Aspiration pneumonia dysphagia after technically successful repair … · Aspiration pneumonia and dysphagia after technically successful repair of oesophageal atresia 107 (a) (b)

Thorax (1966), 21, 104.

Aspiration pneumonia and dysphagia after technicallysuccessful repair of oesophageal atresia

A. R. CHRISPIN, G. W. FRIEDLAND, AND D. J. WATERSTONFrom the Hospital for Sick Children, Great Ormonid Street, Londonz, W.C.J

In the past decade increasingly satisfactory resultshave been achieved in the surgical repair of thecommon congenital abnormality of oesophagealatresia with fistula. However, dysphagia andaspiration pneumonia can occur after operationand may well be related to a stricture at the siteof the anastomosis or to a recurrent fistula. Insome patients, however, no stricture or fistula canbe found to account for an episode or episodesof aspiration pneumonia and dysphagia. Fourteensuch patients with severe symptoms have beeninvestigated. All had previously had a technicallysuccessful one-stage repair of the common typeof oesophageal atresia and fistula (Waterston,Bonham-Carter, and Aberdeen, 1963). Radiologi-cal studies have revealed abnormalities of oeso-phageal flow which can account for these symp-toms. An understanding of the underlying oeso-phageal abnormality enables the clinician to makea logical approach to the management of thesepatients' problems.

CLINICAL DATA

In these 14 patients (Table 1) a primary anasto-mosis of the blind upper pouch to the loweroesophageal segment was achieved, the fistulouscommunication with the trachea being closed atthe same operation. Six of the 14 patientsdeveloped a stricture or diaphragm in the imme-diate post-operative period, but all were satisfac-torily dilated. Later, at oesophagoscopy, three ofthe six were thought to have a mild narrowinginsufficient in itself to cause symptoms. The otherthree (making 11 in all) had a normal lumen.The clinical features that prompted radiological

investigation were aspiration pneumonia anddysphagia. During infancy a common sequencewas an attack of choking and vomiting or rest-lessness at night followed by or associated withcyanosis or even white asphyxia and cardiacarrest and later pneumonia. Young infants also

experienced difficulty with milk feeds, and thiswas sometimes ameliorated by thickening thefeeds. Older infants and children experiencedchoking and discomfort when swallowing solidlumpy foods. Peas seemed particularly liable tocause this. In one patient a peanut obstructed thelower oesophagus and had to be removed atoesophagoscopy. All patients had a characteristiccough.

RADIOLOGICAL INVESTIGATION

OESOPHAGUS Cinefluorographic studies were per-formed on eight patients. In five a recurrence offistula was initially thought to be a possibility,and such a fistula was excluded by radiologicaland endoscopic examinations. Ordinarily cine-fluorographic studies were carried out with thepatient swallowing normally. Usually the patientwas examined supine with either a horizontal orvertical beam. Occasionally the patient was alsoexamined in the erect position.

Conventional fluoroscopy and spot filming wascarried out in 10 patients, usually with the patientsupine but sometimes with the patient erect.

CHEST All patients had chest radiographs. Somepatients required many because of recurrentpneumonia.

RADIOLOGICAL FINDINGS

CONTRAST STUDIES OF OESOPHAGUS Normally, ininfants and children lying in the recumbent posi-tion and following pharyngeal deglutition, astripping wave passes down the oesophagus asfar as the vestibule, which lies immediately abovethe stomach. The oesophagus is completelyemptied of contrast medium by the strippingwave. Contrast medium passes through the vesti-bule into the stomach, and, as flow through thevestibule ceases, its lumen closes.

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Aspiration pneuimonia and dysphagia after technically successftul repair of oesophageal atresia 105

TABLE I

e atio Symptoms Prompting Radiological Radiological Results of Contrast StudiesPatientInvestigatson Investigation TechniqueI (yrs.)

Episodes of choking associated with Fluoroscopycyanosis, rapid respiration, and res- Cinefluorographypiratory infections

Recurrent aspiration infections from Fluoroscopyage 10 days; episodes of vomiting andrestlessness at night; milk feeds only

On three occasions sudden collapse Fluoroscopyassociated with white asphyxia and Cinefluorographysubsequent aspiration pneumonia andcyanosis

Aspiration pneumonia; severe air Cinefluorography ontrapping-age 6 mths; later white two occasionsasphyxia, cardiac arrest, aspirationpneumonia after swallowing solids

Recurrent episodes of choking and res- Fluoroscopypiratory infection from age 2 yrs

6 1 Recurrent respiratory infections; strainedfood taken well but chokes andheaves with lumpy food

7 5 Recurrent bronchitis; vomiting afterfood; struggling and refusal to eatsolids until nearly age 2, thereafterepisodes of choking and food sticking

8 6 Six episodes of aspiration pneumonia;pools of saliva in throat; dribblesfrom mouth; recurrent episodes of"food sticking in throat" and vomiting;still on mashed and strained food

9 2 Noisy cough for one year; lumpy foodsticks in throat

10 10 Recurrent respiratory infections; in-ability to take solids; food sticks inthroat; copious vomiting since birth-severe symptom-described by motheras "frightening"

I I 2 Persistent cough; chronic wet chest;frequent vomiting with mucus; at Iyear, peanut stuck at lower end ofoesophagus-removed at oesophago-scopy

12 2 Recurrent respiratory infections; stillon mashed foods

13 0 2 Aspiration pneumonia severe and per-sisting

14 0 5 Severe aspiration pneumonia

Fluoroscopy

Fluoroscopy

FluoroscopyCinefluorography

Fluoroscopy

Cinefluorography

Cinefluorography

Fluoroscopy

Cinefluorography

CinefluorographyFluoroscopy

Inefficient stripping action; hiatalhernia ; oesophageal residue

Inefficient stripping action; hiatalhernia; oesophageal residue

Inefficient stripping action; hiatalhernia with very free reflux; oesopha-geal residue

Inefficient stripping action, Hiatalhernia; oesophageal residue

Atonic below anastomosis; hiatal heiniawith very free reflux; oesophagealresidue

Atonic below anastomosis; hiatal hernia;oesophageal residue; aspiration ofcontrast medium

Atonic below anastomosis; hiatal hernia;oesophageal residue; prominent leftatrial impression

Atonic from pharynx to vestibule;no hiatal hernia; oesophageal residue;Prominent left atrial impression

Yo-yo movements; absent strippingwave; hiatal hernia-very free reflux;oesophageal residue

Yo-yo movements; absent strippingwave; hiatal hernia; oesophagealresidue

Yo-yo movements; absent strippingwave; hiatal hernia; oesophagealresidue

Yo-yo movements; absent strippingwave; hiatal hernia; oesophagealresidue; prominent left main bronchialimpression

Yo-yo movement resulting in pharyn-geal filling even when erect; absentstripping wave; no hiatal hernia;oesophageal residue

Yo-yo movement on one occasion;no stripping wave visible; oesophagealresidue; hiatal hernia

No significant stricture, diaphragm, or fistulawas present in any of the 14 patients.

A bsence of normal effective stripping waves

Pharyngeal deglutition was normal in all patients.Oesophageal stripping waves were absent in mostpatients, but in some indifferent, inefficient strip-ping waves were seen. However, common to allpatients was the absence of normal efficient strip-ping waves, and in all an oesophageal residuefollowing cessation of deglutition was present.Four patients showed indifferent stripping

action in the oesophagus. Initially stripping waves

emptied the oesophagus, but later the waves were

less efficient and an oesophageal residue remained(Fig. 1). The oesophagus showed little sign ofcontractility at this time.

K

Four patients had an oesophagus which showedlack of contractility. Sometimes a flicker of con-

tractility was seen at the start of swallowing, butthe oesophagus was subsequently completelyatonic. The segment below the anastomosis was

atonic in three (Fig. 2), and in one patient theoesophagus was atonic from the pharynx to thevestibule (Fig. 3).

Six patients exhibited the 'yo-yo phenomenon'(Fig. 4). Oesophageal stripping waves were absentand an oesophageal residue was present after theact or acts of deglutition had ceased. This oeso-

phageal residue was associated with intermittentspontaneous contractions in the lower part of theoesophagus. With each contraction of the loweroesophagus, contrast medium was expelled intothe upper oesophagus. Subsequently the con-

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A. R. Chrispin, G. W. Friedland, and D. J. Waterston

FIG. 1. Patient 2: (left) swallowing, vestibule open; (right) deglutition hasceased. Vestibule contracted; oseophageal residue because of inefficient strippingaction; hiatal hernia.

FIG. 2. Patient 6. About 2 min. after deglutition hadceased. Atonic oesophagus below anastomosis; vestibuleclosed; oesophageal residue; hiatal hernia.

tracted segment relaxed, and contrast mediumreturned into the lower part of the oesophagus.This sequence, when repeated, resulted in con-tinuing upward and downward movement ofcontrast medium, and this feature has becomewidely known as the yo-yo oesophagus. Even inthe erect position the yo-yo movement resulted inreflux into the pharynx in one infant.

There was no evidence of contractility in theoesophagus above the anastomosis as a result ofcontrast medium entering it from below. Only inone patient did the yo-yo type contraction appearto interfere with flow down the oesophagus duringdeglutition, but this interference was transient.

Function of the vestibule Flow of contrastmedium down the oesophagus, through the vesti-bule, and into the stomach occurred as long asacts of deglutition continued. As deglutitionceased so flow through the vestibule ceased. Sinceoesophageal stripping action was defective a resi-due remained in the oesophagus above the closedvestibule. In the erect position gravity-assistedemptying occurred, but this was incomplete (Fig.3d), especially in the younger patients. Duringdeglutition the vestibule opened to the same extentas is seen in normal patients. In achalasia thevestibule never opens in this way.

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Aspiration pneumonia and dysphagia after technically successful repair of oesophageal atresia 107

(a) (b) (c) (d)FIG. 3. Patient 8. Oesophagus atonic from pharynx to vestibule: (a) swallowing, vestibule open; (b) swallowingceasing, vestibule starting to close, prominent left atrial impression; (c) prominent left atrial impression; (d)erect, some time after swallowing had ceased. Vestibule closed, atonic oesophagus containing air and barium.Hiatal hernia In 12 of the 14 patients a slidingtype of hiatal hernia was found. Reflux from thestomach into the oesophagus in the presence of ahiatal hernia is a well-recognized event, and it wasseen in these patients.

Impressions of left main bronchus and leftatrium These were unusually clearly seen insome patients because of the residue in the oeso-phagus (Figs 3 and 4).

CHEST RADIOGRAPHS Pneumonic consolidationwas seen in all patients. Repeated episodes of pul-monary consolidation were seen in some patients.All parts of the lung have been affected andthere has been no predilection for the right upperlobe such as is seen in the patient with a tracheo-oesophageal fistula. In one patient (an infant aged6 months) the pulmonary involvement was asso-ciated with marked air trapping and dilatation ofthe main pulmonary artery, suggesting pulmonaryhypertension and cor pulmonale. Gradually, withtreatment, the appearances returned to normal.

DISCUSSION

REVIEW OF PREVIOUS STUDIES Several observershave reported radiological studies of the oeso-phagus in patients in whom a technically success-ful repair of oesophageal atresia with closure offistula had been achieved. Astley (1956) described'the development of gastro-oesophageal incompe-tence due to operative traction on the cardia'.Haight (1957) noted that in five patients the nor-mal oesophageal contraction or stripping wavefailed to pass through the site of anastomosis, butit reappeared in the lower oesophageal segmentbelow the anastomosis. Kirkpatrick, Cresson, andPilling (1961) conducted cineradiographic studieson 15 patients who had developed dysphagia,impaction of foreign bodies, and aspiration pneu-monia. Contrast medium remained above theanastomosis until a mass contraction propelled amajor part of the bolus past the line of anasto-mosis. Subsequently, after contrast medium hadreached the lower oesophagus, contraction in thatpart of the oesophagus resulted in retrograde but



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studies were carried out pre-operatively on onepatient aged 5 years and on another aged 11 yearsin the prone-oblique position during normaldeglutition. A tracheo-oesophageal fistula (con-firmed at operation) was shown in each patient,but no stripping wave above or below the fistulawas seen. These findings suggest that the absenceof normal stripping waves can be an inherentfeature of the patient with a tracheo-oesophagealfistula. Maybe this is also the case when an atresiahas been present.

Effect of surgical technique It seems unlikelythat surgical techniques are related either to thedevelopment of the yo-yo phenomenon or to theatonic oesophagus. Five surgical teams have beenconcerned with the initial operation in these 14patients. In some patients meticulous care hasbeen taken to achieve primary anastomosis withthe minimum of dissection above and below thesite of anastomosis. In others it has been neces-sary to carry out dissection above and below theanastomosis to produce a sound union.

RELATION OF RADIOLOGICAL FINDINGS TOSYMPTOMATOLOGY

FIG. 4. Patient 12. Deglutition has ceased. Oesophagealresidue. Lower oesophagus contracts, giving upwardphase of yo-yo movement. Prominent left main bronchusimpression.

also some antegrade flow of the contrast medium.Studies on 49 patients, all of whom exhibited ayo-yo phenomenon, were reported by Desjardins,Stephens, and Moes (1964). Six of the 49 oftenhad difficulty with swallowing and had recurrentrespiratory infections. Girdany (1963) has pointedout that the yo-yo phenomenon is not a featurespecific to the patient who has had a repair ofoesophageal atresia.

INTERPRETATION OF RADIOLOGICAL FINDINGS

Evidence favouring an inherent oesophagealabnormality All patients showed an absence ofnormal efficient stripping waves with failure toempty the oesophagus when deglutition ceased.Kirkpatrick et al. (1961) examined a patient with.tracheo-oesophageal fistula and oesophagealstenosis pre-operatively and found an absence ofstripping waves below the stenosis. At theHospital for Sick Children cinefluorographic

Abnormal antegrade oesophageal flow (Fig. 5)Factors affecting flow down the oesophagus inthese 14 patients are (1) inefficient oesophageal

FIG. 5. Abnormal antegrade oesophageal flow. Patienterect. Inefficient or absent stripping wave in the wall ofthe oesophagus (1) and closure of vestibule (2) on cessationof deglutition result in failure of oesophageal content(3) to pass into stomach (4).

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Aspiration pneumonia and dysphagia after technically successful repair of oesophageal atresia 109

contractility; (2) gravity-in the erect positionthis is important, especially when liquids (notablyhigh density mixtures such as barium) are beingswallowed ; (3) repeated acts of deglutition inwhich repeated pharyngeal contractions propelmaterial into and down the oesophagus; (4) vesti-bular closure after cessation of deglutition whichwith (1) results in an oesophageal residue.

In infants in whom sucking and pharyngealdeglutition are normal, intake into the abnormaloesophagus may exceed output through the vesti-bule into the stomach. This imbalance wouldaccount for the episodes of choking in this agegroup.

In older children episodes of choking are lesscommon because they have a larger capacity oeso-phagus of greater length, feeding occurs in theupright posture, and intake is personally con-trolled. Dysphagia with solid foods is then thedominant symptom. Defective oesophageal strip-ping action fails to impel poorly chewed, solid,lumpy food down the oesophagus into thestomach. In the erect position, low density food,such as peanuts, may linger in an oesophagealfluid residue and may impact in the vestibule.

Retrograde oesophageal flow (Fig. 6) Thepresence of a hiatal hernia impairs reflux-preventing mechanisms at the oesophago-gastricjunction. Once reflux has occurred, defectiveoesophageal stripping action can result in unim-peded flow up the oesophagus as far as the

pharyngeal sphincter. The yo-yo movement alsoresults in retrograde oesophageal flow to this level.Horizontal beam cinefluorographic studies withthe infant swallowing supine have shown that thefluid level in the stomach, after even a small feed,is considerably higher than the vestibule, oesopha-gus, pharynx, and larynx. The young infant spendsmuch of its life lying on its back; in this positiononly the pharyngeal sphincters prevent reflux intothe pharynx and vomiting.

Aspiration pneumonia Abnormalities of tran-sit down the oesophagus, reflux with unimpededretrograde flow through the oesophagus, the yo-yomovement in some patients, and an oesophagealresidue after deglutition may all account foraspiration into the respiratory tract.

MANAGEMENT

Erect position In young children this resultsgravity-assisted oesophageal emptying, anddiminishes the chance of reflux.

init

Thickened feeds In infancy these reduce therisk of regurgitation and slow the rate of feeding,thereby avoiding imbalance between the rate ofintake into the oesophagus and the rate of oeso-phageal emptying into the stomach.

Water at the end of a feed Theoretically alittle water given at the end of each feed should

FIG. 6. Retrograde oesophageal flow. Diagrammatic representation based on cine film using h9rizontal x-ray beamwith patient lying supine. Fluid level in stomach (1) is higher than hiatal hernia (2) and vestibule (3), thus permittingreflux from stomach into oesophagus (4). The oesophagus has an inefficient sec?ndary stripping wave. Flowup the oesophagus occurs unimpeded to the lower pharyngeal sphincter (5). When this sphincter relaxes on deglutitionthe oesophageal content can pass into the pharynx (6), larynx (7), and trachea (8), all of which are lower than thelevel in the stomach (1).



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result in a residue of water in the oesophagusrather than a residue of milk or food.

Older children Dysphagia is the major prob-lem in older children, and this may be lessenedby giving minced food and/or water betweenmouthfuls of well-chewed solid food. The vesti-bule opens with each act of deglutition, and thewater provides an intermittent oesophageal lavage.

SUMMARY

Fourteen patients who had previously had atechnically successful repair of oesophageal atresiaand fistula presented with episodic aspirationpneumonia and dysphagia.Conventional fluoroscopy and spot-filming and

cinefluorographic studies of oesophageal functionwere carried out. Significant findings in the oeso-phagus were (1) inefficient stripping action;(2) oesophageal residue after deglutition; (3) slid-ing hiatal hernia with reflux. Chest radiographsshowed evidence of aspiration pneumonia. Onepatient had severe air trapping and probably pul-monary hypertension.

Correlation of symptomatology with radiologi-cal findings suggests that the abnormality ofoesophageal function is causative. Abnormalitiesof transit down the oesophagus, reflux throughthe oesophagus, and the oesophageal residue after

deglutition can account for choking, dysphagia,vomiting, and aspiration into the respiratory tract.Knowledge of these oesophageal abnormalities

provides a logical approach to management. Ininfancy the erect position, thickening of feeds, anda swallow of water at the end of the feed can allbe helpful. Dysphagia in older children can beameliorated by the swallowing of water betweenmouthfuls of well-chewed or minced food.

We wish to thank Professor A. W. Wilkinson, Mr.G. H. Macnab, Mr. H. H. Nixon, and Mr. J. D.Atwell, who have undertaken the care of some ofthe patients in this series. We are indebted to Dr.R. E. Bonham-Carter, of the Thoracic Unit, for aconsiderable amount of help and advice. We mustthank Dr. L. G. Blair for his helpful comments onthe preparation and presentation of the paper.

REFERENCES

Astley, R. (1956). Radiology of the Alimentary Tract in Infancy,p. 7. Arnold, London.

Desjardins, J. G., Stephens, C. A., and Moes, C. A. F. (1964). Resultsof surgical treatment of congenital tracheo-esophageal fistula,with a note on cine-fluorographic findings. Ann. Surg., 160, 141.

Girdany, B. R. (1963). The esophagus in infancy: congenital andacquired diseases. Radiol. Clin. N. Amer., 1, 557.

Haight, C. (1957). Some observations on esophageal atresias andtracheo-esophageal fistulas of congenital origin. J. thorac. Surg.,34, 141.

Kirkpatrick, J. A., Cresson, S. L., and Pilling, G. P. (1961). Themotor activity of the esophagus in association with esophagealatresia and tracheoesophageal fistula. Amer. J. Roentgenol.,86, 884.

Waterston, D. J., Bonham-Carter, R. E., and Aberdeen, E. (1963).Congenital tracheo-oesophageal fistula in association withoesophageal atresia. Lancet, 2, 55.

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Aspiration pneumonia dysphagia after technically successful repair … · Aspiration pneumonia and dysphagia after technically successful repair of oesophageal atresia 107 (a) (b)