transposition of ventricles stems with cerebral abscess · transposition of ventricles and arterial...

TRANSPOSITION OF VENTRICLES ANDARTERIAL STEMS WITH CEREBRAL ABSCESS

By K. L. STUART, M.D., M.R.C.P.Ed., and H. E. FLINT, M.B., B.Chir.From the Departments of Medicine and Anatomy, University College of the West Indies

In the heart to be reported here there are con-genital anomalies allowing communication be-tween the right-heart and left-heart circulations,a feature of most of the other reported cases ofcerebral abscess occurring in association with con-genital heart disease. The anatomical features,however, were unique and of particular interest inrelation to their method of development.The great vessels were completely transposed,

the aorta taking origin from the ventricle on theright side of the heart, and the pulmonary trunkfrom that on the left side. In addition, theauriculo-ventricular valve of the ventricle on theright side had two cusps, while that on the lefthad three. The ventricle on the right had featuresof a normal left ventricle and that on the leftthose of a normal right ventricle. The atria,however, were normal in their connections withthe ventricles and in their venous openings.There would have been isolated right heart-bodyand left heart-lung circulations had there not beena large atrial septal defect which provided com-munication between the circulations. An addi-tional anomaly in the heart was the presence of asingle coronary artery which gave, in its course,branches which approximated to the normalcoronary supply from right and left arteries.The occurrence of cerebral abscess as a com-

plication of congenital heart disease receives onlycursory mention in the standard textbooks ofmedicine, and the number of reported cases isalso relatively small. It is likely that the paucityof reported cases represents failure of recognitionrather than rarity of occurrence, for it is becomingincreasingly evident that the incidence is muchhigher than is at present indicated. Robbins(1945) reported 3 cases from a total of 53 casesof congenital heart disease of all types examinedat post-mortem.Maronde (I950) found ii in a total of 8i post-

mortem cases of congenital heart disease thatsurvived beyond the age of two years and showedat the same time a potential shunt between thesystemic venous and arterial circulations. These

ii cases represented I0.4 per cent. of 105 fatalinstances of brain abscess from all causes,examined in the same series.These abscesses, which are usually solitary

(Hanna, 1941; Gates et al., 1947), are very suitableto surgical treatment; and a number of success-fully treated cases have been reported (Smoliket al., I946; Sweeney and Patton, 1947). Ehniand Crain (I952) reported a patient with Lutem-bacher's syndrome, who developed two cerebralabscesses in six years, diagnosed and treatedsuccessfully on both occasions. It is suggestedthat this condition should no longer be regardedas a medical curiosity; and the frequency of itsoccurrence, the relative ease of diagnosis, once itis suspected, and the good results obtainable byprompt surgery should be re-emphasized.The patient described here had a solitary right

parietal lobe abscess, and died three hours afteradmission before surgery could be attempted.

Case ReportL L., a nine-year-old Jamaican girl of mainly

Chinese extraction, was admitted to hospital inAugust 1954 Her illness started 14 days beforeadmission, when she first complained of graduallyincreasing headaches. Vomiting started within48 hours and continued off and on until heradmission to hospital. The day before admissionthe headache became more severe, her mentalstate deteriorated, and she became drowsy andconfused.

She had been a ' blu'e baby' since birth, andher physical activity had always been somewhatrestricted because of breathlessness on more thanmoderate exertion. The cyanosis had increasedconsiderably since the onset of her illness.

Physical Examination. On admission, she wasacutely ill. Her temperature was I03.50 F. Shewas very cyanosed, and her fingers showed agross' drum-stick ' clubbing. There was markedbulging of the left side of the praecordium whichshowed a diffuse pulsation. There were no signsof congestive cardiac failure. The apex beat

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132 POSTGRADUATE MEDICAL JOU#NAL March 1957

could not be palpated with accuracy, but thepoint of maximum intensity on auscultation wasin the fifth left intercostal space 41 inches fromthe midline, and just outside the mid-clavicularline. Her blood pressure was I10/70 mm. Hg.Careful auscultation revealed no significant cardiacmurmurs, but there was an extremely loud ringingsecond sound in the pulmonary area.Her mental state fluctuated between drowsiness

and a resistant stupor. Her co-operation couldbe obtained for short periods only, and thenmerely to obey simple commands. When arousedshe complained of headache and eyeache, indicat-ing chiefly the right eye. A moderate degree ofneck rigidity was present. Her fields of visionand visual acuity could not be tested accurately,but she appeared to notice movements better inthe left visual half-field than in the right. Herfundi showed a moderate degree of papilloedema.There was ptosis of the right eyelid, and anexternal strabismus with some apparent weaknessof conjugate deviation of the eyes to the left.The right pupil was larger than the left. Bothreacted to light directly and consensually, butsluggishly on the right. The corneal reflex wasdiminished on the right and absent on the left.Some weakness of the left lower face was noted.There was a spastic weakness of the limbs, moremarked on the left and with almost completeparalysis -of left arm movements. Sensation tolight touch could not be tested, but appreciationof pain sensation appeared normal. The reflexesin both upper limbs were normal, but markedlyincreased in the lower limbs, especially on theleft. There was sustained ankle clonus on theleft with a ffw unsustained beats on the right.The plantar reflexes were both extensor.

Laboratory Findings. The following laboratoryresults were obtained on admission. Bloodexamination: Haemoglobin, 2I.0 g. per cent.;packed cell volume, 62.o per cent.; white cellcount, I6,700 per c.m,l. neutrophils 95 per cent.,lymphocytes 3 per cent., smear cells 2 per cent.Two blood cultures were negative. Cerebro-spinalfluid: pressure 300 mm. of water; white cellcount, 6I9 per c.ml. (ioo per cent. polymorphs);protein, 200 mg. per ioo ml.; sugar, 45 mg. perioo ml.; no organisms were seen on direct smear;and aerobic and anaerobic cultures were negative.X-ray chest showed an ill-defined aortic knuckle,

with enlarged pulmonary vessels and increasedperipheral vascular markings (Fig. i). Theappearances suggested congenital heart disease,probably transposition of the great vessels.The electrocardiogram showed marked right

ventricular hypertrophy (Fig. 2).Diagnosis. A diagnosis of cyanotic congenital

heart disease with a rapidly expanding intra-

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FIG. i.-X-ray of chest showing ill-defined aorticknuckle, enlarged pulmonary vessels and increasedperipheral vascular markings.

cranial lesion, probably cerebral abscess, wasmade. The enlargement of the pulmonaryvessels with increased peripheral vascular mark-ings, the absence of a significant murmur, and thevery loud pulmonary second sound were againsta diagnosis of the tetralogy of Fallot. Completetransposition of the great vessels with a shuntbetween the right and left sides of the heart,probably through an atrial septal defect, seemeda reasonable interpretation of the findings. Theother anomalies were not suspected.

Post-mortem ExaminationThe only significant observations pertained to

the brain and heart. There was no transpositionof any of the viscera.The brain weighed 1,300 g. The meminges

were not, thickened; but there was marked con-gestion of the vessels with fattening of the gyri.There was an abscess approximately 3 cm. indiameter in the region of the intraparietal sulcusof the right cerebral hemisphere, immediatelybehind the post-central gyrus (Fig. 3). Approxi-mately 35 c.c. of greyish sanguineous pus wereobtained from this abscess.The paranasal sinuses and middle ear were

clear, showing no evidence of infection. Thevenous sinuses showed nothing remarkable.

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March 1957 STUJART and JFLINT: Transposition of Vlentricles with Cerebral Abscess i3

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FIG. 2.-Electrocardiogram showing marked right ventricular hypertrophy.

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FIG. 3.-Photograph of brain showing right parietal lobe abscess.

Microscopic section of the right cerebral hemi-sphere showed an abscess with a chronic granulo-matous wall. A smear of the pus from theabscess cavity showed a few gram-positive diplo-cocci, gram-positive and gram-negative cocci inchains. There was no growth under aerobic andanaerobic conditions. A blood culture from thecavity of the right ventricle was positive forstreptococcus faecalis.

Description of the HeartThe heart, which was not weighed, had its

normal position in the thorax. Its approximatemeasurements were 8 cm. from base to apex,7 cm. transversely at its broadest part, and 7 cm.antero-posteriorly.

Great Vessels. The great vessels were trans-posed, the aorta taking origin from the ventricleon the right side, the pulmonary trunk from the

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134 POSTGRADUATE MEDICAL JOURNAL March 195 7

NMI? ALUPJODAP. APPSNOWBSliM.ACK?A? Cr*AUOAU*S LA APPOMMat

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FIG. 4.-Diagram of heart seen from sterno-costal surface.

ventricle on the left side of the heart. Theorigin of the aorta thus lay anterior and slightlyto the right of the pulmonary trunk, and theaorta arched over the pulmonary trunk and rightpulmonary artery which lay behind it, and crossedthe right bronchus to become the descendingthoracic aorta (Figs. 4, 5 and 6). From the archof the aorta the three great arterial vessels tooktheir normal origins. A patent ductus arteriosuswas present, 7 mm. long and of the funnel-shapedtype, tapering from an aortic opening 3 mm. indiameter to a narrower pulmonary opening. Asthe aortic and pulmonary trunks lay in the speci-men, the aortic orifice was guarded by twoanterior and one posterior cusp, the pulmonaryorifice by two posterior and one anterior cusp.A single coronary trunk arose from the sinus ofthe posterior cusp of the aorta (Figs. 4 and 5).

Ventricles. The ventricle on the right side,from which the aorta arose, showed features of anormal left ventricle and communicated with theright atrium through a two-cusped valve, whilethe ventricle on the left side showed features ofa right ventricle and communicated with the leftatrium through a valve with three cusps.The interventricular septum was complete.

The cavity of the left-sided ventricle was largerthan that of the right, the septum bulging slightlyto the right, and the wall of the ventricle on theleft side was slightly thicker. The wall of theventricle on the right side was 7 mm. across withcoarse trabeculation extending up to the aorticvestibule. The wall of the ventricle on the leftside was 9 mm. across and, on the inner surface,the trabeculae carneae were more numerous, finer,and more delicately interlaced than on the right.A smooth infundibular region lead to the pul-monary orifice. The crista super-ventriculariswas not prominent, a shallow ridge passing in theroof of the ventricle between the pulmonary and

atrioventricular openings to the septum. Amoderator band was not present.

The Atria. The right and left atria were intheir normal anatomical relationship. The auri-cular appendage of the right atrium was in anunusual position lying on the left side of thepulmonary trunk and the upper side of the left-sided ventricle above the left auricular appendage.The two atria were in free communication througha grossly deficient inter-atrial septum. A partialseptum extended as a crescentic fold from theposterior wall and inferiorly; its free crescenticmargin was directed forwards, and it was 12 mm.wide from base to free margin in its middle part.In the lower part of the crescent was a large ovaldeficiency I5 mm. x 7 mm., bounded anteriorlyby the free edge of the septum which was herewell defined and cord-like. Below this wasanother smaller, fenestrated, deficiency. Thecrescent represented the septumprimum whichwas incomplete and had regressed in an abnormalway. The septum secundum was representedby a crescentic ridge superiorly passing on to theposterior wall on the right side of the septumprimum. This crescentic ridge crossed the upperpart of the septum primum remnant forming theupper sector of the annulus ovalis.The venae cavae and coronary sinus entered the

right atrium, and the four pulmonary veins theleft atrium in the usual way.

There was no evidence either in the gross ormicroscopically of rheumatic fever or subacutebacterial endocarditis.

The Coronary Arteries. A single coronarytrunk took origin from the sinus of the posterioraortic cusp, and, in its subsequent course, gavebranches which approximate to those of thenormal coronary supply from right and leftarteries. A small pin-hole dimple was presentin the sinus of the left anterior cusp, but this

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March 1957 STUART and FLINT: Transposition of Ventricles with Cerebral Abscess I35

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FIG. 5.-Diagram of relationships of great vessels and course of the coronary branches:A: In the normal heart.B: In the heart described in the present report.

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136 POSTGRADUATE MEDICAL JOURNAL March 1957

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FIG. 6.-Anterior view of heart:A. = Aorta.

P.A. = Pulmonary artery.D.A. = Ductus arteriosus.

I. = Innominate artery.L.C.C. = Left common carotid artery.

L.S. = Left subclavian artery.

had no connection with vessels of the coronarysystem. The single coronary trunk lay with theaorta on its left and the pulmonary trunk behindand divided, 4 mm. from its origin, into right andleft branches (Figs. 4 and 5).

The right branch ran between the base of thepulmonary trunk and the right-sided ventricle tothe groove between that ventricle and the rightatrium giving branches to the anterior wall of theventricle. The left branch passed in front of the

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March I957 STUART and FLINT: Transposition of Ventricles with Cerebral Abscess

aorta in the groove between the base of the aortaand the right-sided ventricle giving a branch tothe surface of its anterior wall.

DiscussionThe Heart. Abnormality in the relationship of

the great vessels occurs in Fallot's tetralogy, theEisenmenger complex and the Taussig-Bing com-plex, but those cases in which transposition of thegreat vessels is the main abnormality fall intotwo groups. In both groups the aorta lies infront of, and slightly to the right of, the pulmonarytrunk. In one group the transposition is completeand the aorta arises from the right ventricle, thepulmonary trunk from the left. In the other,rarer group, the transposition is ' corrected' for,despite the abnormal relationship of the twovessels, the aorta arises from the left ventricle,the pulmonary trunk from the right. The venousconnections in both groups are normal, and incomplete transposition life is only possible ifother congenital abnormalities are present allow-ing communication between the left heart-lungcirculation and the right heart-body circulationwhich would otherwise be entirely separate.Hanlon and Blalock (1948), in their study of

complete transposition and the possibilities ofaffording relief by surgery, suggest that thechances of survival improve with the number andextent of the additional abnormalities affordingcommunication between the two circulations. Inthis case the grossly deficient atrial- septum andthe patent ductus arteriosis, provided ample cross-cirulation, and had it not been for the develop-ment of the cerebral abscess, a much longerperiod of survival would have been anticipated.Indeed, there was nothing in her clinical historyup to her final illness to suggest any progressionof her cardiac disability.Gould (I953) states that transposition of the

great vessels is commonly associated with isolateddextrocardia, and that in some cases the trans-position is corrected. The aortic arch usuallycrosses the left bronchus but may, as in the heartrecorded here, cross the right, the descendingaorta lying slightly to the left side in both types.In the present case it would seem that the ven-tricles only have been transposed for the atria arenormal in position, apart from the displaced rightauricular appendage. The abnormal position ofthe right auricular appendage in transposition ofthe great vessels is apparently unusual, althoughMiskall and Fraser (1948) recorded a case oftransposition where the right auricular appendagelay to the left of the great vessels. Ngai (I935)described a heart in which sinitransposition of theright auricle occurred with transposition of thegreat vessels, a ventricular. septal defect, and a

left ventricle with no efferent vessel, both aortaand pulmonary trunk arising from the rightventricle. In this heart, as in the present speci-men, a single coronary trunk took origin from theaorta on the right side.The heart described by Walmsley (I93I) pro-

vides an interesting comparison, for in his speci-men the transposition of the vessels was corrected,the aorta arising from the left-sided ventricle, andthe pulmonary trunk from the right-sided ven-tricle. The left-sided ventricle showed featuresof a normal right ventricle while the right-sidedventricle resembled a normal left ventricle.There were no septal defects present, the ductusarteriosus was closed, and the atria with theirvenous connections were normal.The left-sided atrio-ventricular valve had three

cusps, the right-sided atrio-ventricular valve two.A moderator band and a crista supraventriculariswere present in the interior of the left-sidedventricle but not in that of the right side, and thetrabeculation of the left-sided ventricle was coarselike that of a right ventriLle, while that of theright-sided ventricle was fine like that of a leftventricle. The trabeculation does not show thisfeature in the heart here presented, being finer inthe ventricle on the left side which has no modera-tor band, but the profuse apical trabeculation isapparent and the infundibular region well demar-cated. The atrio-ventricular part of the inter-ventricular septum in our specimen intervenedbetween the right atrium and the left-sided ven-tricle and not between the left atrium and theright-sided ventricle as it did in the heart de-scribed by Walmsley so that, presumably, theposterior part of the interventricular septum met,and fused with, the septum intermedium in theusual manner and was not displaced to the left asWalmsley described for his specimen. Walmsleysuggested that the primary anomaly of the hearthe described was ' an inversion of the ventricularloop, so that what is here the ventricle of theright side is the descending limb of the loop, andmorphologically the left ventricle, and what is theventricle of the left side is the ascending limband morphologically the right ventricle' (Walms-ley, I93I).

This description would seem to apply equallywell to the specimen recorded here for the ven-tricle on the right side is, apparently, morpho-logically the left, the ventricle on the left sidemorphologically the right. Furthermore, Walms-ley suggests that inversion of the ventricular loopwould produce rotation of the proximal bulbarseptum in the reverse of normal so that theinfundibulum is included in the ventricle of theleft side. He suggests that, in the heart hedescribes, the distal part of the bulbar septum

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FIG. 7.-The rotation of the arterio-pulmonarv andventricular bulbar septa:A: In the normal heart.B: In the heart described by T. Walmsley.C: In the heart described in the present report.

Based on a diagram published by Walmsley (I93I).

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March I957 STUART and FLINT: Transposition of Ventricles with Cerebral Abscess

rotated normally and so, in spite of the inversionof the bulbar, loop, a corrected transposition ofthe great vessels resulted, the aorta originating inthe left-sided ventricle.

In the heart here described it is suggested thatthe reverse rotation of the proximal septumdescribed by Walmsley continued in the distalseptum also producing complete transposition(Fig. 7).The occurrence of a single coronary trunk,

while not common, has been reported severaltimes and these cases have been fully reviewedand classified by Krumbhar and Ehrich (I935)and Roberts and Loube (I947). Both reviews referto Hyrtl's criterion for a single coronary arterywhich stipulates that the single vessel must supplythe entire heart without conspicuous anomalousbranches, the anlage of the second coronary arterybeing completely absent in contradistinction to ananomalous joint origin for the two coronaryanlages, or an origin of the one from the other.However, Roberts and Loube suggest that theidentification of cases of single coronary arteryshould be determined by the origin of the coronaryarterial circulation from one trunk rather thanby the true congenital absence of one coronaryartery. White and Edwards (I948) examined thetoronary system of 6oo hearts, which wereprimarily the subject of another study, and foundthree anomalous patterns. In one heart a singlecoronary stem was present, in two others the leftcircumflex artery arose as a branch of the rightcoronary while the anterior descending inter-ventricular artery arose independently from theleft aortic sinus, and in a further heart bothcoronary arteries arose from the left aortic sinus.The single coronary trunk of the heart reportedhere was probably formed by displacement of theanlage of the missing coronary, for its left andright main branches are distributed in a patternsimilar to the distribution of normal left andright coronary arteries. This persisting coronarytrunk was on the right of the, aorta in the specimen,and represents a persistent left coronary artery ofa normally disposed aorta, and the passage of itsleft branch in front of the aorta follows from this.

Cerebral Abscess. The presence, of fever, anelevated white cell count, and pleocytosis of thecerebro-spinal fluid with signs of focal braindisease in a patient with cyanotic congenital heartdisease made the diagnosis in this case compara-tively easy.

It should be realized, however, that this classicalpicture is frequently absent; and it has beenpointed out that it is precisely in the absence ofthese obvious findings that the best results maybe obtained from surgical intervention (Glucket al., 1952). There is usually no demonstrable

source of infection, and the temperature andwhite cell count may remain normal until late.The cerebro-spinal fluid also may remain un-changed until rupture of the abscess into thecerebral ventricular system or subarachnoid spacehas occurred. In a patient with congenital heartdisease, therefore, minimal findings, such as per-sistent and unexplained headaches, drowsiness,slight neck rigidity, and equivocal central nervoussystem signs should be very critically assessed;and the patient should be carefully observed withthis diagnosis in mind, even if the laboratoryfindings are negative. It is likely that in manycases the diagnosis has been missed because it hadnot been considered; and in many others dis-missed' in -the early stages because of lack oflaboratory confirmation. Once the diagnosis issuspected cranial air studies and electro-encepha-lography may be useful aids in the early localiza-tion of the lesion. Even when the diagnosis hasbeen considered, the distinction from subacutebacterial endocarditis with infected embolizationof the brain may yet be difficult.The precise pathogenesis of brain abscesses

occurring in patients with septal defects of theheart is unknown. The time-honoured assump-tion that they are due to paradoxic emboli providesa reasonable mechanical explanation; and it isfeasible that small septic or aseptic emboli maybe shunted from the systemic venous to thesystemic arterial circulation in patients with thistype of heart disease. Maronde (1950) suggeststhat the pulmonary capillaries act as a bacterialbarrier which can be by-passed in the presence ofseptal defects in the heart. A serious objectionto this hypothesis, however, is the absence in ourcase, as in most reported cases, of a demonstrablesource of emboli.

It has been suggested that brain damage pre-cedes* the development of an abscess (Hanna,I941; Robbins, 1945), and 'there is some post-mortem evidence in favour of this (Gluck et al.,1952). The mechanism of production of thisantecedent damage, however, is at present ob-scure; but it seems possible that the slowing ofthe blood secondary to polycythaemia and in-creased viscosity may be an important contributory.factor in cerebral thrombus formation. The fre-quency with which a brain abscess follows a;suppurative process in the lungs suggests thelungs as an alternative source of emboli; and Rich(1948) has drawn attention to the frequency withwhich widespread and progressive thrombosis ofthe pulmonary vessels occurs in congenital heartdisease.

SummaryA case of transposition of the ventricles and

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arterial stems complicated by cerebral abscess hasbeen described. The right ventricle, which hadmorphological features of a normal left ventricleand was guarded by a bicuspid valve, gave originto the aorta. The left ventricle resembled anormal right ventricle, was guarded by a tricuspidvalve and gave origin to the pulmonary trunk.There was a large atrial septal defect, and a patentductus arteriosus. The coronary supply wasfrom a single vessel. The anatomical significanceand development of these findings are suggested.The clinical features and aetiology of cerebral

abscess in congenital heart disease are discussed.The good results obtainable by prompt surgeryare stressed. It is suggested that the develop-ment of even minor signs of focal brain damagein such a patient should arouse suspicions of thediagnosis.

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Proc. Staff Meet. Mayo Clinic, 22, 401.GLUCK, R., HALL, J. W., and STEVENSON, L. D. (1952),

Paediatrtcs, 9, 192.GOULD, S. D. (I953), 'Pathology of the Heart,' Charles Th omas,

Springfield, Illinois.HANLON, G. R., and BLALOCK, A. (I948), Ann. Surg., 127, 385.HANNA, R. (I94I), Amer. J7. Dis. Child., 62, 555.KRUMBHAAR, E. B., and EHRICH, W. E. (1938), Amer. .

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RUTHIN CASTLE, NORTH WALESA Clinic for the diagnosis and treatment of Internal Diseases (except Mental or Infectious Diseases). The

Clinic is provided with a staff of doctors, technicians and nurses.The surroundings are beautiful. The climate is mild. There is central heating throughout. The annual

rainfall is 30.5 inches, that is, less than the average for England.The Fees are inclusive and vary according to the room occupied.

For particulars apply to THE SECRETARY, Ruthin Castle, North Wales.Telegrams: Cstle, Ruthin. Telephone: Ruthia 66

Continuedfrom page i2o-Jack Colover, M.D., M.R.C.P.the capacity to produce a type of autosensitization(Lawrence, 1956) which manifests itself underexperimental conditions as a meningo-encephalo-myelitis with demyelination, having some quitemarked resemblance histologically to some of thehuman demyelinating diseases.

AcknowledgmentsI would like to thank Mr. P. J. Fiske for the

microphotography and Mr. J. Dorling for tech-nical assistance. I am very indebted to Dr. L. E.Glynn of the Department of Pathology at theCanadian Red Cross Memorial Hospital, Taplow,for assistance and advice.

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Sect. VIII, 8, 808.COLOVER, J., and CONSDEN, R. (g956a), Nature, 177, 749.

COLOVER, J., and CONSDEN, R. (I956b), Proc. and Internat.,Cong. of Neuropathology. In press.

COLOVER, J. (i956d), Proc. roy. Soc. Med., 49, IS4.CUMINGS, J. N. (I953), Brain, 76, 55I.DAWSON, J. R. (I934), Arch. Neurol. Psychiat. (Chicago), 31, 685.FREUND, J., and MAcDERMOTT, K. (1942), Proc. Soc. exp.

Biol. (N.Y.), 49, 548.GREENFIELD, J. G. (I933),.7. Neurol. Psychopath., 13, 289.HURST, E. W. (I942), Austral. 7. exp. Biol. med. Sci., 20, 297.HURST, E. W. (I944), Brain, 67, 103.KABAT, E. A., WOLF, A., and BEZER, A. E. (1946), Science, xo4,

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