Bronchobiliary Fistula Detected by

Michael G. Velchik, Gerald M. Roth, William Wegener, and Abass Alavi

Hospital of The UniversityofPennsylvania, Philadelphia, Pennsylvania

the symptoms recurred. She re-presented to her physician who We present a case of a bronchobiliary fistula initially de witnessed the patient coughing up bilious material. Decreased tected by hepatobiliaryscintigraphy. The patient developed breath sounds and dullness to percussion were noted at the bilioptysis 18 mo after undergoing a right hepatic lobec right lung base. Her abdomen was slightly distended and tomy and resection of the common for tholangio shifting dullness could be elicited. carcinoma.The procedurewas complicatedby the devel Laboratoryvalueson admissionshowedan elevatedtotal opment of a subphrenic abscess that required percuta bilirubin of 2.0 mg/dl (normal: 0.0—1.2),with a conjugated neous biliary drainage. fraction of0.7 mg/dl (0.0-0. 1).The GGTP measured 449 UI 1(0—40)and the alkaline phosphatase measured 630 U/I (35— J NucI Med 1991; 32:136—138 125).The transaminase, LDH, and CPK levels were all within the normal range. She had an elevated white blood cell count of 27.4 K/@J,with 92% polymorphonuclear cells, 7% band cells, and 1% lymphocytes on a manual leukocyte differential ronchobiliary fistula is a rare disorder, which is count. Multipleelectrolyteabnormalitieswerealsonotedwith characterized by bilioptysis that may be congenital in a sodium level of 123 mmol/l (133—143), a chloride level of origin, but which is more commonly associated with 89 mmol/l (97—107),a CO2 content of 21 mmol/l (24—32),a glucoselevelof 120mg/dl (70—110),a blood urea nitrogenof trauma, surgery, or with hepatic abscess and/or para 24 mg/dl (10—20),and a creatinine level of 0.5 mg/dl (0.6— sitic disease. Although surgery has been the con 1.0). ventional treatment, recent evidence suggests that per Her chest radiographrevealedan elevated right hemidia cutaneous drainage may also be a valid therapeutic phragm and an infiltrate at the baseofthe right lung consistent option (1). We present a case of bile extravasation with either a subpulmonic effusion or a subphrenic abscess. associated with a bronchobiliary fistula detected by An abdominal radiographshowed mild ascitesand possible cholescintigraphy in a patient with bilioptysis. The gastric outlet obstruction. A hepatobiliary scan, performed physiologic nature ofthe hepatobiliary scan enabled the with 5 mCi of technetium 99m-DISIDA, in order to assess detection, localization, and the differentiation of the the integrity of the patient's hepatobiliary system, revealed a bronchobiliary fistula from bile reflux, the more com perihepatic collection of bile with fistulous tracking through mon cause of bile emesis. the diaphragm to the trachea (Fig. 1 A—C).Percutaneous transhepatic (PTC) confirmed the presence of a collection of bile posterior to the liver with fistulous CASE REPORT tracking through the lung and demonstrated its source to be A 38-yr-old white woman presented complaining of a leak from the anastomotic site (Fig. 1D). A percutaneous “coughingup bile―18 mo after having undergone a right biliary drainage catheter was placed and put on suction. hepatic lobectomy with resection of the common bile duct The etiologyof this bronchobiliaryfistula is unclear but and creation of a Roux-en-Y left hepatico-. Path may be related to the percutaneous drainage ofthe subphrenic ologic examination of the resected specimens revealed the abscessthat the patient developedpostoperatively.Immedi presence of mucinous adenocarcinoma with involvement of ately after the percutaneous drainage, the patient developed a the liver capsule by local extension. Her postoperative course right pneumothorax, indicating that the pleural space and was complicated by a subphrenic abscess, which necessitated perhaps the lung had been traversed before the needle pierced percutaneous drainage. Immediately after the procedure, the the diaphragm, allowing the perihepatic fluid collection to patient developed a right pneumothorax and a chest tube was enter the pleural space.A drainagecatheter was placed into placed. She received two courses ofchemotherapy (5 FU) and the left hepatic duct percutaneously and advanced through 5940 total rads oflocal radiotherapy. the ductal system and then through the leak in the anastomotic She remainedasymptomaticfor 1 mo, after which time site until it reached the collection of bile, at which point it was attached to suction. A few days later, a second catheter ReceivedMar.5, 1990;revisionacceptedJun.11. 1990. was percutaneously introduced into the left hepatic duct and Forreprintscontact:MichaelG.Velchick,MD,DepartmentofRadiOlOgy, DivisionofNuclearMedicine,HospitalofTheLkiiversityof PA,3400Spruce advanced across the anastomotic site to end in the jejunum St./G1, Philadelphia,PA19104. and act as a stent.

136 The Journal of •Vol. 32 •No. 1 •January 1991 A B C D

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FIGURE 1 (A) Hepatobiliaryscan. Anterior projection(60 mm).An abnormalCOlleCtiOnof radioactivityis seen overlyingthe right lobe of the liver (arrowhead)that tracts up over the dome of the liver and enters the bronchialtree (arrow).(B) Hepatobiliaryscan. Rightlateralprojection(60mm).AnabnormalcollectionofradioactMtyisseenposteriorto therightlobeoftheliver(arrowhead) thattractsupoverthedomeof theliverandentersthebronchialtree(arrow).(C)Hepatobiliaryscan.Posteriorprojection(85 mm). An abnormal collection or radioactivity is seen overlying the right lobe of the liver (arrowhead) that tracts up over the dome of the liver and enters the bronchial tree (arrow). (D) Percutaneoustranshepatic cholangiography.Percutaneous transhepatic cholangiogram performed through the left hepatic duct confirms the presence of the bronchobiliary fistula.

DISCUSSION rity of the hepatobiliary system and detect space occu Although bronchobiliary fistula was first described as pying lesions such as bilomas. It can also determine a complication ofhydatid disease ofthe liver (2) and is whether or not the latter communicate with the biliary most often associated with parasitic disease worldwide, system (10), which is not always possible with CT or in America it more commonly occurs in association US, but which has important prognostic and th@rapeu with trauma, surgery, or hepatic abscess formation (3— tic implications. The “physiologic―route of bile flow is 6). A combination ofhepatic infection (hydatid disease, demonstrated using cholescintigraphy, including the amebic, pyogenic) and biliary obstruction is usually presence or absence of bile leaks, fistulas, and obstruc present in the nontraumatic cases. Biliary obstruction tion. In contradistinction, cholangiography is a “non may result in cholangitis with the subsequent develop physiologic―study that requires a pressurized injection ment of an intrahepatic abscess which progressively ofcontrast material into the biliary tree, accessto which ruptures into the right pleural space. Early diagnosis may be difficult in patients with blunt trauma. and treatment is essential due to the high associated Cholescintigraphy has been found to be a sensitive morbidity and mortality. usually consists of a noninvasive technique for the detection and localiza combination of biliary drainage and treatment of the tion of bile leaks (11—18).The case presented here underlying condition. illustrates the important role that hepatobiliary scintig In rare cases, bronchobiliary fistula may be of con raphy plays in the detection and localization of bile genital origin, usually involving the left biliary radicals leaks. Our experience was similar to other previous and the right main stem bronchus ( 7,8). Congenital reports, in that CT was not especially helpful in making bronchobiliary fistula may be due to duplication of the the diagnosis. Cholescintigraphy, however, identified upper or union of an anomalous the fluid as bile and demonstrated the bronchobiliary bronchial bud with an anomalous bile duct (9). fistula. Patients often present with bile stained sputum (bil Interventional has played an increasing ioptysis), which is pathognomonic, and may have fever, role in the percutaneous treatment of abscesses, fluid dyspnea, cholangitis, jaundice, and sepsis. The chest collections, and fistulas that previously required sur radiographusuallyshowsopacification ofthe right lung gery. In this era ofDRGs, this is an important advantage basedue to pleural effusion and/or bronchiolitis. with respect to cost-effective treatment and patient Computed (CT)and ultrasonography morbidity. Finally, cholescintigraphy may be used in (US) are often non-diagnostic. Bronchoscopy and bron order to evaluate the effectiveness of therapy, in this chography usually fail to delineate the fistula. If access case the closure of the fistula and bile leak. Important exists through an external component of the fistula, a principles of therapy include the relief of obstruction pressure contrast injection (fistulogram) may be per and resolution ofany collections by percutaneous drain formed. However, most bronchobiliary fistulas are en age. tirely internal. Percutaneoustranshepatic cholangiog raphy or endoscopic retrograde cholangiography REFERENCES (ERCP)areinvasiveproceduresthat mayfailto detect 1. SchwartzML, Coyle Mi, AldreteiS, Keller F5. Bronchobili small fistulas. Cholescintigraphy can evaluate the integ ary fistula: complete percutaneous treatment with biliary

Bronchobiliary Fistula •Velchik et al 137 drainageand stricture dilation. l988;l68:751—752. Krishnamurphy 01. Detection ofbile leakagefrom traumatic 2. Peacock TB. Case in which hydatids were expectorated and right hepatic duct laceration with technetium-99-m-DISIDA one of suppuration of a hydatid cyst of the liver communi cholescintigraphy. Clin NucI Med 1987;12:589—591. cating with the lungs. Edin Med Surg J 1850;74:33—46. 12. Zeman RK, Lee CH, Stahl R. Strategy for the use of biliary 3. Gugenheim J, Ciardullo M, Traynor 0, Bismuth H. Bron in noniatrogenic biliary trauma. Radiology chobiliaryfistulasin adults. Ann Surg 1988;68:90—94. l984;l51:771—777. 4. Oschner A, Dc Bakey M, Murran 5. Pyogenic abscess of the 13. Sty JR. Starshak Ri, Hubbard AM. hepatobil liver an analysis of47 cases with review ofthe literature. Am iary imagingin the detection oftraumatic biliarytract disease J Surg 1938;40:292—3l9. inchildren.PedRadioll982;12:l15—118. 5. Morton ii, PhillipsEW. Bronchobiliaryfistula. reviewof the 14. PrakashR, Jena A, Behari V. Chopra MK. Intrahepatic bile recorded cases other than those due to Echinococcus and leak producing delayed accumulation of technetium-99-m- amebic abscess.Ann Surg 1928;l6:697—754. DISIDA in an amebic liver abscess. Clin Nucl Med 6. Akinoglu A, BilginI, Erkocak EU. Surgical management of l986;l1:360—361. hydatid diseaseofthe liver. Can J Surg 1985;28:l71—174. 15. SavitchI, EsserJD,SpringoloE,KewMC. Demonstrationof 7. Neuhauser EBD, Elkin M, Landing B. Congenital commu posttraumatic bile leak using Tc-99m-DISIDA. Clin NucI nication between the biliary system and respiratory system. Med 1986;l l(l2):873—874. Am J Dis Child l952;83:654—659. 16. WeissmannHS,ChunKJ,FrankM. Demonstrationoftrau 8. Chang CN, Giulian BB. Congenital bronchobiliary fistula. matic bile leakage with cholescintigraphy and ultrasonog Radiology1985;l56:82. raphy.Radiologyl979;l33:843—847. 9. Sane SM, Sieber WK, Girdany BR. Congenital bronchobiliary 17. Henderson RW, Telfer N, Halls JM. Gastrobiliary fistula: fistula.Surgery197l;69:599—608. pre- and post-operative assessment with Tc-99m-PIPIDA. 10. KuniCC,KlingensmithWC,KoehlerJ,FritzbergAR. Corn AJR 198l;l37:163—l65. munication of intrahepatic cavities with bile ducts: demon 18. Rienzo Ri, Tyler G, Morel DE. Sonographic and scinti stration with Tc-99m-diethyl IDA imaging. Clin Nucl Med graphic detection of a bile leak in a post- 1980;5:349—351. patient. Clin NucIMed l983;8:480—482. I I. SalamM, GlowniakJV, Vetto RM, JarobeJE, HainesJE,

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