Choledochoduodenal fistulas complicating duodenal ulcer

Choledochoduodenal Fistulas Complicating Duodenal Ulcer C,UILLERMO McEWAN-ALVARADO, M.D.,* a n d DONALD N. DYSART, M.D.

HE FIRST REPORT Of b i l i a r y fistula is a t t r i b u t e d to T h i l e s u s in 1670.1 I n 1861, F r e i r i c h s c i t e d several cases,'-" a n d in 1896, N a u n y n a r e p o r t e d 384 cases of s p o n t a n e o u s b i l i a r y fistulas, of w h i c h 184 were e x t e r n a l a n d 200 internal. T h e a d v e n t of g a l l b l a d d e r surgery has g r e a t l y r e d u c e d the f r e q u e n c y of these fistulas. A t present, the f r e q u e n c y of s p o n t a n e o u s i n t e r n a l b i l i a r y fistulas varies f r o m r e p o r t to r e p o r t , b u t it has b e e n said to be 0 . 2 - 0 . 4 % o f a u t o p s i e s a n d 0 . 5 - 7 . 0 % of b i l i a r y tract o p e r a t i o n s , w i t h an average of 1.5%. 4 A t Scott a n d W h i t e M e m o r i a l H o s p i t a l , in 3704 b i l i a r y o p e r a t i o n s d o n e f r o m 1947 to 1966, 14 s p o n t a n e o u s i n t e r n a l b i l i a r y fistulas were f o u n d , an i n c i d e n c e of 0.38%. T h e y have b e e n f o u n d b e t w e e n the d u o d e n u m , stomach, r i g h t h a l f of the colon, a n d - - m o r e r a r e l y - - t h e j e j u n u m , i l e u m , b r o n c h i a l tree, p e r i c a r d i u m , b l a d d e r , uterus, v a g i n a , o v a r i a n cyst, r e n a l pelvis, p o r t a l vein, a n d h e p a t i c artery.a U s u a l l y t h e y are solitary, b u t m u l t i p l e ones have b e e n reported.~ A b o u t 90% of all i n t e r n a l fistulas are d u e to c a l c u l o u s diseased I n c o u n t r i e s w h e r e echinococcosis is e n d e m i c , the c o n d i t i o n is f i ' e q u e n t l y e n c o u n t e r e d f o l l o w i n g r u p t u r e o f a h y d a t i d cyst of the liver, s Less c o m m o n causes are m a l i g n a n t lesions of the g a l l b l a d d e r , stomach, o r pancreas, a n d u l c e r a t i v e colitis2 O f the 14 cases of s p o n t a n e o u s i n t e r n a l b i l i a r y fistulas f o u n d at t h e Scott a n d W h i t e Clinic, 8 were c h o l e c y s t o d u o d e n a l , 2 were cholecystocolic, a n d 4 were c h o l e d o c h o d u o d e n a l d u e to d u o d e n a l nicer. T h e p r e s e n t r e p o r t concerns these 4.

T

REPORT

O F CASES

Case 1 (W. McC.). This 64-year-old white man was seen on Aug. 29, 1960, with epigastric pain present since 1940, relieved by food and antacids and appearing periodically in the fall and spring. In 1948 he had an episode of upper gastrointestinal bleeding with hematemesis and melena, with no recurrence since then. At the time of his admission, the pain was more constant, and he was gelling up 3 or 4 times a night to drink hot milk and relieve the pain. His family history and past medical history were not remarkable. The physical examination revealed a healthy-looking white ,nan in no distress, with a

From the Departments of Gastroenterology and Radiology, Scott and White Clinic, Temple, Tex.

*Fellow in Gastroenterology, Scott and "Whi~e Memorial Hospital, and Scott, Sherwood and Brindley Foundation. New Series, Vol. 12, No. 9, 1967

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McEwan-Aivarado & Dysarf n o r m a l temperature attd pulsc and a blood pressure o[ 148/81). His physical examination was essentially negative. He had a hemoglohin of 15 gm./l()0 nil. and a white blood cell count of 7000, with normal differential. His scdimentation rate (Westergren) was 18 ram. in 1 hr. Results of the Kline test were negative. Exanfination of the stools for occult blood was negative, and urinalysis was normal. A roentgenogram of the chest showed slight increase in tibrous marking along the right lower cardiac margin, probably due to past inflammation. Radiographic study of the colon was negative. R o e n t g e n o g r a m s o[ the stomach and d u o d e n u m showed a small postbulbar ulcer with st tistnlous connection iuto the c o m m o n duct (Fig, 1).

Fig. 1. Roeutgenogram showing ulcer crater at p o s t b u l b a r region with reflux of b a r i u m from ulcer into c o m m o n duct t h r o u g h fistulous tract.

Surgical intervention for complicated dnodenal ulcer was advised. T h e patient went home to r e t u r n in 1 week, b u t no retold of his return wits found.

Case 2 (E. H.). T h i s 63-year-old man was seen on Apr. 21, 1962, with tim chief complaint of chronic cough. He gave a vague history, but apparently some ),ears prior to his admission he had one bout of coffee-ground vomitus with black tarry stools. Besides, he complained of some indigestion and stomach upset at times. X-ray films taker) elsewhere were reported as normal. He had noted no recent signs of blood loss. He complained of dyspnea on moderate effort and of a chronic and, at times, productive, cough for "ahnost all" his life. His family history and past medical history were not remarkable. He used to smoke 1 pack of cigarettes a day. He drank no alcohol. He was a well-developed man with normal temperatnre and pulse, and a blood p~essure of 130/80. His chest was barrel-shaped, n, ith increased AP diameter, and his lungs showed inspiratory rhonchi and wheezing on hoth bases. T h e heart had regular sinus r h y t h m with no murnlurs. T h e a b d o m e n and the remaining physical examination were essentially negative, T h e patient's hemoglobin was 14.0 gin./100 ml.; white hlood cell count, 7800 with normal differential. Urinalysis was normal; the Kline test result was negative. Roentgenogram of the chest showed linear fibrotic markings iu the left lower chest, probably postinllammalory lesidnals. Roentgenologic examination of the stomach and d u o d e n m n showed lnarked duodenal deformity b u t no crater coMd be seen. A tisttdous connection between the posterior portion of the duodenal bulb and the c o m m o n duct was observed, (Fig. 2A), with rellux of b a r i u m reaching the gallbladder and air preset)| in lhe common duet (Fig. 2B). A medical regimen was advised a{ that lime, and tim patient was dismissed wilh medical tleatmenl to r e t u r n for observation.

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American Journal of Diges¢ive Diseases

CholedochoduodenaJ Fistula Case 3 (C. H.). T h i s 67-year-old Negro w o m a n was seen on Feb. 16, 1961, w i t h the chief ~'omplaint of a b d o m i n a l pain, w h i c h started the day before admission. It was sharp, s t a b b i n g in n a t u r e , localized to t h e r i g h t u p p e r q u a d r a n t , a n d was so severe t h a t h e r family doctor hospitalized her with the diagnosis of perforated viscus. She gave a history of repeated attacks of t h e same type of p a i n for t h e last 3 years, b u t this t i m e it was m o r e severe. T h i s r e c u r r e n t p a i n was aggravated by fatty foods, a n d was a c c o m p a n i e d by nausea a n d vomiting. For the last 2 days the p a t i e n t h a d been passing black stools. She gave no history of h e m a t e m e s i s . T h e physical e x a m i n a t i o n revealed a Negro w o m a n older in a p p e a r a n c e t h a n t h e stated age, b u t in no distress. H e r t e m p e r a t u r e a n d pulse were normal. H e r blood pressure was 110/80. Here sclerae showed questionable icterus. H e r l u n g s h a d sibilant rales bilaterally. H e r h e a r t a n d a b d o m e n were n o r m a l , and the r e m a i n d e r of the physical e x a m i n a t i o n showed essentially negative results. T h e p a t i e n t h a d a h e m o g l o b i n of 12.0 gin./100 ml.; a red blood cell c o u n t of 4,170,000; white blood cell c o u n t of 7500, with n o r m a l differential; p r o t h r o m b i n time, 70% (Quick); blood urea nitrogen, 24 gna.; s e r u m bilirubin, direct, 0.30 rag./100 ml.; total, 0.64 rag./100 ml. R e s u l t s Of the Kline test were negative, a n d urinalysis was within n o r m a l limits. A roentgenog r a m of the chest was negative. Roentgenologic s t u d y of the s t o m a c h a n d d u o d e n u m showed m a r k e d d u o d e n a l cap d e f o r m i t y with opacification of the biliary tract (Fig. 3). T h i s p a t i e n t was given a strict medical regimen, b u t on Apr. 19, 1961, she h a d surgical i n v e r v e n t i o n because of severe a b d o m i n a l pain. A n a n t r a l exclusion p r o c e d u r e a n d Billroth II anastomosis was done. T h e p a t i e n t was doing well 1 year after the operation. Case 4 (R. O.). A 56-year-old white f a r m e r was a d m i t t e d on Mar. 3, 1966, w i t h chronic r e c u r r e n t p a i n in the right a b d o m e n w h i c h h a d been p r e s e n t for 2 years before admission.

Fig. 2. A . R o e n t g e n o g r a m showing m a r k e d d u o d e n a l b u l h deformity with fistulous comm u n i c a t i o n from b u l b into c o m m o n duct w h e r e b a r i u m has refluxed. B. Air collection in c o m m o n d u c t (arrows). Note a n o t h e r arrow p o i n t i n g to collection of b a r i u m in gallbladder.

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McEwan-Alvarado & Dysarf T h e p a i n was sharp, mostly localized to the right u p p e r q u a d r a n t , with radiation to the dorsal spine, a n d a c c o m p a n i e d by n a u s e a a n d vomiting, b u t the p a t i e n t h a d no h e m a l e m e s i s or melena. A history of diabetes in the family was rexealed ( m o t h e r a n d sister). T h e past medical history included a cholecystectomy done elsewhere 1 year prior to admission, for the same pain. T h e p a t i e n t smoked 1 pack of cigarettes a day b u t d r a n k no alcohol.

Fig. 3. R o e n t g e n o g r a m clearly shows c o m m o n a n d hepatic ducts filled with b a r i u m froltl fistttlous connection with d n o d e n u n L

He was an obese white m a n in no distress, with a n m m a l t c m p e l a t u r e a n d pulse, a n d a blood pressure of lf~0/85. T h e findings in the general physical e x a m i n a t i o n were essentially negative. H e m o g l o h i n was 16.56 gin.; white blood cell count, 12,5011; neutrophils, 67°4,; lymphocytes. 20%; monocytes, 8°,{,; eosinophils, 4{5o. Results of the Kline test were negative; the van den Bergh direct showed 0.12 rag./100 ml., total 0.50 rag./100 ml.; alkaline phosphatase, 5.4 K-A U.; u r i n a r y p o r p h o b i l i n o g e n , negative; 2-hr. p o s t p r a n d i a l hlood sugar, 142 rag./100 ml. A 5-hr. glucose-tolerance test showed a peak of 186 gin./100 ml. at 11/_, hr.; r o u l i n e urinalysis revealed a specific gravity of 1.01% p H 5.0, a trace of a l b u m i n , no glucose, a n d rare epithelial a n d red and white blood cells. In the roentgenologic studies, a flat plate of the a b d o m e n was negative; i n t r a v e n o u s l)yelogram showed good function, n o r m a l filling a n d o u t l h m bilaterally; esophagus, stomach, d u o d e n u m , and small intestine were negative; colon a n d terminal i l e u m ~.ere also negative. T h e patient was given a diabetic diet. His condition improved a n d hc was dismissed o n Apr. 21, 1966, a s y m p t o m a t i c with the diagnosis of diabetes mellitus. O n July 9, 1966, the patient r e l t u n e d with severe right u p p e r q u a d r a n t pain. P r e l i m i n a r y lihns for an i n t r a v e n o u s c h o l a n g i o g r a m showed air in the biliary ducts (Fig. 4A). R e p e a t roentgenologic e x a m i n a t i o n of t h e s t o m a c h aml d u o d e n u m r m e a l e d an ulcer crater ill the second portion a n d medial aspect: of the d u o d e n u n l (Fig. 4B). P, ellux of bariuln into the c o m m o n duct was obtained in a n o t h e r lihn (Fig. 4(;). T h e p a t i e n t tlndcrwent operation consisting of antrectom~, widl cxchtsiou of Ihe ulcer, a n d vagotonty. On Sept. 10, 1961i, the patient was a s ) m p t o m a t i c .

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American Journal of Dicjesiive Diseases

Choledochoduodenal Fis÷u[e

Fig. 4. A. Air is present an biliary tract (arrows). B. Ulcer crater at the second portion of duodenum, in medial aspect, is in apparent close proximity to expected location of common duct. C. Common duct filled with barium can be seen as vertical column passing behind duodenum. DISCUSSION O f all s p o n t a n e o u s i n t e r n a l b i l i a r y fistulas, 1 0 - 2 0 % a r e o f t h e c h o l e d o c h o duodenal

t y p e . am I n o u r series, t h e p e r c e n t a g e is 28.5. D u o d e n a l

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cause in 80% of the cases. 1° Duodenal ulcer is then the cause in only 6-22.8cfo of all spontaneous internal biliary fistulas. Since these fistulas are found ill about only 0.2% of autopsies and 1.5% of all biliary tract operations, we can see that spontaneous choledochoduodenal fistulas are indeed rare. T h i s may seem surprising in view of the frequency of duodenal ulcers and the proximity of the c o m m o n bile duct to the first p a r t of the duodenum, n Explanation for this may be found in the fact that duodenal ulcers usually occur in the first centimeter or two distal to the pylorus on the anterior or posterior wall rather than laterally. 12 Anterior ulcers may perforate into the peritoneal cavity and very rarely into the gallbladder, which is outside this zone of most frequent ulcer formation. Perforating posterior ulcers more often penetrate into the pancreas a2 and less frequently into the c o m m o n duct, which passes mostly behind the d u o d e n u m at a point lateral to the usual site of ulceration, and is frequently separated from it by the superior edge of the pancreas, n T h e first report of duodenal ulcer fistula with the biliary tract was by Long in 1840.11 Since that time, only about 149 cases had been reported up to 1964. s Usually the diagnosis is made during surgery or necropsy, la Attempts to establish an identifying clinical picture have given few suggestive symptoms. T h e difference in prognosis and treatment of these fistulas makes necessary an accurate diagnosis. Most patients with cholecystoduodenal fistulas due to biliary disease are women in the sixth and seventh decades of life with recurrent severe biliary pain, often associated with jaundice that m a y suddenly disappear. However, Puestow, 14 in a series of 16 cases, found that the symptoms were not relieved but became progressively worse. Jaundice in the absence of biliary obstruction was a frequent finding in this series and indicated cholangitis and hepatitis. T h e bronchobiliary fistulas produce p u l m o n a r y symptoms with expectoration of bile, 2, 15 and hepatic artery fistulas may produce symptoms of u p p e r gastrointestinal bleeding. Fistulas secondary to penetrating duodenal ulcer are more common in men of 40-50 years of age than in w o m e n of any age. T h e patients' histories reveal the presence of duodenal ulcer for an average of 11 years. T h r e e of our 4 patients were men ranging in age from 56 to 64 years. In the first case, the 64-year-old patient had a history of duodenal ulcer for a b o u t 20 years. T h e second patient presented a history compatible with duodenal ulcer, and although an ulcer was not demonstrated previously, it is evident that the patient may have bled at least once. T h e third patient had no history of duodenal ulcer. T h e symptoms, present for only 2 days prior to admission, suggested an acute cholecystitis rather than a duodenal ulcer, but the fact that there was a questionable history of tarry stools suggests a bleeding lesion in the u p p e r gastrointestinal tract. T h e fourth patient presented no history of duodenal ulcer either. His symptomato'ogy was of 2 years' duration and suggested a 952

American Journal of D;ges÷~veDiseases

Choledochoduodenal Fistula

right renal calculus or a common-duct stone, although the right tlank or upper q u a d r a n t pain pattern of postbulbar ulcers was seen in this patient. As in instances of cholecystoduodenal fistulas, a cessation or amelioration of symptoms at the time of fistula formation has been mentioned by others as a characteristic finding in choledochoduodenal fistulas secondary to duodenal ulcer. 11 In none of our patients did we observe that p h e n o m e n o n . T h e second patient was asymptomatic all the time; the other 3, who were symptomatic, became worse. T h e symptoms and clinical course are said not to differ much from uncomplicated duodenal ulcer. T h r e e of our 4 patients, however, presented an atypical picture. I t is i m p o r t a n t to mention that in this type of fistula, symptoms of cholangitis have been an infrequent finding. 1°, la This makes the prognosis and treatment different from the other types of fistulas. Roentgenologic examination is the only m e t h o d for definitive preoperative diagnosis. T h e presence of air in the biliary ducts seen on a flat film of the a b d o m e n - - " p n e u m o b i l i a " (Fig. 2B and 4 A ) - - o r the filling of these channels after a b a r i u m study of the stomach and d u o d e n u m (Fig. 1, 2A, 3 and 4C) or colon is the f u n d a m e n t a l criterion for the diagnosis. 13 It has been m e n t i o n e d that acute emphysematous cholecystitis and regurgitation due to incompetence of the sphincter of Oddi can cause an error in diagnosis. 1~ W a g g o n e r and Le Monde 1~ report t h a t they have not observed the former situation. T h e second has been observed by them in very rare instances, and was due to a pathologic condition in the area of the papilla. Because of the poor prognosis of spon, taneous internal biliary fistulas due to gallbladder disease, the early prophylactic removal of the diseased gallbladder is advocated before fistulas can form. Surgical treatment should include the closure of the fistula and removal of the p r i m a r y site of disease. I n the case of fistulas due to penetration of a peptic ulcer, the prognosis is considerably i m p r o v e d and indications for operation are limited to reducing the complications o.f peptic ulceration. ~7 Partial gastrectomy with Billroth I I anastomosis is advocated, aT, ~s I n our first patient, operation was recommended. Since the second patient was asymptomatic, it was advisable at that time to manage his condition medically, with strict follow-up. Unfortunately, the patient did not return. Because of their severe symptoms, the third and fourth patients were operated upon. In both, an antrectomy with Billroth I I anastomosis was done. Unlike fistulas due to biliary disease, in fistulas due to peptic ulcer it is recommended that the fistula be left intact. 17 Patients with these fistulas have been observed for m a n y years without evidence of cholangitis or hepatitis, 1~ although persistent jaundice associated with a biliary fistula which allows bile to flow out of the bile ducts indicates that hepatitis exists, 14 and the therapy should be directed to correcting this event with the proper antibiotic dosage before any surgical procedure is carried out. New Series, Vol. 12, No. 9, 1967

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Mcl::wan-Alvarado & Dysarf SU M M A R Y

Four patients with spontaneous internal biliary fistttlas o[ the choledochoduodenal type due to duodenal ulcer are presented. A review of the literature and a commentary on the symptomatology and treatment are made. Scott and White Clinic Tent[fie, Tex. 7650l

REFERENCES I. HENRY, (;. L., a n d ORR, T . G. S p o n t a n e o u s external biliary fistulas. Surge~3' 26:641, 1949. 2. DOWSE, J. L. A. S p o n t a n e o u s i n t e r n a l biliary fistnlae. Gut 5:429, 1964. 3. NAUNYN, B. A Treatise on Cholelithiasis, 1892, A. E. Garrod, T r a n s l . New S y d e n h a m Society, L o n d o n , 1896. p. 143. 4. BOCKUS, H. L. Gastroenterology (ed. 2, Vol. I l I ) . Sannders, Philadelphia, 1965, p. 851. 5. TE'rREAULT, A. F., BOWEN, J. R., a n d SAMVMO, N. H e m o b i l i a secondary to intrahep'~tic a n e n r y s m of the h e p a t i c artery. R e p o r t of a case with clinical a n d pathological correlations. JAMA 192:1096, 1965. 6. AMOURY, R. A., a n d BARKER, H. G. Multiple biliary enteric fistulas. Amer ] S,t,g Ill:lEt), 1966. 7. BERC,NER, L. H. I n t e r n a l biliary fistulas. Amer ] Gastroe*~terol 43:11, 1965. 8. KOURIAS, B. A., a n d CnOUUARAS, A. S p o n t a u e o u s gastrointestinal biliary fistula complicating d u o d e n a l tdcer. Surg Gynec Obstet 119:1013, 1964. 9. ORMANDY, L., a n d BARGEN, J. A. T h r o m b o - n l c e r a t i v e colitis associated with cologastric a n d coloduodenal fistulas. Proc Mayo Clin 14:550, 1939. 10. HUTCmNCS, V. Z., WHEELER, J. A., a n d PuEsrow, C. B. C h o l e c y s t o d u o d e u a l fistula complicating d u o d e n a l ulcer. Arch Surg (Chicago) 73:598, 1956. 11. KYLE, J. C h o l e d o c h o d u o d e n a l fistula due to d u o d e n a l ulceration. Brit J Surg 46:124, 1958. 12. ANDERSON, W. A. D. Pathology (ed. 5, Vol. I I ) . Mosby, St. Louis, 1966, p. 852. 13. WACGONER, C. M., a n d LE MONDE, D. V. Clinical a n d roentgen aspects of internal biliary fistulas. Radiology 53:31, 1949. 14. Pules'row, C. B. Spontaneous internal biliary fistula. Ann Surg 115:1043, 1942. 1 5 . ADAMS, H. D. Hepatobiliary i n v o l v e m e n t of the thorax. Surg Clin N A r n e r 38:611, 1958. 16. COt;roLL, C. L., a n d RO'rH, H. P. C h o l e d o c h o d u o d e n a I fistula: R e p o r t of two cases d u e to d u o d e n a l ulcer. Amer Surg 19:480, 1953. 17. JORDAN, P. H., a n d ST1RRETT, L. A. T r e a t m e n t of s p o n t a n e o u s internal biliary fistula caused by d u o d e n a l ulcer. Amer ] Surg 91:307, 1956. 18. Lrvowrrz, B. S. S p o n t a n e o u s internal biliary fistula. Ann Surg 154:241, 196i.

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American Journal of Digestive Diseases