Thorax 1981 ;36:497-501

Hydatid cyst ofthe liver with intrathoracic rupture

DS XANTHAKIS, E KATSARAS, M EFTHIMIADIS, G PAPADAKIS,G VAROUCHAKIS, C ALIGIZAKIS

From the Thoracic Surgical Clinic, Sismanoglion Hospital, Athens, Greece

ABSTRACT In the last 10 years we have operated on 33 cases of hydatid cyst of the liver with intra-thoracic rupture. Twenty-one out of 33 cases ruptured into bronchi, in seven the rupture affected thepleural cavity, and in six simultaneous rupture into the bronchus and pleural cavity occurred.Laboratory tests were not carried out in all cases. The Casoni intradermal test was carried out in 23cases and was positive in 18. A liver scan was performed in 10 and was positive in all. Conservativeoperations were carried out in 22 patients. In these the hepatic cavity was evacuated and separatelydrained. This was followed by suturing the diaphragmatic rupture and also closing the bronchialopening if present. Lung resections were performed in 11 out of 33 cases. In eight lobectomy wascarried out and in three segmental resections. Resection was necessary when suppuration andbronchiectatic changes affected the lung. Ruptured cyst into the pleural cavity requires emergencythoracotomy after the anaphylactic shock is over. Removal of the parasite, re-expansion of the lung,and drainage of the pleural and hepatic cavities is necessary. Immediate and late complicationsoccurred in 13 patients. In two postoperative haemorrhage occurred and in two postoperativeempyema developed. Recurrent haemoptysis was seen in five, persistent bile fistula in one, anddissemination of hydatid cyst in three. In the remaining 20 cases there was no complication. Operat-ive mortality was nil.

Hydatid cysts situated in the upper liver surface havea tendency to grow upwards and if uncomplicatedthey may find their way through the diaphragmand become intrathoracic. Symptoms may becaused either by the size of cyst or, more commonly,if complications arise, by its rupture.Rupture may occur into the pericardium or

mediastinum, the lung and bronchial tree, thepleural cavity, or the peritoneal cavity. Beforerupture into the thorax, the cyst will have alreadyruptured into the biliary tract leading to daughtercyst formation and pyogenic infection. There istherefore a combination of abdominal and thoracicmanifestations. The symptoms from the former mayvary from slight digestive disturbances to epigastricpain with jaundice and enlargement of the liver.Thoracic manifestations vary with the type ofrupture. Involvement of pericardium and media-stinum is very rare and in this paper we will consideronly the more common ruptures into the lung andpleura. Rupture into the chest must be considereda serious complication and there are many publishedreports. 1-12

Address for reprint requests: Dr DS Xanthakis, TheThoracic Clinic, Sismanoglion Hospital, Athens, Greece.

In the last 10 years, we have operated on 33 casesof hydatid cysts of the liver with intrathoracicrupture and we have reviewed our cases.

Review of cases

Of the 33 patients, 23 were male and 10 female;23 came from rural areas. The age incidence isshown in table 1.

Table 1 Age incidence ofpatients

Age Number ofcases

Up to 40 years 1441-50 851-60 9Over 60 2

The sites of rupture are shown in table 2 and theclinical features in table 3.

Table 2 Sites ofruptureSite Number ofcasesRupture into the bronchi and biliary system 21Rupture into the pleural cavity 7Simultaneous rupture into the bronchi and

pleural cavity 4Rupture into the mediastinum

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498 Xanthakis, Katsaras, Efthimiadis, Papadakis, Varouchakis, Aligizakis

Table 3 Clinicalfeatures

Feature Number ofcases

Without symptoms, incidental finding IIrritating cough 24Purulent sputum 24Bile-stained sputum 10Chest pain 16Pyrexia 20Shortness of breath 11Vomique 6Haemoptysis 8Coughing up membranes 6Allergic reaction 2Finger clubbing 2

There is a variety of symptoms (table 3). Only onecase had no symptoms. Irritating cough was themore common symptom (24 patients), with purulentsputum in 24 and bile-stained sputum in 10. Chestpain was a frequent symptom (10 patients) andpyrexia was found in 20. This rise of temperature wasprobably caused by infected cysts. Shortness ofbreath was found in 11 patients. Sudden expectora-tion of pus and membranes of the cyst (vomique)occurred in six and coughing up daughter cysts ofhydatid elements in six. Allergic reactions werenoticed in only two. This is contrary to otherstatistics in which the percentage of allergic reactionis much higher. Finger clubbing was found in twocases of longstanding infected cysts.The results of laboratory investigations are shown

in table 4.

Table 4 Results oflaboratory investigations

Yest Number Positive Negativeofcases

Intradermal test (Casoni) 23 18 5Complement fixation test (Weinberg) 23 16 7Eosinophilia (more than 6%) 33 10 23Pleural fluid (Hooks or Scolices) 12 10 2Liver scan 10 10 -

Table 5 Radiological results

Result Number ofcases

Round or oval shadow ofvarying size in theRLL developing in the right dome ofdiaphragm 5Apparent pleural cyst 5

clearly separated from the diaphragm 3blurred outline 2

High position ofthe right dome 6Pneumonitis ofRLL 14Abscess formation 12

single 8multiple 4

Pleural fluid 8Spontaneous pyopneumothorax 3Pulmonary dissemination 3Bronchograms 8

Figl1Hydatid cyst ofthe right lower lobe after rupture ofa hepatic cyst into the bronchus.

Radiological findings are shown in table 5. Alarge hepatic cyst situated under the diaphragm is ofparticular interest to the thoracic surgeon (figs 1-2).It may be mistaken for a pulmonary cyst or maymimic a pleural effusion and be aspirated, withserious complications. Simple high dome of thediaphragm was noticed in six cases. An apparentpulmonary cyst either clearly separated from thediaphragm or with blurred outline was found inthree and two respectively. Pneumonitis or abscessformation in the right lower lobe was noticed in 14and 12 cases respectively. Pleural fluid was observedin eight. Spontaneous pyopneumothorax caused bysimultaneous rupture of the cyst into the pleura andbronchi was noticed in three (figs 3-5). Pulmonarydissemination caused by daughter cyst formationwas found in three. A bronchogram is a valuableexamination for confirming irreversible bronchectaticchanges.

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Hydatid cyst ofthe liver with intrathoracic rupture

Lung resection was not done often because lunglesions are usually reversible, but in cases where thelung was seriously damaged resection was necessary.In our cases three segmental resections and eightlobectomies were needed. Pneumonectomy was notrequired.

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Fig 2 Thoracic involution ofa hepatic cyst.

Table 6 Methods ofsurgery used

Method Number ofcases

Conservative operation 22Resections 11

Segmental 3Lobectomy 8Pneumonectomy 0

Decortication 5

The operative methods are shown in table 6. Inthe conservative operation the hepatic cavity wasevacuated and drained separately, followed bysuturing of the diaphragmatic rupture and closureof the bronchial opening if present, with drainageof the pleural cavity. If there was a separate cystof the lung this was removed together with a part ofthe adventitia, the fistula was closed, and theremaining pulmonary cavity sutured.

Fig 3 Rupture ofa hepatic cyst into thepleura withhydropneumothorax. A parietal cyst can be seen.

Rupture of a cyst into the pleural cavity necessi-tates emergency thoracotomy. The objects of theoperation then are to remove the parasite, drain thehepatic cavity, and re-expand the lung immediately.Separate drainage of the chest is necessary. Even inneglected rupture with pyothorax and daughtercysts in the visceral or parietal pleura the same typeof operation is indicated. The danger of recurrenceis greater than when cysts rupture into the bronchibut this danger does not justify pleuropneumonec-tomy. In cases with biliary complications opening

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Xanthakis, Katsaras, Efthimiadis, Papadakis, Varouchakis, Aligizakis

Table 7 Outcome ofoperation

Outcome Number ofcases

Immediate postoperative complicationsHaemorrhage 2Empyema 2

Late postoperative complicationsRecurrent haemoptysis 5Persistent bile fistula IDissemination ofhydatic cyst 3

Without complications 20Operative mortality 0

of the common bile duct is necessary with T-tubedrainage.

Early postoperative results are shown in table 7.Postoperative haemorrhage occurred in two casesand thoracotomy was necessary in both. Post-operative empyema occurred in two cases and aftersimple drainage the course was uneventful. Re-current haemoptysis was observed in five cases.Bronchiectatic changes were found in two of thoseand no obvious cause in the remaining three. Allwere treated conservatively. Dissemination ofhydatid cysts was found in three, affecting the rightlower lobe, and lobectomy was carried out in all.There was no operative mortality.

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Fig 4 Rupture ofhepatic cyst into thepleura. Daughtercystsfloating on the surface.

Discussion

Cysts located in the dome of the liver can grow intothe chest and cause severe complications. This exten-sion of a cyst into the thorax is usually caused by itsposition in the superior aspect of the liver and theattendant inflammatory reaction. It is seen inapproximately 3% of the cases.3 Stathatos et alreported 18 cases, in 15 of which rupture into the

Fig 5 After removing thefluida mass ofdaughter cystscan be seen in thepleural andhepatic cavity.

bronchial tree occurred. Tsoufis et a! described thesurgical methods and results in 28 cases of liver cystrupturing into the bronchus.6 Guedj et al'3 reporting600 cases of hydatid cysts of the liver from theperiod 1958-68, observed 15 cases of rupture givingan incidence of 2-5 %. A similar percentage wasreported by Kourias1 in 1334 cases. Bourgeon et all,reported 154 cases, seven of which had ruptured(4 -5 %). Aletras et al 12 reported four ruptures (3%)in 130 cases. Deve and Caniot"6 reported 1743 caseswith rupture in 4-5 0/When the hydatid cyst communicates with the

biliary system and becomes infected, the inflam-matory process extends towards the inferior aspectof the diaphragm and obliterates the space betweenliver and diaphragm. As the size of the cyst increasesthe diaphragmatic fibres get thinner from thepressure and eventually rupture. If there are pre-existing adhesions in the pleural cavity then therupture is directed into the bronchial tree. In caseswhere there are no pleural adhesions the rupturemay appear only in the pleural cavity, but this isless common.3 A true reactive bronchopneu-monitis occurs when the cyst opens into a bronchusand a broncho-biliary fistula is created. This resultsin pain and expectoration of bile-stained pus some-times with daughter cysts or membranes.The diagnosis of rupture presents no difficulty

when bile-stained sputum and daughter cysts areexpectorated. Daughter cysts are found morefrequently in hepatic than in pulmonary cysts.

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Hydatid cyst ofthe liver with intrathoracic rupture

The infected purulent cysts are usually old chroniclesions; communication with the bile ducts isblocked by elements of hydatid cyst and jaundicemay appear.

Primary rupture into the pleural cavity withoutlung involvement is rare. Of our 33 cases only sixruptured into the pleura.By the time the final rupture occurs the diaphragm

has usually become attached to both the liver andthe lung. The symptoms are of a sudden tearingpain, dyspnoea, and anaphylactic shock from whichthe patient may die. If he recovers, the chest radio-graph shows a pleural effusion or a pyopneu-mothorax particularly if the rupture into the pleuraoccurred after rupture into the bronchus.Many authors are in favour of a transpleural

approach.138-10 We have followed the sameapproach. It gives a good exposure and the lungcomplications can be treated. If abdominal com-plications occur the incision can be extended to theabdomen or a separate abdominal incision made. 59

Philippakis reports six cases, in four of whichrupture occurred into the middle lobe.17 A thoraco-abdominal incision was used in four and in two acombination of thoracic and abdominal incisionwas used. Avlamis'1 stresses the importance ofdraining the common bile duct in ruptured cyst withbiliary complications. We prefer to be as conserva-tive as possible as in our cases of hydatid disease ofthe chest. 18

After dissecting the bronchial fistula the openingin the diaphragm is enlarged and the hepatic cavitycleared and drained subdiaphragmatically. Themiddle and lower lobe can undergo an intensesuppurating process that makes the surgeon resortto resection, which can be technically difficult.

It is important to be conservative in dealing withpulmonary complications and we agree withCrausaz3 that lung complications are, in the majority,reversible-Crausaz, in 25 cases, carried out onlyone resection.3 The drainage of the hepatic cavitymust be continued until complete closure of thecavity occurs. The diaphragm is closed in layers and awater seal drainage catheter is left in the pleuralcavity for 48 hours at least.When the common bile duct is obstructed by

elements of hydatid cyst a T-tube has to be insertedat the same operation.A cyst rupturing into the pleural cavity requires

emergency thoracotomy after the anaphylacticreaction is over. The objects of the operation are toremove the cyst which is usually found floating inthe pleural cavity. If there are remnants of thecyst in the hepatic cavity it has to be drained as

already described.Even in neglected rupture with pyopneumothorax

and daughter cysts in the parietal or visceral pleurathe same type of operation is indicated withpleurectomy and decortication of the lung. Thedanger of recurrence is greater than in rupturedcysts into the bronchial tree but even this dangerdoes not justify pneumonectomy.

References

Susman MP. Hydatid disease as it affects the thoracicsurgeon. J Thorac Surg 1953 ;26:111-30.

2 Tool H, Propatoridis J, Pangalos N. Intrapulmonaryrupture of hydatid cyst of the liver. Thorax 1953 ;8274-80.

3Crausaz PH. Hydatid cyst of the lung and hydatid diseaseof the liver with intra-thoracic evolution. J ThoracCardiovasc Surg 1967 ;53:116-29.

4 Kourias B, Mantonakis S. Kystes hydatique du foierompus dans les voies biliaires. Regles therapeutiquesactuelles. J Chir (Paris) 1968 ;96:21-36.

Maragos G. The surgical treatment of complicated hydatidcyst of the liver. Sixth Panhellenic Congress of Surgery1968 ;VI:270-86.

6 Tsoufis E, Agorogiannis S, Kourmadas C, Papaioannou K.Surgical methods and results in 28 cases of hydatid cystof the liver ruptured to the bronchi. Sixth PanhellenicCongress ofSurgery 1968 ;VI:377-85.

7 Stathatos C, Kontaxis A, Zaphiracopoulos P. Hydatidcyst of the liver. Sixth Panhellenic Congress of Surgery1968 ;VI:365-70.

8 Zaphiracopoulos P, Pappas C, Rouskas G. The manage-ment of thoracic complications of hepatic cyst rupturedinto the chest. Sixth Panhellenic Congress of Surgery1968 ;VI:537-46.

9 Giannacopoulos G, Papamichel E. Hydatid disease of theliver. Chest complications. Sixth Panhellenic Congress ofSurgery 1968;VI:508-12.

10 Katsaras E, Zoitopoulos M, Katsara R. Observations oftrans-thoracic surgical approach of hydatid cyst of theupper surface of the liver. Sixth Panhellenic Congress ofSurgery 1968 ;VI:435-40.

Avlamis G, Bonelos K. Problems and surgical treatment ofbroncho-biliary fistulas due to hydatid cyst of the liver.SecondPanhellenic Congress ofSurgery 1961 ;II :960-4.

12 Aletras H, Dadoukis J, Aidonopoulos A. Bronchobiliaryfistulas. Galenus 1971 ;13:657-63.

13 Guedj P, Gairoard G, Morvan F, Bogaert J. Le traitementchirurgical actuel du kyste hydatique du foie et sesprincipales complications. J Chir (Paris) 1967; 93:191-7.

14 Kourias V, Chouliaras C, Gikas D. Thoughts and con-clusions in 1334 operated cases of hydatid of the liverSixth Panhellenic Congress ofSurgery 1968 ;VI:371-6.

15 Bourgeon et al. Reported by Tsoufis E et al. Sixth Pan-hellenic Congress ofSurgery 1968 ;VI:377-85.

16 Deve F. L'ouverture de kysts hydatique du foie dans lespoumons et les bronches. Cinquieme Congres Annuel desSciences Medicales 1935. Oran: 27-32.

17 Philippakis M, Chourdakis C, Alivizatos C, Iconomidis C.Hepato-bronchial fistula due to ruptured hydatid cystof the upper part of the liver. Sixth Panhellenic CongressofSurgery 1968 ;VI:441-5.

1s Xanthakis D, Efthimiadis M, Papadakis G et al. Hydatiddisease ofthe chest. Thorax 1972;27:517-28.

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