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1954;14;254-258PediatricsGEORGE WILLIAM WARE and LOUIS L. CROSS

THE ESOPHAGUSCONGENITAL TRACHEO-ESOPHAGEAL FISTULA WITHOUT ATRESIA OF

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Online ISSN: 1098-4275.Copyright 1954 by the American Academy of Pediatrics. All rights reserved. Print ISSN: 0031-4005.

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CONGEN ITAL TRACHEO -ESO PHAGEAL F ISTULA W ITHOUTATRES IA O F THE ESOPHAGUS

B y GEORGE W iwA rsi W AR .E , M .D ., AN D L ou is L . C no ss, M .D .W a sh in g to n , D .C .

25 4

TRACHEO -ESOPHAGEAL F Isi-u i. AC ONGEN I TA L tracheo-esoph ag eal fistu law itho u t atres ia o f the eso phag us is a

ra re an om a ly . In d is cu ssing th is su b ject,G ross s tate s : W e h av e encoun te red fou ro f th ese ab norm a litie s du rin g th e tim e tha tw e have seen 25 9 bab ie s w ith esoph ag ea la tre sia . H a ig h t, w ith ex pe rience in 6 3 caseso f e sop hageal a tres ia, trea ted tw o cases o ftracheo-e sop hageal fis tu la w ith ou t a tre sia .A t the C h ild ren s H osp ital in C h icag o , o n lyone case w as seen , w h ile 43 cases o f a tre siao f the eso phagu s w ith tracheo-e soph agealf is tu la w ere trea ted .h l W e have rev iew edth e E ng lish lite ratu re an d fo und 26 caseso f th is en tity . To th is g roup , w e w ish to addan o the r case w hich w as treated su ccessfu llyb y su rge ry .

W e have co nside red adu lts rep o rted w ithth e an om a ly as p ossib le case s.2 5 , 26, 2 7 Pa -tien ts desc ribed b y G engenb ach ( C ase 7)and S toeck le in a re a lso p laced in th is pos -s ib le ca tegory . A spon taneous cu re o f acong en ita l trach eo -e so phagea l f is tu la hasb een repo rted , bu t has b een co nsid ereddoub tfu l by us .31 N o poss ib le o r d oub tfu lcase w as used in com piling the s ta tis tic s .

CA SE R EPO RT

F . P . A w hite m a le , w as born a t P ro v iden ceH osp ital on Ju ly 20 , 19 53 . H is b ir th w e igh tw as eig h t p ounds, 1 2 ounces . H e w as the firs tliv ing ch ild o f fou r p regn an cie s . N o th in g ab -norm a l w as no ted a t b ir th .

T he firs t feed in g w as a ttend ed by cough ingan d vom iting . T he n urse la te r reco rded ab-dom ina l d isten tion , dy spn ea w ith ra spy resp ira -tions. The re w as no cyano sis .

O n th e second d ay o f life a d iagno sis o fa te lecta sis o f the le f t low er lobe w as m ade . T heabdom en w as d istend ed , bu t such d isten tion re-

F rom the D ep artm en ts o f T ho rac ic S urg ery andPed ia tric s, P rov ince H osp ital, W ash ing ton , D .C .

spon ded to asp ira tion o f stom ach . P assag e ofL ev ine tub e w as co nside red to ru le ou t th ed iag nosis o f e soph ageal a tres ia w ith trach eo-e sop hagea l f is tu la . P a tien t passed a m econ iumstoo l. T he fo llow ing day , a chest film show edno pu lm on ary p ath o log y bu t tho rac ic h em ive r-teb rae w ere no ted . A n abdom ina l film sh ow edco nsid erab le gas in the stom ach . Th is sam eeven ing , fo llow ing feed ing , rhon ch i w ere n o tedin the chest w ith d im in ished brea th sounds ov erthe rig h t lu ng . Th ere w as d ysp nea and ab -dom ina l d isten tio n . P atien t w as p laced in o xy-g en . F or th e n ex t seve ra l days p atien ts con d i-tio n w as p oor. C ough an d vom iting usua l lyo ccu rred w ith feed ings .

R ad io log ic stud ies o f Ju ly 28 th rev ealedp neum onic conso lida tion occupy ing the rig h tu ppe r lob e w ith heavy congestive chan ges inth e rem a ind er o f the rig h t lung and le f t u pp erlo be . A barium en em a w as nega tive .

A lip iod o l s tudy ru led ou t ca rd iosp asm orh ia ta l he rn ia . A defin ite sea rch w as m ade of atracheo-esoph ag ea l fistu la . D uring exam ina tio nlip io do l w as seen in the trachea , bu t w asth oug h t to b e asp ira ted .#{1 76 }

The pa tien t w as p laced on an tib io tic s andp am en tera l f lu ids . A s h is co nd itio n im proved ,v a riou s fo rm ulae w ere tried w ith va ry ing sue -cess. T he pa tien t w ou ld have no d ifficu lty oned ay , on ly to v om it the m ajo rity o f feed in gs onthe nex t day .

D uring th e firs t w eek of A ugu st an o the rlip io do l sw a llow w as p erfo rm ed . A t th is ex am i-na tion , a qu estio nab le fis tu la w as seen b etw eenthe trachea and esoph agus. Th e pos sib ility o fa sp iratio n cou ld no t be ru led ou t.

F o r the nex t seve ral w eeks g as tric g av ag ew as perfo rm ed . T here w as som e w eigh t ga in .How ever , th e in fan t w as foun d cy ano tic andsevere ly dyspne ic in h is c rib on severa l occa-sions fo llow in g feed in g . F o llow ing one o f th eseep isod es pn eum on ia o f the righ t upp er lo bed ev e loped . T he in fan t w as critic ally ill du ringth is ep iso de .

#{176}R aym ond I. T erra franca , M .D . p erfo rm ed a llco n trast stud ies .

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TRACHEO- ESOPHAGEAL F I S TULA WI THOUT ATRES I A 25 5On September 4, a contrast study w ith

lipiodo l w as repeated and a de finite diagnosiso f t ra ch e o- e so pha g ea l f istula made.

Patient w ent to surgery on the fo llow ing day ,ag e six w eeks, w eight seven pounds and tw oounces. Dr. John Leahy gav e the anesthesia. Arig ht thoraco tomy w as performed under endo -tracheal anesthes ia. The pleural cav ity w asentered through the fo urth intercostal space .

The esophagus w as identif ied and dissec tedcephalad from the lev e l o f the carina. Af is tula w as fo und be low the subc lav ian v e inat the leve l o f the third rib po steriorly . Theesophageal portion w as s lightly low er thanhetracheal opening . The tracheal opening wasc lo sed w ith five interrupted s ilk sutures (5 -0 ).The esophagus w as c losed in tw o lay ers. Themucosa w as c lo sed w ith tw o sutures and thenfour sutures in the musculature. A ro utine chestclo sure was performed.

A t the end of the procedure cerv ical em -physema w as no ted. Ten cc . o f air w as as-pirated w ith nearly complete disappearance o fthe emphy sema. This occurrence w as attributedto the high lev e l o f dissec tion.

On the first post-operative day , there wasate lec tasis o f the right upper and m iddle lobesw hich cleared by coughing and tracheal aspira-t iOIl. There w ere audible rhonchi over bo thlung fie lds w ith normal breath sounds and clearx -ray fo r several day s.

W ater by mouth w as started on the thirdpos t-operative day and formula on the fourth.Improvement w as progressive . Patient w as dis -charged at 10 w eeks of ag e, w e ighing e ightpounds and tw o ounces.

Etio lo gy o f Tra cheo-Esop ha gea l Fis-tula .6 , 15 , 16 , 3 0 There are three theoriespropounded to explain the occurrence oftracheo -esophageal fistula. Ke ith and Spicermaintain that there is an incomplete unionof the lateral tracheo-esophag eal groov es.Such g rooves in the embryo normally forma septum w hich separates the trachea andesophagus.

Pressure ex erted by the anlage of theheart o r by an abnormal vascular system hasalso been propo sed as a cause. A n abnormalblo od vesse l has been recorded in threecases o f proved esophageal f istula6 andin one possible case of congenital f istula.2

The final theory states cellular pro lifera-tion from the embryonic esophagus is de-fic ient; hence , a comple te separation o ftrachea and esophagus do es no t o ccur.

Signs a nd Sympto ms of Tracheo-Esopha-gea l Fistula .6 1O I i , 1 2 These figures arecompiled from 2 7 patients, 11 o f w hom w ereno ted to be males, and nine stated to befemales. (Sex not stated for 7 .) The lesionis more diff icult to diagnose than is atres iaof the esophagus, since symptom s may beunre lated to sw allow ing and there may beasymptomatic periods.

He lmsw orth has po inted out an exce llentdiagno stic triad.1. P aroxysms o f C o ughing w ith Feed ings.Liquids cause the most diff iculty . Suchcoughing is o ften attended by cyanosis . Thedirec tion of the fistula w ill play a part inthis particular instance. If the low er open-ing of the fistula is esophageal, sw allow ingmay be unattended by difficulty . Regurg i-tation of food, how ever, precipitates thetrouble. Hence , coughing and cyano sis mayoccur only after feeding .

Characteristically , symptom s disappearw hen the infant is fed by gavag e. In re -po rted cases the long periods o f g avag e havebeen interes ting . Some infants have beenfed as long as a year by this method.2. Abno rma l D rstentio n of the Abd omen.This is due to div ersion of tracheal air intothe esophagus, which is then sw allow ed intothe s tomach. Rapid direc t inflation of thestomach during coughing is pathognomico f a fistula betw een the tracheo-bronchialtree and upper alim entary t ra c t .h l

In s ix out of nineteen cases w here a com-ple te pro toco l is available , abdominal dis -tention w as a prom inent feature . In our casedistention w as such that intestinal obs truc-lion due to a congenital abnormality w asconsidered. In this patient it w as no ted thata catheter placed into the stomach re lievedthe distention, w hile a rec tal tube did no t.3. P ersistent P neumo nitL Pulmonary pa-tho logy is alw ays present regardle s s o fh eseverity o f g astro-intestinal symptom s. Thepulmonary problem may be the main rea-

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S

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Ferguson

FlussHa i g h tHelmsworthHo l i n g e rI mp e r a t o r iKnoxLeigh

R o b bAu t h o r sAv e r a g e

256 GEORGE WILLIAM WARE AND LOUIS L. CROSSson for seeking medical adv ice . The rightupper lobe is most frequently invo lved.Coarse rhonchi, w hich may be bilateral, areusually present. Chronic cough comple testhe respiratory picture .

General symptom s inc lude poor w eightgain, dehydration, and apathy .

R adiologic A spects.10 21 Pneumonitis w illbe v isible on routine chest f ilm and, as men-tioned, usually invo lv es the right upperlobe.

Lipiodo l is the usual medium for con-trast studies o f the esophagus . The examina-tion should be done in the prone or anterioroblique position so that the o il w ill f lowanteriorly to the trachea. Careful observa-tion is necessary during fluoroscopy .

If the s tudies w ith iodized o il are no tsucces sful, Le igh and his assoc iates recom-mend a tracheogram . The lipiodo l is in-jec ted by laryngo scopic guidance . Films areimmediately taken in the anterior-posterior,posterio -anterior, and bo th lateral pro jec -lions. Such studies may succeed since thetracheal opening is le ss liable to co llapseand clo se the fistula.

I n t h is ser ie s, con t r a s t s t u d ie s h a ve b eendone on 18 patients . In 11 the fistula w asseen on at least one exam ination.

It should be emphasized that one ex -am ination w ith contras t medium is no t

CHART IASSOCIATED ANOMALIES IN CONGEN ITA L TRACHEO-

ESOPHAGEA L FIsTu

Aberrant A rteryP h a r y n gea l D ive r t ic u lu mP a t en t D u ct u s A r te r io su sAnomalous Ribs and/or V ertebraeInterventricular Septal D efectJ e j u n a l D ive r t ic u lu mOrigin of RUL from TracheaM eck el s D iver t icu lu mE sop h a gea l S te n os isMongo l i smCongenital H eart D iseaseLeft-S ided Fused Kidney sMairotation of CecumGastric A berrant PancreasD ouble Tracheo -Esophageal Fis tula

enough. Failure to demonstrate the tract oneven repeated exam inations does not e lim i-nate the diagnos is .

Endoscopy. Such a surg ical maneuvermay prove a valuable aid. Esophag oscopyis usually performed first. If the fistula isseen , bronchosco py w ith the poss ibility o flaryngeal edema w ill no t be necessary .

Leigh and assoc iates hav e recommendedinjec ting methylene blue into the esophagusand then searching for the characteris ticstain in the trachea.

Eight patients have receiv ed endoscopy;the fistulous opening has been identif ied bybroncho scopy on three o ccasions andbroncho scopy plus esophagoscopy on tw ooccasions. I t is ev ident that lack o f v isuali-zatio n w ith endoscopy does no t rule out thediagnosis.

A ssociated A nom alies. These have beenlisted in Chart I. This group has beengathered from the proto co ls o f 1 9 patients.

Treatm ent. Surgery is the o nly cure forthe condition. The one case repo rted as aspontaneous cure is considered a doubtfulcase . Holinger used sodium hydroxide tocauterize the opening of the tract. The fis-tula recurred and surgery w as required forpermanent cure .

The first case treated surg ically w as re -ported by Imperatori in 1939 . The patientw as the son of a doctor and was operated at

CHART IIAG E AT TIME OF SURGERY IN CONGEN ITA L TRAdHEO-

ESOPhAGEAL FISTULA

a. 4 daysb. 5 w eeksc. 5 w eeks

8 months4 ye a r s6 months6 years7 years3 months

a. 7 w eeksb. 1 w eek

11 years6 weeks

3 months

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TRACHEO- ES OP HAGEAL F I S TULA WI THOUT ATRES I A 2 5 7the ag e of seven y ears. H is prev ious historyand tribulations form fasc inating reading .

Thirteen patients have now been oper-ated.6 10, 1 1, 12, 14 , 17, 21, 29Three patientsdid no t surv iv e surg ery .6 21 The age of thepatients at time of surgery is shown in ChartI I .

Post-operative complications inc luded re-currence or pers istence of the fistula/4 17 in-fec tion o f thigh and back,2 and tensionpneumothorax .2 One of the persistent o rrecurrent fistulae c losed spontaneously , butthe second required another operation.14

SUMMARYCongenital tracheo -esophageal f istula

w ithout atres ia o f the esophagus is a rareanomaly . Tw enty-six patients are repo rtedin the literature . The autho rs report an ad-ditional case . The etio logy is still o bscure ,but may be due to the dev elopment of aber-rant blood vesse ls .

Patients present a triad of signs andsymptom s, coughing and cyanosis w ith feed-ing s, abnormal dis tention o f gastro -intesti-nal trac t and persistent pneumonitis .

Radiographic investigation is mo st im-po rtant. Lipiodo l study o f the esophagus inthe oblique or prone position is the mostrew arding diagnostic test.

Surg ery is the only method o f cure .B I B L I O G RAPHY

1. Barkely , H . T., Quoted by Haight: J. Tho-racic Surg ., 17 :600-612, Oct. 1948 .

2. Berman, J. K ., Test, P. S ., M cA rt, B . A .:Congenital esophagobronchial f istula inan adult. J. Thoracic Surg ., 24:4 93 -500 ,N ov . 1952.

3 . B row n, R. K ., B row n, E. C. : Cong eni-tal e sophageal anomalies . Surg . Gynec.Obst., 91 :5 45-550 , N ov . 1951 .

4. Cautley , E. : M alformation o f e so phagus .Brit. J. Child. D is., 1 4:1-10 , Jan. 1917 .

5 . Cardullo , H . M ., B erens, D . L. : Tracheo -e sophageal fistula unassoc iated w ithatresia or s teno sis , N ew Eng land J.M ed., 243:853-856 , N ov . 1950 .

6. Ferguson, C. F. : Congenital tracheo -e so -phageal fis tula no t assoc iated w ith eso -phageal atre sia. Laryng oscope , 61 :7 18 -766 , A ug . 195 1 .

7. Fluss, Z., Poppen, K .J. : Embryogenesiso f tracheo -e sophageal fistula and eso -phageal atre sia. A rch. Path., 5 2 :168 -181 ,Aug . 1951 .

8 . Gengenbach, F.P. , D obos, E. I. Congeni-tal tracheo-esophag eal fis tula. J. Pediat.,.19 :6 44-655 , N ov . 1 941 .

9 . Gross , R. E. : A tresia of the Esophagus, inThe Surgery of Infancy and Child-hood. Philade lphia, W . B . Saunders,1 953 , p. 7 5 .

10 . Haight, C . : Congenital tracheo-esophag ealfistula w ithout esophag eal atresia. J.Thorac ic Surg ., 1 7:600-612 , Oct. 1948 .

11 . Helm sw orth, J. A ., Pry les, C. V .Congeni-tal tracheo -e sophageal fis tula w itho ute sophageal atre sia. J. Pediat., 38 :610 -6 17 , May , 1 951 .

12 . Holinger, P.H ., Johnston, K. C ., Po tts,w . J. : Congenital anomalies of theesophagus. A nn. O to l. Rhin. & Laryng .,60 :707 -731 , S ept. 1951 .

13 . Holt, J. F., Haight, C., Hodges, F. J.:Congenital atresia of esophagus andtracheo-esophag eal f istula. Radio lo gy ,47 :457 -470 , N ov . 1946 .

14 . Imperatori, C. J. Congenital Tracheo-eso-phageal fistula w ithout atre s ia o f e so pha-gus. A rch. Oto laryng ., 30 :352 -360 , S ept.1 939 .

15 . Ingalls , T. H., Prindle , R. A . : Esophagealatres ia w ith tracheo -e so phageal fistula.N ew Eng land J. Med., 2 40:9 87-995 ,June 1949 .

16 . Keith, A ., Spicer, J. E.Three cases ofmalfo rmation o f tracheo-esophag eal sep-tum . J. A nat. & Phys io l., 41 :5 2 -55 , Oct.1906 .

17 . Knox , G. : Congenital tracheo-esophag ealfis tula w itho ut e sophageal atre sia. Sur-gery , 30 :101 6 -102 0 , D ec . 19 51 .

18 . Lamb, D . S . : A fatal case o f co ngenitaltracheo -e sophageal fis tula. Philade lphiaMed. Times, 3 :705 -707 , A ug . 1873 .

19 . Lev en, N . L., Lannin, B . G . : Congeni-tal atre s ia and congenital tracheo -eso -phageal fis tula. Journal-Lance t, 65 : 179 -1 81 , M ay 1945 .

20 . Leven, N . L., V arco , R. L., Lannin, B . C .,Torgen, L. A . : Surg ical management o fcongenital atresia o f esophagus andtracheo -e sophageal f is tula. A nn. S urg .,1 3 6 : 7 0 1 - 7 1 9 , Oc t . 1 9 5 2 .

2 1 . Le igh, T. F., A bbott, 0 . A ., Hopkins , W .A . : Roentg enolog ic considerations intracheo-esophageal f istula w ithout eso-phageal atresia. Radio logy , 5 7:871-877 ,D ec. 1951 .

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25 8 GEORGE WI LLI AM WARE AND LOUI S L. CROS S

90 0 17 th Stree t N.W .

22 . L ohnes , H . R . : R are fo rm of con gen ita ltracheo-esoph ag ea l fis tu la . B u ll. B uffa loG en . H osp ., 1 :109 -1 12 , O c t. 1 92 3 .

23 . M acK enz ie , M . : M alfo rm ations of th eesoph ag us. A rch . L aryng ., 1 :3 01-3 15 ,D ec. 18 80 .

24 . M acK enz ie , M . A M anua l o f D iseases o fth e T hroa t an d N ose. N ew Y ork , W m .W ood and C o ., 188 4 , p . 1 49 .

25 . M cK inn ey , R . : T rach eo -esop hagea l fis tu la ,p ro bab ly congen ita l. A nn . O to l. R h in . &Laryn g ., 4 2 :1234 -1236 , D ec . 1933 .

26 . M orton , D . R ., O sbourne , J . F ., K la ssen ,K . P . : An appa ren tly con gen ita l b ro ncho-e so phagea l f is tu la pe rsis ten t to adu ltlife. J. Thorac ic Surg ., 1 9 :81 1-816 , M ay1950 .

27 . N egu s, V . E . : E sop hag us from a midd le -aged m an sh ow ing co ngen ita l open in gin to trachea. J. Lary n g . & O to l., 4 4 :18 4 ,M ay 1929 .

28 . Po tts , W . J. : A tres ia o f esoph ag us. P os t-g rad ua te M ed ., 10 :304 -3 10 , O c t. 1 951 .

29 . R obb , D . : C ongen ita l tracheo-e sop hagealfistula w itho u t atre sia. A ustralian andN ew Zealand J. Surg . , 22 : 120-12 2 , N ov .1952 .

30 . R osen th a l, H . H . : C ong en ital atre sia o fe sop hagus w ith tracheo-eso ph ag ea l fis-tu la . A rch . P a th ., 12 :756 -7 72 , M arch1 931 .

3 1 . R uck s, W . L . : T racheo-e soph ageal fis tu la .M em phis M ed . J. , 7 :2 09-21 1 , N ov .1 9 3 0 .

32 . S haw , R . : Q uo ted by H aigh t. J . T ho rac icSurg . , 17 :6 00-61 2 , O c t. 1 948 .

33 . S toeck le in , C .J. , N ettrour, W . S . : A cu te

tracheo-eso phagea l fistu la . P en nsy lv an iaM J ., 4 6 :89 8-9 01 , S ep t. 19 43 .

SPAN ISH AB STR AC TFIstu la T r a q u eo E so f g ica C on g#{233}n it a

s in A t r e s ia d e E s#{243 }f agoL a fIs tu la con g#{233 }n ita traqueo-e so f#{2 25}g ica s in

atresia de es# {243 }fago es una anom alIa rara. L arev is io n de la lite ra tu ra al re spec to reve l#{243 } lafecha so lo 26 caso s, a lo s que lo s au to re s ag re -gan uno m #{225} s,u e se tra t#{243}u ir#{241}rg icam en te contodo #{233}x ito .

L a e tio lo g Ia de Ia fIstu la no se ha ex p licadoann sa tisfacto riam ente ; pu ede deberse aa pre-sion que erjecen vasos sanguIn eo s aberran tesduran te e l d esa rro llo em brio l#{2 43}g ico .

D esde e l pu n to de v ista c lm nico H elm sw o rthha sef la la do una tria da d iagn #{243} stic a: pa rox ism osde tos a l alim en ta rse a l n ifio ; d is ten sion an orm alde l tu bo gas tro -in testina l deb id o a l p aso de la ire traq uea l hac ia e l es#{2 43}fag o y neum onitispers isten te q ue por lo com #{241}n se loc aliza en e ll#{24 3}bu lo up er io r d erecho . L os es tud ios rad io l#{243}g i-co s p ara ac la ra r Ia anom alla deben inc lu irsiempre Ia ing estion de lip io do l, co locando a lnino en p osic i#{2 43}n decib ita dorsa l u ob licua ; enes ta fo rm a e l d iagn# {243 }s tico se ob tiene en m #{225}sel50% de los caso s. L a eso fago scop la y /# {2 43}a bro n-cosco pla ayu dan frecu en tem ente a rev elar IafIstula.

E l in ico tratam ien to es el qu iru rg ico ; searea lizado con #{233 }x iton 10 de lo s 1 3 casos tra -tados .

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1954;14;254-258PediatricsGEORGE WILLIAM WARE and LOUIS L. CROSS

THE ESOPHAGUSCONGENITAL TRACHEO-ESOPHAGEAL FISTULA WITHOUT ATRESIA OF

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