endoscopic and surgical management of intrabiliary rupture of...

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Endoscopic and surgical management of intrabiliary rupture

of hydatid liver cyst

SEB,\ql1\N LWNl,, MR.RS, MMED, ()IP, VEN, Yl)uNl,- IN KIM, ML), FRCPC, Rl)BIN (,RAY, Ml), FRCPC, PAUL KURTt\N, MD, FRCPC. CiRH,l)Rr 11:\HI R, M l), FRCPC'

S LEONG, Y-1 KLM, R GRAY, P KORTAN, G HABER. Endoscopic and surgical management of intrabiliary rupture of hydatid liver cyst. Can J Gastroenterol 1992;6(3 ): 135-139. A man with hydatid disease complicare<l by intraab<lomina l cyst rupture 15 years earlier, presented with choLestatic jaundice. There was intrabilia ry rupture of a hydatid liver cyst on cndoswpic rcrrogrnJe cholangiography. Sphinctcrotorny was perfonned wallow clearance of hydmid materia l obstructing the bile ducts and insertion of a nasobiliary catheter for irrigation and drainage. Defini t ive surgery was performed . Wh ile endoscopic management is ga ining recognition for re lieving biliary obstruc tion in hydatid cystobiliary rupture, surgery is still required for patients who continue to pass hydatid debns obstruc ting the biliary tree and inc reasing the risk of cholangitis.

Key Words: Bile duct , Endoscopic retrograde cholangiograpliy. Hydatid cyst, SJJ/iincterowmy

Traitement endoscopique et chirurgical d'un kyste hydatique du foie rompu dans les voies biliaires

RESUME: Un homme a ttc int d 'unc mala<lie hydatiquc compliquce d'un kystc intra-abJo rninal rompu 15 an~ auparavant se pre~ente avec une jaunisse chn lostarique. A la cholangiographie endoscopiquc re trograde, on a pu o b~crver la presence d 'un kystc hyJ a tique du foie rompu J ans les voics biliaires. Un c sphincte rcccomie a ccc pratiquce a fin de perme ttre l'eliminan on des t issus hyJaciques qui obstrua ient ks voics b iliaires ct !'insertion d 'un cathe ter na obiliaire pour !' irrigation ct le dra inage. Le tout a etc suivi d' unc chirurgic definitive. Bien que !'endoscopic soil de plus en plus employee dans le trai ten ent de !'obstruction bilia ire causcc par la prc~ence d'un k.yste hydatiq uc du foie rompu , la chirurgie est toujours nccessai re chez lcs patients qui continuent a climiner de~ debris hyda t iques 4ui obstruent l'arbre bilici irc e t qui presentent un risque accru a l'eg,1rJ de la cholan gite.

Thl' WI dble'i I ((}s/>11al, /J11•,s,on of ( 1tNroc111erol(}gv anJ I ),•parrml'nr of Ri1d1ol(}gV, [lnivcrrn,,v/Tomnw, Tornnw. ()111,1rio

Com:,f><mdl'nu' ,md n·/mnt, · Dr<. ;n:gor:,• I lt1h,•r, I )11•1,1011 of <.,a.,cnk.'1H1:rolo!!)' , cm/osni/1)• l 1nic, /foom 36 1, Tlil' \Xldle.,l.:'i 1-fo,f>Hal, 160 \Xldl.:,li:'i Snw1 r:mr, Toronto, Onrmw M4Y 1.n Tdr/1/wni: (416) 9(1H- 106~. F,i:1. (416) %8-7696

Rec.•11·.:djin p11h/rc,1t1tm Fc:hmm-:,· 14. /992 Acci:/11,:d A/ml !7. 1991

CAN J ( ,.-\STIH )INTI It, )I V, 11 6 N,) \ M M/JUN!' 199 2

H YDAl II ) l)J<..;ff\<..;E 1-; OFTEN /\~Y~ll'

tom,1t1c unul comp l1catiom ov cur and this uncomm\111 l'ntity may hl' overlooked 111 ,m emergency snuau on when ,1 prl'vinusly health) pntit·n t presents frn the first time. I lydaud cyst nf

the liver is the 11111s1 common site and it prodm:e~ symptonb wlwn rhe cyst ht·comes infecred, compresses or ruptures 111to ,1djacen t struuures or gro\\'s ro ,111 cnonnous si:l' (I). A utsl' illusirnting the two mam com pl icar ions \if hyJaud d i:,e,1:,e ( 1,2) rupture ofa hydnt ,d liver cyst into the pL'ritone,11 cnvny and, many years lat<:r, anm her rupture 11110 the biliary tree is presented. Both condm ons may he life threaten mg and prompt management i-, vital for a succcs:,ful outcome.

CASE REPORT A 37-year-old man (if lt ,1 lian de

scent presemcd with jaundice assoc i

ated \\'I th ahdommal disu1mf,1rt for two days. I le had emigrated to Cannda at 12 yean, of age. During his childhood he liwd with hi:, parent, who were fa rmers, rcnd111g cattle, shl·ep, pigs and dog~.

At 22 years, he expcrienct·d severe ahdommal pain ,1fter hemg hit h) a ba ll in the ,1hdrnnen. Emergency laparntomy revealeJ a ruptured hydat id cyst 111 the Liver. Pan ial left he pm ic lobecto m) was performed and he made an uncventfu l i-ccnvl'ry.

LWNG e t al

Figure l) Com(ntted wmogra/)hy scan of ahdomcn showing two hydarid cysts in liver

Figure 2) Cholangiogram showing filling defect due w hydaticl memhranes in the left heparic duct

and common hi/e ducr

Ar 32 years, an ultrasound and computed tnmograpy scan of the ahdomen showed two small I iver cysts (Figure l) at a pre-employment check-up. Nn

treatment was given ,b they were thought to be dead cysts.

At 36 years, the patient presented with lower abdominal pain. Imaging

with ultrasound revealed a new pclvrc cyst. H e underwent a second laparotomy. The cyst was adherent to the bladder wall anterio rly, the rectum posteriorly and laterally to the pelvic wall . ll wa dissected intact and removed. It measured 5 cm in diameter and o n sectioning, multiple daughter cys[S were seen. With the exception of the previously noted liver cysts, no other lesions were found.

At 3 7 years, d,e patient presented with jaundice associated with upper ahdominal discomfort. On abdominal examination , upper and lower midline surgical scars and a tender, enlarged liver were noted. The liver function tests were consistent with cholc~taric jaundice and coagulati()n profi le was normal. Ultrasound of the abdomen revealed a distended gall bladder and a cystic le~ion in the remnant left lobe, measuring 3.9 by 4.5 cm. The common hile duct was dilated to l.6 cm. The intrahepatic ducts were also dilated. Endoscopic retrograde cholangil1graphy showed a dilated hiliary tree with a cystic dilation in a hranch of the left hepatic duct conrnming hydatid materia l (Figure 2). Endoscopic sphincterotomy was performed ,md yellowish gelatinous membrane~ were seen extruding from the ampullary \lrifice. A Donnia basket was passed w evacuatl.' hydarid dehris from the bi le ducts. There was dramatic recovery wi th relief

of biliary obstruction. Two weeks later the jaundice recur

red, assoc iated with fover ,Hld chills, suggesting cholangitis due tn hiliary obstruction from continuing dischnrgc of daughter cysts. At urgent endoscopic retrograde cholangiography, ycl lowish jelly- like hydatid material and pus were seen protruding out of the ampulla of Yater (Figure 3). There was hydatid debris in the d ilated common hile duct and lefc hepatic radical, which was communicating with the cyst ic cavity (Figure 4). After evacuation nf the debris with a basket, a nasnhiliary catheter was inserted into the cyst crwity to improve drainage and allow irrigat ion with saline. The biliary fluid and sediments did not contain any hooklcts or prntoscolices on microscopy. H ence there was no evidence for

136 CAN J GASTROENTERl l l VL)l. 6 NO 3 MAY/JUNE 1992

Figure 3) I lydmid memlnwie proin,c/inl!OIII of o/>cning of am/ml/(l of Vara

via hi Ii ry o f the pmasi Les and medical

thernpy with mehenda:li le was not given. A repeat ultrasnunJ ,i( the ,1h

domen showed a cystic c;1v iry communicating with the kft ma in hepm ic duct whic h was Jilared to 8 mm.

A ltho ugh the feve r and Jaundice

suh~iJcd with a repeat clemance uf the bile duc ts, surgical tre,H mcnt was elected to cradirn te the disease . Resection of the liver cont aining the cyst was performed togethe r with cho lecystec

tomy and imc n inn 1if a T -tuhe to a llow in tra- ,ind posto pc rntive cho la ngio

graphy. Recovery was uneventful and nine nm nths IMer the pa t icnr was asymptomatic and had rct urned to

work without evKlcnce of residual disease o n ,1hdnmina l ultrasound cxaminat io n .

DISCUSSION H ydatid Llisease is a parasitic in fec

tion mo,t Cllmmo nly ca used hy the ces

tode Echinococrns J!.rnnulosus. Very rarely it is Jue 10 Echinornccm multi-

Hydotid liver cyst

Figure 4) Clw/angio,grnm ~howing mewl hml<eL in cvsr c£aoirv crn111n1111icaiing wich 11!/t he/1mic cl11cc

locularis (3) . Humans, sheep a nd cattle

arc inte rmediate husts a nJ the dog is the cnmmon definiti ve host ( I). H ydatid disease is endemic in sheep-rearing wuntries a nd it is like ly thm the present patient acquired the infect ion during his c hilJhn,,d in Italy Crom

handling J ugs ,1r cat ing vegetahlcs C()n taminared with the ovn of the paras ite .

With mc rcasing immigrat ion , hu1m111 echinllcoccosis may be o n t he ri se in N o rth A merica ~incc the infec tion is acquired in the na tive country during childhood. In Canada it is more

c,1111mon in peo ple of G reek a nd Italia n descent (4).

Rupture o f an echinococcal cyst was

the fir~t indicat io n uf hydmiJ disease in this patie nt. Le wall c t al (5) described

three types of rupture: contained , communicating a nd direct , the last hc ing mnsl serious hecause spi 11 ing o f cyst

con tents int o the peri toneal cavity may cause a llergic and infccti ve seque lac. Ir is tho ught that patients with direct rup

t ure shrndJ receive anthe lminthic

drugs to prevent peritoneal hyda tido~is (6 ). T he rupture is C\lmmunicat ing

whe n cyst conten ts escape via b il iary rad icles lead ing to nhstrucrio n o f the bil iary tree ,md cho langitis as in the presen ted patien t. There has heen no

ev idence tha t transhiliary di~pcrsion of sc11licc~ lead~ w d isseminarinn of cch mococcal d isease.

Cyst rupt ure m,1y be due to externa l traum:1 (direct ruptu re) as in the in itia l prcsenta t i11n of th is case, or degene ra tion of t he paras it ic me mbranes (communicating rupture) resulting in the

rcle<1sc of hydatid daughte r cysts .ind J ehris in to the common h ilc duc t.

H ydar iJ cyst rup ture into the b iliary

tree is a ~eri11us complication occurring in 5 10 15% of pat ien ts with hepat ic involvemen t ( 7 ,8). Diagnosis was made only at laparmom y un t il srn1<igraphy hecamc ava ilable (7-9). Sonogrnphic v isualizau o n of ru p tured cyst material in to the h ili ary t ree and communica

t io n be tween a cy~t a nd the hil iary t rnct indicate intrnhi liary rupture ,if a

CAN J GAsrn, lFNTrR()t V()L 6 No 3 M AY/JUNE 1992 I 37

LEl1NG et al

hepatic h ydatiJ cyst ( 10-12). Together

with computeri:cd axia l tomography, which can demonstrate the cyst-bile duct communicat ion sometimes difficult to visuali ze with ultrasound, the diagnostic accuracy can reach 96% for

intrabi lia ry hydatid cyst rupture (13,14).

Cottone ct al (15) first reported the use of endoscopic retrograde cholangiography in the diagnosis of hepatic

hydatid cyst rupture into the biliary tree. The endoscopic pic ture o f yellowish gelatinous me mbranes protrud ing out of the ampulla suggest hydatiu cystobiliary rupture and this is con

firmed by pathological examination of the infected bile which will contain fragmented membranes and daughter

cysts. The presence of pus suggests secondary infectio n and requ ires adm in istrntinn o f apprnrriat c ,rnt ibio t ics. The cholangingram shows a dilated bilia ry tree with filling defects repre

sen ting daughter cysts and h ydarid memhranes. The leaf- like radio logical

appearance of t hese membranes som e

times changes in shape on serial cho langiogram and it differentiates

hydatid materia l from c holcdocholi thias is ( 16, 17). Entry of contrast into a cystic space po inrs t0 a communicat ing

type of ruptu re. Other radiological fca-

REFERENCES I . Sherlock S. Di,ease~ of I he Liver and

Biliary System, 8th edn. Oxfi lrd: Blackwell Scientific Puhlic;nions, 1989:568-75.

2. Al-Ha,hami HM. ln1 rahiliary rupture of hydatid cyst~ ol the liver. Br J Surg l 97 1 ;58:228- 32.

3. Rausch RL, Nelson GS. A review of the genus Echinococcu~. Ann Trop Med Par;isirol 1963;57: 127- 31.

4. umger JC. Rose DB, Kcy,tnne JS. Diagnosi, and management nfhydmid ,li~ca c of the liver. Ann Surg 1984;199:412-7.

5. Lcwall DB, McCnrkcll SJ. Rupture of Echinncoccal cysts: Diagnosis, clas,ific::ninn, and clinical implicm ion,. Am J Rncn1gcn,il 1986; 146: 191-4.

6. Pappalnrdo G. Rcggi,i n. Fr:uwroli FM, Cl al. Cnmhincd medical and surgical then1py: A new appn,ach to ahdominal hydatidnsb. lwl J Surg Sci 1987; 17:'l15-9.

7. Macris GJ, l ~ahnis NN. Rupture of

138

tu res include extrins ic compres ·io n of the biliary tract by a hepatic cyst , complete ohstruc t ion of extrnh cpat ic bi le

ducts and biliary fistula ( 18). Percutaneous transbe patic cholang iography has been used ro demo nstrate the rup

tun.: of a cyst into the bilia ry tree ( 19,20). However, there is the risk of peritoneal seeding or a naphylaxis atho ugh diagnostic and therapeutic percutaneous aspiratio n of an hepatic hyJatid cyst has a lso been described (21 ).

Before che advem of therapeutic endoscopy, surgery was the o n ly effective mode of trca tm.ent for hyJacid disease (15). When the hydatid cyst can

be complccdy removed, surgical exc isio n, partial hcpaceccomy or hepatic

lobectomy is performed (4). ln addition, cysts that have ruptured into the

bi liary tract must be trea ted as infected and the c,>mmon bile duct must be explored and drained with a large calibre

T -tubc whic h would a lso a llow a c ho langiogram to be done to ensure common bile duc e clearance before its rem oval (22).

Decompression of the biliary tree by endoscopic sphincterotomy was fi rst

repo rted in 1986 by S h e mesh ct a I ( 23). Evacuation of h ydacid debris from the obstructed biliary tree was safe and effective in preventing sepsis (23-25 ).

Echinoc,)ccus crt of the liver inlO the biliary duct. Ann Surg 1966;32: 36-44.

8. Al-1 lashimi I IM . lntrabilb1ry rupture of hyda1 id cyst ,if the liver. Br J Surg 197 I ;58:228-32.

9. Harris JD. Rupture of hydatid cyst into the hiliary tract. Br J Surg l 965;52:2 l 0-2.

10. Camunc: F, Simo G, Robledo R, ct al. Ulm1sound diagnosis nf ruptured hydatid cyst of the liver with hiliary nh$lruc1 ion. Gastrointcst R,idiol 1986; 11 :330-3.

I l. Su bra man yam BR, Balthazar EJ, Naidich DP. Ruptured hydatid cyst with hiliary l1hstruction: Di:1gnosis by son,>graphy and computed comogrnphy. Gasrroinrcst Radio! 1983;8: 341 -3.

12. Maru-Bonmau L, Menor F, Ballcsrn A. I lydatid cyM ,){ the liver: Rupture into the biliary tree. Am J Rocntgcnol 1988: l 50: I 05 I -3.

13. Munzer D. Evaluat ion of computerized ax ial t,1mography in the diagno,b of hepa1 ic hydatid disease. J 1-lcpatol 1982;2: l 14-7.

Patients with good clearance of the bile duc ts avoided ~urgery (26,27). In post

o perative patie nts, rhc cause nf jaundice was quickly found a nd endoscopic sphincterutomy with insertion of a nasobi I iary rntheter for irrigation replaced surgery in some patients (26). Scolic ida l agents such as hypertnnic saline can be used co irrigate a nd kill any viable parasites via the nasohilimy

drain (27). This was not used in the p resent patient as t he patho logist found no v iable parasites. However, frequent irrigatio n via a nasohilimy cat heter helps to clea r the h ilc ducts l1f dcbrb

and this migh t have prevented a recurrence of cholangitis a fter the fir~t e ndoscopic retrograde c holangiogrnphy and endoscopic sphinctcrntomy . More likely, it is the continued d ischa rge of daughter cysts t ha t prevents h ilc drainage. O ther possible causes include incomple te extract io n of common hilc

duct debris and inadcquarc sphinctcru romy.

While e ndoscopic treatment of intrabilia ry rupture of hydacid cyst may temporari ly relieve biliary ob truc tion, surgery shou ld he comidered if there 1,

persistent discharge of h ydac id debri,

obstructi ng the b ile duct,. T hi~ will prevent cholangi tis nnd eradicate

hydatid d isease.

14. Mnni-Rnnmau L, Seminll FM. Complic:11 inn, nf hepatic hyd,11 tel cy,1,: Ultra,nund, computed 1omogrnphy and magnet ic re,onance diagno,i,. Gast roint eM Radio! 1990: I 'i: 119-25.

15. Connnc M, Amu,o M, Cotll>n PB. Endoscopic rccrogn1dc cholangio-graphy in hepatic hydat id di,ease. Br J Surg 1978;65: 107-8.

16. Beggs I. The r:1d iology of hyd,1tid disea.,e. Am J R,,entgen<>l 1985; 145:639-48.

17. Shemcsh E, Friedman E. Radi.)logk and endoscopic appearnnce~ of in1rnhiliary rupture ofhyd,11icl liver disea~e. Digestton I 987;'16:96- 100.

18. Munzer n. New perspect ive, in the diagnosis of Echinococcus disc:i-c. J Clin GaslnJenterol 1991; 13:415-23.

19. Tuttle RJ . Ca11,c ol rl'curring oh,trucuve i:iundicl' rl've,11'-d hy percutaneous chPlang1ogr:1phy -hydm id cy,t. N Engl J Med I 970;283:805-6.

20. Farre lly C, Lawril' BW. Diagnosi, of inl rabiliary rupture of hylbtid cyst of the liver hy fi nl' needle J'L'rc111 ,mcou,

CAN J l,ASTR,1ENTEROL VL11 6 No 3 MAY/JUN! 1992

transhepatic ch,1lang1<1graphy. Rr J Radiol 1982;55:172-3.

!l. Mueller PR, l);iwson SL, forrucc1 JT, N.1rd1 UL. I lepauc l'lhmncc:il cyst: Succe,~ful 1x·rcutaneou, dr:1111,1gl'. Am J R.1d1nl 1985; 155:627 8.

Z2. Da,h,h, J. (. ~:111wni, 0. A kt ra, 11. lntrahil,ary rupture nf the hyd,1ud cv,t or the l1VL'r. WPrld j Smg 1984;8: 786-90.

13. Shcmd1 E, Klt:1n E. Ahn11nmv1eh l), ct al. Cummlm hilc duct nh,truct1,m c.n1,ed h1 hyd.111d d.,ughter cy,t,.

Management hy endo,cop1c retmgr,1dc sph1111:teru1nmy. Am J Gastroent eml 1986;8 I :280-2.

24. Magi,rrell1 I', M."etti R, Cnppnla R. Value ,1( ERCP 111 the d1:ig111N, nnd man,1gemenr of pre and po,t nperauve hili:iry .:omplicauons in hyd::iud d,,ea,c uf the liver. Gm,tromte,t Radio! 1989; 14: 1 I 5-20.

25. Al Karaw1 MA, Mohamed ARE, Ym,awy Ml, ct al. Nonsurgicnl cndnscnpK trnn,papillary treat mcnt of ruptured Ec.:h111,1coccus liver cy,1

CANJ GA!sTROENTERlll VOL 6 N\) 1 MAY/jUNF 1992

Hydatid liver cyst

oh,tru<.:1111g the htl1.1ry trel', Endoscopy 1987; 19:81->.

26. bean ~ I, Duren ~ I. Endn,cop11: ,rhinct.:m1Pmy 111 tlw managcm.:nt of J'<i,t npl'r;ll l\·e complir:11 1nn, of hep.11 ic hyd:1ud d1M:,he. End,iscop\ 1991:2LZ82 t

27. Al Karnw, MA, Ya,.111·1 ~Ii. El Sheik M,ihamed AR End,iscopic.: management ,if hiln1ry hyclat1d d1,e,1,L' Report on ,,x c.1,c,. Endll,c,irv 1991 ;2 U78-8 I.

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