12:E2. 10. Gin A, Hong H, Rosenblatt A, Black M, Ristow B, Popper R. Pseudoaneurysms of the mitral‑aortic intervalvular fibrosa: Survival without reoperation Am Heart J 2011;161:130.e1‑5. 11. Romaguera R, Slack MC, Waksman R, Ben‑Dor I, Satler LF, Kent KM, et al. Percutaneous closure of a left ventricular outflow tract pseudoaneurysm causing extrinsic left coronary artery compression by transseptal approach. Circulation 2010;121:e20‑2. 12. Kassim TA, Lowery RC, Nasur A, Corrielus S, Weissman G, Sears‑Rogan P, et al. Pseudoaneurysm of mitral–aortic intervalvular fibrosa: Two case reports and review of literature. Eur J Echocardiogr 2010;11:E7.

Cite this article as: Joshi SS, Jagadeesh AM, Furtado A, Bhat S. Transesophageal echocardiography in surgical management of pseudoaneurysm of mitral-aortic intervalvular fibrosa with aneurysms of right sinus of Valsalva and left main coronary artery. Ann Card Anaesth 2013;16:40-3. Source of Support: Instutional grants. Conflict of Interest: None declared.

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