TRANSPLEURAL CARDIOPLASTY IN ACHALASIA

Thorax (1949), 4, 243.

TRANSPLEURAL CARDIOPLASTY IN ACHALASIAOPERATION RESULTS AND SEQLIELAE

BY

HELGE B. WULFF AND ARNE MALMMalmb, Sweden

The clinical syndrome characterized by dys-phagia, retrosternal or epigastric pain, and, inadvanced stages, by regurgitation of undigestedfood and total obstruction of the oesophagus, haslong been known and has been called by manydifferent names.The Englishman, Thomas Willis, gave a detailed

description in 1672, and after 1882, when v. Mikulicz,who believed it to be caused by a spasm in theregion of the cardia, published his studies on thesubject the syndrome was known as "cardio-spasm"; this name is still in current use in manycountries. When later investigations disprovedthe occurrence of a spasm in the cardiac regionHurst (1914) introduced the name " achalasia,"or lack of relaxation of the cardiac sphincter. Butin the light of our experience and knowledgeto-day it seems that the condition is due to a

combination of factors and not solely to failure ofrelaxation, so that Hurst's suggestion is no longersatisfactory.

Imbalance in the nerve supply to the cardiacsphincter may be responsible for the onset; if so,the sphincter does not respond by relaxing towaves of peristalsis from the oesophagus butremains closed and thereby arrests the descentof the food to the stomach until the cardiacorifice has been opened by dilatation or surgicalintervention or until later on it spontaneouslyrelaxes.Our knowledge of the aetiology is still obscure,

but causal factors do not seem to be uniform.Some authors ascribe the disease to a disorder inAuerbach's plexus (Rake, 1927; Cameron, 1927;Mosher and McGregor, 1928) or to lesions ofthe central nervous system. Others envisage thepossibility of vagotonia (Held and Gross, 1916),degenerative changes in the vagus (Kraus,1902; Heyrovsky, 1913; Pollitzer, 1913) or a

sympathetic nerve derangement (Carlson andLuckhardt, 1921 ; Kurd, 1929; Knight, 1934, andothers). Sauerbruch and v. Haecker (1906) sug-gested that the diaphragm was concerned, andJackson, who shared this view, proposed the nameof " phrenospasm " for the syndrome.

Recent experience from transpleural or trans-abdominal gastric vagotomy for gastric andduodenal ulceration has contributed to the eluci-dation of the role the vagus plays in the occurrenceof achalasia. Judging by the many reports in theliterature no clinical or radiological evidence ofany spastic sequelae of vagotomy has ever beenseen in spite of due attention being directedtowards this point. Neither have we made suchan observation in the course of our experience,which covers some 100 cases of vagotomy. It is,therefore, evident that the oesophageal disordersfrequently seen in vagotomized dogs have nocounterpart in man.*

Psychogenic factors have also been suspected ofplaying a contributory role in causing the condi-tion, and judging by the results of investigationshitherto made, the suspicion does not seem to beentirely unfounded. It ought perhaps to be men-tioned that a psychiatrist is at present making apersonality study of our patients, and the resultswill be the subject matter of a later paper.Some years ago very few cases of achalasia were

admitted to hospitals, but this is no longer thecase. Gray and Skinner (1940), of the MayoClinic, published a report of 1,200 cases, and ina large oesophagoscopic series Moersch (1933), forexample, found the condition in 17.4% of thecases. Guisez (1935) found a similar percentagein his material, and many cases have been reportedfrom Sweden (Gjertz, 1948).The symptoms vary from occasional difficulty in

swallowing, with a temporary feeling of obstruc-tion behind the sternum or in the epigastrium, tocomplete obstruction in the lower section of theoesophagus, marked oesophageal dilatation abovethe functional obstruction, and pulmonary compli-cations in the form of gangrene or abscess of thelungs caused by nocturnal regurgitation and aspira-tion. As a rule medical aid is not sought untitthe disease is advanced.

*Several surgeons in Great Britain have had isolatedcases in which vagotomy has been followed by the typicarradiological and clinical manifes:ations of achalasia. Thesesymptoms have been of temporary duration.-EmToR.

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HELGE B. WULFF AND ARNE MALM

Owing to the many aetiological factors con-cerned in this disease numerou3 treatments havebeen suggested and tried with varying success.Relief has sometimes been achieved by theapplication of antispasmodics, and sometimesthe symptoms pass off spontaneously. In moreadvanced cases dilatation therapy has beenapplied. In a series of 805 patients treated bydilatation Moersch (1933) reported that 71 % weredischarged from hospital without symptoms;Wachs (1940) reported dilatation to be successfulin 70% of his patients, and in a Swedish seriesreported by Gjertz in 1948 the results were justas favourable.

It is evident, both from the literature and fromour own experience, that dilatation by expandingmetal instruments or the introduction of a hydro-static or air-rubber-bag often gives but temporaryrelief and is undoubtedly sometimes followed bycomplications such as rupture of the oesophagusor mediastinitis. We know from our own experi-ence that such ruptures and mediastinitis are morecommon in cases treated previously by dilatationthan might be inferred from statistics. Moerschreported nine cases of death from oesophagealrupture, two from cachexia, and 12 from otherpost-operative complications. In 64 cases, treatedby dilatation, Gjertz reported rupture in three,but there were no deaths. Sometimes the oeso-phagus is distended to such a degree as to contra-indicate dilatation therapy.

In some of these cases surgical interventionmust therefore be carried out, but it is notalways easy to decide when and in which patientsit is indicated. Conservative measures sometimesgive relief even in cases in which at first sight thereseems to be an obstinate constriction, but on theother hand it is not always wise to delay radicaltreatment. Each case must be judged on its ownmerits, due regard being paid to the circumstancesas a whole and not solely to the degree of oeso-phageal dilatation. For instance, when relief canonly be obtained by frequently repeated dilatation,or when the oesophagus is grossly distended andsigmoid in shape so that digestion is disturbedand the patient is cachectic, or when there isevidence of pulmonary complications or media-stinitis, immediate radical treatment is indicated.We would emphasize that delay in remedial

treatment is likely to make an anatomical resti-tutio ad integrum of the oesophagus impossible,because prolonged and extreme distension rendersit less capable of recovering its original form: themuscle fibres separate and degenerate into con-nective tissue with the result that the oesophagus

remains dilated, thin-walled, and non-elastic evenafter the obstruction has been overcome. Similarchanges are seen, for example, in hydronephrosisin which plastic surgery is also necessary to givethe renal pelvis a more or less normal shape afterthe removal of the obstruction.

Several aetiological theories have been put for-ward and almost as many operations have beensuggested to cure the condition.The most popular of these operations are:

(1) " extramucous myotomy," i.e., longitudinalincision of the muscle coats of the oesophaguscarrying the incision down to the mucous mem-brane (Heller, 1913 ; Ropke, 1914); (2) divisionof the fasciae of the diaphragm in relation to theoesophagus and incision of the hiatus, " phreni-cotomy" (Jackson, 1922); (3) establishment of ananastomosis between the oesophagus and fornixventriculi (the cardiac orifice) ad modum Heyrov-sky (1913); (4) cardioplasty with a longitudinalincision through all layers from the oesophagusdown to the stomach with suture at right anglesto the incision (Marwedel, 1903 ; Wendel, 1910);and (5) operations upon the autonomic nervoussystem (Meyer, 1911; Sauerbruch, 1925 ; Knight,1934).Concerning the first four methods, there has

been much discussion as to whether the abdominalor the transthoracic approach should be givenpreference. At first, when the operative mortalityfrom transpleural intervention was relatively high.the abdominal approach was naturally the one ofchoice, but the favourable results obtained withthe transpleural approach during the last decadehave changed the situation. The transpleuralmethods, which provide a better approach and abetter survey of the field of operation, are nowheld to involve but small risks, and our experiencesupports this view.

MATERIALFrom May, 1944, until October, 1948, 21

patients suffering from achalasia have been treatedin our department of thoracic surgery. In themeantime three other cases of achalasia haveundergone cardioplasty with success, but theoperations are too recent to be included in thispaper. One patient was operated upon twice.The youngest was 6 years of age, the oldest 63,and the highest incidence of the disease laybetween the ages of 18 and 37 years. In otherwords, the frequency is greatest in that age periodwhen the struggle for life is hardest, a fact whichshould be borne in mind, because psychogenicfactors are also concerned in the onset of theaffection.

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TRANSPLEURAL CARDIOPLASTY IN ACHALASIA

All the cases, except two, had previously beentreated medically or by dilatation by otolaryngo-logists. The patients were not. transferred to ourdepartment until endoscopic treatments had failedto give relief or had been abandoned on accountof discomfort or the risk of rupture. In generalthe affection was advanced and in some instancesthe shape of the distended oesophagus wasgrotesque.Two patients had not undergone pre-operative

dilatation. One of these was an 11-year-old boywhose history was typical-namely, loss of weight,markedly distended oesophagus, and occasionalaspiration of oesophageal contents into the lungs.The other case was that of a 36-year-old man, inwhom the radiographs revealed an oesophagus asthick as an arm. He presented himself complain-ing of a lung infection, which was found to be alarge pulmonary abscess secondary to aspiration;and he had stagnant food in the dilated oeso-phagus. Both patients were operated on withoutpre-operative dilatation because continuation ofthe conservative treatment would in our opinionhave incurred undue risks and even threatened thepatient's life.

OPERATION METHODS AND TECHNIQUETwenty-two operations were done on 21

patients: cardioplasty in 16 and extramucousmyotomy in six. All operations were done trans-pleurally and in the myotomy cases all layers ofthe oesophagus were incised down to the mucouscoat. In these the operation consisted of a singlelongitudinal incision from the ossophagus down tothe cardia and they were done in the early partof the series, but as the results seemed to beunsatisfactory, we adopted once more the methodused first in the case operated on in 1944-i.e.,transpleural cardioplasty, essentially ad modumMarwedel-Wendel.

Technique.-All patients were operated upon inthe lateral position and under intubation narcosiswith ether-oxygen and nitrous oxide. From 20 to25 cm. of the ninth rib on the left side wereremoved subperiosteally. The pleura was incised,and the mediastinal pleura was opened in thetriangle between the heart, the aorta, and thediaphragm at the site of the inferior pulmonaryligament. The oesophagus, which was often verydilated, was then easy to free from its attachments.A short incision was made in the oesophagealhiatus so that the lower section of the gullet andthe fornix-cardia could be drawn -up for inspec-tion. The longitudinal oesophageal incision wasmade down to the cardiac notch over and across

the ventricular fornix towards the greater curva-ture. The length of the incision varied between4 cm. and 7 cm.

In the cardioplastic operations all the layerswere cut through, whereas in the myotomies theincision was made down to the mucous membraneonly. In the latter operations the cut oesophagealmuscles were not sutured. In the cardioplasticcases the longitudinal incision was sutured trans-versely by means of two rows of stitches, an innercontinuous catgut and an outer interrupted silksuture. The cardia and the upper part of thefornix ventriculi were always left supradia-phragmatically, and fixed to the diaphragm byinterrupted silk sutures (Fig. 1). Only in one

FIG. 1.-Diagram showing the three main steps intranspleural cardioplasty: (1) longitudinal incisionthrough the lower oesophagus, the cardia, theventricular fomix, with (2) transverse suture;(3) fixing the cardia and the superior part of theventricular fomix ibove the diaphragm.

case (the first) was gastrostomy performed beforethe radical operation. The pleural cavity was notdrained during or after the operation, and noinfection of the pleural cavity, the abdominalcavity, or the wall of the thorax occurred in con-nexion with the operations. In most cases 500,000units of penicillin were introduced into the pleuralcavity and one to three aspirations were adequateto remove the usual post-operative effusion. Inthe cardioplastic cases which were operated uponbetween 1944 and 1945 the patients were notallowed to drink for the first four to five daysafter operation, whereas in the latter part of theseries they were given small quantities of liquidon the following day. The patients were generallyallowed to leave their beds five to fourteen daysafter the operation.

IMMEDIATE POST-OPERATIVE RESULTSIn the six cases of extramucous myotomy the

primary post-operative course was uneventful,neither did any post-operative complications super-vene in 15 of the 16 cardioplastic operations. Thesixteenth case was that of a 57-year-old womaninebriate, who was in a very poor condition onadmission; her serum albumin was less than 4%.

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In spite of about one month's pre-operative treat-ment no improvement was observed. This resis-tance to treatment and the severe degree of con-striction which was present demanded, in ouropinion, immediate surgical intervention, and theoperation was carried out in the usual manner.This radical treatment gave complete relief, all theprevious symptoms disappeared, and the patient'sgeneral condition improved. One morning, aboutthree weeks after the operation and after thepatient had been sitting up for some days, shedied suddenly from pulmonary embolism withoutany premonitory elevation of temperature. Anormal operation field and unobstructed oeso-phagus were found post mortem and the fatalembolus was revealed in the pulmonary artery.On discharge from hospital all the other patients

who had undergone cardioplasty were in a goodgeneral condition and free from the symptoms ofachalasia.

Routine radiological examinations were carriedout in all cases three to four weeks after opera-tion, and the films showed a normal passagethrough the operation area in 12 cases and aslightly retarded passage in three. A comparisonof the radiological outline of the oesophagusbefore and after operation showed a markedreduction in dilatation in three cases, a moderatediminution in nine, and practically no improve-ment in three.

LATE RESULTSCases Treated by Cardioplasty.-The 15 patients

who underwent cardioplasty were followed up forperiods varying from five months to five yearsafter operation. When last reviewed radiographi-cally and clinically during the latter part of 1948,the films showed a progressive but moderatereduction in the oesophageal dilatation in five, butno further changes were seen in any of the others.We compared the pre-operative oesophageal radio-logical outlines with those observed some timeafter operation and found a reduction in size inevery case.As to the subjective symptoms, eight patients

were entirely symptom-free, one had very slightdysphagia, and one occasional discomfort fromregurgitation. Four patients had slight tomoderate symptoms not directly related to theaffection, such as nausea, heartburn, and epi-gastric or oesophageal gas formation. As a rulethe symptoms were less troublesome or painfuland of shorter duration than before operation.

In one case dysphagia persisted practicallyunchanged after operation. The patient was a36-year-old unskilled labourer, who stammered.

Personality studies revealed that he was hyper-sensitive and neurotic ; his intelligence quotientwas found to be comparable to that of a 10-year-old child. He was readmitted on-three differentoccasions, and every time the quiet hospitalenvironment and the fact that he could take asmuch time over his meals as he wished had avery good influence on his condition and resultedin rapid recovery.

Cases Treated by Myotomy. -The resultsobtained in two of the six cases of myotomy weresatisfactory. In one case there was a clear-cutrecurrence of symptoms with severe pain so thatre-operation was indicated, and this time a cardio-plasty was done. The patient is now symptom-free (Fig. 2). Evidence of slight or moderateachalasia was seen in the other three patientsafter operation.A comparison of the pre-operative radiological

outline of the oesophagus with that seen post-operatively showed that the degree of dilatationwas unchanged in four cases; one oesophaguswhich pre-operatively was moderately distendedhad returned to normal size, and in another therewas a moderate decrease compared with the sizeshown on the pre-operative films.The results achieved by myotomy were thus

rather discouraging both as to the function andthe morphology of the oesophagus, operationhaving had but little or no effect on the dilata-tion. This induced us to abandon myotomy andto give preference to cardioplasty.

OPERATION SEQUELAESecondary Anaemia.-In 1947, in the course of

the follow-up examination of these cases, a youngboy, who about four months earlier had beenoperated on successfully for achalasia, was foundto have a severe secondary hypochromic anaemia(Hb 51 % ; red count, 3.14 millions). As noexplanation could be found for the anaemia, andas we thought that the operation, which had beendone such a long time before, could hardly be heldresponsible, relatively large amounts of iron wereadministered and the patient recovered. Twoweeks later another patient, who had undergonethe same kind of operation, also appeared withsecondary anaemia of the hypochromic type.This made us suspect some connexion between theoperation and the anaemia, and we directed ourattention to this point, especially as Weber's testwas negative and the blood counts in these twopatients were normal on discharge from hospital.Perusal of the records of the blood counts in thefollow-up examinations of those patients who

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HbX communication B a r r e t t10o .E(1948) told us that his ex-

.V.. perience as regards the like-

,------...... ..--------- . ...lihood of anaemia occurringafter cardioplasty was iden-

/.fi>/\.;<~' ~~ .. ,;>,; , ,, tical with our own. In his80so .. Sn \ ;; ; /./# . - S " 'JX. \ ,/ cases a high proportion of

patients who had undergoneoesophago-gastrostomy -hadto /' <x1 .-- .. ' g .. .wS , -also developed a late second-ary anaemia whereas those60t , . , ' +***;/{.-**+,treatedby Heller's opera-

'* J / tion had a normal post-operative blood picture.

50 t t A perusal of the rele-vant literature revealed that

4 ,_,_,_ ____________________________________________ -..- . .~ lsecondary anaemia had sel-0 1 2 3 I 5 6 7 ?7onM dom been observed before

FIG. 3.-Diagram showing degree and time of onset of anaemia in six cases of and had not been believedachalasia treated by transpleural cardioplasty. The anaemia responded to to be related to the opera-iron medication. Arrows indicate initiation of iron therapy. tion. Grimson and others

(1946) published two caseshad until that time undergone myotomy and and Bell (1946) one. In Bell's case the secondary-ardioplasty showed that the incidence of secon- anaemia appeared 11 months after oesophago-lary anaemia was so high in the cardioplastic gastrostomy; it was severe and was diagnosedoases (Fig. 3) that we considered that this opera- as a symptom of bleeding ulcer, though notion must in some manner be held responsible for clear-cut evidence wvas available to support thethe blood changes. diagnosis.The secondary anaemia in these two cases and Many explanations may be proffered to explain

in those described later did not occur immediately the onset of secondary anaemia in these cases.post-operatively, but during convalescence (Fig. 3). One might be inclined to ascribe the condition toWe found that in all of the cases operated on iron deficiency due to achlorhydria, but in mostluring the last year, with the exception of the one of our cases histamine and insulin tests of thewho died three weeks after operation and another, gastric juice showed normal values so that atwho 10 days after operation had a severe haemor- least in these the root of the evil must be soughtrhage from a gastric ulcer, there was evidence of elsewhere.moderate or severe anaemia with a haemoglobin One theory is that granulation tissue formsvalue of under 50%. during the healing process at the site of theAll the cases were treated successfully by the anastomosis or of the cardioplasty, and that the

exhibition of iron (Fig. 3), and except for a certain numerous traumata caused by the passage of thefeeling of tiredness the patients were soon symp- food might result in small oesophageal lesions andtom-free. In some the disease recurred as soon haemorrhages in this region. We know fromas the medication was discontinued. experience elsewhere.g., rectal haemorrhages-We must stress that secondary anaemia was that small repeated haemorrhages will not produce

observed only after cardioplasty. Judging from symptoms until after a certain latent period. Thisour material, myotomy is not followed by anaemia, theory is supported by observations made in twoand we have not seen this complication post- cases in the present series in which post-operationoperatively or in the follow-up examinations oesophagoscopy revealed a very brittle and readilyduring the latter part of 1948. bleeding granulation tissue at the site of the suture

line. On the other hand, the secondary symptomDISCUSSION did not as a rule occur until two to four months

In this material secondary anaemia occurred as after the operation when haemorrhage from granu-a late complication of cardioplasty. The incidence lation tissue was hardly to be expected, and thiswas such that the operation was suspected of being argues against this belief. Moreover, in four ofresponsible for the blood disease. In a personal our cases with secondary anaemia both Weber

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FIG. 4.-Radiograph of chest of 59-year-old woman (a) before cardioplasty; (b) eight months after cardioplastyshowing no diminution of dilatation but good passage.

( el 1 /h)FIG. 5.-Radiograph of chest of 35-year-old man (a) before cardioplasty; (b) seven weeks later showing rapid

regression of the dilatation.

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tests and Benzidin tests were negative on post-operative review.Another theory is that in these cardioplastic

operations, in which part of the ventricular fornixis drawn up into the thoracic cavity, the bloodsupply in this part of the stomach is therebychanged, with the result that a varying degree ofmucous haemorrhage may be expected. In Franceclinical evidence seen in ventricular herniation ofthe diaphragm (Benard, Rambert, and Canivet,1947) seems to support the idea of haemorrhagesbeing the underlying cause of anaemia followingcardioplasty.A third theory, which we have embraced as a

working hypothesis, is that the underlying mechan-ism is far more subtle and complicated. In thisconnexion it should be pointed out that in all ofthese post-operatively anaemic patients the cardiawas no longer able to function as a normalsphincter because food could empty into, and beregurgitated from, the stomach unhindered. Inthe light of our knowledge of the fact thatabnormal variations in the gastric secretion ofthe mucous membranes of the alimentary tract areliable to affect the blood picture (anaemia inachylia gastrica) this secondary anaemia, encoun-tered after the radical treatment of achalasia, mightbe ascribed to an abnormal gastric secretion in theoesophagus-cardia-stomach region. If this theorybe proved correct it would explain the changes inthe blood picture. For the purpose of investiga-ting this possibility we are at present performing aseries of experiments on dogs which have beensubjected to cardioplasty and other transpleuraloperations. Judging from preliminary resultscardioplasty does not produce secondary anaemiain dogs.

SUMMARYTwenty-one patients were operated on for

achalasia (myotomy in six cases, cardioplasty in16 cases; one patient had two operations-namely,myotomy and cardioplasty).

All cases were operated upon transpleurally.No post-operative complications were observed,except in a single patient, who died from pulmon-ary embolism three weeks after operation.

In our material myotomy did not produce satis-factory results, with respect both to the functionand the morphology of the oesophagus, the opera-tion having had but little or no effect on thedilatation.

Cardioplasty gave functionally satisfactoryresults. The condition of most patients three

to five years after operation was still satisfactoryand the pre-operative degree of dilatationdiminished considerably.

Secondary anaemia developed in several casestreated by cardioplasty transpleurally one to fourmonths after operation. In all cases the anaemiaresponded to iron therapy. In-our cases treatedby myotomy no such anaemia was observed.The possible causation of the anaemia is

discussed.REFERENCES

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37, 194.Beattie, W. J. H. M. (1931). St Bart's Hosp. Rep.,

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by Julius Springer, 2, 572.and Haecker, R. (1906). Dtsch. med. Wschr.,

32, 1263.Wachs, E. (1940). Arch. klin. Chir., 200, 259.Wendel, W. (1910). Ibid., 23, 311.Willis, T. (1672). Pharmaceutica Rationalis (quoted by

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