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Image of the Month David M. Warshauer, Section Editor Question: A 74-year-old woman with a remote history of polycystic kidney disease and left renal transplantation presented with com- plaints of a chronic cough productive of irides- cent green sputum of 2 years’ duration. She reported gradually increasing bronchorrhea to the point where she would expectorate copi- ous amounts and described it as “very bitter tasting.” Multiple previous sputum cultures were negative for acid-fast bacilli, fungi, and bacteria other than normal flora. She had been treated with various courses of oral antibiotics, none of which altered her clinical course. Ad- ditionally, she reported a green discoloration to her tongue, which did not improve with ag- gressive oral hygiene (Figure A ). A 30-lb weight loss was noted, but she denied fever, chills, or abdominal discomfort. On physical examina- tion, she was found to be afebrile and cachec- tic with an irregular liver edge palpable 15 cm below the costal margin. Laboratories revealed a white blood cell count of 5.4 H11003 10 9 , hemo- globin of 11.2 g/dL, platelets of 120/mm 3 , total bilirubin of 0.8 mg/dL, and normal coagulation studies. A chest radiograph revealed left upper lobe scarring and a large subdiaphragmatic cal- cified hepatic cyst (Figure B ). A sputum gram stain was negative for polymorphonuclear leu- kocytes, but the culture of sputum obtained at this time revealed Bacteroides fragilis . What is the diagnosis? Look on page 385 for the answer and see the Gastroenterology website (http://www. gastrojournal.org) for more information on submitting your favorite image to Image of the Month. LCDR E DITH R. L EDERMAN , USNR, MC LCDR K ESHAV R. N AYAK , USNR, MC CAPT D ENNIS E. A MUNDSON , USN, MC Infectious Diseases Division Internal Medicine Department Pulmonary Division Naval Medical Center San Diego San Diego, California The views expressed in this article are those of the authors and do not reflect the official policy or position of the Department of the Navy, Department of Defense, or the United States Government. Dr. Lederman’s current affiliation is: Naval Medical Research Unit No. 2, Jakarta, Indonesia. © 2004 by the American Gastroenterological Association 0016-5085/04/$30.00 doi:10.1053/j.gastro.2003.06.006 GASTROENTEROLOGY 2004;126:7
Answer to the Image of the Month Question (page 7): Bronchobiliary Fistula Secondary to Polycystic Liver Disease Subsequent computed tomographic and HIDA-biliary scans confirmed the presence of a bronchobiliary fistula connecting a large subdiaphragmatic hepatic cyst to the patient’s right mainstem bronchus. Enhancement of the tongue on the HIDA-biliary scan was especially noteworthy (Figure C , arrow ). An attempt to surgically correct the fistula was planned; however, the patient expired secondary to thrombotic complications associated with a traumatic hip fracture. Bronchobiliary fistula (BBF) is a rare entity usually seen as a complication of conditions that create chronic subdia- phragmatic irritation and pressure. First described in the context of amoebic liver abscess, it has since been associated with pyogenic and tuberculous liver abscesses, hydatid liver disease, trauma, chronic pancreatitis, and choledocholithiasis. It may also be observed as the result of congenital abnormalities and postsurgical complications. We report the first case of BBF secondary to polycystic liver disease. Patients with BBF characteristically have insidious weight loss and chronic cough productive of copious amounts of bilious sputum. 1 Biloptysis is pathognomonic for the presence of a BBF. Sputum cultures may be unremarkable or contain typical biliary flora, as in our case. The most sensitive and noninvasive diagnostic test to confirm the presence of a BBF is the HIDA-biliary scan, 2 although computed tomographic scans, bronchoscopy, and fistulography have all been used with varying degrees of success. Surgical correction is the standard of care, although newer techniques using percutaneous catheters and stents are gaining popularity. 3 HIDA-biliary scans may be used for postintervention surveillance to ensure enduring success of the procedure. References 1. Al-Mezem SS, Al-Jahdali HH. Chronic cough due to bronchobiliary fistula. Respiration 1999;66:473–476. 2. Kinaci C, Akgul E, Sire D, Ersoy G, Dolapcioglu N, Beyci F. Scintigraphic detection of bronchobiliary fistula in a patient with bile in bronchial secretions. Clin Nucl Med 2001;26:359. 3. Deshmuks H, Prasad S, Patankar T, Patel V. Percutaneous management of a bronchobiliary fistula complicating ruptured amebic liver abscess. Am J Gastroenterol 1999;94:289–290. For submission instructions, please see the Gastroenterology website (http://www.gastrojournal.org). January 2004 CORRESPONDENCE 385
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