SHIU: SPONTANEOUS INTERNAL BILIARY FISTULAE 969

ileal reduplications were described as ‘ granulation tissue with primitive epithelial structures similar to that observed in “ teratomata” and “ a primitive form of intestinal epithelium” ’. Whereas there is no explanation for the appearance of heterotopic tissue in reduplications, such primitive tissues and, indeed, cases of dual heterotopia lend weight to the suggestion of an origin in multi-potent cells sequestrated here during abnormal development.

There is no argument as to the treatment-it is excision of the reduplication and the adjoining gut, for to attempt to dissect the reduplication from the gut would be to inflict certain damage on the normal gut or its vessels. Only when a diverticulum has become ‘pinched off’ from the gut wall can such a course be contemplated. The only circumstances when this form of treatment is impracticable is where a massive part of the gut is reduplicated, when anastomosis of reduplication to normal channel must be considered. The case described by Levack (1963) is instructive in that it demonstrates that closure of a perforation in a reduplication is not effective-resection must be performed. The case described by Basu, Forshall, and Rickham (1960), where a second reduplication was overlooked, emphasizes that when one area of reduplication has been found, the whole gut must be examined very carefully to exclude such further pathology.

SUMMARY A rare case of massive rectal bleeding in a child is

described. The underlying pathology of the reduplica- tion with diverticula represents abnormality derived from two embryonic stages. The dual heterotopia described is excessively rare and supports the theory of an origin of heterotopic tissue in sequestrated pluripotent cells.

BIBLIOGRAPHY ANDERSEN, M. C., SILBERMAN, W. W., and SHIELDS,

BASU, R., FORSHALL, I., and RICKHAM, P. P. (1960), B Y . 3. BREMMER, J. L. (1944), Archs Path., 38, 132. BURIAN, J., and others (1958j, D t . Z . Verdau.-u. Stoffwechs

T. W. (1962), Archs Surgery, Chicago, 85, I.

surg., 47, 477.

Krankh., 18, 48. EDWARDS, H. (1929), BY. 3. Surg., 17, 7. FISHER, H. C. (IgSO), Archs Surg., Chicago, 61, 957. FITZ, R. H. (1884), Am. 3 . med. Sci., I 14, 30. GROSS, R. E. (1964), Surgery of Znfancy and Childhood.

LADD, W. E. (1937)~ Sth med.3., Nashville, 30, 363. LEVACK, J. H. (1963), Br. 3. Surg., 50, 805. OECONOMOPOULOS, C. T., and SWENSON, 0. (1962), 3. SWENSON, 0. (1958), Pediatric Surgery. New York:

TAYLOR, A. L. (1927),3. Path. Bact., 30, 415. VEENEKLAAS, G. M. H. (I952), Am.3 . Dis. Child., 83,500.

Philadelphia : Saunders.

Pediat., 60, 3.

Appleton-Century-Crofts Inc.

AN UNUSUAL CASE OF SPONTANEOUS INTERNAL BILIARY FISTULAE

BY M. H. SHIU

DEPARTMENT OF SURGERY, UNIVERSITY OF HONG KONG

SPONTANEOUS internal biliary fistulae are not surgical curiosities. Common causes include cholelithiasis, peptic ulceration, and malignant neoplasm. Wag- goner and Le Mone (1949)~ in their review of 819 cases, found that 51 per cent of such fistulae were cholecystoduodenal, 21 per cent cholecystocolic, and 19 per cent choledochoduodenal, with cholecysto- gastric, choledochogastric, cholecystocholedochal, and other types forming the rest. Occasionally a single fistula involves two different but adjacent parts of the alimentary canal, a well-known example being the cholecystoduodenocolic fistula (Puestow, I942 ; Pitman and Davies, 1962). The occurrence of two separate spontaneous fistulae involving different parts of the biliary tract in the same patient must be very rare, and a search of the literature has failed to reveal any recorded case. The following report concerns a patient who presented with two such fistulae, cholecystoduodenal and choledochoduodenal. Apart from its rarity, the case has also some features which appear to merit discussion.

CASE REPORT A 43-year-old male prisoner, serving a sentence for

possession of narcotics, was admitted to Queen Mary Hospital, Hong Kong, because of acute abdominal pain and haematemesis, under the diagnosis of bleeding peptic

ulcer. He gave a history of having had, on and off, epigastric pain for 20 years. The pain was often colicky in nature and accompanied by vomiting. It had usually been relieved by antacids, but recently it could only be relieved by heroin. Fourteen years previously, at the height of a severe attack, he was admitted to another hospital in which he underwent an operation. He bore an upper midline abdominal scar, but could not recall what the operation was, nor could his hospital record be obtained. He had been symptom-free since the operation until 8 years ago, when similar attacks again occurred. A year prior to admission he had had an episode of jaundice, diagnosed and treated as virus hepatitis in the prison hospital.

Examination on admission showed that he was very pale, but apart from the presence of some tenderness in the upper abdomen and the operation scar, there was nothing remarkable. He was managed under a con- servative regime for bleeding peptic ulcer, with sub- sidence of pain and no further haematemesis.

A barium-meal examination was then performed (Fig. I). The stomach was normal. The duodenal cap was smoothly outlined and contained a spherical filling defect which was constant in size, shape, and position. The second part of the duodenum appeared kinked, and here a blotch of barium was seen passing out to outline a small paraduodenal pocket. Air was seen in the biliary tree and later barium was visualized in the common bile-duct, hepatic ducts, and the cystic duct. The latter appeared dilated and the barium was arrested after filling two spiral turns of Hartmann’s pouch. The radiological findings thus suggested a cholecystoduodenal fistula,

970 BRIT. J. SURG., 1967, Vol. 54, No. 11, NOVEMBER

through which a large gall-stone had escaped into the duodenum, and, as there was no peptic ulcer, the haema- temesis was probably caused by the stone as it eroded

channel, then, did the air and barium enter the bile- ducts ?

The patient was operated on soon after the barium meal. Unfortunately the gall-stone could no longer be discovered, despite thorough palpation of the whole gastro-intestinal tract; it had probably passed out through the rectum. Many adhesions were encountered under the right lobe of the liver. After dividing these, a contracted, grossly thickened, and chronically inflamed gall-bladder was found, the fundus of which was stuck to the second part of the duodenum. Upon opening the gall-bladder, the lining epithelium was found to be haemorrhagic, oedema- tous, and partially denuded. A short fistulous tract (Fig. 2, see arrow), 1.5 cm. in diameter, led from the gall- bladder into the duodenum. No stones were seen. The opening of the cystic duct could not be identified; the region which had previously been the cystic duct and the free edge of the lesser omentum was replaced by a mass of dense fibrous tissue.

T o solve the mystery of the air and barium in the biliary tree, the second part of the duodenum was opened and inspected through a longitudinal incision on its anterior aspect. There was no gall-stone, nor duodenal ulcer, and the sphinctkr of Oddi was normal in appear- ance. But just 1.5 cm. superior to the sphincter orifice was found another opening (Fig. 3, see upper arrow), about 3 mm. in diameter and rather punched out in appearance. Through this opening, bile and a little debris were seen coming out. A bougie could easily be introduced into the common bile-duct through the sphincter of Oddi and made to come out again through this abnormal orifice, which was now realized to be a choledochoduodenal fistula. The two separate fistulae having been examined beyond doubt, the duodenotomy and then the cholecystoduodenal

F ~ ~ , I,-~arium meal showing a round filling defect (black arrow) in the duodenum due to a stone; fistula between the fundus of the gall-bladder and the duodenum; and air (white arrow) and barium in the intrahepatic bile-ducts.

FIG. 2.-Note the grossly thickened and inflamed wall of the gall-bladder. The arrow points at the cholecystoduodenal fistula leading into the second part of the duodenum.

through the intestinal wall. The air and barium in the bile-ducts could have made their entry via the chole- cystoduodenal fistula, but this seemed unlikely as the cystic duct was apparently blocked and neither air nor barium filled the interval between Hartmann’s pouch and the fundus of the gall-bladder. Through what

FIG. 3.-The second part of the duodenum has been opened. A bougie passes through the lower part of the common bile-duct, traversing the sphincter of Oddi (lower arrow) and a choledocho- duodenal fistula (upper arrow).

fistula were closed. Cholecystectomy was performed and a T-tube inserted into the common bile-duct for drainage.

The patient made an uneventful recovery. The bile grew Escherichia coli and Pseudomonas aeruginosa on bacteriological culture. It also showed actinomyces filaments on direct smear, though subsequent culture

SHIU: SPONTANEOUS INTERNAL BILIARY FISTULAE 97 1

failed to yield the fungus. Chloramphenicol and strepto- mycin were administered in accordance with the sensi- tivity report, and to these were added a course of penicillin. A postoperative retrograde cholangiogram showed mild dilatation of the intrahepatic bile-ducts, no residual stones, and free flow of contrast medium into the duodenum. The cholangitis having been controlled, the T-tube was removed at the end of 5 weeks, and the patient discharged soon afterwards.

DISCUSSION The clinical features of spontaneous internal

biliary fistulae have been reviewed by numerous authors (Borman and Rigler, 1937; Puestow, 1942; Pitman and Davies, 1962; Ong, Lee, Low, and Iu, 1962). The symptoms vary according to the primary condition (cholelithiasis, peptic ulceration, etc.) and to the organs involved by the fistulae (duodenum, stomach, colon, etc.). Charcot’s triad of pain, fever, and jaundice occurs in a large proportion of cases, due either to obstructive cholangitis prior to the establishment of a fistula, or to reflux cholangitis after- wards. In those cases due to cholelithiasis, Ong and others (1962) have observed that many of the patients remembered an unusually severe episode of pain, followed by complete relief, with subsidence of fever and jaundice soon afterwards. Alimentary haemor- rhage is occasionally a feature of spontaneous internal biliary fistulae. The present case was actually admitted under the diagnosis of bleeding peptic ulcer after a massive haematemesis. The bleeding was presumably due to erosion of blood-vessels in the bowel wall as the stone worked its way through. The case lends illustration to the difficulty sometimes met with in trying to diagnose clinically the cause of gastro-intestinal haemorrhage. Spontaneous internal biliary fistula is a rare but well-documented cause of such haemorrhage (Carlson, Gates, and Novacovich, 1955; Meeroff, Arlowski, Bilesio, and Subira, 1961).

Although the clinical features are numerous, the single most reliable diagnostic sign of a spontaneous internal biliary fistula is the radiological demonstra- tion of air or barium in the biliary tree. Depending on the site of the fistulous tract, the gall-bladder, common bile-duct, or the hepatic ducts will be out- lined. I n the present case radiographs showed a cholecystoduodenal fistula with air and barium also in the bile-ducts. This was thought to be rather unusual

as the occurrence of air in the bile-ducts due to cholecystoduodenal fistulae is known to be uncommon. Thus Marshall and Polk (1958) noted its presence in only I case out of their series of 30. One reason for this rarity may be that many of these fistulae close as soon as the stones have passed through. Another possible explanation may be blockage of the cystic duct after repeated episodes of severe cholecystitis which have culminated in the development of the fistula. Under such circumstances, air and barium can fill the gall-bladder but will be unable to enter the common bile-duct. For this reason the present case was suspected to have another abnormal channel apart from the cholecystoduodenal fistula, and, indeed, a choledochoduodenal fistula, quite distinct and sepa- rate, was discovered at operation. The blockage of the cystic duct, which had been demonstrated by the radiographs, was also confirmed.

SUMI\IARY A case of spontaneous double internal biliary

fistulae, cholecystoduodenal and choledochoduodenal, is reported. The presenting symptom is haemate- mesis. The radiological findings include a spherical filling defect in the duodenum, barium filling of the fundus of the gall-bladder via the duodenum, block- age of the cystic duct, and air and barium in the common bile-duct and hepatic ducts.

Acknowledgements.-I wish to thank Professor G. B. Ong for his advice and helpful criticism of this paper. My thanks are also due to Mr. James Ho who prepared the illustrations.

REFERENCES BORMAN, C. N., and RIGLER, L. G. (1937)~ Surgery, St

Louis, I , 349.

Surgery Gynec. Obstet., 101, 321. CARLSON, E., GATES, C. Y., and NOVACOVICH, G. (1955),

MARSHALL. S. F.. ~ ~ ~ P o L K . R. C. (1958), Surf. ClinsN. . _ _ I _ - Am., 38; 679. .

C. (1961), Prensa med. argent., 48, 2198.

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MEEROFF, M., ARLOWSKI, I., BILESIO, E. A., and SUBIRA,

ONG, G. B., LEE, T. C., Low, G., and Iu, I?. Y. (1962),

PITMAN, R. G., and DAVIES, A. (1962), Ibid., 50, 414. PUESTOW, C. B. (1942), Ann. Surg., 115, 1043. WAGGONER, C. M., and LE MONE, D. V. (1g49), Radiology,

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