Quadricuspid aortic valve (QAV) is a rare congenital lesion, generally      manifesting with valve regurgitation. Standard treatment involves valve      replacement, though anecdotal cases of successful repair by means of valve      tricuspidization have been reported. Here, the successful application of a repair      technique previously unreported in the setting of QAV is described

Alessandro Mazzucco

Giovanni Battista Luciani

Giuseppe Faggian

Mohammed Morjan

Interactive Cardiovascular and Thoracic Surgery

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Repair of quadricuspid aortic valve by bicuspidization: a novel technique

LUCIANI, GIOVANNI BATTISTA

MORJAN, MOHAMMED

FAGGIAN, Giuseppe

MAZZUCCO, Alessandro

Catalogo dei prodotti della ricerca Università degli Studi di Verona

ARTICLE IN PRESSwww.icvts.orgdoi:10.1510/icvts.2010.237404Interactive CardioVascular and Thoracic Surgery 11 (2010) 348–350 2010 Published by European Association for Cardio-Thoracic SurgeryCase report - CongenitalRepair of quadricuspid aortic valve by bicuspidization: a noveltechniqueGiovanni Battista Luciani*, Mohammed Morjan, Giuseppe Faggian, Alessandro MazzuccoDivision of Cardiac Surgery, University of Verona, O. C. M. Piazzale Stefani 1, 37126 Verona, ItalyReceived 9 March 2010; received in revised form 1 June 2010; accepted 2 June 2010AbstractQuadricuspid aortic valve (QAV) is a rare congenital lesion, generally manifesting with valve regurgitation. Standard treatment involvesvalve replacement, though anecdotal cases of successful repair by means of valve tricuspidization have been reported. Here, the successfulapplication of a repair technique previously unreported in the setting of QAV is described. 2010 Published by European Association for Cardio-Thoracic Surgery. All rights reserved.Keywords: Quadricuspid aortic valve; Bicuspid aortic valve; Aortic valve repair; Bicuspidization; Tricuspidization; Aortic insufficiency; Valve repair1. IntroductionQuadricuspid aortic valve (QAV) is a congenital anomalyrarely seen in clinical practice with incidence rates rangingfrom 0.008% to 0.04% w1, 2x. Most cases have been discov-ered as incidental finding at surgery or autopsy. Valveinsufficiency is the most frequent abnormality in patientswith QAV. The management of regurgitant QAV commonlyinvolves valve replacement. More recently, a few cases ofvalve repair have been reported w3–5x: in all these cases,tricuspidization of the aortic valve was used to restorecompetence. Here, we report is a case of QAV, which wasrepaired using an alternative technique.2. Case reportA 68-year-old-man was referred to our center for severeaortic incompetence identified during his admission forheart failure at a nearby hospital. He complained of severedyspnea and presented hypertension and diabetes as co-morbidities. The chest film showed moderate cardiomegaly.Surface EKG demonstrated regular signs rhythm with signsof left heart hypertrophy. Trans-thoracic echocardiographicexamination showed severe aortic regurgitation with unde-fined valve morphology, left ventricular dilatation withmoderate systolic dysfunction (ejection fractions42%).The results were confirmed at angiography, which alsoshowed mild ascending aortic ectasia (42 mm), while cor-onary lesions were excluded. Trans-esophageal echocardi-ography performed during surgery, revealed QAV anatomy(Fig. 1a) with severe regurgitation, due to a larger centraljet caused by lack of coaptation and multiple finer para-*Corresponding author. Tel.: q39-45-8122485; fax: q39-45-8123308.E-mail address: giovanni.luciani@univr.it (G.B. Luciani).commissural jets. Via median sternotomy, under normo-thermic cardiopulmonary bypass, using retrograde andselective antegrade blood cardioplegia, the aortic valvewas inspected through a transverse aortotomy. The valvecomprised of four unequal sized cusps: a larger-sized leftcoronary, a medium-sized right coronary and two smallernon-coronary leaflets, classified as type G according toHurwitz and Roberts w2x (Fig. 2a,b). The latter two, inaddition, were proplapsing and large fenestrations werepresent at the commissural level in all leaflets. Repair wasperformed by detachment of the inter-non-coronary com-missure and plication of the new non-coronary leaflet usinga locking 5-0 polypropylene suture (Fig. 2c). Due to per-sistence of prolapse and the presence of large fenestrationsat the left-to-non-coronary commissure, the latter commis-sure was demolished (Fig. 2d) and the non-coronary leafletwas then anchored to the left coronary leaflet, therebyturning the valve from quadricuspid to bicuspid (Fig. 2e,f).Furthermore, all remaining fenestration were closed withrunning 7-0 polypropylene suture. Finally, sub-commissuralanuloplasty with interrupted pledgeted 4-0 polypropylenesutures was carried out to allow insertion of a size 20 Hegardilator. Post-repair trans-esophageal echocardiographyshowed trivial residual central regurgitation and minimalstenosis (mean pressure gradients16 mmHg), with normalorifice area (s3.80 cm or 1.73 cm ym ) (Fig. 1b,c).2 2 2Patient recovery was uneventful with discharge on postop-erative day 5. Discharge trans-thoracic echocardiographicexamination confirmed intraoperative findings. At 18-month follow-up, the patient is symptom-free and trans-thoracic echocardiographic examination showed mild aorticregurgitation, with effective height of coaptation of 11 mm(Fig. 1d), preserved leaflet mobility and improved leftventricular function (ejection fractions61%).ARTICLE IN PRESS349G.B. Luciani et al. / Interactive CardioVascular and Thoracic Surgery 11 (2010) 348–350NewIdeasInstitutionalReportWorkinProgressReportESCVSArticleNegativeResultsState-of-the-artBestEvidenceTopicBriefCommunicationCaseReportFollow-upPaperEditorialProtocolProposalforBail-outProcedureNomenclatureHistoricalPagesFig. 1. (a) Pre-repair intraoperative trans-esophageal short-axis view of the aortic valve during diastole. Four unequal size cusps are present; (b) post-repairintraoperative trans-esophageal short-axis view of the aortic valve during systole showing bicuspid morphology and normal aortic orifice area (measured3.80 cm ); (c) post-repair intraoperative trans-esophageal long-axis diastolic view showing trivial central jet regurgitation at Doppler interrogation; (d) follow-2up trans-thoracic para-sternal long-axis view during diastole documenting effective height of leaflet coaptation (measured 11 m).Fig. 2. Operative anatomy and surgical steps. (a) Quadricuspid aortic valve is present, with four unequal-sized leaflets (type G): the two smaller non-coronaryleaflets also present multiple large fenestrations; (b) schematic of operative anatomy; (c) schematic of operative steps: detachment of inter-non-coronarycommissure; (d) after non-coronary leaflet reconstruction, detachment of left-non-coronary commissure; (e) completed repair: the right coronary leaflet hasbeen preserved, while the non-coronary leaflets have been joined and then anchored to the left coronary leaflet. The former now represents the smaller anteriorleaflet, the latter the larger posterior leaflet; (f) operative view of completed bicuspidization.ARTICLE IN PRESS350 G.B. Luciani et al. / Interactive CardioVascular and Thoracic Surgery 11 (2010) 348–3503. DiscussionSeven anatomical variants of QAV have been described byHurwitz and Roberts w2x, based on the relative size of thefour cusps. Over 85% of the reported cases belong to typeA, B, or C. Most patients with this anomaly have requiredaortic valve surgery, which has traditionally consisted ofprosthetic valve replacement. Valve repair has seldom beenreported, mostly applied to type A, B or C w3–5x and hasthus far entailed tricuspidization. Tricuspidization has orig-inally been proposed for the repair of regurgitant tricuspidas well as bicuspid aortic valves in children and youngadults w6x. However, when compared to bicuspidization orpreservation of native bicuspid valve, tricuspidization hasbeen found to be a risk factor for late repair failure w6x.On the contrary, bicuspidization has been applied success-fully to tricuspid, unicuspid and bicuspid aortic valve anat-omy w6, 7x. Similar to the high success rate observed withmitral valve repair, the experience of Scha¨fers and col-leagues suggests that valve competence and stability arebetter achieved in bicuspid rather than tricuspid valves dueto the presence of fewer coaptation lines w6, 7x. The casereported here supports this hypothesis by showing thatbicuspidization may successfully be applied even to farmore complex and rare morphologies, such as type G QAV,where the presence of four unequal cusps makes tricupi-dization geometrically difficult to apply. In agreement withprior clinical evidence on bicuspidization of unicusp aorticvalves w7x, this technique allows restoration of valve com-petence without significant residual trans-valvar pressuregradients, as is confirmed by mid-term follow-up in thepresent case. Although long-term observation after repairof QAV, particularly for the more uncommon variants, isunavailable, incidental findings of normally functioning QAVin the elderly would suggest this aortic valve anatomy isnot necessarily pathological. Identical considerations applyfor native (and, possibly, surgically created) bicuspid aorticvalves. Therefore, theoretical grounds exist for a durablevalve function after bicuspidization of QAV.Referencesw1x Timperley J, Milner R, Marshall AJ, Gilbert TJ. Quadricuspid aorticvalves. Clin Cardiol 2002;25:548–552.w2x Hurwitz LE, Roberts WC. Quadricuspid semilunar valve. Am J Cardiol1973;31:623–626.w3x Naito K, Ohteki H, Yunoki J, Hisajima K, Sato H, Narita Y. Aortic valverepair for quadricuspid aortic valve associated with aortic regurgitationand ascending aortic aneurysm. J Thorac Cardiovasc Surg 2004;128:759–760.w4x Jeanmart H, de Kerchove L, El Bitar F, Glineur D, Vandyck M, NoirhommeP, El Khoury G. Tricuspidation of quadricuspid aortic valve: case reports.J Heart Valve Dis 2007;16:148–150.w5x Schmidt KI, Jeserich M, Aicher D, Scha¨fers HJ. Tricuspidization of thequadricuspid aortic valve. Ann Thorac Surg 2008;85:1087–1089.w6x Langer F, Aicher D, Kissinger A, Wendler O, Lausberg H, Fries R, Scha¨fersHJ. Aortic valve repair using a differentiated surgical strategy. Circu-lation 2004;110(11 Suppl 1):II67–II73.w7x Scha¨fers HJ, Aicher D, Riodionycheva S, Lindinger A, Ra¨dle-Hurst T,Langer F, Abdul-Khaliq H. Bicuspidization of the unicuspid aortic valve:a new reconstructive approach. Ann Thorac Surg 2008;85:2012–2018.

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Repair of quadricuspid aortic valve by bicuspidization: a novel technique

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