Marfan s S drome: Combined Composite Valve GrAeplacement of the Aortic Root and Transaortic Mihal Valve Replacement

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1 Marfan s S drome: Combined Composite Valve GrAeplacement of the Aortic Root and Transaortic Mihal Valve Replacement E. Stanley Crawford, M.D., and Joseph S. Coselli, M.D. ABSTRACT Echocardiographic studies in patients with Marfan s syndrome indicate that 95% eventually have aortic root dilatation, mitral valve prolapse, or a combination of these. Both lesions can progress and require operation for aortic root aneurysm, aortic valve insufficiency, and mitral valve insufficiency. This report reviews the simplified treatment of 6 patients by combined composite valve graft replacement of the aortic root and transaortic mitral valve replacement at one operation. All patients survived and are well for periods extending from 1 month to 12 months. It is estimated that death occurs in one-third of patients with Marfan s syndrome during the first 32 years of life and in two-thirds by the age of 50 years [l]. Death in most is due to cardiovascular manifestations, the most common of which are dilatation of the aortic root and mitral valve prolapse. Echocardiographic studies indicate that 95% of these patients eventually have one or both abnormalities to varying extent [2]. The time of their appearance and natural course are variable and unpredictable. Aortic root dilatation begins early in life, even in utero, but its progression is extremely variable, perhaps being dependent on collagen chemical activity and aortic wall stress. Aortic root complications are rare in the early stages but acute dissection, aortic valve insufficiency, rupture, cardiac tamponade, and sudden death occur with increasing frequency in later life as the aortic root diameter increases to more than 5.0 cm [2]. Aortic valve regurgitation usually begins when annular dilatation reaches 6.0 cm. The latter condition worsens, thereby increasing heart strain, and the incidence of heart failure from abnormal valve function and the incidence of simple aortic rupture or dissection increase with progressive dilatation [2, 31. Several studies indicate that treatment of this condition by either composite valve graft replacement or radical aortic resection, graft replacement, and separate valve replacement is associated with low initial mortality and a significantly better long-term survival than can be expected in the natural course of the disease [%6]. It is From the Department of Surgery, Baylor College of Medicine and The Methodist Hospital, Houston, TX. Accepted for publication Oct 15, Address reprint requests to Dr. Crawford, 6535 Fannin St, MS 8-405, Houston, TX now advised that this form of treatment be routinely recommended when the aortic root diameter is double the size of the uninvolved distal aorta or when the diameter reaches 5.5 to 6 cm [3]. Mitral valve prolapse is ultimately present in 80% of patients with Marfan s syndrome [2]. This abnormality is usually benign in its earlier stages and does not require special treatment other than follow-up and prophylactic antibiotic therapy for infection. Serious valve dysfunction occurs in 25% of the patients with this defect [2]. The rate of progression is unpredictable; however, serious mitral regurgitation may be observed at all ages. In fact, this complication is the most common indication for cardiac operation in children with Marfan s syndrome. Generally, the indications for operation for this condition are similar to those in patients who do not have Marfan s syndrome. The indications and the technique of operation vary according to the presence of other lesions requiring treatment as well as the severity and nature of the mitral valve lesion or lesions. The aortic root and mitral valve abnormalities just described can coexist, as indicated earlier, and the complications with which they are associated can emerge simultaneously. Surgical correction of both sets of abnormalities at one operation is desirable, and the incidence of previous application of this concept in patients with Marfan s syndrome ranges from 4% (2/50) in the series of Gott and colleagues [3], to 21% (5/24) in the series of Kouchoukos and associates [5], and 22% (4/18) in the report of Cabrol and co-workers [4]. These authors treated the aortic root complications with composite valve grafts. Gott [3], Cabrol [4], and their co-workers dealt with the mitral valve regurgitation either by repair or replacement. Kouchoukos and colleagues [5] opted for mitral valve replacement. Although it is not clear from the reports, it appears that these authors performed the mitral operation through separate atrial incisions. Of the 130 patients with Marfan s syndrome referred to us for operation, most had complicated aortic problems of varying segments up to and including the entire aorta. Nine (7%) of these patients, however, required combined treatment of aortic root complications and mitral valve regurgitation. Transaortic mitral valve replacement as described by Carmichael and associates [7] for valvular endocarditis was employed in combination with aortic root reconstruction in the more recently treated patients. This report is concerned with the indications for operation and the technique of a simplified combined procedure. 296 Ann Thorac Surg 45:29&302, Mar Copyright by The Society of Thoracic Surgeons

2 297 Crawford and Coselli: Combined Operation for Marfan s Syndrome A B Fig 1. (A) Anteroposterior and (B) lateral roentgenograms showing the nature of the cardiac enlargement in Patient 6 with an aneurysm of the aortic root, aortic insufficiency, and mitral insufficiency. The presence of the aortic root aneurysm is not evident. Material and Methods There were 4 men and 2 women whose ages ranged from 26 to 39 years. All patients had the four classic manifestations of Marfan s syndrome. Aortic root dilatation and mitral valve regurgitation were known to have been present for varying periods of time. The patients were referred for surgical treatment because of increasing size of the aortic root and/or onset of complications related to worsening mitral regurgitation. The latter were manifested by progressive enlargement of the left atrium and onset of atrial fibrillation, which became difficult to control even by electroshock therapy. Recent deterioration of heart function was evident by a change in New York Heart Association Functional Class from 1 to 2 in 2 patients, 1 to 3 in 1 patient, and 2 to 4 in 1 patient. Plain roentgenograms of the chest at the time of admission showed cardiac enlargement but, as expected, no signs of aortic root dilatation (Fig 1). Echocardiographic examination, however, revealed aortic root diameters that ranged from 4.6 to 7.0 cm (mean, 6.0 cm). Cardiac Doppler studies indicated the presence of severe aortic valve and mitral valve regurgitation. These findings were confirmed by angiographic examination performed both for this purpose and for determining the extent of aortic involvement and the status of the coro- nary arteries (Fig 2). This method of study showed that the aortic root in the patient with enlargement to 4.6 cm by echocardiography was more than twice the diameter of the uninvolved distal ascending aorta. Operative Technique The aortic dilatation and the incompetency of the aortic and mitral valves were treated at one operation. The ascending aorta and aortic valve were replaced with prefabricated St. Jude Medical composite valve grafts (albuminized just before insertion) ranging in size from 25 to 31 mm. The valve end of the graft was attached proximally to the aortic annulus and the distal end, to the transected uninvolved distal ascending aorta. Two methods for coronary artery reattachment were employed depending on the method of dealing with the aorta. In one, used in the first 4 patients in this series, the aortic root and ascending aorta were totally excised except for islands of aortic tissue from which the coronary arteries arose. The mobilized coronary arteries were reattached by suturing the islands of aortic tissue to openings made in the graft and incorporating a circular strip of Teflon felt to add strength and protect the anastomosis (Fig 3). The aortic root was not completely excised in the last 2 patients, and the coronary arteries were reattached with an albuminized 10-mm woven Dacron graft (Fig 4). One end of this graft was sutured around the origin of the left coronary artery (see Fig 4F). The composite valve graft was inserted next, and then the small graft was placed behind and alongside the right side of the composite valve graft and its end sutured around the origin of the right coronary

3 298 The Annals of Thoracic Surgery Vol 45 No 3 March 1988 A Fig 2. (Patient 6.) Cineangiogram and line interpretation showing the aneu ysm (A) of the aortic root and the mitral valve insufficiency. (LA = left atrium; LV = left ventricle.) artery. Coronary artery reattachment was completed by side-to-side anastomosis of both grafts (see Fig 4H). Inclusion technique was not employed. Regardless of the method chosen for treatment of the aorta and aortic valve abnormalities, the mitral valve was replaced first through the aortic annulus opening after the aortic valve leaflets were excised (see Fig 4C-E). The anterior leaflet of the mitral valve and its chordae were removed first; a 10-mm rim of valve leaflet was left near its aortic annulus attachment (see Fig 4C). The posterior leaflet was next partially excised, leaving its posterior segment with attached chordae-papillary muscle complex. St. Jude Medical valves ranging from 25 to 31 mm in size were inserted with interrupted pledgeted sutures first placed in the edges of residual valves and then through the sewing ring of the prosthetic valves (see Fig 4D, E). The operation was completed by inserting the composite valve graft as described. The average total aortic clamp time was 133 minutes for operations that included total aortic excision and 102 minutes for the two operations that did not. The average time for mitral valve replacement was 20 minutes. Moderate (26 C rectal temperature) systemic hypothermia and frequent, intermittent (total, 6 to 8 L) coronary artery perfusion with cold (8 C) hyperkalemic dilute blood cardioplegic solution were used during aortic occlusion. Pathology All lesions grossly and histologically appeared to be degenerative in origin and characteristic of those observed in Marfan s syndrome. The mitral annuli were extremely dilated, both valve leaflets were massively redundant, and all chordae were elongated and attenuated. All patients had multiple ruptured chordae involving either one leaflet or both leaflets. In most cases, rupture was confined to a cluster of chordae (two to four) that inserted into the central edge of the posterior leaflet. Thus, repair would have required partial excision and reconstruction of leaflets, shortening or transferring multiple chordae or both, and reduction in diameter of the annulus. Results All patients survived and were discharged in normal sinus rhythm 10 to 15 days (average, 11 days) after operation. Warfarin sodium therapy was started 4 days after operation, and prothrombin times ranged from 19 to 22 seconds at the time of discharge. Studies performed prior to discharge indicated that the valves were functioning normally and that all suture lines were intact without perivalvular leaks or false aneurysms (Fig 5). Current follow-up ranging from 1 month to 12 months has been completed. All patients are living and have resumed normal activities. All have maintained normal sinus rhythm, and none have experienced either thromboembolic disturbances or complications of anticoagulant therapy. Comment The good early results in this small group of patients are related to a number of factors. All of the patients were young and free from associated noncardiovascular disease. Although the cardiovascular lesions were producing substantial abnormal hemodynamic change and their nature had approached the degree that more serious disturbances were certain, none had advanced sufficiently to impose major risk to operation or to the point of irreversibility. The aortic root lesions had not dis-

4 299 Crawford and Coselli: Combined Operation for Marfan s Syndrome a Fig 3. Method of aneu ysm removal and coronary artery reattachment in Patients 1 through 4. (a) The sinus and tubular segments of the ascending aorta have been excised, leaving islands of aorta from which the corona y arteries arise. The aortic valve leaflets have been removed, and the valve end of the graft has been sutured to the aortic annulus and proximal residual aorta in one bite. (b) The coronary artery is mobilized to gain length and is reattached to an opening made in the graft. Reattachment is reinforced with a washer of Teflon felt. (c) The right coronay artery is reattached with a similar technique. sected or ruptured, and the valve lesions had not produced irreversible left ventricular changes. Consequently, to date, operation has restored cardiac function to normal, and risky aortic root complications have been virtually eliminated. We believe the early indications used in recommending operation in these patients seem justified, and suggest that they be adopted for future use. The indications for aortic operation in these patients are well established, and surgeons who operate on the mitral valve have always insisted that the results of mitral valve surgery are best if the operation is performed when atrial fibrillation can be reversed (operation performed within one year of onset) and before irreversible left ventricular changes develop. The results also argue that the techniques employed were a favorable factor in outcome. A one-stage operation correcting all abnormalities, performed with an economy of time, and lacking in complications, satisfies all technical requirements. We have used the various operations designed to treat aortic root disease in patients with and without Marfan s syndrome. Currently, the technique described using a 10-mm Dacron graft to reattach the coronary arteries according to the method of Cabrol and associates [4] is preferred regardless of the location of the coronary artery ostia or the etiology except in patients with acute dissection extending into the coronary arteries. In these patients, the coronary arteries are best reattached by separate ascending aorta (grafttdistal coronary artery vein bypass grafts. The advantages of the former procedure include the ability to reattach without tension in all instances, routine hemostasis, and less time for its performance. The transaortic approach provided excellent exposure for easy, rapid, accurate mitral valve replacement without the need for excess retraction and without the risk of injury to the heart and bleeding from a cardiac suture line, as can occur after replacement through separate atrial incisions. Staging the operation (performing the mitral valve procedure at one operation and the aortic root operation at another in patients who need both) is unnecessary and contraindicated in most instances. The aortic root lesion may rupture during the recovery period after the mitral valve operation, and persistent mitral valve disease may retard or prevent recovery from the aortic root operation. Moreover, the mitral valve procedure is more difficult to perform as a second operation. A final and important point is the selection of the proper mitral valve operation in these patients. The current trend is to repair degenerative valves to avoid thromboembolic disease and complications of anticoagulant therapy, and possibly to reduce the need for reoperation. We recommend this approach when it is possible, and suggest the techniques advocated by Carpentier

5 300 The Annals of Thoracic Surgery Vol45 No 3 March 1988 Fig 4. Method of aortic reconstruction in Patients 5 and 6 and method of mitral valve replacement in all patients. (A, B) The ascending aortic aneu rysm is opened longitudinally, the walls are retracted to expose the sinus segment, and the aortic valve leaflets are excised. (C, D) The anterior or aortic leaflet of the mitral valve is removed first. The posterior or mural leaflet is then partially removed, sparing the posterior chordae-papillary complex and associated leaflet. lnterrupted pledgeted sutures are placed through the sewing ring of the prosthetic valve. (E, F) The prosthetic mitral valve is seated, and its sutures are tied. Similar sutures are placed into the aortic valve annulus. One end of an albuminized 10-mm woven Dacron tube is sutured about the origin of the left coronary artery. (G, H) The aortic annulus sutures are placed through the sewing ring of the composite valve graft and tied. The distal end of the composite valve graft is cut to the desired length and sutured end-to-end to the distal uninvolved ascending aorta. The 10-mm Dacron tube is placed behind and alongside the aortic graft on the right, and the free end is sutured around the origin of the right coronary artery. Coronary artery reattachment is completed by a sideto-side anastomosis of both grafts.

6 301 Crawford and Coselli: Combined Operation for Marfan s Syndrome n

7 302 The Annals of Thoracic Surgery Vol45 No 3 March 1988 Fig 5. (Patient 6.) Aortograms and line interpretations showing the composite valve graft in place and functioning. [8] in the patient with isolated mitral valve insufficiency; however, the stated indications for repair become moot in the patient who requires both composite valve graft replacement of the aortic root and correction of mitral insufficiency because the former requires long-term anticoagulation. This eliminates most of the advantages of mitral valve repair. Last, repair requires a degree of expertise that permits completion of the mitral repair and allows time for the aortic root reconstruction, which is the more immediate life-threatening lesion. Even experts may require two to three hours for repair, and they suggest that this operation may not be possible in 10% of patients and is contraindicated in patients requiring multiple valve replacement [9, 101. Until these matters are settled, com- bined aortic root replacement and transaortic mitral valve replacement with the St. Jude Medical valve at one operation performed at a stage when normal sinus rhythm can be maintained is the preferred operation for the average surgeon. Its application in other situations in which the method of mitral valve operation is not an issue has been covered by Carmichael and associates (71. References 1. Murdoch JL, Walker BA, Halpern BL, et al: Life expectancy and causes of death in the Marfan syndrome. N Engl J Med 286:804, Pyeritz RE: Heritable disorders of connective tissue. In Fortuin NJ (ed): Current Therapy in Cardiovascular Disease. Philadelphia, Decker, 1987, pp Gott VL, Pyeritz RE, Magovern GJ Jr, et al: Surgical treatment of aneurysms of the ascending aorta in the Marfan syndrome: results of composite-graft repair in 50 patients. N Engl J Med 314:1070, Cabrol C, Pavie A, Mesnildrey P, et al: Long-term results with total replacement of the ascending aorta and reimplantation of the coronary arteries. J Thorac Cardiovasc Surg 91:17, Kouchoukos NT, Marshall WG Jr, Wedige-Stecher TA: Eleven-year experience with composite graft replacement of the ascending aorta and aortic valve. J Thorac Cardiovasc Surg 92:691, Miller DC, Stinson EB, Oyer PE, et al: Concomitant resection of ascending aortic aneurysm and replacement of the aortic valve. J Thorac Cardiovasc Surg 79:388, Carmichael MJ, Cooley DA, Favor AS: Aortic and mitral valve replacement through a single transverse aortotomy: a useful approach in difficult mitral valve exposure. Tex Heart Inst J 10415, Carpentier A Cardiac valve surgery: the "French correction.'' J Thorac Cardiovasc Surg 86:323, Spencer FC: Discussion of Angel1 WW, Oury JH, Shah F: A comparison of replacement and reconstruction in patients with mitral regurgitation. J Thorac Cardiovasc Surg 93:672, Spencer FC, Colvin SB, Culliford AT, Isom OW: Experiences with the Carpentier techniques of mitral valve reconstruction in 103 patients ( ). J Thorac Cardiovasc Surg 94341, 1987 Addendum Since submission of this manuscript, we have employed graft replacement of the ascending aorta for mycotic aneurysm and separate replacement of aortic valve (No. 23 St. Jude Medical) and transaortic mitral valve (No. 25 St. Jude Medical) in a non-marfan patient with chronic subacute bacterial endocarditis and acute heart failure. All 7 patients continue to do well 5 to 17 months after operation.

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