Biliary-bronchial fistula demonstrated by endoscopic retrograde cholangiography LAURENCE WATKINS,* B SC; IGOR LAUFER,t md, frcp[c]; Geoffrey Evans,J mb, frcs; J. Edwards Mullens,$ md, frcs[c]

Summary: Endoscopic retrograde Biliary-bronchial fistula is a rare com¬ cholangiography is valuable in the plication of liver abscess.1,2 The diag¬ evaluation of biliary tract disorders. A nosis is usually made from a history of the coughing up of bile ("biloptysis"); 50-year-old Italian woman developed biloptysis 1 year after radiographic demonstration of the fis¬ cholecystectomy because of intrabiliary tulous tract is rare preoperatively. We rupture of a hydatid cyst with report a case in which the pathway of the fistulous tract was demonstrated by secondary infection, which resulted in intrathoracic rupture and communication with the bronchial tree.

Endoscopic retrograde cholangiography showed the cause and pathway of the fistulous tract by outlining the biliary tree, abscess cavity and communication with the right upper lobe bronchus. This technique appears to be well suited to the investigation of patients with biliary-bronchial

endoscopic retrograde cholangiography.

Case report

bercle bacilli. A chest radiograph showed right pleural effusion with elevation of the right diaphragm, consolidation in the anterior segment of the right upper lobe, and a rounded rim of calcification in the right lobe of the liver just below the diaphragm. This rim was also evident on the preoperative chest film and corresponded in location to the firm area in the liver described by the surgeon. On the 3 rd hospital day the sputum became bright yellow and tests for bili¬ rubin and bile acids were positive. A chest radiograph showed an air-fluid level within the calcified lesion in the liver. Bronchoscopy showed bile in the right upper lobe bronchus. A broncho¬ gram did not demonstrate a communicaa

A 50-year-old woman who had immigrated to Canada from Italy 20 years before was admitted to her local hospital with right upper quadrant pain and jaundice. She had previously been well. The gallbladder was not visualized by intra¬ fistula. venous cholangiography. A presumptive diagnosis of cholelithiasis was made and a cholecystectomy performed; no stones were found in the gallbladder. A firm Resume: Fistule biliobronchique area was noted in the right lobe of the demontree par cholangiographie liver. An operative cholangiogram showed retrograde endoscopique no evidence of stones but the right hepatic duct was not filled; a postoperative T-tube La cholangiographie retrograde had similar features. The cholangiogram endoscopique est une methode T-tube was removed after 6 weeks. She was then well for 13 months, until precieuse pour diagnostiquer les the sudden onset of right anterior chest troubles des voies biliaires. Une pain and shortness of breath. Pneumonia Italienne de 50 ans presenta des was diagnosed and she was treated with expectorations de bile 1 an apres antibiotics but her symptoms did not subavoir subi une cholecystectomie, side. She began to produce bloody sputum par suite de la rupture d'un kyste and was readmitted to hospital. The pre¬ avec infection secondaire, hydatique sumptive diagnosis was pulmonary em¬ laquelle rupture intrathoracique avait bolism and she was treated with heparin etabli la communication avec I'arbre intravenously. The chest pain decreased but she still had daily fever, unresponsive bronchique. Une cholangiographie to penicillin. When clindamycin and genta¬ retrograde endoscopique revela la micin were administered there was some cause et la voie du trajet fistuleux, en clinical improvement. Two weeks later mettant en evidence les voies biliaires, la cavite de I'abces et la communication she started to produce copious purulent sputum. A chest radiograph revealed a avec la bronche du lobe superieur right pleural effusion and thoracentesis droit. Cette methode radiographique 40 ml of blood-stained fluid. The yielded convient parfaitement a I'observation volume of purulent sputum increased to de malades porteurs de fistule approximately 1 litre per day for 3 days and she was transferred to McMaster Uni¬ biliobronchique. versity Medical Centre. She was pale and obese and was cough¬ ing frequently. The sputum contained fresh blood and later became rust-coloured and frothy. The respiratory rate was 24/ min. The lungs were dull to percussion and the breath sounds were decreased at the right lung base. Abdominal examina¬ tion yielded no abnormalities. The ab¬ From the departments of surgery and normal laboratory findings were as fol¬ radiology, McMaster University Medical Centre lows: leukocyte count, 14.2 x 109// with 71% neutrophils and 2% eosinophils; FIG. 1.Intravenous cholangiogram ?Medical student tAssistant professor of radiology erythrocyte sedimentation rate, 133 mm/h; with tomography: common bile duct ^Associate professor of surgery results of liver function tests, mildly ab¬ (arrowheads) and air-containing cavity Reprint requests to: Dr. Igor Laufer, normal. Sputum cultures grew Escherichia with calcified wall in right lobe of liver of Department radiology, McMaster University coli and a Klebsiella species but no tu- (arrow). Medical Centre, Hamilton, ON L8S 4J9 868 CMA JOURNAL/NOVEMBER 8, 1975/VOL. 113

tion between the bronchial tree and the

liver. Tuberculin skin test, Casoni's skin test, Entamoeba histolytica hemagglutination, gel diffusion and Echinococcus comple¬ ment fixation tests all yielded negative re¬ sults. Intravenous cholangiography showed air in the biliary ducts and within the cal¬ cified lesion in the liver (Fig. 1); the common bile duct was of normal calibre and contrast material drained freely into the duodenum. Endoscopic retrograde cholangiography showed a bile duct of normal calibre; contrast material entered the cavity in the right lobe of the liver through a stenosed branch of the right hepatic duct (Fig. 2), then tracked through the pleural cavity and outlined the right upper lobe bronchus (Fig. 3). Operation revealed a stenotic branch of the right hepatic duct leading to an abscess cavity with a calcified wall in the right lobe of the liver. Through a hole in the right leaf of the diaphragm air escaped into the abdomen, indicating a communication with the bronchial tree. The abscess cavity was drained of inspissated bile and granulation tissue that yielded Klebsiella and Enterobacter organ¬ isms on culture. Histologic examination showed no evidence of an echinococcal

the right pleural cavity. Postoperatively her cough gradually subsided and the drains were removed. A postoperative chest radiograph showed complete clear¬ ing of the right pleural effusion.

Discussion

Endoscopic retrograde cholangio¬ graphy has proved to be of value in evaluating the biliary tract, particularly in the jaundiced patient,3,4 and Classen and colleagues5 have used it postopera¬ tively to evaluate surgical fistulas be¬ tween the biliary tract and duodenum. The technique requires only local anes¬ thesia for the throat and sedation with diazepam, given intravenously. A sideviewing duodenoscope is passed into the duodenum and the papilla of Vater is located and cannulated. By manipulating the tip of the endoscope and cannula, one can selectively cannulate the pancreatic duct or the common bile duct and thus obtain excellent ra¬ diographic detail of these ducts.6 This appears to be the first reported case in which endoscopic retrograde cholangiography has been used to cyst. Drains were left in the abscess cavity, demonstrate a biliary-bronchial fistula. right hepatic and common bile ducts and The diagnosis of biliary-bronchial fis¬

FIG- 2.Endoscopic retrograde cholangiogram, lateral view: narrowing of branch of right hepatic duct (arrowheads) and extravasation of contrast material into calcified lesion in right lobe of liver (arrow).

tula is

usually made clinically in a pa¬ biloptysis. This is generally a complication of a liver abscess that ruptures through the diaphragm into the pleural cavity. Of the usual causes of liver abscess, namely pyogenic, amebic and echinococcal, the latter two are more commonly complicated by biliary-bronchial fistula.1 The Italian origin of this patient, the rim of cal¬ cification and the firm area in the right lobe of the liver noted at the original operation all suggested an echinococcal cyst. These cysts are known to rupture into the biliary tree,7 where they may cause either no complications, or ob¬ structive jaundice or suppuration.8 This patient's original illness, characterized by right upper quadrant pain and jaundice, was probably due to intrabiliary rupture of the cyst, whereas the subsequent events were due to second¬ tient with

ary infection of the cyst. Intrathoracic rupture of the abscess may have been spontaneous or may have been induced by inadvertent puncture of the liver during thoracentesis because of the

elevated right diaphragm. The negative results of Casoni's skin test and the complement fixation test can be explained by the calcification of the cyst, which indicates that it had been inactive.9 In addition, secondary infection may destroy the membrane

FIG. 3.Contrast material escaping through hole in diaphragm (white arrows) and outlining branches of right upper lobe bronchus (black arrows).

CMA JOURNAL/NOVEMBER 8, 1975/VOL. 113 871

Beclo vent Additional prescribing information Dosage and administration (cont'd.) In the presence of excessive mucus secretion, the drug may fail to reach the bronchioles. Therefore, if an obvious response is not obtained after ten days, attempts should be made to remove the mucus with expectorants and br with a short course of systemic corticosteroid treatment. Careful attention must be given to patients previously treated for prolonged periods with systemic corticosteroids, when transferred to Beclovent. Initially Bec/ovent and the systemic steroid must be given concomitantly while the dose of the latter is gradually decreased. The usual rate of withdrawal of the systemic corticoid is the equivalent of 2.5 mg of prednisone every four days if the patient is under close observation. If continuous supervision is not feasible, the withdrawal of the systemic steroid should be slower, approximately 2.5 mg of predriisone (or equivalent) every ten days. If withdrawal symptoms appear, the previous dose of the systemic drug should be resumed for a week before further decrease is attempted. There are some patients who cannot completely discontinue the oral corticosteroid. In these cases, a minimum maintenance dose should be given in adddition to Beclo vent Inhaler. Contraindications Active or quiescent untreated pulmonary tuberculosis, or untreated fungal. bacterial and viral infections, and in children under six. Status asthmaticus and in patients with moderate to severe bronchiectasis. Warnings In patients previously on high doses of systemic steroids, transfer to Beclovent Inhaler may cause withdrawal symptoms such as tiredness, aches and pains, and depression. In severe cases, acute adrenal insufficiency may occur necessitating the temporary resumption of systemic steroids. "The development of pharyngeal and laryngeal candidiasis is cause of concern because the extent of its penetration of the respiratory tract is unknown. If candidiasis develops the treatment should be discontinued and appropriate antifungal therapy initiated" Precautions 1 The replacement of a systemic steroid with Beclovent Inhaler has to be gradual and carefully supervised by the physician. The guidelines under Dosage and Administration should be followed in each case. 2 Unnecessary administration of drugs during the first trimester of pregnancy is undesirable. Corticosteroids may mask some signs of infection and new infections may appear. A decreased resistance to localized infection has been observed during corticosteroid therapy. During long-term therapy, pituitary-adrenal function and hematological status should be periodically assessed. 3 Fluorocarbon propellants may be hazardous if they are deliberately abused. Inhalation of high concentrations of aerosol sprays has brought about cardiovascular toxic effects and even death, especially under conditions of hypoxia. However, evidence attests to the relative safety of aerosols when used properly and with adequate ventilation. 4 It is essential that patients be informed that Beclo vent Inhaler is a preventive agent, must be taken at regular intervals, and is not to be used during an asthmatic attack. 5 There is an enhanced effect of corticosteroids on patients with hypothyroidism and in those with cirrhosis. 6 Acetylsalicylic acid should be used cautiously in conjunction with corticosteroids in hypoprothrombinemia. 7 Patients should be advised to inform subsequent physicians of the prier use of corticosteroids. Adverse reactions No major side-effects attributable to the use of recommended doses of Beclo vent Inhaler have been reported. No systemic effects have been observed when the daily dose was below 1 mg (twenty puffs). Above this dose, reduction of plasma cortisol, indicating adrenocortical suppression, may occur. Therapeutic doses may cause the appearance of Candida albicans in the mouth and throat. The replacement of systemic steroids with Beclovent Inhaler may unmask symptoms of allergies which were previously suppressed by the systemic drug. Conditions such as allergic rhinitis and eczema may thus become apparent during Beclovent therapy after the withdrawal of systemic corticosteroids. Symptoms and treatment of overdosage Overdosage may cause systemic steroid effects such as adrenal suppression and hypercorticism. Decreasing the dose will abolish these side-effects.

and the daughter cysts, resulting in a negative skin test.10 The echinococcal hemagglutination test, the most sensitive technique for detecting echinococcal infection1' was not performed. The presence of a biliary-bronchial fistula can be inferred from biloptysis. Occasionally the fistulous tract has been shown preoperatively by percutaneous transhepatic cholangiography,'2'4 but this technique is not recommended in these patients because of the risk of disseminating infection. The fistula is generally not demonstrated by bronchography. Therefore, until the development of endoscopic retrograde cholangiography, there was no safe and reliable technique for the anatomic demonstration of the fistula; this technique is ideal because it avoids the risks associated with puncturing the liver and it provides accurate preoperative anatomic detail. Although biliary-bronchial fistula is rare, this case illustrates the value of endoscopic retrograde cholangiography in evaluating abnormalities of the biliary tract. We wish to thank Dr. Harry Richardson of the department of bacteriology for reviewing the manuscript, and Miss Carole Johnstone for her secretarial assistance.

References 1. AMLR-JAHED AK, SADRIEH M, FARPOUR A,

et al: Thoracobilia: a surgical complication of hepatic echinococcosis and amebiasis. Ann Thorac Surg 14: 198, 1972 2. RUBIN RH, SWARTZ MN, MALT R: Hepatic abscess: changes in clinical, bacteriologic and therapeutic aspects. Am I Med 57: 601, 1974 3. KASUGA! T, KUNO N, Kizu M, et al: Endoscopic pancreatocholangiography. II. The pathological

endoscopic

pancreatocholanglo-

gram. Gastroenterology 63: 227, 1972 4. OGosm K, NIWA M, HARA Y, et al: Endoscopic pancreatocholangiography in the evaluation of pancreatic and biliary disease. Gastroenterology 64: 210, 1972 5. CLAsSEN M, FRUHMORGAN P, Kozu T, et al:

Endoscopic-radiologic demonstration of billodigestive fistulas. Endoscopy 3: 138, 1971 6. KAsUGAI T, KUNO N, KoBAYAsHI 5, et al: Endoscopic pancreatocholangiography. I. The normal endoscopic pancreatocholangiogram. Gastroenterology 63: 217, 1972 7. HARRis JD: Rupture of hydatid cysts of the liver into the biliary tracts. Br I Surg 52: 210, 1965 8. AL-HAsHIMs HM: Intrabiliary rupture of hydatid cyst of the liver. Br I Surg 58: 228, 1971 9. KAGAN IG: A review of serological tests for the diagnosis of hydatid disease. Bull WHO 39: 25, 1968 10. ALasilo K, HOLM C, NYSTROM G, et al: Biliobronchial fistula secondary to echinococcus abscess of the liver. Acta Chir Scand 138: 90, 1972 11. MArossiAN RM, KANE GJ, CHANTLER SM,

Supplied Bec/ovent Inhaler is a metered-dose aerosol delivering 50 micrograms of beclomethasone dipropionate with each depression of the valve. There are two hundred doses in a container. Official product monograph on request.

AS AIIen&Hanburys 11 Toronto, Canada

874 CMA JOURNAL/NOVEMBER 8, 1975/VOL. 113

et al: The specific immunoglobulin in hydatid disease. Immunology 22: 423, 1972

12. FLEMMA RJ, ANLYAN Wi, DURHAM NC: Tu-

berculous bronchobiliary fistula: report of an unusual case with demonstration of fistubus tract by percutaneous transhepatic cholangiography. I Thorac Cardiovasc Surg 49: 198, 1965 13. MIwA 5, WADA T, SOGAwA S, Ct al: (Case of liver abscess with biliobronchial fistula observed by percutaneous cholangiography.] Jap I Gastroenterol 69: 1320, 1972 14. VAN DE WavER KH, LEICHT E: Nachweis einer billo-bronchialen Fister durch perkutane transhepatische Cholangiographie. Fortschr Geb Roentgenstr Nuklearmed 109: 106, 1968

Biliary-bronchial fistula demonstrated by endoscopic retrograde cholangiography.

Endoscopic retrograde cholangiography is valuable in the evaluation of biliary tract disorders. A 50-year-old Italian woman developed biloptysis 1 yea...
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