REVIEW
Hemobilia from Ruptured Hepatic Artery Aneurysm Report of a Case and Review of the Literature
Nickolas N. Harlaflis, MD, Atlanta, Georgia John 1. Akin, MD, Atlanta, Georgia
Hemobilia, bleeding through or in the biliary tree, may originate in the liver, in the intrahepatic or extrahepatic biliary tract, or in the pancreas. It may be secondary to trauma, inflammatory disorders, gallstones, or tumors. Rupture of hepatic artery aneurysm is a rare cause of the clinical entity. The purpose of this communication is to present a case of hemobilia due to rupture of an aneurysm of the right hepatic artery and to review the syndrome of hemobilia. Case Report A sixty-two year old white female (AMD) was admitted to Piedmont Hospital on January 1,1973, with a history of right upper quadrant pain, nausea, and vomiting of two weeks’ duration. A cholecystogram showed nonvisualization of the gallbladder and an intravenous cholangiogram revealed normal common bile duct and obstruction of the cystic duct. She underwent operation on January 4,1973, and a gangrenous gallbladder with multiple stones was resected. The patient’s postoperative course was without complications. She did well until three months after surgery (April 2, 1973) when she was readmitted to the hospital because of nausea, hematemesis, tarry stools, and cramping discomfort in the right upper quadrant and epigastrium. Admission vital signs were within normal limits, serum bilirubin level was 2 mg/lOO ml, and SGOT level was 822 mu/ml. Abdominal examination showed nothing abnormal. Bleeding continued and she was given 2,000 cc of blood over the next 48 hours. Upper gastrointestinal series and gastroscopy showed nothing abnormal. Bleeding stopped and From the Department of Surgery, Piedmont Hospital, and the Emory University Affiliated Surgical Services, Atlanta, Georgia. Reprint requests should be addressed to John T. Akin, MD. 35 Collier Road, NW, Suite 655, Atlanta, Georgia, 30309.
Volume 133, February 1977
she was discharged April 10,1973. She was readmitted to Piedmont Hospital ten days later with right upper quadrant pain, nausea, and maroon colored stools. Again, vital signs were within normal limits. Gastroscopy, upper and lower gastrointestinal series, intravenous cholangiogram, and liver scan disclosed no abnormality. During this admission serum bilirubin level was 0.5 mg/lOO ml, alkaline phosphatase 132 mu/ml, and SGOT 102 mu/ml. Bleeding ceased and on May 3,1973, she was discharged without subjective complaints. On May 10, 1973, she again had crampy right upper quadrant pain, nausea, and hematemesis and was readmitted t.o Piedmont Hospital. Physical examination again showed nothing abnormal, and hematocrit was 36.1 per cent, SGOT 262 mu/ml, and serum bilirubin 2.6 m&O0 ml. The night of admission she vomited large quantities of bright red blood and required 5 units of whole blood. Emergency selective celiac arteriogram revealed an aneurysm of the right hepatic artery measuring 16 mm in diameter, communicating with the common hepatic duct. (Figures 1 and 2.) Emergency laparotomy was performed. The stomach and the entire small bowel and colon contained blood. The common bile duct was found dilated to approximately 1.4 cm in diameter. The hepatic artery was located posteromedially to the common bile duct and the aneurysm of the right hepatic artery measuring 1.6 by 1.2 cm was firmly adherent to the common hepatic duct. Choledochotomy was then performed, and active arterial bleeding encountered coming through a 5 mm communication of the aneurysm with the posterior wall of common hepatic duct just distal to the bifurcation. The right hepatic artery was ligated with silk proximal and distal to the aneurysm and the fistulous opening was closed with continuous 5-O arterial sutures. A T tube was placed in the common bile duct about which the duct was closed with interrupted fine silk sutures. A Penrose drain was placed in the foramen of Winslow and the abdominal wound was closed in layers.
229
Harlaftis
Figure 7. Selective ceiiac arteriogram demonstrating an aneurysm from arising the right hepatic artery cm 1.5 distal to the origin of the left hepatic artery.
Figure 2. Selective ceiiac arteriogram, delay Him, stiii demonstrating the aneurysm as we/i as extravasation of contrast media from the aneurysm into the common bile duct.
During the operation, the estimated blood loss was 2,000 cc, and 8 units of whole blood were given to maintain the patient’s vital signs within normal limits. The postoperative course was complicated by elevation of serum bilirubin
Sandblom [4] in 1972. The most common vascular lesion causing hemobilia is aneurysm of the hepatic artery. Review of eighty cases (including our own) showed the arterial location of the aneurysm to be the right hepatic artery (42 cases), common hepatic artery (26), left hepatic artery (6), cystic artery (5), and gastroduodenal artery (1). Forty-eight of these eighty aneurysms were true and thirty-two were false. The most common sites of communication between the aneurysm and biliary tract are the common bile duct or hepatic ducts (76.2 per cent). (Table I.) The male to female ratio is 1.6:1, the average age being 41.6 years. Clinical Manifestations. Quincke [5] was the first to establish the three main features of hemobilia, namely, gastrointestinal bleeding, biliary colic, and jaundice. The bleeding into the biliary tree secondary to a ruptured aneurysm may be slow, leading to melena and secondary anemia, or rapid, leading to hematemesis and shock. This extreme variation in blood loss is illustrated by our patient, requiring no transfusions on one admission and many transfusions on another. When the hemorrhage is in the gallbladder and if clotting prevents emptying, the distended gallbladder may be palpated. Because of limited space in the biliary tract and the resistance of the sphincter of Oddi, recurrent bleeding is char-
level to 4 to 6 mg/lOO ml, with return to normal by the twelfth postoperative day. On May 23,1973, T-tube cholangiography showed no intraductal filling defects. The T tube was removed and she was discharged on the fifteenth postoperative day. The patient was seen one and a half years postoperatively with no symptoms referable to the gastrointestinal or biliary systems. Comments
Hemorrhage through the biliary tract was first described by Glisson [1] in 1654 and such bleeding from rupture of a hepatic artery aneurysm into the common duct was first reported by Jackson [2] in 1821. The term “hemobilia” was first proposed by Sandblom [3] in 1948. Approximately 10 per cent of gross hemobilia is secondary to vascular disease. This was the most common cause of hemobilia during the past century. However, because of the near disappearance of the mycotic aneurysm and the increased incidence of accidents, accidental or iatrogenic trauma is now the most common cause of hemobilia, accounting for 55 per cent of 355 fully documented cases reviewed by
230
The American Journal of Surgery
Ruptured
TABLE
I
Site of Communication and Biliary
between
bile duct or hepatic
No. of Cases ducts
Gallbladder Cystic duct Gallbladder and common bile duct Gallbladder and peritoneal cavity Gallbladder and duodenum Hepatic ducts and duodenum Unknown Total
acteristic
61 9 3 1 1 1 1 3 80
of hemobilia. In addition, the biliary obstruction from the clots may tamponade for a time the bleeding source [4]. When an aneurysm communicates with the biliary tract it may abruptly distend the ducts, exposing them to arterial pressure and resulting in severe pain. The passage of clots may act as gallstones causing biliary colic. The pain from hepatic artery aneurysm is in the epigastrium or right upper quadrant or may radiate into the back. With a slow flow of blood, pain may be absent. Jaundice is caused by obstruction of the biliary tree from blood clots or from compression by the aneurysm. Fever is usually associated with obstructive jaundice, and peritonitis secondary to perforation of the gallbladder has been reported [6]. Diagnosis. The classic triad of hemobilia is gastrointestinal bleeding, right upper quadrant or epigastric pain, and jaundice. Other clinical manifestations as reported by Guida and Moore [7] are a palpable gallbladder or a palpable pulsating mass with bruit. At present the most useful diagnostic test is selective arteriography of the celiac axis, the hepatic artery, or the superior mesenteric artery. The arteriography, as in the case reported herein, may reveal the specific lesion, the precise source of bleeding and the anatomy of the hepatic artery. The first preoperative arteriography in a case of a ruptured hepatic artery aneurysm causing hemobilia was performed by Kirklin [8] in 1955. Twenty of the reviewed eighty cases have been diagnosed preoperatively by this method (Table II.) Other methods of diagnosis include: barium studies of upper gastrointestinal tract, intravenous cholangiography, duodenoscopy with retrograde cholangiography, percutaneous splenoportography, and scintigraphy of the liver [4,9]. Unsuspected
Volume 133, February 1977
TABLE
II
Methods
Artery Aneurysm
of Diagnosis
System
Site Common
Aneurysm
Hepatic
Method Preoperatively Umbilical portography Arteriogram Clinical signs During operation Macroscopy Operative arteriogram Postoperatively Autopsy Arteriogram Not recorded Total
No. of Cases
1 20 2 20 2 29 2 4 80
hemobilia in patients operated upon for gastrointestinal bleeding may be found at the time of surgery by (1) palpation of the cystic and hepatic vessels for aneurysms or tumors, (2) palpation of the liver for masses or bruit, (3) cholecystostomy or aspiration of gallbladder, (4) choledochotomy, or (5) operative arteriogam [4,10,11]. Treatment. Hemobilia secondary to rupture of hepatic artery aneurysm has been managed surgically with increasing success during the past twenty-five years. Of sixty patients who underwent surgery, thirty-five (58.33 per cent) survived. Mester [12] in 1895 was the first to suggest ligation of the hepatic artery or one of its branches to treat a hepatic artery aneurysm ruptured into the biliary tract. Kehr [13] in 1903 was the first to successfully treat a patient with hemobilia secondary to a ruptured right hepatic artery aneurysm communicating with the cystic duct, by ligation and division of the hepatic artery proper. Ligation of the hepatic artery distal to the gastroduodenal artery interrupts the collateral circulation through the gastroduodenal and right gastric arteries with a higher degree of mortality and accordingly should not be done without reconstruction except when the aneurysm has completely occluded the artery [7]. Ligation or excision of the hepatic artery or its branches has been successfully accomplished in twenty-two cases [8,9,13-301. Other surgical methods that have been used in cases of hemobilia secondary to hepatic artery aneurysm are (1) excision of the aneurysm and restoration of the arterial blood flow with a prosthetic graft [31,32], (2) obliterative or reconstructive endoaneurysmorrhaphy [33-371, and (3) hepatic resection, which has been used chiefly in traumatic hemobilia and in cases of intrahepatic aneurysms [I 7,38-401.
231
tiarlaftis
Summary
Hepatic artery aneurysm is a rare vascular disease assqciated with high mortality caused by massive hemorrhage or by complications following surgical treatment. Over the past twenty-five years it has been managed surgically with increasing success. Eighty reported cases of hepatic artery aneurysms ruptured into the biliary tree were reviewed and the etiology, clinical signs, diagnosis, and treatment of such an aneurysm are discussed. A personal case with an aneurysm of the right hepatic artery ruptured into the common hepatic duct is reported. The diagnosis was made before surgery by arteriography and the aneurysm was successfully managed by ligation of the right hepatic artery both proximal and distal to the aneurysm, closure of the communication between the common hepatic duct and the aneurysm, and choledochal drainage. Hemobilia secondary to hepatic artery aneurysm must be considered in the differential diagnosis of unexplained gastrointestinal hemorrhage. References
5. 6. 7.
8.
9.
10.
11. 12. 13.
14.
15.
232
Glisson F: Anatomia hepatis. 1654. Cited in [ 41. Jackson JBS: Aneuysm of the hepatic artery bursting into the hepatic duct. A&d h&wine (Boston) 3: 115, 1834. Sandblom P: Hemontrage into the biliary tract fotlowlng trauma: “traumatic hemobilia.” Surgery 24: 571, 1948 Sandblom P: Hemobilia (biliary tract hemorrhage). History, Pathotogy, Diagnosis, Treatment. Springfield, Illinois, Charfes C. Thomas, 1972, p 5. Quincke H: Ein Fall von Aneurysma der Leberarterie. Bert k/in Wschr 30: 349. 1871. Siegel E: Ein Fall von traumatischer Gang&t der Gallenblasse. h&Inch Med Wschr 56: 341. 1909. Guida PM, Moore SW: Aneurysm of the hepatic artery: report of 5 cases with a brief review of the previously reported cases. Surgery 60: 299. 1966. Kirklin JW. Shocket E. Comfort MW, Huizenga KA: Treatment of aneurysm of the hepatic artery by excision: report of a case. Ann Surg 142: 110. 1955. Berenson MM, Freston JW: lntrahepatic artery aneurysm associated with hemobilia: report of a case. Gsstrcenterotogy 66: 254, 1974. Brown KL, Hcpwood HH. Engle JC: Hemobilia caused by rupture of an aneurysm of an anomalous right hepatic artery. Am Surg 39: 229. 1973. Guynn VL. Reynolds JT: Surgical management of hemobilia. Arch Surg 83: 89. 1961. Mester B: Das Aneurysma der Arteria Hepatica. Ztschr K/in Med 28: 93. 1895. Kehr H: Der erste Fall von erfolgeicher Unterbindurg dsr Arteria Hepatica Propia wegen Aneurysma. A&Inch Med Wschr 50: 1881.1903. Colmers F: lntrahepatisches Aneurysma und Gallenfistel nach Leberzerreissung. Heilung durch Unterbindung der Arteria Hepatica Communis. Bruns Beitr K/in Chir 122: 324, 1921. Devin R, Mambrini A, Winninger A, Febvey M. D’Drso M, Bensussan: Un cas d’ ar&rysme de I’art&e cystiqua rompu dans la vesicule biliaire. Chirurgie 97: 332, 1971.
16. Dreze C, Lombard R, Jacquet N, Hen/e P: H&ncbilie seccndaire a la fistulisation d’ un faux anevrisme d’ une bran&e de I’ art&e hepatique droit darts la voie biliaire. Diagnostic preoperatoire par I’ arteriographie selective. Acta Gastroenterol Be/g 36: 397. 1973. 17. Gupta S. Cope V: Hepatic artery aneurysm as a cause of gastrointestinal blood loss. 8r J Radio/ 45: 726, 1972. 18. Hepp J, Caroli J, Moreaux J, Opolon P. Bismuth H: Les h&mobilies par lesion traumatiqus de I’ art&e hepatique darts les contusions de I’ abdomen. Ann Chir 20: 359, 1966. 19. Hughes JH. Guzman JG. Roberts SS: Massive hemobilia from ruptured hepatic artery aneurysm. JAMA 214: 913. 1970. 20. lnui FK, Ferguson TA: Aneurysm of the right hepatic artery: preoperative diagnosis and successful excision. Ann Surg 144: 235, 1956. 21. Jesipowicz M. Klamut M. Pochylski T: Tetniak tetnicy watrobowej jako przyczyna hemobilii. PO/ Przegl Chir 10: 1117. 1968. 22. Jesseph JE. May KJ Jr: False aneurysm of the hepatic artery with massive hemobilia and jaundice. Arch Surg 81: 646. 1960. 23. Jewett TC Jr: Aneurysm of the hepatic artery in a child. Ann Surg 150: 951, 1959. 24. Kading K: Ein geheilter Fall von intrahepatischem Aneurysma mit besonderer Berucksichtigung der traumatischen Leberarterienaneurismen. Msch ZChir 150: 82, 1919. 25. Kunzli HF, Schmidt HE: lntrahepatisches Aneurysma dar m&ten Leberarterie mit posttraumatischsr l-ktmobilie. He/w Chir Acta 37: 559. 1970. 26. Larmi TKI: Hemobilia associated with cholecystitis, postcholecystectomy conditions and trauma: review of 12 cases. Ann Surg 163: 373, 1968. 27. Mctachlin AD, Coles JCG: Hemobilia: a case of unusualetiology and its management. Surg C//n North Am 50: 107, 1970. 28. Mackay AG, Page HGzHematemesis associated with hemobilta: report of a c&e due to an intrahepatic artery aneurysm with survival. N Errol J A&d 260: 468. 1959. 29. Schildberg FW, Stucker FJ: Die Verletzungen der A. Hepatica und ihrer intrahepatischen Aufzweigungen. Bruns t?ettr K/in Chir218: 193, 1970. 30. Stack BHR. Ramkin JT, Bentley RJ: Hepatic artery aneurysm after staphylococcal endocarditis. Br kd J 3: 659. 1968. 31. Hufnagel CA: In discussion of Posttethwait RW, Hemandez RR. Dillon ML: Hepatic artery lesions. Ann Surg 159: 895, 1964. . 32. Santiago-Delpin EA. Marquez E, Rodriquez OL, Oliveras FE, Baldizon C. Martinez-Cabruja R: Perforated hepatic artery aneurysm and muttiple aneurysms in incomplete Marfan syndrome. Ann Surg 176: 772, 1972. 33. Fekete F: Personal communication. Cited in [ 31. 34. Gordon-Taylor G: A rare cause of severe gastrointestinal haemorrhaga with a note on aneuysm of the hepatic artery. BrMedJ 1: 504. 1943. 35. Hess W. Celia A: Das Aneurysms del Arteria Hepatica. Ein durch Aneurysmorrhaphie geheitter Fall. He/v Chir Acta 22: 286. .l955. 36. Paul M: A large traumatic aneurysm of the hepatic artery. Br J Surg 39: 278. 1951. 37. Winchester DP. Seed RW. Bergan JJ, Conn J: Jaundice hemobilta, and hemoperitrmeum. Consequences of rupture of hepatic artery aneurysm. Am J Surg 120: 384, 1970. 38. Cohen Y, Chow KW. Seah CS: lntrahepatic aneurysm preoperatively diagnosed and treated by resection. 8r J Surg 53: 602, 1966. 39. Lataste J, Albou JC. Melet J: Anhrysme intrah@atique complique d’twmobilie: gueri par hepatectomie droite. Resse hkd79: 1809, 1971. 40. Owen CE, Owen MB: Hemobilia: a case with massive repeated bleeding from the biliary tract. Proc Roy Sot Med 53: 223. 1960.
TheAmerican
Journal of Surttary
Hemobilia from Ruptured Hepatic Artery Aneurysm Report of a Case and Review of the Literature
Nickolas N. Harlaflis, MD, Atlanta, Georgia John 1. Akin, MD, Atlanta, Georgia
Hemobilia, bleeding through or in the biliary tree, may originate in the liver, in the intrahepatic or extrahepatic biliary tract, or in the pancreas. It may be secondary to trauma, inflammatory disorders, gallstones, or tumors. Rupture of hepatic artery aneurysm is a rare cause of the clinical entity. The purpose of this communication is to present a case of hemobilia due to rupture of an aneurysm of the right hepatic artery and to review the syndrome of hemobilia. Case Report A sixty-two year old white female (AMD) was admitted to Piedmont Hospital on January 1,1973, with a history of right upper quadrant pain, nausea, and vomiting of two weeks’ duration. A cholecystogram showed nonvisualization of the gallbladder and an intravenous cholangiogram revealed normal common bile duct and obstruction of the cystic duct. She underwent operation on January 4,1973, and a gangrenous gallbladder with multiple stones was resected. The patient’s postoperative course was without complications. She did well until three months after surgery (April 2, 1973) when she was readmitted to the hospital because of nausea, hematemesis, tarry stools, and cramping discomfort in the right upper quadrant and epigastrium. Admission vital signs were within normal limits, serum bilirubin level was 2 mg/lOO ml, and SGOT level was 822 mu/ml. Abdominal examination showed nothing abnormal. Bleeding continued and she was given 2,000 cc of blood over the next 48 hours. Upper gastrointestinal series and gastroscopy showed nothing abnormal. Bleeding stopped and From the Department of Surgery, Piedmont Hospital, and the Emory University Affiliated Surgical Services, Atlanta, Georgia. Reprint requests should be addressed to John T. Akin, MD. 35 Collier Road, NW, Suite 655, Atlanta, Georgia, 30309.
Volume 133, February 1977
she was discharged April 10,1973. She was readmitted to Piedmont Hospital ten days later with right upper quadrant pain, nausea, and maroon colored stools. Again, vital signs were within normal limits. Gastroscopy, upper and lower gastrointestinal series, intravenous cholangiogram, and liver scan disclosed no abnormality. During this admission serum bilirubin level was 0.5 mg/lOO ml, alkaline phosphatase 132 mu/ml, and SGOT 102 mu/ml. Bleeding ceased and on May 3,1973, she was discharged without subjective complaints. On May 10, 1973, she again had crampy right upper quadrant pain, nausea, and hematemesis and was readmitted t.o Piedmont Hospital. Physical examination again showed nothing abnormal, and hematocrit was 36.1 per cent, SGOT 262 mu/ml, and serum bilirubin 2.6 m&O0 ml. The night of admission she vomited large quantities of bright red blood and required 5 units of whole blood. Emergency selective celiac arteriogram revealed an aneurysm of the right hepatic artery measuring 16 mm in diameter, communicating with the common hepatic duct. (Figures 1 and 2.) Emergency laparotomy was performed. The stomach and the entire small bowel and colon contained blood. The common bile duct was found dilated to approximately 1.4 cm in diameter. The hepatic artery was located posteromedially to the common bile duct and the aneurysm of the right hepatic artery measuring 1.6 by 1.2 cm was firmly adherent to the common hepatic duct. Choledochotomy was then performed, and active arterial bleeding encountered coming through a 5 mm communication of the aneurysm with the posterior wall of common hepatic duct just distal to the bifurcation. The right hepatic artery was ligated with silk proximal and distal to the aneurysm and the fistulous opening was closed with continuous 5-O arterial sutures. A T tube was placed in the common bile duct about which the duct was closed with interrupted fine silk sutures. A Penrose drain was placed in the foramen of Winslow and the abdominal wound was closed in layers.
229
Harlaftis
Figure 7. Selective ceiiac arteriogram demonstrating an aneurysm from arising the right hepatic artery cm 1.5 distal to the origin of the left hepatic artery.
Figure 2. Selective ceiiac arteriogram, delay Him, stiii demonstrating the aneurysm as we/i as extravasation of contrast media from the aneurysm into the common bile duct.
During the operation, the estimated blood loss was 2,000 cc, and 8 units of whole blood were given to maintain the patient’s vital signs within normal limits. The postoperative course was complicated by elevation of serum bilirubin
Sandblom [4] in 1972. The most common vascular lesion causing hemobilia is aneurysm of the hepatic artery. Review of eighty cases (including our own) showed the arterial location of the aneurysm to be the right hepatic artery (42 cases), common hepatic artery (26), left hepatic artery (6), cystic artery (5), and gastroduodenal artery (1). Forty-eight of these eighty aneurysms were true and thirty-two were false. The most common sites of communication between the aneurysm and biliary tract are the common bile duct or hepatic ducts (76.2 per cent). (Table I.) The male to female ratio is 1.6:1, the average age being 41.6 years. Clinical Manifestations. Quincke [5] was the first to establish the three main features of hemobilia, namely, gastrointestinal bleeding, biliary colic, and jaundice. The bleeding into the biliary tree secondary to a ruptured aneurysm may be slow, leading to melena and secondary anemia, or rapid, leading to hematemesis and shock. This extreme variation in blood loss is illustrated by our patient, requiring no transfusions on one admission and many transfusions on another. When the hemorrhage is in the gallbladder and if clotting prevents emptying, the distended gallbladder may be palpated. Because of limited space in the biliary tract and the resistance of the sphincter of Oddi, recurrent bleeding is char-
level to 4 to 6 mg/lOO ml, with return to normal by the twelfth postoperative day. On May 23,1973, T-tube cholangiography showed no intraductal filling defects. The T tube was removed and she was discharged on the fifteenth postoperative day. The patient was seen one and a half years postoperatively with no symptoms referable to the gastrointestinal or biliary systems. Comments
Hemorrhage through the biliary tract was first described by Glisson [1] in 1654 and such bleeding from rupture of a hepatic artery aneurysm into the common duct was first reported by Jackson [2] in 1821. The term “hemobilia” was first proposed by Sandblom [3] in 1948. Approximately 10 per cent of gross hemobilia is secondary to vascular disease. This was the most common cause of hemobilia during the past century. However, because of the near disappearance of the mycotic aneurysm and the increased incidence of accidents, accidental or iatrogenic trauma is now the most common cause of hemobilia, accounting for 55 per cent of 355 fully documented cases reviewed by
230
The American Journal of Surgery
Ruptured
TABLE
I
Site of Communication and Biliary
between
bile duct or hepatic
No. of Cases ducts
Gallbladder Cystic duct Gallbladder and common bile duct Gallbladder and peritoneal cavity Gallbladder and duodenum Hepatic ducts and duodenum Unknown Total
acteristic
61 9 3 1 1 1 1 3 80
of hemobilia. In addition, the biliary obstruction from the clots may tamponade for a time the bleeding source [4]. When an aneurysm communicates with the biliary tract it may abruptly distend the ducts, exposing them to arterial pressure and resulting in severe pain. The passage of clots may act as gallstones causing biliary colic. The pain from hepatic artery aneurysm is in the epigastrium or right upper quadrant or may radiate into the back. With a slow flow of blood, pain may be absent. Jaundice is caused by obstruction of the biliary tree from blood clots or from compression by the aneurysm. Fever is usually associated with obstructive jaundice, and peritonitis secondary to perforation of the gallbladder has been reported [6]. Diagnosis. The classic triad of hemobilia is gastrointestinal bleeding, right upper quadrant or epigastric pain, and jaundice. Other clinical manifestations as reported by Guida and Moore [7] are a palpable gallbladder or a palpable pulsating mass with bruit. At present the most useful diagnostic test is selective arteriography of the celiac axis, the hepatic artery, or the superior mesenteric artery. The arteriography, as in the case reported herein, may reveal the specific lesion, the precise source of bleeding and the anatomy of the hepatic artery. The first preoperative arteriography in a case of a ruptured hepatic artery aneurysm causing hemobilia was performed by Kirklin [8] in 1955. Twenty of the reviewed eighty cases have been diagnosed preoperatively by this method (Table II.) Other methods of diagnosis include: barium studies of upper gastrointestinal tract, intravenous cholangiography, duodenoscopy with retrograde cholangiography, percutaneous splenoportography, and scintigraphy of the liver [4,9]. Unsuspected
Volume 133, February 1977
TABLE
II
Methods
Artery Aneurysm
of Diagnosis
System
Site Common
Aneurysm
Hepatic
Method Preoperatively Umbilical portography Arteriogram Clinical signs During operation Macroscopy Operative arteriogram Postoperatively Autopsy Arteriogram Not recorded Total
No. of Cases
1 20 2 20 2 29 2 4 80
hemobilia in patients operated upon for gastrointestinal bleeding may be found at the time of surgery by (1) palpation of the cystic and hepatic vessels for aneurysms or tumors, (2) palpation of the liver for masses or bruit, (3) cholecystostomy or aspiration of gallbladder, (4) choledochotomy, or (5) operative arteriogam [4,10,11]. Treatment. Hemobilia secondary to rupture of hepatic artery aneurysm has been managed surgically with increasing success during the past twenty-five years. Of sixty patients who underwent surgery, thirty-five (58.33 per cent) survived. Mester [12] in 1895 was the first to suggest ligation of the hepatic artery or one of its branches to treat a hepatic artery aneurysm ruptured into the biliary tract. Kehr [13] in 1903 was the first to successfully treat a patient with hemobilia secondary to a ruptured right hepatic artery aneurysm communicating with the cystic duct, by ligation and division of the hepatic artery proper. Ligation of the hepatic artery distal to the gastroduodenal artery interrupts the collateral circulation through the gastroduodenal and right gastric arteries with a higher degree of mortality and accordingly should not be done without reconstruction except when the aneurysm has completely occluded the artery [7]. Ligation or excision of the hepatic artery or its branches has been successfully accomplished in twenty-two cases [8,9,13-301. Other surgical methods that have been used in cases of hemobilia secondary to hepatic artery aneurysm are (1) excision of the aneurysm and restoration of the arterial blood flow with a prosthetic graft [31,32], (2) obliterative or reconstructive endoaneurysmorrhaphy [33-371, and (3) hepatic resection, which has been used chiefly in traumatic hemobilia and in cases of intrahepatic aneurysms [I 7,38-401.
231
tiarlaftis
Summary
Hepatic artery aneurysm is a rare vascular disease assqciated with high mortality caused by massive hemorrhage or by complications following surgical treatment. Over the past twenty-five years it has been managed surgically with increasing success. Eighty reported cases of hepatic artery aneurysms ruptured into the biliary tree were reviewed and the etiology, clinical signs, diagnosis, and treatment of such an aneurysm are discussed. A personal case with an aneurysm of the right hepatic artery ruptured into the common hepatic duct is reported. The diagnosis was made before surgery by arteriography and the aneurysm was successfully managed by ligation of the right hepatic artery both proximal and distal to the aneurysm, closure of the communication between the common hepatic duct and the aneurysm, and choledochal drainage. Hemobilia secondary to hepatic artery aneurysm must be considered in the differential diagnosis of unexplained gastrointestinal hemorrhage. References
5. 6. 7.
8.
9.
10.
11. 12. 13.
14.
15.
232
Glisson F: Anatomia hepatis. 1654. Cited in [ 41. Jackson JBS: Aneuysm of the hepatic artery bursting into the hepatic duct. A&d h&wine (Boston) 3: 115, 1834. Sandblom P: Hemontrage into the biliary tract fotlowlng trauma: “traumatic hemobilia.” Surgery 24: 571, 1948 Sandblom P: Hemobilia (biliary tract hemorrhage). History, Pathotogy, Diagnosis, Treatment. Springfield, Illinois, Charfes C. Thomas, 1972, p 5. Quincke H: Ein Fall von Aneurysma der Leberarterie. Bert k/in Wschr 30: 349. 1871. Siegel E: Ein Fall von traumatischer Gang&t der Gallenblasse. h&Inch Med Wschr 56: 341. 1909. Guida PM, Moore SW: Aneurysm of the hepatic artery: report of 5 cases with a brief review of the previously reported cases. Surgery 60: 299. 1966. Kirklin JW. Shocket E. Comfort MW, Huizenga KA: Treatment of aneurysm of the hepatic artery by excision: report of a case. Ann Surg 142: 110. 1955. Berenson MM, Freston JW: lntrahepatic artery aneurysm associated with hemobilia: report of a case. Gsstrcenterotogy 66: 254, 1974. Brown KL, Hcpwood HH. Engle JC: Hemobilia caused by rupture of an aneurysm of an anomalous right hepatic artery. Am Surg 39: 229. 1973. Guynn VL. Reynolds JT: Surgical management of hemobilia. Arch Surg 83: 89. 1961. Mester B: Das Aneurysma der Arteria Hepatica. Ztschr K/in Med 28: 93. 1895. Kehr H: Der erste Fall von erfolgeicher Unterbindurg dsr Arteria Hepatica Propia wegen Aneurysma. A&Inch Med Wschr 50: 1881.1903. Colmers F: lntrahepatisches Aneurysma und Gallenfistel nach Leberzerreissung. Heilung durch Unterbindung der Arteria Hepatica Communis. Bruns Beitr K/in Chir 122: 324, 1921. Devin R, Mambrini A, Winninger A, Febvey M. D’Drso M, Bensussan: Un cas d’ ar&rysme de I’art&e cystiqua rompu dans la vesicule biliaire. Chirurgie 97: 332, 1971.
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