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Gastroesophageal reflux is quitecommon in infants and mostcan be treated with conserva-

tive measures. This report is of a new-born infant who presented with severegastroesophageal reflux, which re-quired surgical intervention. Aftersurgery, bile was seen in the endotra-cheal tube and investigation revealed abronchobiliary fistula. Such a condi-tion is rare; fewer than 20 cases havebeen reported in the literature.

CASE REPORT

A 35-year-old gravida 2, para 1

white woman was delivered of a termfemale infant after an uncomplicatedpregnancy. The infant’s birth weightwas 3325 g and Apgar scores were 9at 1 and 5 minutes. Mother and infantwere discharged home at 24 hours.The infant was readmitted to hospital2 days later because of poor feeding,regurgitation, mild respiratory distressand mild cyanosis. The child was pass-ing stool and there was no abdominaldistension.On admission to the neonatal in-

tensive care unit the infant was in nodistress and on physical examinationthe only abnormality was mildly in-

creased respiratory effort. A chest ra-diograph demonstrated early consol-idative changes suggestive of aspira-tion. Intravenous antibiotics weregiven. A cardiologist was consultedbecause of mild cyanosis. The electro-cardiogram and echocardiogram werenormal and the cardiac examinationgave normal results. Upper gastroin-testinal examination demonstratedmarked gastroesophageal reflux withno evidence of a tracheoesophagealfistula. Antireflux measures werestarted, but the infant continued tovomit and show signs of respiratorydistress. A 24-hour pH probe demon-

Case ReportÉtude de cas

BRONCHOBILIARY FISTULA: A CASE REPORT

James D. Fischer, MD

From the Department of Surgery, King Fahad National Guard Hospital, Riyadh, Saud Arabia

Presented at the Pacific Association of Pediatric Surgeons meeting, Singapore, May 1996

Accepted for publication Sept. 9, 1997

Correspondence to: Dr. James D. Fischer, Chairman, Department of Surgery, King Fahad National Guard Hospital, PO Box 22490, Riyadh, Saudi Arabia 11426

© 1998 Canadian Medical Association (text and abstract/résumé)

Fewer than 20 cases of bronchobiliary fistula have been reported in the literature. In this report a newbornfemale infant was referred for investigation of gastroesophageal reflux. Upper gastrointestinal endoscopyand 24-hour pH monitoring revealed severe reflux. A fundoplication was carried out. In the immediatepostoperative period bile was noticed coming from the endotracheal tube. Bronchoscopy revealed an ab-normal opening just to the left of the carina. Fistulography and HIDA scanning confirmed the presence ofa bronchobiliary fistula. A right thoracotomy was used to divide the fistula. The child made an uncompli-cated recovery. Pathological examination of the excised specimen demonstrated tissue most consistent withan esophageal origin.

Moins de 20 cas de fistule bronchobiliaire ont fait l’objet d’un rapport dans les écrits. Dans ce compterendu, un nouveau-né de sexe féminin a été soumis à une investigation pour reflux gastro-oesophagien.Une endoscopie oeso-gastro-duodénale et la surveillance du pH pendant 24 heures ont révélé un refluxgrave. On a procédé à fundoplicature. Au cours de la période qui a suivi immédiatement l’intervention, ona constaté que de la bile sortait du tube endotrachéal. Une bronchoscopie a révélé une ouverture anormaleimmédiatement à gauche de l’éperon trachéal. Une fistulographie et une scintigraphie de la vésicule biliaire(HIDA) ont confirmé la présence d’une fistule bronchobiliaire. On a procédé à une thoracotomie du côtédroit pour diviser la fistule. L’enfant s’est rétabli sans complication. L’examen pathologique du spécimenexcisé a démontré la présence de tissus dont la nature correspond le plus à une origine oesophagienne.

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BRONCHOBILIARY FISTULA

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CJS, Vol. 41, No. 6, December 1998 471

strated significant reflux in spite of ag-gressive medical measures. On the16th day of life a fundoplication wascarried out. In the immediate postop-erative period bile was noted in theendotracheal tube. At bronchoscopyan abnormal opening to the left of thecarina was noted (Fig. 1).Bronchography revealed a connec-

tion to the left lobe of the liver (bron-chobiliary fistula) (Fig. 2). HIDA scan -ning showed a connection betweenthe left lobe of the liver and the leftmain-stem bronchus, with normal flowof bile into the duodenum. At opera-tion, a Fogarty catheter was placed inthe fistula and the right chest wasopened through a posterolateral tho-racotomy incision in the fifth costal in-terspace. The fistula was easily identi-fied by palpation of the catheter, andafter removal of the Fogarty catheterthe fistula was excised from the regionof the left main-stem bronchus to thediaphragm (Fig. 3). Pathological ex-amination of the excised specimen re-vealed tissue most consistent withesophagus. The postoperative coursewas uncomplicated. The child fed welland was discharged 6 days after thesecond procedure. The child had nosymptoms at 36 months and was nor-

mal for size and development. Therewas no evidence of liver abnormality atfollow-up.

DISCUSSION

Bronchobiliary fistula is a rareanomaly; fewer than 20 cases havebeen reported in the literature. In thefirst case, reported in 1952,1 the childdid not survive, but in most cases sincethen the children have been treatedsuccessfully with surgical excision2–6 aswas the infant reported here. Thechild in this case report appeared tohave severe gastroesophageal reflux,and this has also been reported in theliterature.6 Although it is usually anisolated anomaly, bronchobiliary fis-tulae have been reported in associa-tion with esophageal atresia and tra-cheoesophageal fistula7 and withbiliary atresia.8

The embryology of this rare anom-aly is still unclear.1 One theory is thatthese fistulae develop from the unionof an anomalous bronchial bud with

an anomalous bile duct. The fact thatsome fistulae are ringed by cartilagesimilar to bronchus gives support tothis concept. The second theory isthat the fistula represents a duplicationof the gastrointestinal tract. Supportfor this theory is the fact that some fis-tulae, as in this case, are found, histo-logically, to be of esophageal origin.

FIG. 1. Bronchoscopic view shows the fistula (ar-rowhead) as an abnormal opening to the left ofthe carina. FIG. 2. A bronchogram showing the bronchobiliary fistula (arrowheads).

FIG. 3. Operative view of the fistula during exci-sion.

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FISCHER

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472 JCC, Vol. 41, No 6, décembre 1998

Although most reported casesshowed the fistula arising from theright side of the tracheobronchial tree,in the present case it arose from the leftside. All cases reported in the literatureterminated in the left lobe of the liver.Investigation of a child with a sus-

pected bronchobiliary fistula shouldinclude bronchoscopy, bronchogra-phy and HIDA scanning. It is impor-tant to ascertain whether the affectedlobe of the liver drains normally, ashepatic resection of the involved lobemay be necessary in addition to resec-tion of the fistula.6 A right thoraco-tomy can be used in every case, and inthe present case a Fogarty catheterplaced via bronchoscopy was helpfulin identifying the fistula. Bronchobil-iary fistula should be suspected in the

child with bilious reflux without evi-dence of obstruction.

References

1. Neuhauser EB, Elkin M, Landing B.Congenital direct communication be-tween biliary system and respiratorytract. Am J Dis Child 1952;83:654-9.

2. Wagget J, Stool S, Bishop HC, KurtzMB. Congenital broncho-biliary fis-tula. J Pediatr Surg 1970;52:566-9.

3. Weitzman JJ, Cohen SR, Woods LOJr, Chadwick DL. Congenital bron-chobiliary fistula. J Pediatr 1968; 73:329-34.

4. Sane SM, Sieber WK, Girdany BR.Congenital bronchobiliary fistula.

Surgery 1971;69:599-608.

5. Lindahl H, Nyman R. Congenitalbronchobiliary fistula successfullytreated at the age of three days. J Pedi-atr Surg 1986;21:734-5.

6. Gauderer MW, Oiticica C, Bishop HC.Congenital bronchobiliary fistula: man-agement of the involved hepatic seg-ment. J Pediatr Surg 1993;28:452-5.

7. Kalayoglu M, Olcay I. Congenitalbronchobiliary fistula associated withesophageal atresia and tracheo-esophageal fistula. J Pediatr Surg 1976;11:463-4.

8. Chan YT, Ng WD, Mak WP, KwongML, Chow CB. Congenital bron-chobiliary fistula associated with biliaryatresia. Br J Surg 1984;71(3):240-1.

This list is an acknowledgement of books and other media received. It does not precludereview at a later date.

Cette liste énumère les livres et autres documents reçus. Elle n’en exclut pas la critiqueà une date ultérieure.

Atlas of Retinal and Vitreous Surgery. Travis A. Meredith.244 pp. Illust. Mosby Inc., St. Louis; Harcourt Brace & Co.Canada, Ltd., Toronto. 1999. Can$263. ISBN 0-8151-2834-7

Concise Surgery. An Illustrated Guide. Edited by Kevin Laf-ferty and John Rennie. 498 pp. Illust. Arnold, a member of the

Hodder Headline group, London, UK; Oxford University PressCanada, Toronto, 1998. Can$59.50. ISBN 1-340-70611-2

Medical Management of the Surgical Patient. 2nd edition.Edited by Geno J. Merli and Howard H. Weitz. 410 pp. Illust.W.B. Saunders Company, Philadelphia; Harcourt Brace & Co,Canada, Ltd., Toronto. 1998. Can$59.95. ISBN 0-7216-6976-X

Oxford Textbook of Sports Medicine. 2nd edition. Edited byMark Harries, Clyde Williams, William D. Stanish and Lyle J.Micheli. 957 pp. Illust. Oxford University Press, Oxford, UK;Oxford University Press Canada, Toronto. 1998. Can$269.50.ISBN 0-19-262717-1

Books and Other Media ReceivedLivres et autres documents reçus

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