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B e cau se o f p re m a tu re lab o r a nd a b ree ch pre se n ta tio n , a 1 ,20 09 w h ite b o y , a firs t-b o rn

S to m a c h R u p tu reA s s o c ia te d w ith E s o p h a g e a lA t r e s i a T r a c h e o e s o p h a g e a lF is tu la a nd V en tila to ryA s s i s t a n c e

T h o m a s B . Jo n e s 1S a n d ra G . K irch n e r1

F re d A . L e e 2R ic h ard M . H e lle r1

W he n in fan ts w ith trac h eo e s op h a g ea l f is tu la req u ire v en tila to ry as s is ta n ce g a str icd is te n tio n m a y u ltim a te ly res u lt in p e rfo ra tio n a n d p n e u m o p e rlto n e u m . T h is repo r td e scr ib e s fo u r n e o n ate s w ith es o p h a g e a l a tre s la a n d tra ch e o e so p h a g e a l fis tu la w h oex per ienc ed s uc h a c om p lic at io n

P e rfo ra tio n o f th e g a s tro in te s tin a l tra c t a n d pn e u m o p e rito n e u m c a n re su lt fro mve n tila to ry ass is ta nce in a n ew b orn w ith an a n a to m ic a lly n o rm a l ga s tro in tes tina ltra c t [1 , 2 ]. T he re fo re , it is n o t su rp ris in g tha t ve n tila to ry a ss is tan ce in a ne w b o rnw ith e sop h ag ea l a tres ia an d d is ta l tra ch eo es op ha g ea l fis tu la m ig h t le a d tom a rke d g a s tric d is te n tio n , e sp e c ia lly in the p re se n ce o f d u o d e n a l a tre s ia [3 ].G a str ic ru p tu re in a n e w b o rn w ith a tra c h e o es o p h a g e a l fis tu la h a s a ls o b e enre p o rte d [4 , 5 ]. W e re p o rt fo u r a d d itio n a l ca se s th a t e m p h a s iz e th e n e ce ss ity o fco n s id e ring tra c h eo e s o p h a g e a l fis tu la w h e n a n e w b o rn re ce iv in g v e n tila to rya s s is ta n ce d e ve lo ps m a rk e d g as tric d is te n tio n o r a p n e u m o p erito n e u m .

C a s e Repor tsC a se

R e ce ive d Ju ly 17 , 1 9 7 9 ; a cc ep ted a fte r re vi-s ion D e c e m b e r 1 2, 19 79 .

D e pa rtm en t o f R ad io log y an d R ad io lo g ic a l S c i-enc es , v an de rb il t U n ive rs ity M ed ica l C e n te r,N as hv il le TN 3 72 32 . A dd re ss r ep ri nt r eq ue st s t oT . B . Jo nes .

2 Dep ar tm en t o f R ad io lo gy , C hild re ns H osp ita lo f Lo s A ng e les , U n iv e rs ity o f S o u the rn C a lifo rn iaS ch oo l o f M ed ic ine , Lo s A nge le s , C A 90 0 33 . JR 1 34 :67 5 -6 77 , A pril 19 8003 61 -8o3x/8o/1 344-0675 00.00 A m e ric an R oe n tg en R ay S o c ie ty

A 1 .60 0 9 w h ite b oy w as d e live re d a t 35 w e e ks g e s ta tio n d ue to p rem atu re la b o r.U ltraso u nd exa m ina tio n 1 m o n th b e fo re de live ry reve a le d h yd ram nios . A fte r d e liv e ry , A p g arsco re s w e re 2 a t 1 m m a nd 1 a t 5 m m , ne ce ss ita tin g in tub a tion in th e de live ry ro o m . A nin it ia l rad io g rap h sh ow e d hya line m em bran e d ise as e a nd a n o ro ga s tric tu be e n d ing in th eup p er eso p ha gu s . T he s tom ac h a nd p ro x im a l d uo d en um w ere d is te nd ed w ith a ir (d o ub leb u b b le ) w ith o u t a n y d is ta l s m all b o w e l g a s (fig . 1 A). A b d o m in a l d is te n tion in cre a se d a n d asu b se q u e n t ra d io g ra p h re ve a le d a p n e u m op erito n e u m (fig . I B ). T he d ia g n o s is o f e so p h a -g e a l a tre s ia , tra ch e o e s o ph a g e a l fis tu la , d u o d en a l a tre s ia , a n d g a s tric ru p tu re w a s m ad e .A t la pa ro to m y a la rg e p e rfo ra tio n a lo n g th e le sse r c u rv a ture w as rep a ire d a n d a g a stro s to m ytu b e in s erte d . T h e in fa n ts co n d itio n p re c lu d e d m ore e x te n s ive s u rg e ry . P o s to p e ra tive lyth e p a tien t d e ve lop e d m as s ive in tra pe rito n ea l b le ed in g a n d e xp ire d .

C a s e 2

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Fi g. 1.-Case 1. A , A n te ro po ste rio r ra d io g ra ph s o f ch es t a nd ab do m e n . A , O ro ga stric tub e w ith tipin u ppe r es oph ag us . E n do trac he a l tub e a nd um b ilica l ven ou s ca th e te r a lso se en . P u lm o na ry pa ren chy -m a l pa tte rn co ns is te n t w ith hy a line m em bra ne d ise as e ; abd om in a l bo w e l g as pa tte rn sug ge sts du od en a lo bs tru c tio n . B , 8 h r la te r . La rge p neu m ope ritone um no w s ee n .

F ig . 2 .-C ase 2 . A n te ro p os te rio r ra d io g ra p h o fch es t a n d ab do m en . G as d is ten ds s to m ach an d fil lsla rg e an d s ma ll bo we l. L arge p neu mo pe rito neu m;d iffu se pu lm on ary in fil tra tes . E nd o trac hea l tub e a ndu mb il ica l a rte ria l ca th ete r.

67 6 J O N E S E T A L . A J R : 1 34 A p r il 1 9 8 0

tw in , w a s d e liv e re d by ces a re an se ct ion a t 33 w e eks g es ta t ion . H eh ad A p gar s co res o f 4 a t 1 m m a nd 5 a t 5 m m , ne ces s ita tin gin tu ba t ion in th e de live ry room . A fte r s ev e ra l h ou rs he de ve lop edpro g ress ive a b do m ina l d is te n tion an d ra d iog ra ph s re ve a led agrea tly d is ten de d s to m a ch w ith s om e sm a ll b o w e l g as . S u b seq u en tra d io g ra ph s rev ea le d a pn eu m o p erito n eu m (fig . 2 ). A n o rog a s trictu be w o u ld n o t p as s in to the s to m a ch . T h e d ia g no s is o f es op ha g ea la tre s ia , trac he oe so ph a ge a l fis tu la , a nd ga s tric ru p tu re w as m a de .A t su rge ry , a g as tr ic ru p tu re a lo ng the le sse r cu rv a tu re w a s rep a ire da nd a g a s tros to m y tu be p la ced . T h e in fan t d eve lo p ed res p ira to ryd is tre ss s ynd ro m e , a p a te n t d uc tu s a rte rios us , an d e xp ire d a fte r am a ss ive in tra ven tricu la r h em orrha ge .

C ase 3A 1 .2 50 9 w h ite m a le b o y , a sec on d -bo rn tw in (b ro the r o f ca se

2 ), w a s a lso d e liv e re d by c es a re an se c tio n . H is A p ga r sco re s w e re1 a t 1 m m a n d 7 a t 5 m m , ne ce ss ita tin g in tub a tion in th e de live ryro om . H e ha d inc rea s in g ab do m ina l d is te n tio n an d a n oro ga strictu b e c ou ld n o t b e pa sse d . R a d iog ra ph s sh ow e d th e o ro g as tric tub e ,e nd in g in th e p ro x im a l e so ph ag u s , a n d a p n eu m o pe rito ne u m . A tsu rg e ry , a ga s tric pe rfo ra tio n a lo ng th e les se r c u rv a tu re w a s re -pa ired a nd a ga s tro s tom y tu be inse rted . P o s to pe ra tive ly th e in fan tde ve lo pe d in traa bd o m in a l b lee d in g , sh o ck , an d res p ira to ry fa ilu re ,a nd h e sub se qu e n tly ex p ired .

Case

A 1 ,2 199 w h ite g irl w a s d e live re d a t 31 w ee ks g es ta tio n b eca useo f p rem atu re la bo r. S h e de ve lop ed res p ira to ry d is tres s s ynd ro m e

an d w a s p la ced o n v en tila to ry a ss is ta nc e . H er a bdo m en bec am em arked ly d is te nd ed an d an o rog as tric tu be c ou ld no t be pa ss ed .R ad iog rap h s reve a le d a p n eu m o pe rito n eu m . A la rg e g as tr ic p e rfo -ra t ion a lo ng the le ss e r c u rv a tu re w a s rep a ire d a t su rg e ry an d aga s tro s tom y tub e inse rte d . T h e tra ch eo e sop h ag ea l fis tu la w a s Ii-g a te d . Th e in fa n t ha d a d iff icu lt co u rs e w ith resp ira to ry d is tres ssy nd ro m e , sep s is , an d se izu re s . A t ag e 3 m on ths , sh e req u ire d atra ch eo s tom y d u e to la ryng o m a la c ia , sub g lo ttic an d s up ra g lo ttics te no s is , a nd le ft vo ca l co rd p a ra lys is , b u t s he h a s su rv ive d .

D i s c us s i onO f th e five typ es o f es op ha g ea l a tre s ia a nd trach e oe so ph -

a g ea l fis tu la , e so p ha ge a l a tres ia w ith a fis tu la be tw ee n th etra ch ea an d th e d is ta l e sop h ag ea l p o uch is the m os t co rn -rn on , co n s titu ting ov e r 8 0 o f a ll cas es a nd inc lu d ing a llfou r o f ou r ca se s . W he n the se in fa n ts re qu ire in tub a tion a ndvent i iatory a ss is tan ce , th e inc re a se d p re ssu re in th e a irw ayha s a d ire c t ro u te to the ga s tro in te s tin a l tra c t v ia th e tra ch e -oe so ph a ge a l fis tu la . W h ile th e de ve lo pm en t o f a p n eu m o -p e riton eu m m a y re su lt from d is sec tion o f a p n eu m o m e d ias -tin um [6 ], p e rfo ra tion o f th e ga s tro in tes tina l tra c t w ith itsa sso c ia ted h ig h m o rta lity m u s t b e ex c lu d ed [7 , 8 ].

T he w ork o f H e lle r e t a l. [9 ] su gg es ts tha t a c om b in a tio no f in c re ase d p ress u re a n d im m a tu re m us cu la r d eve lop m e n tp red is po se s to g as tr ic rup tu re . A ll fou r o f o u r pa tie n ts w e rep re m a tu re a nd h ad e le va ted g as tric p ress u re s sec on da ry toa rtif ic ia l v en tila tio n a nd trac he o eso p ha ge a l fis tu las . T he a s-so c ia te d d uo de n a l a tre s ia in c as e 1 ve ry like ly re s u lte d in

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A JR :13 4 , A p ril 1 98 0 S T O M A C H R U P T U R E W IT H A S S IS T E D V E N T ILA T IO N 677

e ve n g re a te r g as tric in tra lu m in a l p re ssu re . In a ll fo u r ca se s ,th e g as tric ru p tu re w a s a lo ng the less e r cu rva tu re , a s iso fte n th e cas e w ith ru p tu re se co nd ary to o ve rd is ten tio n .S p on tan eo u s rup tu re o f the s tom ach can a lso o ccu r, u su a llyw ith in a fe w d ay s a fte r b irth . It h a s b e e n re p o rte d th a t su chp er fo ra tio ns oc cu r a lon g th e g re a te r cu rva tu re [10 ]. W h e na n in fan t rec e iv in g ve n tila to ry ass is ta nce de ve lo ps se ve reg as tric d is te n tion o r p ne um op e rito ne um , tra ch eo e so ph a-g ea l fis tu la w ith o r w ith ou t es op ha g ea l a tre s ia m us t beconsidered.

ACKNOWLEDGMENTSW e th an k B e tty B u rn s id e fo r h e lp in m a nu sc rip t p re pa ra tion a n d

Y uk io H ira ta fo r he lp in tra n s la tio n .

R E F E R E N C E S1 . Le on id a s J , B e rdo n W E , B ak er D H , A m o ury R . P e rfo ra tio ns o f

th e g as tro in te s tin a l tra c t a nd p ne um op e rito ne um in n ew b orn stre a te d w ith co n tinuo us lung d is ten d in g p re ssu res . Ped ia t rR a d io 19 74 ;2 :2 41 -2 4 6

2 . K irkp a tr ick B V , F e lm an A H , E itzm an D V . C om p lica tio ns o fv en tila to r the ra py in res p ira to ry d is tress s ynd ro m e . A m J D isCh i ld I 9 74 ; 1 2 8 : 49 6- 50 2

3 . M c C o ok T A , F e lm an A H . E s op ha gea l a tres ia , du ode na l a tres ia ,a nd g as tric d is ten tio n : R e po rt o f tw o c a s e s . A JR 1 97 8; 1 3 1 :167-1 68

4 . M ika m i K , T e raza k T , K im ura T . C as e o f c on ge n ita l trac he o-eso p ha ge a l fis tu la co m p lica te d by ga s tric p e rfo ra tion . lryo1 96 7; 21 : 13 27 -1 33 0

5 . O th e rson H B , G reg or ie H B J r. P neu m atic rup tu re o f th e s to m -ach in a ne w b orn in fa n t w ith e sop ha ge a l a tres ia an d trac he o-eso pha ge al fis tu la . Sur ge r y 1 9 63 ;5 3 : 3 62 -36 7

6 . C a m pbe ll R E , B og gs R R , K irkp a trick JA . E arly n eo na ta l p ne u -m o pe rito ne u m fro m p ro g ress ive m as s ive te ns io n p n eu m o m e -d ias t inum. Rad io logy 1 9 7 5 ; 114 :121 -126

7 . P a rke r JJ , M ik ity V G , J aco bs on G . T ra um atic pn eu m o pe rito -ne um in th e ne w b orn . JR 1965 ;95 :203 -207

8 . K au fm a n R A , K uh ns U R , P oz na ns k i A K , H o lt JF . G a s tro in te s -t ina l p e rfo ra tion s w itho u t in tra pe r ito nea l a ir- flu id lev e l in n eo -n a ta l p n eu m op e ri to n eu m . A JR 1 9 7 6 ; 1 2 7 :9 1 5 -9 2 1

9 . H e lle r R M , W h ite JJ , S ha ke r IJ , Ja m e s A E . S to m a ch ru p tu re inth e n ew bo rn . A p p R a d io 1 9 7 5 ;4 :43 -44

10 . P ertsem lid is D . N e on a ta l g as tric pe rfo ra t ion . J M ou n t S ina iH o s p 19 64 ;31 :9 7 -1 23