echinococcal tension pneumothorax

Thorax (1969), 24, 547.

Echinococcal tension pneumothoraxFARHAN BAKIR AND MUAYYAD M. AL-OMERI

From the Medical Department and Cardio-thoracic Unit, Medical College anzd Republic Hospital,University of Baghdad, Baghdad, Iraq

Hydatid cyst is rarely mentioned among the causes of pneumothorax in text-books or monographs,especially those written in English. Five examples of tension pneumothorax secondary to rupturedhydatid cyst of the lung are reported: the mechanism of this tension effect and helpful diagnosticpoints are discussed. We think that surgical correction is the only satisfactory treatment of tensionpneumothorax due to ruptured hydatid cyst: surgery is advocated in any suspected cyst as soonas it is discovered so as to avoid any such serious complication.

Hydatid cyst is rarely listed among the aetiologicalcauses of spontaneous pneumothorax in theEnglish literature (Perry, 1939; DuBose, Price, andGuilfoil, 1953; Mills and Baisch, 1965).

References to echinococcus as a cause of pneu-mothorax are scanty even from countries in whichthe disease is known to be prevalent or endemic(Sinclair, 1955). Among 478 cases of pulmonaryhydatid cyst seen in Australia and New Zealand,hydropneumothorax occurred in 244% of the 246cases from Australia but was not reported amongthe 232 New Zealand cases (Waddle, 1950). In aprevious study on pulmonary hydatid cyst in Iraqonly one case of pneumothorax was listed amongthe 189 cases reported (Kelly and Izzi, 1959).However, we found some references in the

Spanish medical literature to pneumothorax com-plicating pulmonary hydatids (Sala de Pablo andSan Joaquin, 1954). Tomalino (1959), in his paperon pleural complications, gave 16 examples ofpneumothorax, with mediastinal shift in five cases.Agustoni, Taboada, Berinstein, and Chorne (1960)have reported a single case of tension pneumo-thorax.The purpose of this paper is to present five cases

of tension pneumothorax due to pulmonaryhydatid cysts. All the five patients were seen with-in a period of one year.

CASE REPORTS

CASE 1 A 12-year-old boy complained of suddenshortness of breath which was preceded by right-sidedpleuritic pain, stabbing in n-ature, together with a dryirritative cough. Physical examination revealed aseriously sick child with severe respiratory distressand orthopnoca. Respiration was shallow and therate was 42 per minute; the pulse rate was 116 per

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minute and the temperature 380 C. The trachea waswell shifted to the left and the apex beat was pushedto the mid-axillary line. The classical signs of fluidwere demonstrated in the lower part of the right hemi-thorax, and signs of air were found in the upper partof the right pleural cavity. A chest radiograph con-firmed the presence of hydropneumothorax (Fig. la).The W.B.C. ranged between 9,000 and 10,000/cu. mm.There were 760/% polymorphs and 6% eosinophils. Onaspiration of the chest, air under high pressure gushedout, and was followed by turbid fluid. A repeat chestradiograph failed to show improvement, so a water-sealed drainage tube was inserted, and suction applied.The symptoms improved, the trachea became central,and a further radiograph showed correction of themediastinal shift, though the lung was still collapsed.There was an ill-defined shadow at the right cardio-phrenic angle with an irregular thin wall border (Fig.lb). The Casoni skin test was positive. Because of thefailure of conservative measures a thoracotomy wasperformed, when a huge ruptured hydatid cyst wasfound destroying and replacing the middle lobe, whichwas therefore removed. Lung expansion was completeand recovery was satisfactory (Fig. Ic).

CASE 2 A 26-year-old girl had been in normal healthuntil four days prior to admission, when she fell downstairs on her right side. She developed severe distres-sing pleural pain with irritative cough and hae-moptysis. The pain increased on coughing andbreathing. After the chest had been strapped for asuspected fractured rib, rigor, fever, and progressiveshortness of breath developed.

Physical examination revealed a sick woman whowas in pain and had severe dyspnoea. The respiratoryrate was 38/minute, the pulse rate 110/minute, thetemperature 39° C, and the trachea was pushed tothe left. Signs of hydropneumothorax were elicited inthe right hemithorax, and the liver was enlarged toabou,t 3 cm. below the costal margin. Her chest radio-graph demonstrated a partial collapse of the rightlung with air at the top and fluid at the bottom of

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Farhan Bakir and Muayyad M. Al-Omeri

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Echinococcal tension pneumothorax

(c)FIG. 1. Case 1. (a) Hydropneumothorax with marked mediastinal shift. (b) Aftera water seal drainage and suction, double wall rounded shadow is seen at the baseof the right lung. (c) Post-operative film. The lung is fully expanded. The shadowat the right base is a residual pleural reaction.

the right pleural cavity. A round shadow wasvisualized in the collapsed right lobe and a secondone was seen in the upper zone of the left lung (Fig.2a). The W.B.C. was 10,200/cu. mm. and there were8%° eosinophils. The Casoni skin test and theWeinberg complement-fixation test were negative.Serosanguineous fluid came through a water sealdrainage, but this last procedure did not succeed inrelieving the collapse (Fig. 2b). A thoracotomy wasperformed and a ruptured hydatid cyst in the rightlower lobe was found. Lobectomy was done andanother cyst was enucleated from the right upper lobe,after which recovery was uneventful and the lungfully expeanded (Fig. 2c).

CASE 3 A 40-year-old woman experienced asharp, stabbing, tearing pain in the left hemithoraxfollowing a simple sneeze. Her past history was notsignificant except for removal of a hydatid cyst fromthe kidney a few years previously.

Examination revealed a seriously ill woman whowas orthopnoeic with a respiratory rate of 35/minuteand a pulse rate of 110/minute. The temperature was38° C., the trachea was shifted to the right, the apexbeat was felt in the right hemithorax, and the heartsounds were audible in the right chest. The left chestwas hyper-resonant, the breath sounds were absent,and the vocal resonance was diminished. A chestradiograph showed complete collapse of the left lung,with shift of the upper and lower mediastinum towardthe opposite side (Fig. 3a). The W.B.C. count was

9,000/cu. mm. and the eosinophils were 5-11%. TheCasoni skin test was positive. Aspiration revealed thehigh tension quality of the pneumothorax. Continuousair leak and failure of expansion of the lung in spite ofcontinuous suction confirmed the presence of a fairsized broncho-pleural fistula. When thoracotomy wascarried out, a ruptured hydatid cyst was removed fromthe lingula, and the fistula was closed. Recovery wassatisfactory and the lung expanded fully (Fig. 3b).

CASE 4 A 33-year-old woman was seized suddenlywith severe left-sided chest pain. The pain was asso-ciated with shortness of breath, cough, and expectora-tion of a large amount of salty fluid. She was referredto a county hospital where a chest radiograph wastaken, after which she was referred to our thoracicsurgical unit for management.Examination revealed a well-built woman who was

short of breath and perspiring. The pulse rate was110/minute. There was hyperresonance in the lowerpart of the left hemithorax, and the cardiac dullnessshifted to the right hemithorax. The chest radiographon admission showed a large air space in the lowerpart of the left chest with what seemed to be a water-lily appearance. Moderate shift of the lowermediastinum to the opposite side was present. Therewere linear opacities at the upper border of the airspace reaching the chest wall (Fig. 4a, b).Under-water seal drainage was placed in the mid-

axillary line, where there was a considerable air leakand some fluid came out. This procedure was of no

549





(hILegend to Fig. 2 (a) and (b) on page 551.

550





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help in expanding the lung. Examination of the fluidwas negative for scolices but the Casoni skin test waspositive.Thoracotomy was performed, when a large ruptured

hydatid cyst was found occupying the anterolateralaspect of the left lower lobe, and a 0-3-cm. tear wasfound in the pleura. The whole upper lobe wasadherent to the chest wall. The cyst was removed, theleaking bronchus was closed, the space was obliterated,and the chest was closed with basal and apical drain-age. The post-operative course was uneventful.

CASE 5 An 18-year-old girl complained of severepleuritic pain of seven days' duration. The pain wasof gradual onset and was assoiated with cough,expectoration of whitish sputum, and a continuouslow-grade fever. The patient gave a history of hae-moptysis six years previously, for which she had beenadmitted to a sanatorium.On physical examination the patient was actually

ill and in pain, the pulse rate was 120/minute, thetemperature was 38 5' C., and the respiratory rate was

40/minute. The trachea was central on admission. Theleft upper chest was slightly more resonant than theright, but there were signs of a small effusion in theleft axilla. The lower border of the liver was felt3 cm. below the right costal margin and was nottender. The spleen was felt 5 cm. below the costalmargin: the uterus was the size of a 32 weeks'pregnancy. The admission chest radiograph showeda small pneumothorax on the left side and a cavity-like appearance in the collapsed lung with pleuralthickening and effusion (Fig. 5a). The W.B.C. countranged from 15,000 to I 1,000/cu. mm. The polymorphsranged from 90 to 73°,' and the eosinophils were with-in normal limits. The Casoni skin test was positive.Two days after admission to hospital the patient

developed severe respiratory distress. The tracheashifted to the opposite side and signs of tension pneu-mothorax were evident. A further chest radiographshowed obvious mediastinal shift, a collapsed lungwith an empty cavity in it, and a fluid level at thelower part of the pleural cavity (Fig. Sb). A water-sealdrainage was placed in the second anterior space.

551




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FIG. 3. Case 3. (a) Massive left pneumothorax with severe mediastinal shift andcomplete atelectasis of the left lung. (b) Post-operative film. Full expansion with anormal chest radiograph.





FIG. 4. Case 4. P.A. and lateral views showing selectivepneumothorax, inediastinal shift, and water-lily appearance.

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FIG. 5. Case 5. (a) Small pneumothorax and pleural thickening and effusion inthe collapsed lung. (b) Tension pneumothorax two days after admission.

554





Unmistakable hydatid material was recovered fromthe fluid aspirated posteriorly. The patient gave birthto a normal infant prematurely, and she is still inhospital under conservative measures for the infectedcavity.

DISCUSSION

Hydatid cyst is a continuously growing parasite.Its expansion is usually resisted by the tissue andorgan of the host, though this reaction of the lungparenchyma is minimal and poor as comparedwith that of other organs (Cleland, 1963). Pul-monary echinococcal cysts may reach a huge sizeduring growth, the eroding blood vessel oftengiving rise to haemoptysis. The bronchial passagescould be eroded as well and air escape around orinto the cyst, this accounting for the various radio-logical features in this disease. The erodedbronchus does not function as a fistula to startwith, due to the pressure of the tense cyst againstthe fistula opening. Progressive enlargement willlead to its rupture either into the bronchial tree orinto the pleura. Intrapleural rupture more fre-quently presents as pneumo- or hydropneumo-thorax. Occasionally such an accident may pre-sent in one of the allergic manifestations such asurticaria, asthma, or dramatic anaphylactic shock.The pneumothorax may be tension in type, as

in the above cases, where it is quite easy tovisualize the tension or the valvular mechanism.The collapsed or crumpled wall of a ruptured cystsimply acts as a curtain or check valve, admitting

air into the pleura during inspiration, then fallingback and closing the opening or outlet duringexpiration, and thus allowing air to enter but notto escape from the pleural cavity (Fig. 6). Apositive pressure will continuously build up insidethe pleural cavity which will ultimately shift themediastinal structures to the opposite side, inter-fering with the haemodynamics of the respiratoryand cardiovascular systems. These changes areresponsible for the clinical picture usually seen intension pneumothorax which may be fatal if thetension is not relieved.

All the five cases reported here illustrate thetension quality of the pneumothorax bothclinically and radiologically. Because of the ratherdramatic clinical picture, manometric measure-ment was not attempted. A sudden rise in theintrapulmonary pressure is the usual precipitatingfactor in rupture of the cyst: the cause could be atrivial one, such as coughing or sneezing, thoughsometimes it may follow an increase in intra-abdominal pressure as in pregnancy or ascites.Rupture may occur spontaneously without anypredisposing factor. As a result, secondary hyda-tidosis of the pleura may result from seeding ofthe scolices into the pleural cavity (Deve, 1925;Tomalino, 1959). Pleural infection and empyemais frequently met with, as in cases 1, 2, and 5. It isquite possible that the infection reaches the spacevia a trans-bronchial spread: infection andempyema are always an additive insult to thepatient and increase the seriousness of the illness.

(a) (b)FIG. 6. Mechanism of echinococcal tension pneumothorax: (a) during inspiration; (b) during expiration.

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Since pulmonary hydatid cysts are liable to anumber of serious complications (Bakir, 1967), weshould not leave such cysts waiting for spon-taneous evacuation or natural cure. Coughing upthe cyst is usually associated with complications(Brown, 1958; Cleland, 1963), and in our opinioncomplete evacuation with a satisfactory naturalcure is exceptional.

Since the natural course in any single case can-not be foreseen, we strongly advocate surgicalremoval of any pulmonary hydatid cyst or sus-pected cyst as soon as it is discovered. The recentadvances made in thoracic surgery and anaesthesiaare an additional justification for a surgicalapproach. Earlier operation is easier and less de-vastating and a cure may be achieved by simpleenucleation of the cyst. Conservative measuressuch as needling and water-seal drainage with orwithout suction have only succeeded in alleviat-ing the distressing emergency symptoms but neverin closing the fistula and expanding the lung: webelieve that thoracotomy is mandatory after re-lieving the tension by these measures.Conventional diagnostic measures such as the

Casoni skin test, the Weinberg complement fixa-tion test, and an eosinophil count are not veryreliable when the cyst has ruptured or is infectedor calcified. Some features made us suspect anechinococcal nature or aetiology in our cases,such as the radiological shadow of a cyst wall incase 1 (Fig. lb), the multiple rounded shadows incase 2 (Fig. 2a), a previous history of a hydatidcyst removed from a kidney in case 3, the expec-

toration of salty fluid and a water-lily appearancein case 4, and an empty cavity with aspiration ofhydatid material in case 5. Other helpful diag-nostic points are urticaria, asthma, or anaphylaxisas the allergic manifestation of a ruptured cyst,while scolices in the pleural aspirate are patho-gnomonic. A tension pneumothorax with persistingbroncho-pleural fistula in an endemic area of thedisease should suggest a ruptured hydatid cyst.

We should like to thank Dr. Kwakab M. Hickmatfor Figure 6 illustrating the mechanism of tensionpneumothorax.

REFERENCESAgustoni, C. B., Taboada F. F., Berinstein, E., and Chorne, I. (1960).

767 Espontaneo hidatideo. Sem. med. (B. Aires). Fio Neumo-thorax, 116.

Bakir, F. (1967). Serious complications of hydatid cyst of the lung.Amer. Rev. resp. Dis., 96, 483.

Brown, C. J. 0. (1958). Surgical pathology of hydatid cysts of the lung.Postgrad. med. J., 34, 195.

Cleland, W. P. (1963). Pulmonary hydatid disease. In Chest Diseases.Vol. 2, p. 386. Ed. Perry, K. M. A., and Sellors, T. H. Butter-worths, London.

DuBose, H. M., Price, H. J., and Guilfoil, P. H. (1953). Spontaneouspneumothorax: medical and surgical management. Analysis of75 patients. New Engl. J. Med., 248, 752.

Kelly, T. D., and Izzi, N. (1959). Pulmonary hydatid disease in Iraq,with a review of history and life history of the parasite. J. Fac.Med. Baghdad, n.s., 1, 115.

Mills, M., and Baisch, B. F. (1965). Spontaneous pneumothorax. aseries of 400 cases. Ann. thorac. Surg., 1, 286.

Perry, K. M. A. (1939). On spontaneous pneumothorax. Quart. J.Med., n.s., 8, 1.

Sala de Pablo, J., and San Joaquin, R. Araiz (1954). Neumotoraxhidatidico. Cirurg. Ginec. Urol., 7, 171.

Sinclair, J. D. (1955). Spontaneous pneumothorax. A review of seventycases. N.Z. med. J., 54, 51.

Tomalino, D. (1959). Pleural complications of hydatidosis, pleuralhydatid accident, secondary pleural hydatidosis (study of 47personal observations). T6ra;., 8, 73.

Waddle, N. (1950). Pulmonary hydatid disease. A review of 478 casesreported in the Louis Barnett Hydatid Registery of the RoyalAustralasian College of Surgeons. Aust. N.Z. J. Suirg., 19, 273.

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pneumothoraxEchinococcal tension


doi: 10.1136/thx.24.5.5471969 24: 547-556 Thorax

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