CASE REPORT
Hemosuccus Pancreaticus Secondary to Ruptured Splenic Artery Aneurysm J A M E S R. S T A R L I N G , MD, and A N D R E W B. CRUMMY, MD
Hemorrhage through the pancreatic duct into the duodenum is a rare presentation of upper gastrointestinal bleeding. It is most commonly associated with inflammatory disorders of the pancreas with bleeding originating from an area of hemorrhagic pancreatitis, abscess, or p s e u d o c y s t . A pseudoaneurysm can form as a result of erosion and autodigestion by the inflammatory process of a major blood vessel (1, 2). Sandblom (3) designated this condition " h e m o s u c c u s p a n c r e a t i c u s " in his report of two cases of splenic artery aneurysms which ruptured into the pancreatic ducts. There have been only eleven reported cases of upper gastrointestinal bleeding due to rupture of a splenic artery aneurysm into a pancreatic duct (4). More frequently splenic artery aneurysms rupture into the greater or lesser peritoneal sacs, stomach (5, 6), retroperitoneum (7), colon (8), or splenic vein (7). We present a patient with a prolonged history of upper gastrointestinal bleeding in which the diagnosis of hemosuccus pancreaticus secondary to a ruptured splenic artery a n e u r y s m was diagnosed p r e o p e r a t i v e l y . Cure was obtained with bipolar ligation of the splenic artery and ligation of the area of erosion into the pancreatic duct. This approach is safer and simpler than distal pancreatectomy and splenectomy (4), CASE REPORT A 59-year-old male was first admitted to the hospital in February 1972 complaining of epigastric pain, belching, From the Departments of Surgery and Radiology, Veterans Administration Hospital, and University of Wisconsin Hospitals, Madison, Wisconsin 53792. Address for reprint requests: Dr. James R. Starling, Department of Surgery, Room H4/328, Clinical Sciences Center, 600 University Avenue, Madison, Wisconsin 53792.
726
intermittent hematemesis, and weight loss. He had a history of moderate alcohol intake. Abdominal x-rays revealed pancreatic calcification. An upper gastrointestinal series showed a hiatal hernia and minimal duodenal bulb deformity. Fiberoptic upper endoscopy showed the hiatal hernia and a normal stomach and duodenum. Acute and chronic esophagitis was found on biopsy. His hematocrit was 39%. He improved on antacid therapy. In March 1973 he was readmitted because of severe epigastric pain and hematemesis. His hematocrit was 32% with hypochromic, microcytic indices. Fiberoptic endoscopy revealed only minimal esophagitis without a specific bleeding point. During the next two years, he was hospitalized three additional times because of intermittent epigastric pain and persistent anemia. Endoscopy repeated twice was nondiagnostic. On one admission his serum amylase and lipase levels were elevated. The patient was hospitalized for the seventh time in February 1975 with daily epigastric pain which did not respond to food, antacids, or abstinence from alcohol and caffeine. His hematocrit was 34% and hemoglobin value 10.5. His stools were positive for occult blood. An ultrasonogram and x-rays suggested a 7 • 10-cm pseudocyst in the head of the pancreas. Although cystogastrostomy was performed, his epigastric pain and anemia persisted. The patient returned to the hospital in October 1977 with severe intermittent epigastric pain, melena, anemia, and a 10-kg weight loss. Fiberoptic endoscopy did not show a bleeding site. Shortly after admission he had a 1000-ml bright red emesis with severe abdominal pain and orthostatic hypotension. Repeat endoscopy showed blood in the duodenum and stomach, but failed to reveal a specific bleeding site. The patient responded favorably to transfusions and his pain subsided. Visceral angiography demonstrated a 3-cm splenic artery aneurysm as well as vascular changes consistent with chronic pancreatitis, a thrombosed splenic vein, and duodenal varices (Figure 1). At surgery, chronic pancreatitis with peripancreatic scarring was found. A 3-cm splenic artery aneurysm was palpated along the superior margin of the mid-body of the pancreas. Proximal and distal control of the splenic artery was obtained, and the aneurysm was opened. Inspection revealed a splenic artery pseudoaneurysm eroding into the pancreas with evidence of recent hemorrhage and orDigestive Diseases and Sciences, Vol. 24, No. 9 (September 1979)
0163-2116/79/0900-0726503.00/19 1979DigestiveDiseaseSystems, Inc.
HEMOSUCCUS PANCREATICUS
Fig 1. Splenic arteriogram; arrow indicates 2-cm splenic pseudoaneurysm.
ganized thrombi of various dates. The splenic artery was ligated proximally and distally. The area of erosion into the pancreatic duct was suture ligated with fine wire and the area drained. The patient did well and one year later is asymptomatic, has a normal hematocrit, and has gained 15 kg. DISCUSSION The incidenCe of splenic aneurysm rupture reported in the literature ranges from 5.3% to 46% (913). Excluding pregnant and multiparous women, no single predisposing factor has been determined to account for splenic artery rupture (10, 13, 14). Severe upper gastrointestinal hemorrhage secondary to pancreatitis has been recognized for years (2). The occurrence of visceral artery aneurysms as a complication of chronic pancreatitis is increasingly recognized with the use of angiography. According to White, 10% of patients with chronic pancreatitis have visceral pseudoaneurysms (15). In the series of 60 splenic artery aneurysms reported by Stanley et al, two of the six aneurysms that ruptured had coexistent chronic pancreatitis (13). We recently have had two patients with chronic pancreatitis who died because of exsanguinating bleeds from ruptured dorsal pancreatic and pancreaticoduodenal artery aneurysms. Ten of the 11 previously reported cases of hemorrhage through the pancreatic duct from a ruptured splenic artery aneurysm had coexistent chronic pancreatitis (4). Our patient is typical, with chronic Digestive Diseases and Sciences, Vol. 24, No. 9 (September 1979)
recurrent epigastric pain, coexistent chronic pancreatitis, anemia, with intermittent hematemesis and melena. It is highly likely, but not absolutely certain, that the bleeding was from the aneurysm. Repeated upper gastrointestinal evaluations, including seven endoscopies, were unsuccessful in diagnosis. It is apparent that this patient should have had angiography earlier. Considering the juxtaposition of the splenic artery and the pancreas, it is not surprising that splenic artery pseudoaneurysms resulting from audiodigestion by pancreatic enzymes are frequently associated with, or secondary to, pancreatitis (Figure 2). From the previous case reports, it appears that most cases of hemosuccus pancreaticus secondary to a ruptured splenic artery aneurysm involved the bifurcation of the splenic and pancreatica magna artery. Only one case report specifically localized the ruptured aneurysm to the caudal pancreatic artery (16), The operation suggested for a ruptured splenic artery aneurysm into a pancreatic duct is distal pancreatectomy, resection of the aneurysm, and splenectomy (4). Unless technically impossible because the normal anatomy is altered in the presence of intense peripancreatic inflammation, it may not be necessary to undertake such a large procedure. For r u p t u r e d or u n r u p t u r e d proMmal splenic aneurysms, bipolar ligation and excision of the aneurysm will spare the patient splenectomy. The extensive blood supply from the stomach via the short
727
STARLING AND CRUMMY
~ ~:~
. ~
~ ~
pan~-.uocl, a. 4 DorSal pancreatic a. 5--Pancreatica magna a. 6 Caudal pancreatic a. 7 Splenic a 8 Left gastroepipioica a. 9 Short gastric a.
Fig 2. Course of splenic artery and arterial supply of pancreas. gastric arteries (vasa brevia) and gastroepiploic artery will prevent infarction and maintain function of the retained spleen. Indeed, for years surgeons have occasionally ligated the splenic artery or even attempted utilization of the splenic artery for revascularization of other organs without adverse sequelae (17). If the area of erosion into the pancreatic ducts can be localized, simple suture ligation with fine wire will eliminate the need for pancreatic resection. We report the twelfth instance of upper gastrointestinal bleeding secondary to rupture of a splenic artery aneurysm into the pancreatic duct. Visceral artery aneurysms are common in patients with pancreatitis and, therefore, those with obscure or atypical bleeding should have early angiography rather than costly, multiple, upper gastrointestinal series, endoscopy, and transfusions. SUMMARY A case is presented of a ruptured splenic artery aneurysm communicating with the main pancreatic duct which resulted in obscure upper gastrointestinal bleeding. Bipolar ligation of the splenic artery and excision of the pseudoaneurysm was curative. The location of the splenic artery makes it especially vulnerable to aneurysmal formation in patients with pancreatitis. It is recommended that
728
early arteriography be obtained in patients with obscure upper gastrointestinal bleeding. If the splenic artery aneurysm is located in the mid-portion of the vessel, partial pancreatectomy and splenectomy are not necessary to effect a cure.
REFERENCES 1. Buckman CA: Arterial hemorrhage in pseudocyst of pancreas. Arch Surg 92:405-409, 1966 2. Hailer JD, Pena C, Dargan EL: Massive upper gastrointestinal hemorrhage due to pancreatitis. Arch Surg 93:567-572, 1966 3. Sandblom P: Gastrointestinal hemorrhage through the pancreatic duct. Ann Surg 171:61-66, 1970 4. Bivins BA, Sachatello CR, Chuang VP, Brady P: Hemosuccus pancreaticus. Arch Surg 113:751-753, 1978 5. Hootkin LA, Corco AE: Ruptured splenic artery aneurysm with resultant upper gastrointestinal bleeding. Am J Dig Dis 15:848-850, 1961 6. Schecter LM, Gordon HE, Passard E: Massive hemorrhage from the celiac axis in pancreatitis. Am J Surg 128:301-305, 1974 7. Jones EL, Finnay GG: Splenic artery aneurysm. Arch Surg 97:640-647, 1968 8. Moore SW, Lewis RJ: Splenic artery aneurysm, Ann Surg 153:1033-1045, 1961 9. Babb RR: Aneurysms of the splenic artery. Arch Surg 111:924-925, 1976 10. Biijsen E, Efsing HO: Aneurysm of the splenic artery, Acta Radiol 8:29-41, 1969 11. Carlisle BB, Lawler MR: Aneurysm of the splenic artery. Am J Surg 114:443-447, 1967 Digestive Diseases and Sciences, Vol. 24, No. 9 (September 1979)
HEMOSUCCUS PANCREATICUS 12. Owens JC, Coffey RJ: Aneurysms of splenic artery, including a report of six additional cases. Int Abst Surg 97:313335, 1953 13. Stanley SC, Thompson NW, Fry WJ: Splanchnic artery aneurysms. Arch Surg 101:689-697, 1970 14. MacFarlane JR, Thorbjarnarson B: Rupture of splenic artery aneurysm during pregnancy. Am J Obstet Gynecol 95:10251037, 1966
Digestive Diseases and Sciences, Vol. 24, No. 9 (September 1979)
15. White AF, Baum S, Buranasir S: Aneurysm secondary to pancreatis. Am J Roentgenol 127:373-396, 1976 16. Koehler PR, Nelson JA, Berenson MM: Massive extra-enteric gastrointestinal bleeding. Angiographic diagnosis. Radiology 119:41-44, 1976 17. Edwards WS, Lewis CE, Blakely WR, Napoiitano L: Coronary artery bypass with internal mammary and splenic artery grafts. Ann Thorac Surg 15:35-40, 1973
729
Hemosuccus Pancreaticus Secondary to Ruptured Splenic Artery Aneurysm J A M E S R. S T A R L I N G , MD, and A N D R E W B. CRUMMY, MD
Hemorrhage through the pancreatic duct into the duodenum is a rare presentation of upper gastrointestinal bleeding. It is most commonly associated with inflammatory disorders of the pancreas with bleeding originating from an area of hemorrhagic pancreatitis, abscess, or p s e u d o c y s t . A pseudoaneurysm can form as a result of erosion and autodigestion by the inflammatory process of a major blood vessel (1, 2). Sandblom (3) designated this condition " h e m o s u c c u s p a n c r e a t i c u s " in his report of two cases of splenic artery aneurysms which ruptured into the pancreatic ducts. There have been only eleven reported cases of upper gastrointestinal bleeding due to rupture of a splenic artery aneurysm into a pancreatic duct (4). More frequently splenic artery aneurysms rupture into the greater or lesser peritoneal sacs, stomach (5, 6), retroperitoneum (7), colon (8), or splenic vein (7). We present a patient with a prolonged history of upper gastrointestinal bleeding in which the diagnosis of hemosuccus pancreaticus secondary to a ruptured splenic artery a n e u r y s m was diagnosed p r e o p e r a t i v e l y . Cure was obtained with bipolar ligation of the splenic artery and ligation of the area of erosion into the pancreatic duct. This approach is safer and simpler than distal pancreatectomy and splenectomy (4), CASE REPORT A 59-year-old male was first admitted to the hospital in February 1972 complaining of epigastric pain, belching, From the Departments of Surgery and Radiology, Veterans Administration Hospital, and University of Wisconsin Hospitals, Madison, Wisconsin 53792. Address for reprint requests: Dr. James R. Starling, Department of Surgery, Room H4/328, Clinical Sciences Center, 600 University Avenue, Madison, Wisconsin 53792.
726
intermittent hematemesis, and weight loss. He had a history of moderate alcohol intake. Abdominal x-rays revealed pancreatic calcification. An upper gastrointestinal series showed a hiatal hernia and minimal duodenal bulb deformity. Fiberoptic upper endoscopy showed the hiatal hernia and a normal stomach and duodenum. Acute and chronic esophagitis was found on biopsy. His hematocrit was 39%. He improved on antacid therapy. In March 1973 he was readmitted because of severe epigastric pain and hematemesis. His hematocrit was 32% with hypochromic, microcytic indices. Fiberoptic endoscopy revealed only minimal esophagitis without a specific bleeding point. During the next two years, he was hospitalized three additional times because of intermittent epigastric pain and persistent anemia. Endoscopy repeated twice was nondiagnostic. On one admission his serum amylase and lipase levels were elevated. The patient was hospitalized for the seventh time in February 1975 with daily epigastric pain which did not respond to food, antacids, or abstinence from alcohol and caffeine. His hematocrit was 34% and hemoglobin value 10.5. His stools were positive for occult blood. An ultrasonogram and x-rays suggested a 7 • 10-cm pseudocyst in the head of the pancreas. Although cystogastrostomy was performed, his epigastric pain and anemia persisted. The patient returned to the hospital in October 1977 with severe intermittent epigastric pain, melena, anemia, and a 10-kg weight loss. Fiberoptic endoscopy did not show a bleeding site. Shortly after admission he had a 1000-ml bright red emesis with severe abdominal pain and orthostatic hypotension. Repeat endoscopy showed blood in the duodenum and stomach, but failed to reveal a specific bleeding site. The patient responded favorably to transfusions and his pain subsided. Visceral angiography demonstrated a 3-cm splenic artery aneurysm as well as vascular changes consistent with chronic pancreatitis, a thrombosed splenic vein, and duodenal varices (Figure 1). At surgery, chronic pancreatitis with peripancreatic scarring was found. A 3-cm splenic artery aneurysm was palpated along the superior margin of the mid-body of the pancreas. Proximal and distal control of the splenic artery was obtained, and the aneurysm was opened. Inspection revealed a splenic artery pseudoaneurysm eroding into the pancreas with evidence of recent hemorrhage and orDigestive Diseases and Sciences, Vol. 24, No. 9 (September 1979)
0163-2116/79/0900-0726503.00/19 1979DigestiveDiseaseSystems, Inc.
HEMOSUCCUS PANCREATICUS
Fig 1. Splenic arteriogram; arrow indicates 2-cm splenic pseudoaneurysm.
ganized thrombi of various dates. The splenic artery was ligated proximally and distally. The area of erosion into the pancreatic duct was suture ligated with fine wire and the area drained. The patient did well and one year later is asymptomatic, has a normal hematocrit, and has gained 15 kg. DISCUSSION The incidenCe of splenic aneurysm rupture reported in the literature ranges from 5.3% to 46% (913). Excluding pregnant and multiparous women, no single predisposing factor has been determined to account for splenic artery rupture (10, 13, 14). Severe upper gastrointestinal hemorrhage secondary to pancreatitis has been recognized for years (2). The occurrence of visceral artery aneurysms as a complication of chronic pancreatitis is increasingly recognized with the use of angiography. According to White, 10% of patients with chronic pancreatitis have visceral pseudoaneurysms (15). In the series of 60 splenic artery aneurysms reported by Stanley et al, two of the six aneurysms that ruptured had coexistent chronic pancreatitis (13). We recently have had two patients with chronic pancreatitis who died because of exsanguinating bleeds from ruptured dorsal pancreatic and pancreaticoduodenal artery aneurysms. Ten of the 11 previously reported cases of hemorrhage through the pancreatic duct from a ruptured splenic artery aneurysm had coexistent chronic pancreatitis (4). Our patient is typical, with chronic Digestive Diseases and Sciences, Vol. 24, No. 9 (September 1979)
recurrent epigastric pain, coexistent chronic pancreatitis, anemia, with intermittent hematemesis and melena. It is highly likely, but not absolutely certain, that the bleeding was from the aneurysm. Repeated upper gastrointestinal evaluations, including seven endoscopies, were unsuccessful in diagnosis. It is apparent that this patient should have had angiography earlier. Considering the juxtaposition of the splenic artery and the pancreas, it is not surprising that splenic artery pseudoaneurysms resulting from audiodigestion by pancreatic enzymes are frequently associated with, or secondary to, pancreatitis (Figure 2). From the previous case reports, it appears that most cases of hemosuccus pancreaticus secondary to a ruptured splenic artery aneurysm involved the bifurcation of the splenic and pancreatica magna artery. Only one case report specifically localized the ruptured aneurysm to the caudal pancreatic artery (16), The operation suggested for a ruptured splenic artery aneurysm into a pancreatic duct is distal pancreatectomy, resection of the aneurysm, and splenectomy (4). Unless technically impossible because the normal anatomy is altered in the presence of intense peripancreatic inflammation, it may not be necessary to undertake such a large procedure. For r u p t u r e d or u n r u p t u r e d proMmal splenic aneurysms, bipolar ligation and excision of the aneurysm will spare the patient splenectomy. The extensive blood supply from the stomach via the short
727
STARLING AND CRUMMY
~ ~:~
. ~
~ ~
pan~-.uocl, a. 4 DorSal pancreatic a. 5--Pancreatica magna a. 6 Caudal pancreatic a. 7 Splenic a 8 Left gastroepipioica a. 9 Short gastric a.
Fig 2. Course of splenic artery and arterial supply of pancreas. gastric arteries (vasa brevia) and gastroepiploic artery will prevent infarction and maintain function of the retained spleen. Indeed, for years surgeons have occasionally ligated the splenic artery or even attempted utilization of the splenic artery for revascularization of other organs without adverse sequelae (17). If the area of erosion into the pancreatic ducts can be localized, simple suture ligation with fine wire will eliminate the need for pancreatic resection. We report the twelfth instance of upper gastrointestinal bleeding secondary to rupture of a splenic artery aneurysm into the pancreatic duct. Visceral artery aneurysms are common in patients with pancreatitis and, therefore, those with obscure or atypical bleeding should have early angiography rather than costly, multiple, upper gastrointestinal series, endoscopy, and transfusions. SUMMARY A case is presented of a ruptured splenic artery aneurysm communicating with the main pancreatic duct which resulted in obscure upper gastrointestinal bleeding. Bipolar ligation of the splenic artery and excision of the pseudoaneurysm was curative. The location of the splenic artery makes it especially vulnerable to aneurysmal formation in patients with pancreatitis. It is recommended that
728
early arteriography be obtained in patients with obscure upper gastrointestinal bleeding. If the splenic artery aneurysm is located in the mid-portion of the vessel, partial pancreatectomy and splenectomy are not necessary to effect a cure.
REFERENCES 1. Buckman CA: Arterial hemorrhage in pseudocyst of pancreas. Arch Surg 92:405-409, 1966 2. Hailer JD, Pena C, Dargan EL: Massive upper gastrointestinal hemorrhage due to pancreatitis. Arch Surg 93:567-572, 1966 3. Sandblom P: Gastrointestinal hemorrhage through the pancreatic duct. Ann Surg 171:61-66, 1970 4. Bivins BA, Sachatello CR, Chuang VP, Brady P: Hemosuccus pancreaticus. Arch Surg 113:751-753, 1978 5. Hootkin LA, Corco AE: Ruptured splenic artery aneurysm with resultant upper gastrointestinal bleeding. Am J Dig Dis 15:848-850, 1961 6. Schecter LM, Gordon HE, Passard E: Massive hemorrhage from the celiac axis in pancreatitis. Am J Surg 128:301-305, 1974 7. Jones EL, Finnay GG: Splenic artery aneurysm. Arch Surg 97:640-647, 1968 8. Moore SW, Lewis RJ: Splenic artery aneurysm, Ann Surg 153:1033-1045, 1961 9. Babb RR: Aneurysms of the splenic artery. Arch Surg 111:924-925, 1976 10. Biijsen E, Efsing HO: Aneurysm of the splenic artery, Acta Radiol 8:29-41, 1969 11. Carlisle BB, Lawler MR: Aneurysm of the splenic artery. Am J Surg 114:443-447, 1967 Digestive Diseases and Sciences, Vol. 24, No. 9 (September 1979)
HEMOSUCCUS PANCREATICUS 12. Owens JC, Coffey RJ: Aneurysms of splenic artery, including a report of six additional cases. Int Abst Surg 97:313335, 1953 13. Stanley SC, Thompson NW, Fry WJ: Splanchnic artery aneurysms. Arch Surg 101:689-697, 1970 14. MacFarlane JR, Thorbjarnarson B: Rupture of splenic artery aneurysm during pregnancy. Am J Obstet Gynecol 95:10251037, 1966
Digestive Diseases and Sciences, Vol. 24, No. 9 (September 1979)
15. White AF, Baum S, Buranasir S: Aneurysm secondary to pancreatis. Am J Roentgenol 127:373-396, 1976 16. Koehler PR, Nelson JA, Berenson MM: Massive extra-enteric gastrointestinal bleeding. Angiographic diagnosis. Radiology 119:41-44, 1976 17. Edwards WS, Lewis CE, Blakely WR, Napoiitano L: Coronary artery bypass with internal mammary and splenic artery grafts. Ann Thorac Surg 15:35-40, 1973
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