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=Abstract = Esophageal Atresia and Tracheoesophageal Fistula in Korea - A National Sur very of Its Members by the Korean Association of Pediatric Surgeons -
WH Park, SI Kwon, SC Kim, SK Kim, WK Kim, IK Kim, JE Kim, HH Kim, KW Park, YS Park, YT Song, JW Yang, SM Oh, SY Yoo, DS Lee, MD Lee, SC Lee, SK Lee, TS Lee, SI Chang, SY Chun, ES Chung, SY Chung, SE Chung. PM Chung, MH Cho, JS Joo, SO Choi, SH Choi, YS Huh, and CHong
The Korean Association of Pediatric Surgeons
The first national survey on esophageal atresia and tracheoesophageal fistula was conducted to access the current status of its incidence. clinical manifestation, preop erative diagnosis and management, type of its anomaly, associated anomalies, and surgical results and course. The 43 members of the Korea Association of Pediatric Surgeons received questionnaires and registration forms to be filled out on each pa tient who were born during the three years from January I, 1992 through December 31, 1994. Questionnaires composed of six broad areas which include 1) preoperative diagnosis and management, 2) surgical technic, 3) long gap, 4) postoperative manage ment, and 5) complications and courses. A total of 148 cases was returned by 28 members working at 23 institutions and 27 members returped questionnaires. We ob tained the following results by analysis of the 148 cases of tracheoesophageal anoma lies. The incidence of tracheoesophageal anomaly was about 1/10,000- 11,000 in 1994, which is one third of that of anorectal malformations in Korea and the distribution of the patients was almost proportionate to the size of each province. Both sexes are about equally affected. Majority of the members make diagnosis of tracheoesopha geal anomaly by taking a simple infantogram with a radiopaque tube in upper pouch and a little under half(46%) prefers to perform echocardiography as a part of preop erative management to identify congenital heart disease and lateralize the aorta.
- 149 - - :t.0 l-.21}!j- : ;IJ]l i'! ;IJ] 2 ~ 1995\1 -
Esophageal atresia with distal TEF(87.50/0) was by far the most common and threre were pure esopahgeal atresia(5.6%), H-type TEF(2.l%), and so on. About half(49%) of the patients had one or more associated anomalies in addition to tracheoesopha geal anomalies. Congenital heart disease was associated in 46 cases(31 %), anorectral malfomations in 19 cases(13%), musculoskeletal anomalies in 15 cases(10%), gen itourinary anomalies in 10 cases(7%) and gastrointestinal anomalies in 7 cases(5%). Postoperatively, parenteral nutrition and assisted ventilation were given in 66% and 52% of patients respectively. Ninety three(74%) of 126 cases who underwent sug:ical procedure, experienced one or more complications such as respiratory complication (65%), leak(22%), stricture(21%) and so on. The survival rates related to the Waterston risk categories were 90.2% in grpup A, 71.4-75% in BI, B2, and C groups, and 28% in group C2 and the overall survival rate was 71.4%. Thirty six(28.6%) of 126 cases died of pneumonia/ sepsis(12 cases), respiratory failure(l2 cases), and con genital heart disease(4 cases). With short term follow-up, 69% of patients have been excellent whereas remainders of the cases have suffered from some sort of morbidi ty related to gastroesophageal reflux, recurrent respiratory infection, and esophageal stricure.
Index Words: Esophageal atresia, Tracheoesophageal fistula, National survey, Korea
(Registration forms) ~ .!i!.t.l1~c}- . ~o} 'g~ rJl->-J ,~ 1992\1 1-%1. 191oj\Al 1994\1 12~ 3191 71\7.\ ~ 3\'1{} 7]-t!-~x7]-&2.Q.£ ~,\~~ ~o}£ -6}';'ctJl 1994\'15:. rJl~5:o}9.j~~§j -&-oa~~§joj] A-l. ~ T~~Jl §j~~ ~0}5:. Y"y 15mc}-. ~-lf-7.]-c Z{ c:jy-2} 7]-t!-~5:.7]-&2oj] -t!-"0 ~~~, ~1±, ~~~-l §j~o]
- 150- 43"3 ~~ % 23t!J~$.J 30"3°] A}R ~ ..li.41 T Table 3. History of Maternal Polyhydramnios jr:J-. 7]:>&~.£7]~~0l- ;~ A}.li-C 28"30) ..li.4J Polyhydramnios No. of patient Percent( 0/0) r-932.-rlj % 148"3 01 ;:~S:19ir:t. ;:~Al~ ..li.41~ Yes 46 31 ~~ol 'V,': AI~E1 ~~T~ ..li.tB, .+~- 10, i'-~ No 13 9 ), q(-('- 3, /iftJ z, 7J~, 7J.*, -"Lhi-°] 44
Tflble 5. Diagnostic Procedures (N =:= 148) - 143~ojH A(j~ *~ol 7}~"6}S1,~o:j 'ito} 69 Methods No. 0/0 Infantogram with a radioopa· 128 86 ~, cxjo} 73~.2..s. 'it1.-1$.J l:l17} 1: l.l01 9it+. Al~ que NG lube ~ -l't!f~ .!t.1i'1 '?l~ 7cl71 39~ (26.4%), AH: 37 Infantogram with constrast 40 27 ~ (25%), ?g'if 23~ 05.6%), I11~ ?g.Ji1- 19 ¥-{} media in upper pouch (12.8%)tt'- o]9i~vj, ~.::il~ 1993\'15:. ~J.H}-8 l~chocardiography 51 34 ",~ot$.J ~*%4 71=t!-615:.71t§$.J 7,1~~ 1l.J1*%~ Itenal ultrasonogram 51 34 l:ll~~ o,VJ% .!t.S1,t+OE 1). ~.s::.~ 71=t!615:.71t§ I~sophangogram 7 13 9.J ~"'~-{t3l.,': If 29} iJq. Others 5 3
A Table 2, Annual Incidence by Year S:)9i~vj. 9'F J.g. T7J~l:l1% J1}t+, ~*~'Cl. ;<,3 ~~, y*~'Cl~ 9.) tt'-°)':(lqUI 3, 4). ~td.2..£ Year Birth No. Patients Incidence 1992 723,927 39 1/18,562 13JA}-{:l l:l11f-4"'(j 1l'tl- .1- tdtt'-~od~ ~4-7l- 128~1 1993 702,546 43 1/16,338 (86%),6Js:.~03~ol 40~](27%), {l.&g31j-{lA} 1994 (702,546) 66 1/10,645 51~1(34%);:ol ol%soJ':(lr:}(lf 5). 148 1/14,385 * Hirschaprung's disease in 1992: about 190 cases reported in Korea * Imperforate anus in 1993 " 143 caSes reported in -615:.71t§$.J %~ ~ ~~, 6j£4L~~ '3c! '6Pf 7] Korea( estimated in cidence : 1/3,456) W61.s::.T--%(Esophageal atresia with distal tra cheoesophageal fistula) 0] 126~ (87.5%), tr-'T ~5:..!j'--%~o] 8~ (5.6%) H-~ 71~-61.s::.T-~ol 3 ~ (2.} %)%$.J tt'-°l~.2..vj, l:ll13AJ?;j~ ~ (unusu al type) 3~~ 2t§ t AJlj!·.!j'--tt~ol 9J.::il i;}1jL71~
-151- 6js:.-r-tt 0] .y 7 ]~ A ] ~ A]1fL ~ (carina )oj] ~ ~Jl , Table 7. Incidence of Other Congenita l Anomalies t-j-Pj A] 1 tg ~ "J1fL6J s:.1¥--tt -'6- 1fL~ ~ .y 7] ~ A] ~ Anomaly No. KAPS series Holder series
1fL7} ~%~ (common wa ll) -£- 5:* -6} ~ -r-tt 0 ] \it CHD 46 31 % 19% ~ ~Ell~q(lE 6). Imperforate anus 19 13% 9% 148tg% 73tg(49%)oJ]Ai ~7}A ] o] "J~ 7 ]~0] Musculoskeletal 15 10% 9% Genitourinary 10 7% 10% %It!~ ~:: t~], 11 ::'B"a {l 7.J7] t§ 0] 46 ill] (31 %), "B'J-l!: Gastrointestinal 7 5% 13% -3J AJ7]~ 19 ill] (13%), -ctt2j 7 ]~ 15 ill] (10%)% CNS 6 4% 6% 9.J 'i'°]~q(lE 7). %~ 7 ]~~ '\HJ ~ "B'J*.g. lE8 Face 6 4% 5% oj] ~ 7]~ ~Oi ~q . Others 7 7% 9% * 75 (51 %) patients had no associated anomalies. Table 6. Types of Esophageal Anomalies * 73 (49%) patients had one or more associated KAPS* series Holder 1964 anomalies. EA + distal TEF 126(87.5%) 86.5% Pure EA 8( 5.6%) 7.7 % H-type TEF 3( 2.1 %) 4.2% EA + Double TEF 2( 1.4% ) 0.7% A + Proximal TEF 1( 0.7%) 0.8% ~ ~ ~ oJ 0J:~ (total parenteral nutrition) 0] 83 1( Esophageal stenosis 0.7%) 0.0% tg (66% )oJH A]t;~~~Jl, .!i!.~:3j ~*.R~o] 65 Unusual type 3( 2.1 %) 0.0% tg(52%)oJ]Ai A]t;~~~~ t~], ~ + 3~ o]-6} A]t;~ * KAPS: Korean Association of Pediafric Sur- ~ ~~7} 25tg~.£. 7}7.J ~~q(lE 9). geons
Table 8. Types of Associated Anomalies T'ye Number T'y2e Number 1. CHD 46 6. Face 6 2. Imperforated anus 19 Cleft lip 2 3. Muskuloskeletal 15 Cleft palate 2 Polydactyly 5 Tongue-tie 1 Syndactyly 3 Cboanal atresia 1 Vertebral anomaly 1 Hip dislocation 1 7. CNS 6 Others 5 Hydrocephalus 1 Microcephaly 2 4. GUT 10 Brain atrophy 1 Cryptorchidism 4 Facia N palsy 1 Hydronephrosis 2 IVH 1 Horse-shoe kidney 1 Renal agenesis 1 8. Abdominal wall 3 Vesicoureteral reflux 1 Omphalocele 1 Vaginal prolapse 1 Abd. muscle deficiency 1 Inguinal hernia 1 5. GIT 7 Hypertrophic pyloric stenosis 4 9. Miscellaneous 7 Duodenal atresia 1 Diaphragmatic hernia 1 Others 2 of Bochdalek Biliary atresia 1 Down's dyndrome 1 Hirsutism 1 Single umbilical artery 2 Sclerema 1
-152-- Table 9. Postopera ti ve Care( N =: 126) Table 11 . Survivai Relatetl to Waterston Risk Cate· gory TPN Assisted Ventilation Category No.of pis. Lived Percent of Survival Yes 83(66% ) 3'?J °l()~ 25 A(2.5kg 1 ) 51 46 90.2 No 43(j4% ) 4~ 7'?J 17 B, (1.8 ~ 2.5kg) 14 10 71.4 8~ 14'?J 5 E}(2.5kg r +*Pn/ Ano) 32 24 75 15~27 '?J 8 C,(1.8kg ) 4 3 75 Others 10 C,(any kg + Severe 25 7 28 *Pn/ Ano) Table 10. Complications Total 126 90 71.4% Respiratory complications 65(52%) * Pn/ Ano : pneumonia or associated anomaly Pneumonia (35) (12) Atelectasis Table 12. Survival Related to Operative Procedure Pneumothorax (12) Operation Patient Lived Tracheal injury (3) Primary repair 96 71(74.0%) Hyaline membrane disease (2) Delayed primary repair 13 8(61.5%) Anastomotic leak 28(22% ) Staged repair 17 11(64.7%) Anastomotic stricture 27(21%) Total 126 90(71.4%) Recurrent fistula 3( 2%) Miscellaneous 33(26% ) Table 13. Causes of t.tlortality Sepsis (23) Wound problem (4) Pneumonia/ Sepsis 19 53% GER (4) Respiratory failure 12 33% Other (2) Pneumothorax (5) * Complication in 93 (74%) of 126 patients Hyaline membrane dis. (2) Asphyxia (1) Not specified (4) 148tg ~ ~ .zi A ~0J- x::: 7:~~t!- ~o~~ ~].21()~ Congenital heart disease 4 11% .Jl ~~% ~{?:- 126tg ~ 93tg (74% )0] t!-7~7:] Unknown 1 3% °VJ-9.1 ~"l'l~% 7Cl~om.Q..t9, ~.B-t!- ~"l'l'6'-.Q..-.£ * Mortality: 36(28.6%) of 126 patients §.%~"l'l'6'-° 1 65il1] (52%), -li-~-¥- T~ 28il1] (22
%), -li-~-¥- ~~ 27il1](21 % )%9.1 ~.Q..-.£ y.E}J;); Table 14. Postoperative Course(N=75) l:HK 10). Course Doing well 56 75% Doing well but GER 6 8% Recurrent respiratory infection 9 12% Waterston risk category oj] o:t~ J.~~% .!i!.l?! A Esophageal stricture with bougie 4 5%
-ir-~ 90.2%, B" B2, Cl-ir-{?:- 71.4%~.Jl, C2-ir-, ~ ~1]'l5-.l2J- ~71]~ol ~'6'-9.1 lll]~.l2J-, 'l5-~9.1 E} %1(}-7] ~).a {JAJ-"l'l 4~(ll%)~omq(lI. 13). ~~t:!-7] cSS 0] ~.c -ir-oJ] Ai 28 % -.£ J.~ ~% 9.1. 1t Zl t!- o~7J- ~ .zl- 7Cl.l2J-~ .!i!.l?! ~~o] 7}'ot!- 75~'l5- 56~ (75%) .!i!.~.Q..t9 ~~1]~~ J.~~%~ 71.4%~q(K ll). 0] °J:§:.o}~.Jl, A~~ ).a "J-71.s:. ~~ol ~::: 7Cl~7} +~ llJ~oJl o:t.e Ag~%;: 61.5%-74.0%-.£ Zf 9~ (12%), .!fl6J.s:.Q;j*~ol ~.Q..y. .:z.'iitJl-.£ ~ 7:1 llJ'tJ ZloJ] -e- ~tO]7} ~~q(K 12). 126tg'l5- 36 y..c 7Cl~7} 6~ (8%), 6J.s:.~:3}.Q..-.£ ~AJ-~~ ~ ~(28.6%)o ] A}°J-o~~.Q..t9, 'l5-.B-t!- ~~.Q..-.£ llll~ .Jl ~.c 7Cl~7} 4~(5%)Ol~q(lI. 14). / llll ~'6'-0] 19t9 (53%), §.%-¥-~ 12tg (33%), ~
- 153- ~~ 5. .q..~ rJl%~-'6- ~ H-~ 71~-6J5:.T.:g-oJl rJl II. ~~(Quesdonnaires)otl ~'M ~,~ ~.l!/- ~ ~~.g.?(N=26) Experience with right aortic arch 9(35% ) Experience with H-type TEF 8(31 %)
~~ 8. ~ 'F -5-}.lj!--6J5:. ~;;:!°l <>1aj~Jt! 7;:l.q..7} ~ (N=6) ~cAl ~.Q.1?! <>1tg!711 "8ll~-5-}S1cAl?(7l{!01 .:J.to~~ -154- c. Stitch number $j 7.jli "J~-€:-? CD 5 1(4%) ® 6 - 8 11(42%) CD Esophageal anastomosis 16 ® 9- 11 11(42%) @ 12 - 18 3(12%) Primary repair + / - myotomy or bougie (8) Gastrostomy and 3 months later (4) ~-li:- 11. U} jiF~a T~A] (primary repair) A] T Gastrostomy + fistula division and ~ ~~-€:-? 3 months later (2) CD Esophageal repair only 13(50% ) Fistula division only and 2~6 months ® Esopahgeal repair plus 13(50%) later (2) Gastostmy (3), ® Interposition graft(stomach or colon) 7 Gastrosttmy + Centrallline ( 4 ), Centralline(6) Gastrostomy and1 year later (1) Gastrostomy + fistula division and 1 year later (3) ~-li:- 12. 78.g.~1f.:;;:!(Transanastomotic tube)A}.§ Esophagostomy + gastrostomy ~1f..2l A}.§-A] J 7]Z!: ~ A}.§- o]%e? + fistula divison and 1 year later (3) a. A }.§-~1f. CD A}.§- 21(81 %) ® OO~ 1 7°,J : 14, 4~5~ : 3, ~.g. 16. ~T ~ s:.lj'-.:g.~Al 7.1li "Hl-€:-? 2~3~ : 2, 10~12~ : 2 CD Gastrostomy + primary repair 12 ( 48 % ) ® A}.§- ~~ 5(19%) in 2~3mo:7, in 4~6mo:2, in 7~12mo:3 b. A}.§-o]%(18~o] ~7}Aj °VJ %'it~) ® Primary repair 3 (12 % ) CD Stent 10 ® Decompression 6 ® Esophagostomy and gastrostomy and colon ® feeding 5 @ GER J 1 graft in 1 ~ 2 years 6(24% ) ® Guide in stricture 1 @ Gastrostomy and stomach/ colon graft in lyear 4(16 %) ~ -li:- 13. t:!:ll] ~ ~~ (staged procedure) $j ~ g ~-€:-?(N=26) ~.g. 17. ~ s:. 7.1~~ (esophageal replacement) A1 CD Long gap(3.0cm 1 , or carina fistula) 15 {!§."5"}e :: - 155 - b. Assisted ventilation a. Factors in stricture formation(N = 23 ) CD Routine 8(31 %) ® Sometimes 16(62% ) CD Leak 14( 61 % ) @ No 2( 8%) ® Tension 10(43% ) @ Surgical skill 8(35% ) ~ * 19. ~ +- ~~] ~ s:.~oa~% ;"]i>~<'>H=-A l? ::I @ GER 2(9% ) c.j Jl '{i~] T % ~ ;..] -3}<'>H::-A] ? @ Others 3(13%) a. Esophagogram b. Management for stricture(N= 23 ) CD 5 d 3(12% ) ® 7~8d 19(76%) CD Forgaty balloon catheter 12(52%) @ 9~14d 3(12%) @ No 1(4% ) ® Other type of balloon catheter 4(17%) b. Feeding @ Esophagoscopic dilatation 4(17%) CD 2~3d 2(8%) ® 5~6 d 4(15% ) @ Rubber esphageal bougie 2(9% ) @ 7~8d .17(65%) @ 10 ~ 14d 3(12% ) @ Tucker dilator via gastrostomy 1(4% ) c. Prophylactic dilatation CD Yes 1 ® No 24 (N=25) ~* 23. A~e- oj] °a'O'.,1:% upl-c Jl'tl-€:-? (N=22) CD Associated anomaly 19 (86 % ) CD Anastomotic leak 23(92% ) ® Low birth weight 11(50%) ® Anastomotic stricture 21(84%) @ Long gap 6(27%) @ Respiratory complication 18(72%) @ Surgical skill 5(23% ) GER (13) Tracheomalacia (5) @ Preop pulmonary problem 4(18%) Not mentioned (6) ® Postoperative complication 3(14%) @ Recurrent fistula 14(56% ) IJ) Nursing care quality 1(4.5%) @ Pneumothorax 2(8% ) @ Age at surgry 1(4.5% ) ~* 21. *~-¥- T-~ ~'tl~ o] ~ ~o ] 7 ] 5fl~ tlJ ~* 24. ~ ~ AJ71s:. ~~.Q.5!. ArT ~~ojl '?J~ ~,c:? a. Factors (N = 26 ) W rrJl A~Zf8jJ o t W ~'tl ~~-€:-?(N = 22) CD Gastroesophagal reflux 10 ( 45 % ) CD Tension & long gap 20(77%) ® Recurrent or missed TEF 10(45%) ® Surgicall skill 7(27%) @ Esophageal stricture 6(27%) @ Infection 3(12%) @ Esophageal dysmotility 3(14%) @ Poor distal pouch 2(8%) @ Swallowing problem 2(9% ) @ Other 5(19%) Tracheomalacia 2(9% ) b. To lessen leak(N=25) ® IJ) Others 1(5%) CD Neck flexion 11 (44 % ) ® Transanastomotic 9(36% ) ~* 25. ~ ~ {J~ 5fl~£Q:j*% 5'::-c 7 1:t!~§} @ Assisted ventilation 5(20% ) % (tracheomalacia) % 7J ~ ~ ~ 0 I ~ -CAl? @ Gastrostomy 3(12%) a. GER probably requiring antireflux proce @ Others 3(12%) dure CD Yes 6 ® No 19 22. 7.]li-c? ~* *~-¥- ~:3}9.j 'tl;z,} ~ ~:3} ;.. ] b. Experience with tracheomalacia .:Lc.jJl ~ ~ oj]tlJ~ ~s:. ~ AJ~ ;..1i>~'6}-cAI? CD Yes 5 ® No 20 -156- 7]~9.j %*.£..~ 11. ~Acl {J ~~ 0] 31 %, "8"J-~3J AJ71 ~ 13%, :C~~f1 1 7 1~ 'g.9.j 'i?°J~1=-t:ll. 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A1] 2 ~ 1995\1 - ~ ~lli ~ il}%oJ1 -\:l:~ %-~11 ~ '?l 7S 1';.,1=% .21C:;;-il} III NA: Influence of co ngenital heart disese on .iil ~-":jcS"}O:j ~c.l ,7.}li~ TO] .!i!.Jl~.9.j .*}1ll. .Aej ~ management of oesophageal atresia. Pediatr %-A}.Aej g ~-":jcS"}Jl A~ ~.Q. oj, q~ cS"}4~ ~~A~ Surg Int 7: 90-93, 1992 (questionna ir es) ~ -":j% £.rJl£ 1U} rJl~:t.0}.21il} 9. Goh DW, Brereton RJ: Triangle of ausculta ~§] ~"'€rJl§]oJ1 A~ £..9.j~ 41%% 71&£ §]-'tl%.9.j tion thoractomy for esophageal atresia. ~ £71 ~.9.j ~ Ttl: ~lli, "'€+ -\:l:c.l, ~~ % %oJ1 rJl Esopageal artesia. J Thora Cardiovasc Surg ~ 7S ~il} .9.j {1% "5-~cS"}O:j Jl'i1% 4.Aej cS"}$1 q. 103(1): 14-16,1992 10. Spitz L. Kiely E, Brereton RJ : Esophageal atresia; Five year experience with 148 cases. J Pediatr Surg 22(2): 103-108. 1987 1. Spitz L. Hitchcock RJ: Oesophageal atresia 11. Chittmittrapap S, Spitz L. Kiely EM. 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