FUNCTIONAL DISEASE THE OESOPHAGUS · BELSEY:Functional Disease of the (Esophagus spasm at any point. Failure of the lower end of the gullet to relax during swallowing leads to the

POSTGRAD. MED. J. (I963), 39, 290

FUNCTIONAL DISEASE OF THE OESOPHAGUSRONALD BELSEY, M.S., F.R.C.S.

Surgeon-in-Charge, Thoracic Unit, Frenchay Hospital, Bristol; Consultant in Thoracic Surgeryto the South West Region

THIS term is used to embrace all those causes ofdysphagia dependant upon a disorganization ofthe normal, co-ordinated, neuro-muscular functionof the oesophagus. All forms of dysphagia due toorganic stenosis of the gullet, obstruction of itslumen by foreign bodies, or compression byextrinsic pressure are excluded.

Consideration of the causes of functionaldysphagia is complicated by a lack of certainty inour knowledge of the normal activity of the gullet.This organ is not a mere inert drainpipe orpassive conduit. Not only does it function withefficiency and consistency as a pump to propelthe bolus or fluid from mouth to stomach undernormal conditions in man, but can also overcomethe forces of gravity, as demonstrated by thesportsman who, for a wager, imbibes a pint ofbeer whilst standing on his hands in the invertedposition. Anyone who has witnessed a giraffe inthe act of drinking will appreciate the dynamicpropensities of this organ.

Peristalsis, as normally accepted, probably doesnot occur in the oesophagus.The oesophagus is essentially a muscular tube

with a sphincteric mechanism at both ends. Thelumen normally contains a small quantity ofsaliva, and a larger quantity of air under a mildnegative pressure reflecting and varying with thenegative pressure in the pleural cavities. Thesphincter at the upper end is controlled by thecricopharyngeal muscle in a state of tonic contrac-tion. The vigilance of this sphincter is testifiedby the infrequency with which sudden inversionis accompanied by regurgitation of oesophagealcontents. Were it not so, then every patient withachalasia of cardia and a resting oesophagealresidue of up to two pints, or more, of fermentingfood would run the risk of drowning every night.During normal deglutition the upper sphincterrelaxes as the pharyngeal muscles voluntarilypropel the bolus from the mouth. The lowersphincter is more complex. There is considerabledoubt as to whether any intrinsic sphinctermechanism exists. Probably the control of gastro-

BELSEY: Functional Disease of the (Esophagus

spasm at any point. Failure of the lower end ofthe gullet to relax during swallowing leads to thewell-known condition of achalasia, with diffusehypertrophy and dilatation of the whole organ.

Failure of the lower sphincter and the reflux ofgastric secretion into the esophagus is the essentialfunctional disturbance responsible for most ofthe symptoms in hiatus hernia, and will not beconsidered further in this communication.

Finally, a rare form of dysfunction in whichacute spasm of the mid-third of the organ accom-panies the attempt to vomit large quantities offood and fluid after an alcoholic debauch, maylead to a rise in intra-luminal pressure sufficientto result in spontaneous rupture of the lowerthird with catastrophic results.

This communication is mainly concerned withthe management of the three commoner forms offunctional dysphagia amenable to surgical treat-ment-spasm of the upper sphincter, 'corkscrewesophagus', and achalasia of the cardia. The 132cases reviewed were all admitted to a RegionalSurgical Thoracic Unit under the care of onesurgeon over a period of 20 years. The reasonfor investigation was either dysphagia or thepulmonary complications of oesophageal dysfunc-tion. Non-surgical functional disorders, such asthose due to basilar artery thrombosis ormyasthenia gravis, will not be considered furtheralthough they complicate the problem ofdifferential diagnosis.

TABLE IRELATIVE FREQUENCY OF THREE PRINCIPAL FORMS

OF FUNCTIONAL DISORDER(i) Upper oesophageal spasm, with pouch

formation .. .. .. .. 20(ii) Corkscrew oesophagus .. .. I8(iii) Achalasia of cardia .. .... 94

Achalasia of Pharyngo-Esophageal Junctionwith Pouch Formation

Globus hystericus has been a popular diagnosisin any case where intermittent symptoms ofdysphagia have been referred to the cervicaloesophagus. In fact, the majority of these patientsare suffering not from hysteria but from organicobstruction of the cervical oesophagus by muscularspasm, or achalasia, in the region of the pharyngo-cesophageal junction (Fig. I). The spasm may besufficiently severe to lead to the formation of apulsion diverticulum through the posterior wallof the pharynx. When the patient presents withthe well-established picture of a pharyngealdiverticulum, the underlying obstructive elementis usually overlooked, and completely ignored inplanning treatment. This omission undoubtedlyaccounts for the unsatisfactory results of treatmentand the high incidence of recurrent diverticulum

FIG. I.-Severe spasm of the cervical oesophagusDysphagia relieved by myotomy.

Iformation. Attention is diverted from the under-lying cause of the dysphagia by the bizarreradiological manifestations of the sequelse. Thediagnosis is made largely on the symptoms, thelocalization of the obstruction, the radiologicaldemonstration of a zone of persisting spasm in thisarea, and the exclusion of other possible causesof obstruction. A barium swallow examinationmay reveal a small pulsion diverticulum of thelower pharynx, too small to be able to contributeto the dysphagia. (Esophagoscopy will contributelittle to the diagnosis except to exclude otherpossible causes for the symptoms, e.g. a malignantstricture. Apart from the distress caused by thedysphagia, the importance of this lesion lies in thefact that it is the precursor and cause of apharyngeal diverticulum. Before the onset of thiscomplication the dysphagia is variable, andinfluenced by emotional stress-hence the term'globus hystericus'. Once a pouch has developedand assumed a dependant position the symptomsincrease in severity and are more constant. Thesesymptoms are well recognized and will not beelaborated here, except to stress the frequencywith which these patients present with chronic orrecurring aspiration pneumonitis due to thenocturnal inhalation of the debris that mayaccumulate in the pouch.The development of a pulsion diverticulum is a

reversible process in its early stages, and relief of

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FIG. 2.-(a) Spasm of the cervical esophagus with early pulsion diverticulum. (b) Followingmyotomy; the spasm and dysphagia have been relieved and the diverticulum has disappeared.

I

FIG. 3.-Pulsion diverticulum of the pharyngo-cesophageal junction. The diverticulum disappearedcompletely following an upper cesophageal myo-tomy.

FIG. 4.-Lipiodol accumulating in the mediastinumfollowing instrumental perforation of a pharyngealdiverticulum. Spontaneous healing occurred, andat a later date successful myotomy and diverticulo-pexy were carried out.

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/3 Ho!...FIG. 5.-(a) Pharyngeal diverticulum.

cesophageal myotomy.

the obstruction may lead to rapid disappearanceof the pouch (Fig. 2). Hence the importance of earlyrecognition of this condition and its promptrelief.Negus (I950) has advocated dilatation of the

spastic pharyngo-oesophageal junction, repeated asnecessary. Even in the hands of an expert,endoscopic dilatation of the cesophagus is notdevoid of risk of perforation or abrasion of thegullet, the risk being directly proportional to thefrequency with which it has to be performed.If the dysphagia recurs after the initial dilatation,then other methods are indicated. That preferredby the author consists of an extramucous myotomysimilar to the classical Heller operation performedon the lower oesophagus for the relief of achalasia.A vertical myotomy incision is made over theantero-lateral aspect of the lower half-inch of thepharynx and the upper two inches of the cervicalesophagus, usually on the right side. Thisprocedure has proved satisfactory and has led tothe disappearance of early diverticula (Fig. 3).

It is logical to assume that destruction of thesuperior oesophageal sphincter might expose thepatient to the hazards of recurring aspirationpneumonitis. Provided that one of the cesophagealsphincters remains competent, in practice therisk appears to be small, and no instance has beenencountered so far in this small series. In factthere has been in this series no instance of'oesophageal breathing' as anticipated by Negus(1950) in discussing the principles of thisprocedure.Once a pulsion diverticulum larger than a

grape has developed then dilatation becomes evenmore dangerous owing to the difficulty of gainingaccess to the lumen of the esophagus beside theneck of the pouch and the risk of perforating the

(b) Following diverticulopexy and

FIG. 6.-'Corkscrew oesophagus' associated with aType I hiatus hernia.

bottom of the pouch and causing mediastinitis(Fig. 4). In addition to the myotomy alreadydescribed, either excision of the pouch, or itsinversion and suture to the anterior longitudinalligament of the cervical spine in the up-endedposition is necessary. The latter method has beenused by the author in i8 cases with completerelief of symptoms and no recurrence of thepouch formation. It is preferred to excision

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FIG. 7.-(a) 'Corkscrew esophagus' with a small diverticulum of the lowerthird. (b) Following Heller's operation. Dysphagia completely relieved,but the diverticulum persists.

owing to the risk of a salivary fistula and thedifficulty of performing a satisfactory repair ofthe thin posterior pharyngeal wall.A direct attack on the pouch with no attempt

to relieve the underlying cause will fail to relievecompletely the dysphagia and will be followed bya high rate of recurrent pouch formation (Fig. 5).The longest follow-up period following myotomyand diverticulopexy has been ten years in thisseries. As yet there has been no recurrence of thepouch formation or dysphagia.Diffuse (Esophago-spasm

This condition is commoner in the lower halfof the oesophagus. It is not peculiar to anyparticular age group or sex. It occurs in twoforms-primary and secondary. The primaryform appears to be closely allied to achalasia of thecardia in that examination of the oesophagusreveals diffuse muscular hypertrophy indis-tinguishable from that which characterizes thelatter condition. The secondary form is frequentlyassociated with the presence of a hiatus herniaand gastro-oesophageal reflux (Fig. 6). The assumedassociation between the two is based upon theobservation that surgical control of the gastro-cesophageal reflux leads to relief of the spasticcondition of the gullet. The degree of muscularhypertrophy is less in the secondary form.The radiological appearances presented by this

condition are bizarre and have acquired thedesignation of 'corkscrew oesophagus'. A seriesof contraction rings occurs intermittently through-

out the lower half of the oesophagus. They havenot been observed above the level of the aorticarch. The rings always occur at the same levels,and are not produced by an exaggerated form ofperistalsis. Solitary or multiple pulsion diverticulaare commonly associated with this form ofcesophago-spasm and are undoubtedly 'blow-outs'occurring in the zones of increased intra-luminalpressure.Much of our knowledge of the abnormal

physiology of this condition has been elucidatedby Good (working at the Mayo Clinic), by meansof serial pressure recordings from balloons locatedat various levels in the gullet. By this methodpressure changes at various points within thelumen can be recorded during swallowing and atrest, and the disordered activity of the gullet corre-lated withthe symptoms. The commonest symptomof this condition is dysphagia, but in some casesthe presenting symptom is chest pain of ananginal distribution. During swallowing the con-traction of the various spastic zones or rings mayoccur in one of two ways: either synchronously,or progressively from above downwards. In theformer instance the presenting symptom is dys-~phagia; in the latter, substernal chest pain.The indication for surgical treatment in this

condition is the severity of the symptoms. Thepresence of pulsion diverticula in the lower'oesophagus rarely aggravates the symptoms andin contrast to those at the pharyngo-aesophagealjunction, these do not need surgical treatment,except in the rare instances when they develop

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FIG. 8.-(a) Achalasia of the cardia complicated by gross oesophageal dilatation.Note fluid level above the right clavicle. (b) Two years following modifiedHeller's operation. (Esophageal dilatation persists, but there is now noretention of fluid in the organ.

to a large size, and cause extrinsic pressure on thegullet below the level of the origin of the diverti-culum (Fig. 7). Dilatation is rarely of value in thiscondition and if diverticula are present can bedangerous. Relief of the dysphagia can be achievedby a Heller type of myotomy performed on thelower oesophagus as in the treatment of achalasiaof the cardia. A vertical extra-mucous myotomyincision is made over the lower three inches ofesophagus at least, and extended for a furtherhalf inch over the cardia of the stomach to ensurethat all the circular muscle fibres of the lowercesophagus have been divided. If a hiatushernia is present this should be repaired and afunctioning valvular mechanism restored to thecardia to prevent any further gastro-cesophagealreflux. Minor degrees of aesophagospasm arefrequently encountered complicating reflux andeffective control of this reflux leads to prompt andcomplete relief of the spasm.

Excision of pulsion diverticula of the loweroesophagus, on the mistaken assumption that theyare responsible for the patient's symptoms,without relief of the functional obstruction willlead to catastrophic and often fatal complicationsin the form of broken-down suture lines andmediastinal and pleural suppuration.Achalasia of the CardiaThe cause of this condition remains obscure.

Commonly the obstruction is confined to thecardia but in the early stages of the conditionthere may be diffuse spasm of the lower half of theorgan; in the later stages this is superseded bydiffuse progressive dilatation and lengthening of

the organ. The irregular spasm already describedas 'corkscrew oesophagus' may be associated withachalasia and there appears to be a close patho-logical affinity between the two conditions.However, pulsion diverticula are rarely seen inachalasia. There is diffuse uniform hypertrophyof the muscle layers of the oesophagus, maximalin the lower half.At the cardia, the point of obstruction, the

architecture of the muscle layers suddenlybecomes normal and in this region there is nohypertrophy, supporting the thesis that theobstruction is due to failure of normal relaxationrather than true spasm. Pathological changeshave been observed in the nerve plexes in themuscle layers but whether these are primary orsecondary, congenital or acquired, is not known;nor is their significance in the aetiology of thiscondition. In longstanding cases the degree ofdilatation or mega-oesophagus that develops isprodigious and this undoubtedly jeopardizes thefunctional result following surgical relief of theobstruction. Moderate dilatation will disappearfollowing operation; gross dilatation will diminishbut some degree will remain permanently (Fig. 8).Considerable quantities of ingested food andfluid, some taken several days previously, arecommonly retained in the dilated gullet. In theerect position a fluid level can be seen in theregion of the clavicles on radiological examination.Conditions are ideal for alcoholic fermentation,a fact which may explain the unnatural euphoriaenjoyed by many patients suffering from thiscondition. Of greater importance is the diffuseretention oesophagitis that may occur in advanced

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cases. The mucosa is then reddened and oedema-tous, and has a characteristic granular appearance,bleeding readily if touched. There is no resemb-lance to the oesophagitis caused by gastro-oesophageal reflux. The significance of theoesophagitis is twofold. First, it appears to be adefinite pre-malignant condition; reference tothis will be made later. Second, the presence ofgross retention oesophagitis is a contra-indicationto radical surgical treatment and some preliminaryform of drainage is necessary to control it beforeany form of Heller procedure is attempted,owing to the risk of perforation or spontaneousrupture of the softened oedematous mucosa whosetexture can rightly be likened to that of theproverbial wet blotting paper.Diagnosis

Although dysphagia is the presenting symptomin approximately 90% of cases, the patient maycome under observation in other ways. Thedysphagia is of such long duration, and so insidiousin onset and progress that the patient may accepthis disability as natural and inevitable. Only byasking leading questions can the clinician elicita true picture of the patient's disability. Thesepatients are often diagnosed as hysterical in theearly stages before obvious dilatation of thegullet has occurred. Four patients with achalasiawere referred to the author from mental hospitalswith a diagnosis of chronic depression and 'anoesophageal abnormality'. On questioning it waslearnt that the patients were depressed merelybecause they could not swallow. Relief of thedysphagia resulted in a dramatic psychiatric cure.Pulmonary complications due to recurring

aspiration pneumonitis, following regurgitation ofoesophageal contents at night when the upperoesophageal sphincter is caught off its guard, arecommon, and may result in extensive, diffuse,pulmonary fibrosis. The dyspnoea and pulmonarysymptoms then overshadow the dysphagia. It issurprising that more patients do not drown intheir own cesophageal residue. The youngestpatient in the author's series, a boy of 8 years, wasadmitted to hospital with a diagnosis of bron-chiectasis for further investigation. It was thenoise caused by the aspiration of fluid into thetrachea and bronchi at night that led to thecorrect diagnosis. As long ago as I943 Hurstdescribed four cases with respiratory complicationsfollowing regurgitation, and a fifth, fatal, case ofasphyxia.

Achalasia may be brought to light as the resultof a routine mass miniature radiography examina-tion. The opaque, fluid-filled, dilated oesophaguspresents many of the radiological appearances ofa mediastinal tumour. When the dilatation is

greatest in the aircontaining upper third of theorgan, a diagnosis of lung cyst, lung abscess, oreven pneumothorax may be made.

In one instance the patient was admitted to amedical ward with a diagnosis of acute rheu-matoid arthritis. Examination suggested acutepulmonary osteoarthropathy and further investi-gation confirmed the presence of achalasia of thecardia. The association of pulmonary osteo-arthropathy and oesophageal disease is nowrecognized. In this case the retention oesophagitiswas so severe that all mouth feeding was stoppedand a preliminary gastrostomy performed: within48 hours all pain and swelling had disappearedfrom the joints. One month later a Heller operationwas performed and the gastrostomy allowed toclose.

Investigation is carried out by means of radio-logical examination and oesophagoscopy. Theradiological appearances are well recognized,especially the smooth, conical, constrictionconfined to the cardia that characteristicallyrelaxes after the inhalation of octyl nitrite. Theprotean appearances presented by the dilatedoesophagus on plain radiological examinationmimic all forms of mediastinal pathology. Inno other form of oesophageal obstruction is acomparable degree of oesophageal dilatation seen.

CEsophagoscopic examination is unsatisfactoryowing to the difficulty of clearing the gullet of itsretained debris. The examination is safer whenperformed in the sitting position under localanesthesia; induction of general anaesthesia maylead to sudden flooding of. the air passages withoesophageal contents. The main object of thisexamination is to inspect the mucosa and determinethe severity of the retention oesophagitis. Acareful inspection is carried out for any signs ofearly malignancy. It is rare for any convincingview to be obtained of the cardia and the exclusionof a malignant or benign stricture at the cardiamay be impossible.Relationship to Carcinoma of the (Esophagus

In the author's series of 94 cases of achalasiaof the cardia there were eight cases of cesophagealcarcinoma. In four cases the patients presentedwith severe obstruction due to advanced carcino-mata; radiological examination revealed theunderlying achalasia and megaoesophagus. Theremaining four patients had previously beentreated for achalasia and returned with recurrentobstruction due to malignant strictures 15 monthsto five years following the Heller operation. In alleight cases the growths were squamous cell intype, were situated in the mid-third of the gullet,and had reached an advanced stage before obstruc-tive symptoms occurred owing to the previous

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FIG. 9.-Achalasia of the card'a complicated by anadvanced carcinoma of the middle third of theoesophagus.

dilatation of the oesophagus (Fig. 9). In one casemultiple carcinomata were present. Inaseriesofover500 cases of hiatus hernia complicated by refluxcesophagitis observed and treated by the author,there was no instance of a squamous cell carcinomacomplicating the oesophagitis although severalco-existing adenocarcinomata of the cardia wereencountered. This evidence suggests that theretention cesophagitis complicating achalasia is apre-malignant condition involving a high risk ofmalignant degeneration; that the reflux cesophag-itis found in association with a hiatus hernia isnot a pre-malignant condition. Of great interestare the four patients who developed malignantstrictures subsequent to Heller operations, andin one case five years later. It is probable inretrospect that malignant degeneration had alreadycommenced at the time of the original operation.As already stated it is notoriously difficult toconduct a thorough examination of the wholecesophageal mucosa, and when grossly inflamedand obscured by rotting food debris and barium-an early carcinoma can easily escape detection.In four of the cases determined but ultimatelyfutile attempts were made to resect and reconstruct

the gullet, but in no instance was the growthoperable by normal standards. It is logical toassume that the sooner steps are taken to relieve theobstruction and oesophagitis in achalasia, the lessthe risk of subsequent malignant degeneration.

TreatmentPre-operative care is important. If gross

oesophagitis is present a course of octyl nitriteinhalations following low residue meals willdrain the osophagus from below. It may benecessary to pursue this regimen for three tofour weeks before it is safe to proceed withsurgical treatment. In two cases in the author'sseries it was necessary to perform temporarygastrostomies and stop all mouth feeding tobring the inflammation under control. CEsopha-geal washouts are dangerous owing to the risk ofdrowning the patient.An intensive course of physiotherapy to improve

the pulmonary complications is usually called for.The patient's nutritional state may be severelydepleted from chronic starvation; this againmust be corrected and blood transfusion may benecessary.Numerous operations have been described for

the relief of this condition. Repeated dilatationsenjoyed a short vogue of popularity which was notjustified by the results, and has now beenlargely abandoned by even its most ferventadvocates. Various forms of oesophago-gastro-stomy have been performed, to be followed in ahigh percentage of cases by disastrous gastro-oesophageal reflux and ulceration. An extra-mucous myotomy (Heller operation) dividing allthe circular muscle fibres over the lower two tothree inches of esophagus is the most satisfactoryoperation available at the present time. Theoperation is best performed through the chestas only an inadequate myotomy incision is possiblethrough the abdominal approach. Franklin hasrightly said that it is an easy operation to performbut a difficult operation to perform well. In itsoriginal form the operation was not entirelysuccessful, the occasional poor results being dueto the development of fibrous strictures secondaryto gastro-cesophageal reflux. It has not beengenerally recognized that any surgical inter-ference with the region of the cardia may weakenthe already precarious valvular mechanism, andprecipitate the development of a hiatus herniaor patulous cardia and its sequelae. The authorhas in recent years employed a modification ofthe Heller procedure. Recognizing the risk of ahiatus hernia developing, the cardia is boldlyfreed from its attachments and following themyotomy a formal hernia repair is carried out

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298 ~~POSTGRADUATE MEDICAL JOURNAL My16

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FIG. io.-(a) Mega-cesophagus complicating achalasia of the cardia. (b) Nineyears following modified Heller myotomy; the cesophageal dilatationhas disappeared.

by creating an acute angle of entry at the cardia,restoring an intra-abdominal segment of cesopha-gus, and approximating the two halves of theright crus behind the hiatus to form a buttressagainst which the intra-abdominal segment ofoesophagus can be compressed by the abdominalpressure. In a personal series of go cases treatedsurgically, in the first 56 a formal Heller operationwas carried out with satisfactory initial results,but in six cases, or 11%, fibrous strictures subse-quently developed of sufficient severity to needesophago-gastric resection and reconstruction(Fig. Io). In the last 34 cases the modified operationhas been performed and as yet there has been noinstance of stricture formation or subsequentnecessity for further surgery. There was onepost-operative death in this series, from uraemiadue to co-existing chronic nephritis.The surgical relief of functional cesophageal

obstruction is justified by the gratitude of thepatient, by the satisfactory long-term results, and

by the relative safety of the surgical proceduresindicated.

SummaryI. A series of 132 cases of functional disorders

of the oesophagus has been described. Of these 120required surgical treatment for relief of dysphagia,or the pulmonary complications of oesophagealobstruction.

2. No operation for pharyngeal diverticulum iscomplete unless the underlying spasm of theupper sphincter is relieved.

3. Pulsion diverticula of the lower esophagusrarely need surgical treatment; the underlyingfunctional obstruction frequently does.

4. Achalasia of the cardia is a pre-malignantcondition.

5. The Heller operation must be accompaniedby the reconstruction of a functioning valvularmechanism at the cardia to prevent subsequentgastro-cesophageal reflux and fibrous stenosis.

REFERENCESFRANKLIN, R. H.: Personal communication.HURST, SIR A. (1943): Respiratory Complications of Achalasia of the Cardia with Mega-CEsophagus, Guy's Hosp. Rep.,

92, 68.NEGUS, V. E. (1950): Pharyngeal Diverticula: Observations on Their Evolution and Treatment, Brit. J. Surg., 38, 129.

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FUNCTIONAL DISEASE THE OESOPHAGUS · BELSEY:Functional Disease of the (Esophagus spasm at any point. Failure of the lower end of the gullet to relax during swallowing leads to the