Accessory causing aortic regurgitation outflow obstruction Case … · Accessorymitalvalveleaflet...

7
Br Heart J 1988;59:491-7 Accessory mitral valve leaflet causing aortic regurgitation and left ventricular outflow tract obstruction Case report and review of published reports JUN SONO,* ROXANE McKAY, ROBERT M ARNOLD From the Royal Liverpool Children's Hospital SUMMARY Arrhythmias, aortic regurgitation, and symptoms of severe intermittent ventricular outflow obstruction developed in a 14 year old boy with a heart murmur who had been followed from infancy. These were caused by an accessory mitral leaflet, which was successfully removed at open heart operation. A review of 21 previously reported cases found a high incidence of associated cardiac malformations, appreciable subaortic obstruction in most patients, and a consistent attachment of the accessory tissue to the ventricular aspect of the anterior mitral leaflet. The characteristic echocardiographic appearance of a mobile mass arising from the area of aortic-mitral continuity is sufficient for the diagnosis of accessory mitral leaflet and echocardiographic examination will facilitate the surgical management of this condition. Among the more rare causes of subaortic obstruction are several mitral valve anomalies, including abnor- mal insertion within the left ventricular outflow tract, prolapse of redundant chordae or a leaflet, and accessory tissue in the subaortic area. Such accessory tissue is uncommon in normally connected hearts. It is a condition singularly amenable to repair without valve replacement. We report a case with several unusual clinical features that illustrates the value of real time echocardiography in diagnosing left ventricular outflow tract obstruction with aortic regurgitation caused by an accessory mitral leaflet. Case report A 14 year old boy was admitted to hospital with a two month history of a central stabbing chest pain. He had been seen first at 11 months for an asymptomatic heart murmur that was present at birth, and right heart catheterisation subsequently confirmed the clinical diagnosis of a small ventricular septal defect. Routine outpatient follow up found no change in his Requests for reprints to Dr Roxane McKay, FRCS, The Royal Liverpool Children's Hospital, Myrtle Street, Liverpool L7 7DG. *Present address: Kobe City General Hospital, Minatojima, Chuo-Ku, Kobe 650, Japan. Accepted for publication 19 November 1987 physical signs until an early diastolic murmur was heard when he was 14. The patient then recalled several episodes of sharp precordial pain precipitated by strenuous exercise, as well as palpitation and giddy spells unrelated to effort. The relevant physical findings included an irregularly irregular waterhammer pulse at 80 beats/ minute, a systemic blood pressure of 130/70 mm Hg, and an easily palpable thrill along the left sternal border. This corresponded to a harsh systolic ejec- tion murmur radiating widely over the precordium as well as to the patient's neck and back. In addition, there was a grade 2/4 immediate diastolic murmur. The electrocardiogram showed a bizarre pattern of multifocal ventricular extrasystoles, some periods of sinus rhythm with intermittent right bundle branch block, and other periods of junctional rhythm. There was no evidence of ventricular hypertrophy or ischaemia. Chest x ray showed a cardiothoracic ratio of 0-53 with pulmonary vasculature. The remainder of the patient's physical examination and laboratory investigations were within normal limits. Cross sectional echocardiography showed a pedunculated mass originating just beneath the non- coronary cusp. The mass had prolapsed through the aortic valve during systole (fig 1). In addition, there was a large aneurysm of the membranous septum. Pulsed Doppler examination showed moderately 491 on August 6, 2020 by guest. Protected by copyright. http://heart.bmj.com/ Br Heart J: first published as 10.1136/hrt.59.4.491 on 1 April 1988. Downloaded from

Transcript of Accessory causing aortic regurgitation outflow obstruction Case … · Accessorymitalvalveleaflet...

Page 1: Accessory causing aortic regurgitation outflow obstruction Case … · Accessorymitalvalveleaflet causingaortic regurgitation andleft ventricular outflow tract obstruction Table Summaryofreportedcases

Br Heart J 1988;59:491-7

Accessory mitral valve leaflet causing aorticregurgitation and left ventricular outflow tract

obstructionCase report and review ofpublished reports

JUN SONO,* ROXANE McKAY, ROBERT M ARNOLD

From the Royal Liverpool Children's Hospital

SUMMARY Arrhythmias, aortic regurgitation, and symptoms of severe intermittent ventricularoutflow obstruction developed in a 14 year old boy with a heart murmur who had been followedfrom infancy. These were caused by an accessory mitral leaflet, which was successfully removed at

open heart operation. A review of21 previously reported cases found a high incidence ofassociatedcardiac malformations, appreciable subaortic obstruction in most patients, and a consistentattachment of the accessory tissue to the ventricular aspect of the anterior mitral leaflet. Thecharacteristic echocardiographic appearance ofa mobile mass arising from the area ofaortic-mitralcontinuity is sufficient for the diagnosis of accessory mitral leaflet and echocardiographicexamination will facilitate the surgical management of this condition.

Among the more rare causes of subaortic obstructionare several mitral valve anomalies, including abnor-mal insertion within the left ventricular outflow tract,prolapse of redundant chordae or a leaflet, andaccessory tissue in the subaortic area. Such accessorytissue is uncommon in normally connected hearts. Itis a condition singularly amenable to repair withoutvalve replacement. We report a case with severalunusual clinical features that illustrates the value ofreal time echocardiography in diagnosing leftventricular outflow tract obstruction with aorticregurgitation caused by an accessory mitral leaflet.

Case report

A 14 year old boy was admitted to hospital with a twomonth history of a central stabbing chest pain. Hehad been seen first at 11 months for an asymptomaticheart murmur that was present at birth, and rightheart catheterisation subsequently confirmed theclinical diagnosis of a small ventricular septal defect.Routine outpatient follow up found no change in his

Requests for reprints to Dr Roxane McKay, FRCS, The RoyalLiverpool Children's Hospital, Myrtle Street, Liverpool L7 7DG.

*Present address: Kobe City General Hospital, Minatojima, Chuo-Ku, Kobe650, Japan.

Accepted for publication 19 November 1987

physical signs until an early diastolic murmur washeard when he was 14. The patient then recalledseveral episodes ofsharp precordial pain precipitatedby strenuous exercise, as well as palpitation andgiddy spells unrelated to effort.The relevant physical findings included an

irregularly irregular waterhammer pulse at 80 beats/minute, a systemic blood pressure of 130/70mm Hg,and an easily palpable thrill along the left sternalborder. This corresponded to a harsh systolic ejec-tion murmur radiating widely over the precordium aswell as to the patient's neck and back. In addition,there was a grade 2/4 immediate diastolic murmur.The electrocardiogram showed a bizarre pattern ofmultifocal ventricular extrasystoles, some periods ofsinus rhythm with intermittent right bundle branchblock, and other periods of junctional rhythm. Therewas no evidence of ventricular hypertrophy orischaemia. Chest x ray showed a cardiothoracic ratioof 0-53 with pulmonary vasculature. The remainderof the patient's physical examination and laboratoryinvestigations were within normal limits.

Cross sectional echocardiography showed a

pedunculated mass originating just beneath the non-coronary cusp. The mass had prolapsed through theaortic valve during systole (fig 1). In addition, therewas a large aneurysm of the membranous septum.Pulsed Doppler examination showed moderately

491

on August 6, 2020 by guest. P

rotected by copyright.http://heart.bm

j.com/

Br H

eart J: first published as 10.1136/hrt.59.4.491 on 1 April 1988. D

ownloaded from

Page 2: Accessory causing aortic regurgitation outflow obstruction Case … · Accessorymitalvalveleaflet causingaortic regurgitation andleft ventricular outflow tract obstruction Table Summaryofreportedcases

beneath the commissure between non-coronary andright cusps there was a wide-mouthed aneurysm (20mm in diameter) of the membranous septum extend-ing into the right ventricle. There was no ventricularseptal defect; but, at the lower margin of theaneurysm, a discrete ring of fibrous tissue extended

_ * into the outflow tract for about 5 mm and around halfits circumference. This did not correspond with theechocardiographic appearances, however, and ac-cordingly a search was made deeper within the leftventricle. This disclosed a mass of soft white tissueattached in several places to the ventricular surface of

_~fi--- . the anterior mitral leaflet and, on the other side, bywell defined chordae, to a small papillary muscle nearthe ventricular septum (fig 2). This tissue wascompletely separate from both the subaortic mem-brane and the aneurysm of the membranous septum(fig 3).

Fig 1 Cross sectional long axis echocardiograms in systole(a) and diastole (b) showing the prolapse of a large mass of...................tissue (small arrows) through the aortic valve duringventricular ejection. This tissue fell back against theaneurysm of the membranous septum (large arrow) duringdiastole. VS, ventricular septum; LV, left ventricle;AO, aorta; LA, left atrium; AM, accessory mitral leaflet.

severe aortic regurgitation and a velocity of 2-3metres/second across the left ventricular outflowtract., indicating a gradient of approximately 21mm Hg. Repeat cardiac catheterisation andangiography confirmed the echocardiographic find-ings and excluded any intracardiac shunt.

Despite the absence of clinical or laboratory find-ings to suggest bacterial endocarditis, the echocar-diographic appearances were thought to be consis-tent with a large vegetation and the patient wastreated with intravenous antibiotics. Four days after Fig 2 Transluminated surgical specimen showing the aorticcatheterisation his giddy spells recurred, and urgent (a) and ventricular (b) sides of the accessory mitral leaflet.

tranaortceplortionof he lft vntrcula outlowThe papillary muscle arose near the normal anterolateral

trantwsaotcarrexplorationofaheletienrulmoary outflow papillary muscle and the upper lip was attached to the back

wtrath aoearredhyouthemo n dcardiopulmon caryrypss. of the anterior mitral leaflet in several places. A rudimentarywith oderaehyotheria an cariopleic arest. chordal architecture and cusp differentiation is apparent but

The aortic valve was normal, and immediately the leaflet was imperforate.

Sono, McKay, Arnold492

on August 6, 2020 by guest. P

rotected by copyright.http://heart.bm

j.com/

Br H

eart J: first published as 10.1136/hrt.59.4.491 on 1 April 1988. D

ownloaded from

Page 3: Accessory causing aortic regurgitation outflow obstruction Case … · Accessorymitalvalveleaflet causingaortic regurgitation andleft ventricular outflow tract obstruction Table Summaryofreportedcases

Accessory mital valve leaflet causing aortic regurgitation and left ventricular outflow tract obstruction

Fig 3 Diagram of the operativefindings showing therelation of the accessory mitral leaflet to the aortic valve andto the aneurysm of the membranous septum.

The discrete subaortic membrane was enucleated,'and the accessory mitral leaflet was excised by sharpdissection. The aneurysm of the membranous sep-tum was plicated on its right ventricular side throughthe tricuspid valve. Weaning from cardiopulmonarybypass was smooth and uneventful, as was thepatient's subsequent postoperative recovery. He wasdischarged from hospital seven days after operation,when pulsed Doppler echocardiography showedonly a trace of aortic regurgitation. The electrocar-diogram had returned to normal sinus rhythm.

(fig 4) but these have been accompanied by com-plaints ofexercise intolerance, chest pain on exertion,or syncope in less than one third ofpatients. Usually,the obstruction is severe, with gradients of >50mm Hg measured by cardiac catheterisation or Dop-pler echocardiography. There were major associatedcardiac -defects in 13 of 21 previously describedcases2 512 and in our patient, but these did not suggestany particular developmental pattern (table).Our patient was unusual because he presented with

dominant aortic regurgitation and arrhythmias,which, in view ofhis past history ofventricular septaldefect, suggested bacterial endocarditis. It is possiblethat the aneurysm of the membranous septumprovided a bypass around the obstructing accessorymitral tissue until this tissue became sufficiently largeto prolapse through the aortic valve. The only otherpatient to have such a mild gradient across the leftventricular outflow tract also had an aneurysm of themembranous septum.6 Sudden complete blockage ofaortic outflow would account for the patient's chestpain and giddy spells, but the aetiology of hisarrrhythmias is less clear. One possibility is thatincreased left ventricular pressure caused tension onthe aneurysm and adjacent conduction tissue, result-ing in bundle branch block and the episodes ofnodalrhythm. Alternatively, the accessory leaflet itselfmayhave damaged the conduction tissue as it movedwithin the subaortic area. In either event, theelectrocardiogram would be expected to revert tonormal, as it did, after operation.The diagnosis of this and other types of subaortic

obstruction has been facilitated greatly by real timeechocardiography."3 "1 Although M mode studiesshowed multiple echoes in the subaortic area,'5 sectorscanning clearly differentiates between fixed fibroustissue and movement of an accessory leaflet into theoutflow tract. Whereas a tumour mass or vegetation,theoretically, might produce a similar appearance,

3Discussion

Accessory mitral valve tissue producing subaorticobstruction was first described by MacLean et al in19632; and, in his precise classification, Edwardsincluded this anomaly as a rare cause of subaorticstenosis.3 During the past decade one or two caseshave been reported each year, and it is now possibleto characterise this unusual condition in some detail.The condition usually presents as an asymp-

tomatic heart murmur, although the two youngestpatients had low cardiac output from aortic obstruc-tion at three days of age,4 and congestive heart failureat 14 months.5 Signs of left ventricular outflow tractobstruction generally develop during the first decade

v)'E 2

0..40QL a

o 1*z

oJ WBIVWW'WIVWWVWVI,. , , , , , I , , ,-. , I , ,

c< 1 2 3 4 5 6 7 8 9 10 11 12 13'14>14Age (yr)

Fig 4 Age distribution of 22 patients who werefound tohave a subaortic accessory mitral valve leaflet at operation ornecropsy.

493

on August 6, 2020 by guest. P

rotected by copyright.http://heart.bm

j.com/

Br H

eart J: first published as 10.1136/hrt.59.4.491 on 1 April 1988. D

ownloaded from

Page 4: Accessory causing aortic regurgitation outflow obstruction Case … · Accessorymitalvalveleaflet causingaortic regurgitation andleft ventricular outflow tract obstruction Table Summaryofreportedcases

494

Table Summary of reported cases of accessory mitral leaflet or tissue in normally connected heartsSono, McKay, Arnold

Age (yr)/sex of Gradient Surgical Associated

Author case Presentation (mm Hg) approach Accessory tissue Histology lesions Results

Maclean et al2 29/M Exercise intolerance, 70 Aortotomy 2 parachute-like Situs inversus, Good(1963) murmur since structures (5 mm and 15 right SVC

childhood mm) attached to anteriormitral leaflet

Dealetal'(1963) 11/F Seizures; heart 70 Aortotomy 5 balloon-like masses of Connective Membranous Died POD4.Sellars et al murmur, "Known to spherical tissue tissue similar subaortic stenosis. Accessory(1964) have subaortic to normal Supravalvar mitral tissue not

stenosis" mitral valve ring; bicuspid aortic excisedvalve; single sinusorigin of coronary

Kelly et al'7 (1972) 7/M 130 Ventriculo- On anterior mitral Dextrocardia, Goodtomy leaflet subaortic ring

Cooperberg et al'5 9/M Asymptomatic 80 Aortotomy Parachute-shaped, Good(1976) then attached to anterior

ventriculo- mitral valve ring;tomy chordae attached to

septum and papillarymuscles

Mathewson et al 4 3 d/F Murmur; low cardiac 62 Aortotomy Pedunculated mass Died in(1976) output then originating from operating

ventriculo- midpoint of junction of theatretomy mitral annulus and

anterior leaflet

Freedom et al 12 8 mnths/ 40 None (Post- Nodular gelatinous mass Ventricular septal Died(1977) not mortem of undifferentiated defect, coarctation

known specimen) endocardial tissue fromaortic leaflet of mitralvalve and contiguousmembranous septum

Kohda et al 2/F Heart murmur at 14 50 Aortotomy Accessory tissue attach- Fibrous Mild mitral(1979) months ed to anterior mitral tissue regurgitation

leaflet with 3 chordae

Kuribayashi 3/M Heart murmur at 1 100 Aortotomy White tissue with Fibrous Dextrocardia Goodet al 2' (1979) month; syncope and then left chordae attached to tissue

chest pain on exertion atriotomy anterior leaflet of mitralvalve and septum

Nanton et al 9/F Murmur from 1st year 100 Aortotomy Balloon-like, 2-2 cm x Discrete subaortic Died in low(1979) 1-3 cm. Attached in ridge, muscular cardiac

concave arc below left VSD, accessory output.coronary leaflet and to tricuspid tissue, 2 AccessoryLV wall. Chordae to coronary ostia in tissue notanterolateral papillary left sinus excisedmuscle and lateral wallofLV

Gomes et al 14 Murmur from birth, 90 1.2 cm x 4 cm nodular, Valve-like Partial AV septal Mitral(1980) mnths/ congestive heart flap-like fibrous tissue structure defect; left SVC insufficiency,

M failure attached to base of otherwiseanterior mitral leaflet satisfactory

Tanimoto et al6 10 Heart murmur from 26 Aortotomy Chordae attached to Fibrous Supravalvar aortic Good(1981) mnths/ birth mitral valve ring, tissue stenosis; septal

F aortic annulus, and LV aneurysmfree wall

Hatem et al" 10/F Asymptomatic heart 195 Aortotomy Sheet-like mass under Fibromyoid Bicuspid aortic Residual 110(1981) murmur at 5 weeks aortic valve with tissue valve mm Hg

chordae to septum and consistent gradient;papillary muscles with valve reoperated

tissue via leftatrium;similar massremovedfrom base ofmitral leaflet

on August 6, 2020 by guest. P

rotected by copyright.http://heart.bm

j.com/

Br H

eart J: first published as 10.1136/hrt.59.4.491 on 1 April 1988. D

ownloaded from

Page 5: Accessory causing aortic regurgitation outflow obstruction Case … · Accessorymitalvalveleaflet causingaortic regurgitation andleft ventricular outflow tract obstruction Table Summaryofreportedcases

Accessory mital valve leaflet causing aortic regurgitation and left ventricular outflow tract obstruction

Table Summary of reported cases of accessory mitral leaflet or tissue in normally connected hearts

495

Age (yr)/sex of Gradient Surgical Associated

4uthor case Presentation (mm Hg) approach Accessory tissue Histology lesions Results

Furuta et al 5/M Murmur from birth. 100 Aortotomy 25 cm x 30 mm Myxoidal Ring of fibrous Good(1983) Tumour-like mass then left accessory leaflet with dysplasia. tissue on atrial side

LVOTO on echo atriotomy multiple chordal Normal of anterior mitralattachments to anterior endothelium leafletmitral leaflet and free of mitral cuspwall ofLV

Hotta et al 21 14/M Easy fatiguability; 50 Aortotomy Attached to anterior Collagen fibre Mild aortic(1983) heart murmur at 2j then left mitral leaflet and with myxoid regurgitation

years atriotomy ventricular septum. stroma andChordae connected to endotheliummitral valve chordae

Zin'kovsky and 6/F Breathless on exertion; 100 Aortotomy 20 x 15 mm, arising Like a mitral GoodIgnatow 19 (1984) temporary heart pains from anterior leaflet of valve

mitral valve

Losay et al 14* 9/M Aortic stenosis and 34 Aortotomy 1-5 x 2 cm Situs inversus, Mild aortic(1985) regurgitation "membrane" mitral regurgitation regurgitationBinet et al 22* 4/M Aortic stenosis and 112 Aortotomy Good(1985) regurgitation

2/M Aortic stenosis and 44 Aortotomy VSD Goodregurgitation

Alboliras et al 16 15/M Exertional dizziness 100 "Hood" of accessory Discrete subaortic Residual 23(1985) and dyspnoea; mass tissue on anterior mitral membrane mm Hg

on echo valve leaflet gradient;accessorytissue notexcised

Meldrum-Hanna 7/F Asymptomatic 80 Aortotomy Attached around Reoperated 2et al 23 (1986) murmur leftward part of anterior years later for

mitral leaflet and on to discreteseptum. Short chordae subaorticon free edge stenosis

Ascuitto et al'3 4/M Heart murmur, mass 100 Aortotomy Folds of leaflet-like fib- Myxomatous Trace of(1986) on anterior mitral then left rous tissue attached to dysplasia; aortic

leaflet on echo atriotomy base of anterior mitral abnormal regurgitation.leaflet; chordae to both mitral valve Dopplerpapillary muscle tissue gradient 25

mm Hg

*Three cases reported in two papers.AV, atrioventricular; SVC, superior vena cava; VSD, ventricular septal defect; LVOTO, left ventricular outflow tract obstruction; POD, postoperativeday.

such lesions more commonly originate from cardiacmuscle or build up directly upon the low pressure

side of a heart valve respectively. The degree ofsubaortic obstruction may be estimated from Dop-pler echocardiography,`6 which may avoid the needfor catheterisation. Indeed, recently operation hasbeen recommended without invasive studies.'4Angiography can visualise a mass in the subaorticarea but adds little to the diagnosis, and cardiaccatheterisation probably is required only to inves-tigate associated cardiac malformations.The indication for operation was severe left

ventricular outflow obstruction in eighteen patientsand explorations of an intracardiac mass in two.One neonate who had repair of aortic coarctationsubsequently died and was found to have accessorymitral valve tissue at necropsy." In another patientthe accessory leaflet was discovered during reopera-

tion for residual subaortic obstruction.17All operations were performed on cardiopulmon-

ary bypass, with a variety of approaches to theaccessory leaflet. While aortotomy provides goodaccess,'8.9 identification of the accessory tissue maybe difficult when it is accompanied by subaorticstenosis or has collapsed into an empty left ventricle.8This is because the accessory mitral valve tissuealways lies below any discrete fibrous obstruction.When the relation ofthe accessory leaflet to the mitralvalve cannot be clearly seen through the aorta, orwhen additional malformations are present on theatrial side of the valve,7 a left atrial approach has beenuseful. `520 Although a systemic ventriculotomy alsoprovides good access, . 21 it is likely to compromisecardiac function and is probably unnecessary.The gross appearances of the subaortic accessory

mitral leaflet have been remarkably constant and are

on August 6, 2020 by guest. P

rotected by copyright.http://heart.bm

j.com/

Br H

eart J: first published as 10.1136/hrt.59.4.491 on 1 April 1988. D

ownloaded from

Page 6: Accessory causing aortic regurgitation outflow obstruction Case … · Accessorymitalvalveleaflet causingaortic regurgitation andleft ventricular outflow tract obstruction Table Summaryofreportedcases

496 Sono, McKay, Arnoldnot unlike those of accessory mitral valve tissuedescribed in ventriculoarterial discordance with2324or without25 26 atrioventricular discordance. In allpatients the accessory tissue was attached on theventricular surface ofthe anterior mitral leaflet and inmost it extended to the septal surface or free wall ofthe left ventricle. Both in relative and real terms, theaccessory leaflet tends to be large. In our patient itsdiameter exceeded 25 mm, compared with an aorticannulus of 20 mm; and in all other operated cases thewidth was > 15 mm. Occasionally, a second mass oftissue originates from the membranous septum.81'The extent of differentiation has been variable and

may reflect haemodynamic forces applied to theaccessory tissue within the left ventricular outflowtract. The most primitive, nodular, gelatinous massof undifferentiated tissue was found in an eightmonth old infant'2; but most leaflets are parachute orsail shaped with a variable number of well definedchordae anchoring the ends to either or bothpapillary muscles of the mitral valve, to normalchordae, to the left ventricular free wall, or, as seen inour case, to an accessory papillary muscle. The hood-like structure thus produced presents a concavesurface towards the left ventricle, so that in ven-tricular systole this pouch is distended with bloodand is carried into the subaortic area. Histologicalexamination of the accessory leaflet has generallyshown fibrous tissue with myxoid dysplasia,analogous to dysplastic mitral valve tissue.71320

Because the leaflet is accessory tissue, its removalshould in no way compromise the function of anotherwise normal mitral valve, and the results ofoperation usually have been satisfactory. In fifteenpatients in whom the mass was removed completelyas a primary procedure, there was one death. Thiswas a three day old baby who had a left ven-triculotomy after the tissue could not be identifiedthrough the aorta.4 Two patients had mild mitralinsufficiency attributed in one to simultaneous repairof a partial atrioventricular septal defect5 18; and threepatients (including the one reported here) had trivialaortic regurgitation." 22 All survivors remained orbecame symptom free and none has shown signs ofrecurrent subaortic obstruction. Failure to removethe tissue, however, has been associated with con-siderable early mortality"'0 and a high number ofreoperations,"' 6 as has partial or incomplete relief ofthe subaortic obstruction."12' Since the developmentof real time echocardiography to demonstratereliably the accessory leaflet before operation, therehave been no operative deaths attributed to thislesion.

Little is known about the aetiology and coursewhen an accessory mitral leaflet is present. It hasbeen suggested that anomalies of the mitral valve and

fibrous subaortic stenosis both result from abnormaldevelopment of endocardial cushion tissue.27 Yet,considerable evidence supports the view that discretesubartic stenosis is an acquired malformation,2"3while the anlaga of an accessory leaflet probably ispresent at birth, since the murmur is present early inlife, even when there are no other cardiac lesions.27 Itis likely that the degree of left ventricular outflowobstruction is progressive, because a gradient of 65mm Hg at five years increased to 196 mm Hg by 10years in the only patient who underwent serial leftheart studies." Whether this results from enlarge-ment ofthe accessory leaflet or from narrowing oftheoutflow tract by a discrete membrane is a questionthat echocardiography may answer in the future;certainly when the leaflet eventually prolapsesthrough the aortic valve it produces symptoms ofsevere left ventricular outflow tract obstruction andaortic regurgitation, as seen in our patient.More widespread availability of sector scanning

and Doppler echocardiography may lead to theidentification of an accessory mitral leaflet before thedevelopment of symptoms or important obstructionto left ventricular outflow. While elective, prophylac-tic removal of the tissue does not seem to be justified,careful excision ofthe accessory leaflet can be accom-plished without morbidity or mortality. Accordingly,accessory tissue probably should be removed duringopen heart surgery for any associated cardiac defectsto avoid residual and, possibly, progressive leftventricular outflow obstruction.'6

J S was supported by the National Heart ResearchFund. We thank Dr Audrey Smith for the prepara-tion of figs 1 and 2, Dr Maciej Karolczak fortranslation of reference 19 from the original Russian.

References

1 McKay R, Ross DN. Technique for the reliefof discretesubaortic stenosis. J Thorac Cardiovasc Surg 1982;84:917-20.

2 MacLean LD, Culligan JA, Kane DJ. Subaorticstenosis due to accessory tissue on the mitral valve. JThorac Cardiovasc Surg 1963;45:382-7.

3 Edwards JE. Pathology of left ventricular outflow tractobstruction. Circulation 1965;31:586-99.

4 Mathewson JW, Riemenschedier TA, McGough EC,Condon VR. Left ventricular outflow tract obstruc-tion produced by redundant mitral valve tissue in aneonate. Clinical, angiographic, and operative find-ings. Circulation 1976;53:196-9.

5 Gomes AS, Nath PH, Singh A, et al. Accessory flapliketissue causing ventricular outflow obstruction. JThorac Cardiovasc Surg 1980;60:21 1-6.

6 Tanimoto T, Kajino T, Hirose 0, Kamiya T, Naito T,Kozuka T. A case of left ventricular (LV) outflowtract obstruction caused by accessory endocardial

on August 6, 2020 by guest. P

rotected by copyright.http://heart.bm

j.com/

Br H

eart J: first published as 10.1136/hrt.59.4.491 on 1 April 1988. D

ownloaded from

Page 7: Accessory causing aortic regurgitation outflow obstruction Case … · Accessorymitalvalveleaflet causingaortic regurgitation andleft ventricular outflow tract obstruction Table Summaryofreportedcases

Accessory mital valve leaflet causing aortic regurgitation and left ventricular outflow tract obstruction 497cushion tissue with supravalvular aortic stenosis(SVAS). Japanese Society of Ultrasound MedicineProceedings 1981;56:311-2. [In Japanese]

7 Furuta N, Luhmer I, Hetzer R, Kallfelz HC. Abnormalaccessory mitral leaflet simulating left ventricularoutflow tract tumour. Thorac Cardiovasc Surg 1983;31:249-53.

8 Nanton MA, Belcourt CL, Gillis DA, Krause VW, RoyDL. Left ventricular outflow tract obstruction owingto accessory endocardial cushion tissue. J ThoracCardiovasc Surg 1979;78:537-41.

9 Deal CP, Trummer MJ, Bellamy JC, Timmes JJ. Anewdisease entity: leaflet redundancy of the mitral valve.Am Heart J 1963;65:441-5.

10 Sellars RD, Lillehi CW, Edwards JE. Subaortic sten-osis caused by anomalies of the atrioventricularvalves. J Thorac Cardiovasc Surg 1964;48:289-302.

11 Hatem J, Sade RM, Taylor A, Usher BW, Upshur JK.Supernumerary mitral valve producing subaorticstenosis. Chest 1981;79:483-6.

12 Freedom RM, Dische MR, Rowe RD. Pathologicanatomy of subaortic stenosis and atresia in the firstyear of life. Am J Cardiol 1977;39: 1035-44.

13 Ascuitto RJ, Ross-Ascuitto NT, Kopf GS, KleinmanCS, Talner NS. Accessory mitral valve tissue causingventricular outflow obstruction (two-dimensionalechocardiographic diagnosis and surgical approach).Ann Thorac Surg 1986;42:581-4.

14 Losay J, Binet JP, Piot JD, Lucet P, Petit J. Stenoseaortique sous-valvulaire par tissu mitral accessoire.Arch Mal Coeur 1985;78:737-40.

15 Cooperberg P, Hazell S, Ashmore PG. Parachuteaccessory anterior mitral valve leaflet causing leftventricular outflow tract obstruction. Report ofa casewith emphasis on the echocardiographic findings.Circulation 1976;53:908-1 1.

16 Alboliras ET, Tajik AJ, Puga FJ, Ritter DG, SewardJB. Accessory mitral valve tissue in association withdiscrete subaortic stenosis: a two dimensionalechocardiographic diagnosis. Echocardiography 1985;2:191-5.

17 Kelly DT, Wulfsberg E, Rowe RD. Discrete subaorticstenosis. Circulation 1972;46:309-22.

18 Kohda Y, Ueno Y, Yoshino T, et al. A case report ofsubaortic stenosis due to accessory tissue of anteriormitral leaflet. Kyobu Geka 1979;32:707-10. [InJapanese]

19 Zin'kovsky MF, Ignatow PI. Rare case of congenitalsubaortic stenosis caused by hyperplasia of theanterior cusp of the mitral valve. Grudn Khir1984;6:83. [In Russian]

20 Kuribayashi R, Imai T, Yagi Y, Gomi H. Subaorticstenosis caused by an accessory tissue of the mitralvalve. J Cardiovasc Surg (Torino) 1979;20:591-6.

21 Hotta T, Kobayashi Y, Ohnishi K, et al. A case ofsub-valvular aortic stenosis caused by an accessoryflap-like tissue of the mitral valve. Nippon KyobuGeka Gakkai Zasshi 1983;31:1607-12. [In Japanese]

22 Binet JP, Losay J, Piot JD, Lucet P, Petit J. Stenoseaortique sous-valvulaire secondaire a du tissu mitralaccessorie. Ann Chir: Chir Thorac Cardiovasc1985;39:424-5.

23 Meldrum-Hanna WG, Cartmill TB, Hawker RE,Celermajer JM, Wright CM. Accessory mitral valvetissue causing left ventricular outflow obstruction. BrHeart J 1986;55:376-80.

24 Levy MJ, Lillehei CW, Elliott LP, Carey LS, Adams PJr, Edwards JE. Accessory valvular tissue causingsub-pulmonary stenosis in corrected transposition ofthe great vessels. Circulation 1963;27:494-502.

25 Rastelli GC, Wallace RB, Ongley PA. Complete repairof transposition of the great arteries with pulmonicstenosis. A review and report of a case corrected byusing a new surgical technique. Circulation1969;39:83-95.

26 Martin EC, La Corte MA, Steeg CN, Bowman FO Jr.Accessory mitral valve tissue causing left ventricularoutflow tract obstruction in D-transposition of thegreat arteries. Cardiovasc Intervent Radiol 1981;4:124-7.

27 Van Praagh R, Corwin RD, Dahlquist EH Jr, FreedomRM, Mattioli L, Nebesar RA. Tetralogy of Fallotwith severe left ventricular outflow tract obstructiondue to anomalous attachment ofthe mitral valve to theventricular septum. Am J Cardiol 1970;26:93-101.

28 Rosenquist GC, Clark EB, McAllister HA, Bharati S,Edwards JE. Increased mitral-aortic separation indiscrete subaortic stenosis. Circulation 1979;60:70-4.

29 Somerville J. Congenital heart disease-changes inform and function. Br Heart J 1979;41:1-22.

30 Freedom RM, Fowler RS, Duncan WJ. Rapid evolu-tion from "normal" left ventricular outflow tract tofatal subaortic stenosis in infancy. Br Heart J1981;45:605-9.

on August 6, 2020 by guest. P

rotected by copyright.http://heart.bm

j.com/

Br H

eart J: first published as 10.1136/hrt.59.4.491 on 1 April 1988. D

ownloaded from