Pancreaticoduodenal Artery Aneurysms Associated with Celiac Axis Stenosis: Report of Two Cases and Review of the Literature Pierre Quandalle, MD, Jean-Pierre Chambon, MD, Philippe Marache, MD, Alain Saudemont, MD, Benoit Maes, MD, Lillc, France
We have endeavored to define the incidence of pancreaticoduodenal artery aneurysm associated with stenosis of the celiac axis and to address modalities in this setting. This association was found in 23 of 34 cases. Aneurysmal dilation of the pancreatic arteries associated with celiac axis stenosis is not accidental and seems to be due to increased flow through the peripancreatic arteries. Hemorrhagic complications, i.e., peripancreatic hematoma, hernoperitoneum, ductal hemorrhage, were the initial manifestation in 16 of 34 cases. Isolated aneurysms were treated by resection, exclusion or embolization without any complications. For associated aneurysms, resection and exclusion were employed. Pancreatoduodenectomywas required in three cases in which bleeding was uncontrollable. In six cases, the celiac axis was restored either by arterial reconstruction, aortohepatic bypass, or division of the arcuate ligament. Four patients died. All had been operated on for ruptured aneurysm. None of the deaths was directly due to ischemic compromise of the celiac artery bed. Because of the risk of rupture, all pancreaticoduodenal artery aneurysms should be treated surgically after appropriate investigation to detect associated celiac axis stenosis. Revascularization of the celiac axis is indicated whenever blood flow is reduced or, routinely, to reduce high peripancreatic collateral flow, particularly when collaterality is due to arcuate ligament-related stenosis. (Ann Vasc Surg 1990;4:540-545). KEY WORDS: Pancreaticoduodenal artery aneurysm; celiac artery; celiac axis stenosis; median arcuate ligament division.
Pancreaticoduodenal artery aneurysms represent 3% of all visceral artery aneurysms [I]. They differ from posttraumatic pseudoaneurysms and aneurysms occurring during the course of acute or chronic pancreatitis [2-4] and should be considered From the Service de Chirurgie, HGpitul Claude Huriez and Service de Radiologie, Hfipital Cardiologique, Centre Hospitalier et Universitaire de Lille, France. Presented at the Annual Meeting of the Sociktk de Chirurgie Vasculaire de Langue Franfaise, June 23-24, 1989, Strasbourg, France. Reprint requests: P . Quandalle, M D , Service de Chirurgie, H6pital Claude Huriez, C H U , 59037 Lille CP'dex. France.
as separate entities. The association of pancreaticoduodenal aneurysms with celiac axis stenosis has often been cited since the first publication by Sutton in 1973 [ S ] . The aim of this study was to describe the incidence, the possible anatomic and clinical consequences, and the treatment of this association.
MATERIAL AND METHODS Case reports
Patient N o . I : A 42-year-old man, with known alcoholic cirrhosis, was admitted on September 3, 1987, for hematemesis due to ruptured esophageal varices. Endo-
540
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PANCREATICODUODENAL ARTER Y A N E U R Y S M S
Fig. 1. Computerized tomography showing prepancreatic hematoma (Patient No. 2).
scopic sclerotherapy was performed. Celiac and mesenteric arteriograms documented a 5 mm nonruptured aneurysm located on the inferior pancreaticoduodenal artery. The celiac axis was tightly compressed by the arcuate ligament whereas the penpancreatic arteries were dilated. The hepatic bed was visualized by back flow originating from reversed flow through the gastroduodenal artery. Child's classification was C (10). As the patient was unfit for surgery. the aneurysm was not operated and the patient died on December 16, 1988, of hepatic failure. Patient N o . 2: A 62-year-old man experienced sudden pain in the upper right quadrant and flank in January 1987. associated with a transient drop in systolic blood pressure of 75 mmHg. Hemoglobin was 8.5 gidl. After emergency supportive care, a computed tomographic (CT) scan was obtained and showed an anterior homogenous collection in the pancreatic lodge (Fig. I ) . Arteriograms demonstrated a fusiform aneurysm of the infrarenal abdominal aorta. Because of stenosis, it was impossible to catheterize the celiac axis. Visualization of the superior mesenteric artery showed dilation of the pancreaticoduodenal arteries and an aneurysm located on an anterior branch of the dorsal pancreatic artery. Extravasation of contrast medium was seen (Figs. 2 , 3), attesting to rupture of the aneurysm. At operation, initial control of the supraceliac aorta was obtained. After evacuation of hemoperitoneum and a hematoma on the anterior aspect of the pancreas, the remaining pancreatic parenchyma was seen to be intact. A small anfractuous cavity, filled with a blood clot, was sutured through-and-through. The pressure gradient in the splenic artery compared to the aorta was 80 mm. The celiac axis was then approached and the stenosis was found to be due to compression by the arcuate ligament. Once the arcuate ligament was divided, the pressure gradient dropped to 40 mmHg. The patient's postoperative course was uneventful. In March 1988, the patient underwent surgical cure of his aortic aneurysm. Review of the literature
ThirtY-two cases of P a n c r e a t i c o d u o d e n a l a r t e r y aneurysm have been published since 1973 when this
541
Fig. 2. Superior mesenteric arteriogram: peripancreatic arteries are dilated and revascularize the liver. Aneurysm is located on dorsal pancreatic artery (Patient No. 2).
association w a s first described 151. O n l y reports with d o c u m e n t e d arteriographic o r a n a t o m i c studies will be c o n s i d e r e d h e r e . T h e y h a v e b e e n divided into t w o g r o u p s according t o w h e t h e r they were a s s o c i a t e d or not with s t e n o s i s of t h e celiac axis ( T a b l e s 1 a n d 11).
RESULTS Of t h e total of 34 r e p o r t e d cases of pancreaticoduodenal a n e u r y s m s , 23, including o u r two
Fig. 3. Superior mesenteric artery arteriogram: arrow indicates extravasation attesting to aneurysmal rupture (Patient No. 2).
P A N C R E A T I C O D U O D E N A L ARTERY A N E U R Y S M S
542
TABLE I.-Reported
Author Sutton [5] 1973 Mora [6] 1976 Schefflan [7] 1977 Kadir [8] 1978 Proud [9] 1978 Roback [ l o ] 1979 Ho [ l l ] 1979 Vermynck [12] 1979 Mariano [13] 1981 Samson [14] 1981 Vernhet [15] 1982
Sex/age F121 F169 Fl44
Aneurysm status Nonruptured Nonruptured Nonruptured
Aneurysm diameter (mm) 13 15 20
Management No surgery Exclusion Revascularization of celiac axis
Outcome Unknown Favorable Favorable*
Pancreatoduodenectomy Revascularizationof hepatic artery Colectomy
Favorable
Favorable Favorable Favorable*
Ruptured into pancreas
20
MI72
Ruptured into hollow viscus Nonruptured Nonruptured Nonruptured
35
Ruptured into peritoneal cavity Ruptured into peritoneal cavity Nonruptured
20
Resection Resection Section of arcuate ligament of diaphragm Exclusion
40
No surgery
Died
20
Favorable
Ruptured into peritoneal cavity Nonruptured
NA
Resection Aortohepatic bypass Exclusion
30
Resection
Favorable
15
Exclusion
Favorable
NA
Exclusion
Favorable
F137
Ruptured into peritoneal cavity Ruptured into pancreas Nonruptured
30
Favorable
MI38
Nonruptured
30
Resection vascular reconstruction Resection
Fl25
Nonruptured
25
Resection
Favorable
Mi61
Ruptured into Wirsung’s canal Ruptured into pancreas Ruptured into peritoneal cavity
70
Resection vascular reconstruction Pancreatoduodenectomy
Favorable
Pancreatoduodenectomy Section of median arcuate ligament of diaphragm No surgery Exclusion section of median arcuate ligament of diaahraam
Died
MI26 Fl63 Fl32 Fl49 MI64 Fl44 Fl67 Fl56 MI53
MI58 F159
Quandalle (this report) 1990
cases of pancreaticoduodenalartery aneurysms associated with celiac axis stenosis (Group I)
Mi56
MI65 Thevenet [16] 1983 Partensky [17] 1984 Foster [18] 1985 Gangahar [19] 1985 Ambrosetti [20] 1987
ANNAISOF VASCULAR SURGERY
MI42 Mi62
Nonruptured Ruptured
20 15 10
NA NA 5 4
I
.
Died
Died
Favorable
Favorable
Favorable
Died Favorable
,
‘Spontaneous thrombosis of aneurysm
cases, were associated with celiac axis stenosis (group I). In the I 1 other cases (group II), the causes of aneurysm were classic, including atheroma, angiodysplasia, and Takayasu’s disease. Patients’ ages at the time of discovery ranged from 21 to 69 years (mean 51 years) for group I and from 46 to 75 years (mean 53 years) for group 11. The male/female sex ratio was close to 1 : 1 within the two groups of 34 patients. The cause of celiac axis stenosis was not always clear. According to arteriographic or operative findings, arcuate ligament compression was the cause in two of our cases and in five others [5,9,15,16,20]. Other mechanisms such as atheroma and agenesis of the celiac axis, have been mentioned [I 1,171.
Revascularization of the hepatic artery through the gastroduodenal artery and development of anastomoses between the pancreaticoduodenal arteries are features that have always been underscored. The diameter of the aneurysm was evaluated in 23 cases, ranging from 4 to 70 mm (mean = 25 mm) without any difference being noted between the two groups. In 18 cases, (12 in group I , 6 in group 11), the aneurysm was discovered before any complication occurred, either by chance or during work-up for abdominal pain, which was occasionally accompanied with epigastric bruit upon auscultation. Aneurysmal calcifications on plain abdominal films combined with ultrasound and CT scans contributed to
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PANCREATICODUODENAL AKTER Y ANEUR YSMS
543
TABLE 11.-Reported cases of pancreaticoduodenalartery aneurysms nonassociated with celiac axis stenosis (Group II)
Author Murase [21] 1973 Spanos [22] 1974 Harris [23] 1975 Allegaert [24] (1976) Keehan [25] 1978 Noyes [26] 1979 Colluzza [27] 1981 Eyskens [3] 1982 Harbin [28] 1983 Thevenet [261 1983 Hubens [31] 1984
Sexlage MI48
Aneurysm status Nonruptured
MI58
Ruptured into peritoneal cavity Nonruptured
Aneurysm diameter (mm) 30
Management Resection
Outcome Favorable
10
Exclusion
Favorable
NA*
Exclusion
Favorable
NA
Exclusion
Favorable
NA
Resection
Favorable
Fl49
Ruptured into peritoneal cavity Ruptured into retroperitoneum Nonruptured
NA
Resection
FavorabIe
MI54
Nonruptured
NA
Exclusion
Favorable
Fl52
15
Resection
Favorable
Fl63
Ruptured into peritoneal cavity Nonruptured
20
Ernbolization
Favorable
F146
Nonruptured
NA
No surgery
NA
F147
Ruptured into peritoneal cavity
NA
Resection
Favorable
Fl54 MI45 MI75
*Not available
correct diagnoses. Arteriograms were obtained either as part of initial evaluation or to further delineate an anomaly found during other investigations. In the 16 other cases (1 1 in group I, 5 in group ll), rupture was the initial clinical manifestation. The consequence was hemoperitoneum, anterior or posterior peripancreatic or retroperitoneal hematoma, or intraductal hemorrhage, according to whether the aneurysm was anterior, posterior or intrapancreatic. Emergency clinical, sonographic and CT scan findings were easily diagnostic of hemorrhage and its localization. Arteriograms were used to identify the aneurysm and the eventual stenosis of the celiac axis. In four cases, arteriograms were obtained after initial laparotomy during which the aneurysm had been missed [6-8,19]. In three of these cases, the lesions were cured by reoperation [6,7,19]. Therapeutic decisions varied according to whether the celiac axis was stenotic or not, and whether the aneurysm had ruptured or not (Tables 111 and IV). The technique employed to treat the aneurysm was either resection or exclusion. These procedures were difficult to perform when the aneurysm was located behind the pancreas, impinged on the pancreas, or had already ruptured. Emergency pancreaticoduodenectomy was required to stop hemorrhaging in three instances in this group of patients. As this procedure interrupted the pancreaticoduodenal anastomoses, hepatic artery revascularization was necessary in two instances, requiring either division of the arcuate ligament or a bypass procedure [7,201.
In group I, after resection or exclusion performed in 14 cases, restoration of the hepatic artery was deemed necessary in four instances, either by reconstruction of the peripancreatic arterial network [16,19], aortohepatic bypass [12], or by division of the arcuate ligament (our Patient No. 2). In two cases of nonruptured aneurysms, 10 and 20 mm in diameter, the arcuate ligament was simply divided or the celiac artery was revascularized with complete disappearance of lesions on postoperative repeat arteriograms [6,91. In four instances, the patient was not operated on. The reasons for therapeutic abstention included
TABLE Ill.-Management of pancreaticoduodenal artery aneurysms associated with celiac artery stenosis (Group I)
Manaaement No surgery Colectorny Section of median arcuate ligament Revascularization of celiac axis Resection or exclusion Pancreatoduodenectomy Pancreatoduodenectorny with revascularization Resection or exclusion with revascularization (
). Perioperative mortality
Ruptured Nonruptured aneurvsms aneurvsms
2
2
544
PANCREAI’ICODUODENAL ARTERY ANEURYSMS
TABLE IV.-Management of pancreaticoduodenal artery aneurysms associated with celiac artery stenosis (Group II*)
ANNALS OF VASCULARSURGERY
cular reconstruction. Although results of surgery have generally been satisfactory, embolization could be an alternative to surgery in the poor risk Ruptured Nonruptured patient or when the aneurysm is easily accessible by Manaaement aneurysms aneurvsms catheter [28,32]. No surgery For aneurysms associated with stenosis of the 1 Embolization 1 celiac axis, exclusion or resection have been emResection or exclusion 5 4 ployed. In three cases, however, these procedures ‘There was no perioperativemortality in this group of patients failed and pancreatoduodenectomy was necessary to stop the bleeding. The effects of these procedures on the celiac circulation must be evaluated by rapidly lethal, sudden vascular collapse due to measurement of the aortohepatic arterial pressure intraperitoneal rupture [ 111, refusal of surgery [ 161, gradient. Revascularization did not appear to be poor risk patient (case I), and deliberate abstention necessary in 9 of 14 cases of exclusion or resection and in one of three pancreatoduodenectomies. in a patient with a 13 mm diameter lesion. Four patients died after surgical treatment, all in Good tolerance could be explained by multiple group I and all having been operated on for compli- collateral supply. In the absence of complications, when the aneucated lesions. No complications due to ischemia in the celiac axis vascular bed were noted. The aneu- rysm is small or difficult to approach, treatment of rysm never recurred when the celiac axis was not the celiac artery stenosis alone appears to be an attractive alternative. This proved to be successful reconstructed. for Mora and Proud [6,9]. In addition to local treatment of the aneurysm, complementary revascularization of the celiac terDISCUSSION ritory can be considered to preclude recurrence by The association of pancreaticoduodenal artery reestablishing normal circulation. To this intent, an aneurysm with celiac stenosis is not coincidental. aortohepatic bypass or the division of the arcuate According to Sutton IS], the dilation of the arterial ligament should be preferred to reconstruction of walls leading to aneurysmal formation is the result the aneurysmal arterial segment left in place. of increased flow through these arteries to vascularize the celiac axis bed through the superior mesenteric artery. Atheroma may be a promoting CONCLUSION factor [3]. The reports of Mora [6] and Proud [9] are The study of aneurysms of pancreaticoduodenal remarkable demonstrations of this theory because the aneurysms disappeared on follow-up arterio- arteries shows that stenosis of the celiac axis is a grams after they revascularized the celiac axis. The determining but not exclusive factor in their pathoprecise incidence of this association with regard to genesis. This association should always be searched all celiac axis stenoses is not known. Thevenet [30] for, even in the emergency. setting, by preoperative reported one case in a series of 35. These aneu- arteriography whenever feasible. Treatment should rysms may be due to other causes, such as ather- strive to cure the aneurysm and restore normal oma and angiodysplasia, as mentioned in other blood flow to the celiac territory. studies [ 1,3,31]. Irrespective of their origin, the clinical features of aneurysms of the pancreaticoduodenal arteries are REFERENCES similar. In the absence of complications, symptoms I . STANLEY JC. Splanchnic artery aneurysms. In: RUTHare not very characteristic. In half of cases, rupture ERFORD RB (ed). Vascular Surgery. Philadelphia, W B is the initial manifestation and leads to severe 1977: pp. 673-684. hemorrhage. Arteriography is essential to the iden- 2. Saunders. ECKHAUSER FE, STANLEY JC. ZELENOCK GB, et al. tification and location of the lesion, as well as to the Gastroduodenal and pancreaticoduodenal artery aneurysms: safety of treatment. Arteriography is the only way a complication of pancreatitis causing spontaneous gastrointestinal hemorrhage. Surgery 1980;88:335-344. of identifying stenosis of the celiac axis. Surgical intervention can then be planned before a compli- 3. EYSKENS E. L’anevrysme des arttres pancrtaticoduodh a l e s : prksentation d’un cas personnel, revue et analyse de cation arises because even small aneurysms less la littkrature. Chirurgie 1982;/08:734-743. than one centimeter in diameter can rupture [3]. It 4. LERUT JP, GIANELLO PP, OTTE JB, et al. Les anevrysmes des branches du tronc coeliaque. J Chir (Paris) 1985;/22:93-100. must be remembered that rupture is a serious com5 . SUTTON D. LAWTON G. Celiac stenosis or occlusion with plication since death occurred in 4 of 16 cases aneurysm of the collateral supply. C/in Radio/ 197324:4%53. reported. 6. MORA JD. OBST D. Celiac axis artery stenosis with aneuAneurysms without stenosis of the celiac axis are rysmal calcification of the collateral supply. Ausr Radio1 best treated by exclusion or resection without vas1976;20:252.
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PANCREATICODUODENAL ARTERY A N E U R YSMS
7. SCHEFLAN M, KADl R S. ATHANASOULIS CA. et al. Pancreaticoduodenal artery aneurysm simulating carcinoma of the head of the pancreas. Arch S w g 1977:/12:1201-1203. 8. KADlR S. ATHANASOULIS CA. YUNE HY. et al. Aneurysms of the pancreaticoduodenal arteries in association with celiac axis occlusion. Curdiovusc Rtrdiol 1978:/: 173-177. 9. PROUD G , CHAMBERLAIN J. Aneurysm formation on the small pancreatic arteries in association with celiac axis compression. Ann R Coll Surg Engl 1978:60:294297. 10. ROBACK DL. Epigastric pain with failing hematocrit. J A M A 1979;242:463-464. 11. HO KL. Aneurysm of pancreaticoduodenal artery: report of a case and review of the literature. Inr Surg 1979;64:35-39. 12. VERMYNCK JP. BERTOUX JP, REMOND A. et al. Anevrysme des arteres pancreatico-duodenales associe B une thrombose du tronc coeliaque. Ann Chir 1979;13:43&432. 13. MARIANO EG. GIEGO RS. Aneurysm of the pancreaticoduodenal artery. J Med So(. N J 1981;78:191-193. 14. SAMSON ID. Successful resection of an aneurysm of the pancreaticoduodenal artery. Vasc S l u g 1981:15: 157-162. 15. VERNHET J , CORCOS J . Anevrysmes des artkres pancreaticoduodenales. Chirrrrgie 1982:/08:617424. 16. THEVENET A. JOYEUX A. Anevrysmes des arteres pancreatidoduodenales: B propos de deux cas. Chirrtrgic, 1983: 109:668-670. 17. PARTENSKY G , DESGOS L. CHAMPETIER P, et al. Anevrysme d'une arcade arterielle pancreatique "anormale" avec agCnCsie du tronc coeliaque. Gus/roenrerol Clin B i d 1987;11:260-261. 18. FOSTER DR. Inferior pancreaticoduodenal artery aneurysm presenting in a young woman. Br J Rudiol 198558: 1127-1129. 19. GANAHAR DM. CARVETH SW. REESE HE. et al. True aneurysm of the pancreaticoduodenal artery: a case report and report and review of the literature. J Vusc Surg 1985:2: 741-742. 20. AMBROSETTI P, MEYER P, MENTHA G. et al. Anevrys-
21.
22. 23. 24.
25. 26. 27. 28.
29. 30.
3I.
32.
545
mes des arteres pancreaticoduodenales et hepatiques. Chirurgie 1987;/1:3616. MURASE H. NAKAJIMA M, I T 0 Y, et al. Aneurysms of pancreaticoduodenal artery: a case successfully diagnosed and excised. Nippori Ntrika Gakkai Za.sslii 1973;62:765-769. SPANOS PK, KLOPPEDAL EA, MURRAY CA. Aneurysms of the gastroduodenal and pancreaticoduodenal arteries. A m J Srtrg 1974:127:345-348. HARRIS RD. ANDERSON JE, COEL MN. Aneurysms of the small pancreatic arteries: a cause of upper abdominal pain and intestinal bleeding. Radiology 1975;/ IS: 17-20. ALLEGAERT W. BOSTOEN H , PIL J . Geruptureerd aneurysma van der arteria pancreaticoduodenalis inferior. A
We have endeavored to define the incidence of pancreaticoduodenal artery aneurysm associated with stenosis of the celiac axis and to address modalities in this setting. This association was found in 23 of 34 cases. Aneurysmal dilation of the pancreatic arteries associated with celiac axis stenosis is not accidental and seems to be due to increased flow through the peripancreatic arteries. Hemorrhagic complications, i.e., peripancreatic hematoma, hernoperitoneum, ductal hemorrhage, were the initial manifestation in 16 of 34 cases. Isolated aneurysms were treated by resection, exclusion or embolization without any complications. For associated aneurysms, resection and exclusion were employed. Pancreatoduodenectomywas required in three cases in which bleeding was uncontrollable. In six cases, the celiac axis was restored either by arterial reconstruction, aortohepatic bypass, or division of the arcuate ligament. Four patients died. All had been operated on for ruptured aneurysm. None of the deaths was directly due to ischemic compromise of the celiac artery bed. Because of the risk of rupture, all pancreaticoduodenal artery aneurysms should be treated surgically after appropriate investigation to detect associated celiac axis stenosis. Revascularization of the celiac axis is indicated whenever blood flow is reduced or, routinely, to reduce high peripancreatic collateral flow, particularly when collaterality is due to arcuate ligament-related stenosis. (Ann Vasc Surg 1990;4:540-545). KEY WORDS: Pancreaticoduodenal artery aneurysm; celiac artery; celiac axis stenosis; median arcuate ligament division.
Pancreaticoduodenal artery aneurysms represent 3% of all visceral artery aneurysms [I]. They differ from posttraumatic pseudoaneurysms and aneurysms occurring during the course of acute or chronic pancreatitis [2-4] and should be considered From the Service de Chirurgie, HGpitul Claude Huriez and Service de Radiologie, Hfipital Cardiologique, Centre Hospitalier et Universitaire de Lille, France. Presented at the Annual Meeting of the Sociktk de Chirurgie Vasculaire de Langue Franfaise, June 23-24, 1989, Strasbourg, France. Reprint requests: P . Quandalle, M D , Service de Chirurgie, H6pital Claude Huriez, C H U , 59037 Lille CP'dex. France.
as separate entities. The association of pancreaticoduodenal aneurysms with celiac axis stenosis has often been cited since the first publication by Sutton in 1973 [ S ] . The aim of this study was to describe the incidence, the possible anatomic and clinical consequences, and the treatment of this association.
MATERIAL AND METHODS Case reports
Patient N o . I : A 42-year-old man, with known alcoholic cirrhosis, was admitted on September 3, 1987, for hematemesis due to ruptured esophageal varices. Endo-
540
VOLUME4 N O 6 - 1990
PANCREATICODUODENAL ARTER Y A N E U R Y S M S
Fig. 1. Computerized tomography showing prepancreatic hematoma (Patient No. 2).
scopic sclerotherapy was performed. Celiac and mesenteric arteriograms documented a 5 mm nonruptured aneurysm located on the inferior pancreaticoduodenal artery. The celiac axis was tightly compressed by the arcuate ligament whereas the penpancreatic arteries were dilated. The hepatic bed was visualized by back flow originating from reversed flow through the gastroduodenal artery. Child's classification was C (10). As the patient was unfit for surgery. the aneurysm was not operated and the patient died on December 16, 1988, of hepatic failure. Patient N o . 2: A 62-year-old man experienced sudden pain in the upper right quadrant and flank in January 1987. associated with a transient drop in systolic blood pressure of 75 mmHg. Hemoglobin was 8.5 gidl. After emergency supportive care, a computed tomographic (CT) scan was obtained and showed an anterior homogenous collection in the pancreatic lodge (Fig. I ) . Arteriograms demonstrated a fusiform aneurysm of the infrarenal abdominal aorta. Because of stenosis, it was impossible to catheterize the celiac axis. Visualization of the superior mesenteric artery showed dilation of the pancreaticoduodenal arteries and an aneurysm located on an anterior branch of the dorsal pancreatic artery. Extravasation of contrast medium was seen (Figs. 2 , 3), attesting to rupture of the aneurysm. At operation, initial control of the supraceliac aorta was obtained. After evacuation of hemoperitoneum and a hematoma on the anterior aspect of the pancreas, the remaining pancreatic parenchyma was seen to be intact. A small anfractuous cavity, filled with a blood clot, was sutured through-and-through. The pressure gradient in the splenic artery compared to the aorta was 80 mm. The celiac axis was then approached and the stenosis was found to be due to compression by the arcuate ligament. Once the arcuate ligament was divided, the pressure gradient dropped to 40 mmHg. The patient's postoperative course was uneventful. In March 1988, the patient underwent surgical cure of his aortic aneurysm. Review of the literature
ThirtY-two cases of P a n c r e a t i c o d u o d e n a l a r t e r y aneurysm have been published since 1973 when this
541
Fig. 2. Superior mesenteric arteriogram: peripancreatic arteries are dilated and revascularize the liver. Aneurysm is located on dorsal pancreatic artery (Patient No. 2).
association w a s first described 151. O n l y reports with d o c u m e n t e d arteriographic o r a n a t o m i c studies will be c o n s i d e r e d h e r e . T h e y h a v e b e e n divided into t w o g r o u p s according t o w h e t h e r they were a s s o c i a t e d or not with s t e n o s i s of t h e celiac axis ( T a b l e s 1 a n d 11).
RESULTS Of t h e total of 34 r e p o r t e d cases of pancreaticoduodenal a n e u r y s m s , 23, including o u r two
Fig. 3. Superior mesenteric artery arteriogram: arrow indicates extravasation attesting to aneurysmal rupture (Patient No. 2).
P A N C R E A T I C O D U O D E N A L ARTERY A N E U R Y S M S
542
TABLE I.-Reported
Author Sutton [5] 1973 Mora [6] 1976 Schefflan [7] 1977 Kadir [8] 1978 Proud [9] 1978 Roback [ l o ] 1979 Ho [ l l ] 1979 Vermynck [12] 1979 Mariano [13] 1981 Samson [14] 1981 Vernhet [15] 1982
Sex/age F121 F169 Fl44
Aneurysm status Nonruptured Nonruptured Nonruptured
Aneurysm diameter (mm) 13 15 20
Management No surgery Exclusion Revascularization of celiac axis
Outcome Unknown Favorable Favorable*
Pancreatoduodenectomy Revascularizationof hepatic artery Colectomy
Favorable
Favorable Favorable Favorable*
Ruptured into pancreas
20
MI72
Ruptured into hollow viscus Nonruptured Nonruptured Nonruptured
35
Ruptured into peritoneal cavity Ruptured into peritoneal cavity Nonruptured
20
Resection Resection Section of arcuate ligament of diaphragm Exclusion
40
No surgery
Died
20
Favorable
Ruptured into peritoneal cavity Nonruptured
NA
Resection Aortohepatic bypass Exclusion
30
Resection
Favorable
15
Exclusion
Favorable
NA
Exclusion
Favorable
F137
Ruptured into peritoneal cavity Ruptured into pancreas Nonruptured
30
Favorable
MI38
Nonruptured
30
Resection vascular reconstruction Resection
Fl25
Nonruptured
25
Resection
Favorable
Mi61
Ruptured into Wirsung’s canal Ruptured into pancreas Ruptured into peritoneal cavity
70
Resection vascular reconstruction Pancreatoduodenectomy
Favorable
Pancreatoduodenectomy Section of median arcuate ligament of diaphragm No surgery Exclusion section of median arcuate ligament of diaahraam
Died
MI26 Fl63 Fl32 Fl49 MI64 Fl44 Fl67 Fl56 MI53
MI58 F159
Quandalle (this report) 1990
cases of pancreaticoduodenalartery aneurysms associated with celiac axis stenosis (Group I)
Mi56
MI65 Thevenet [16] 1983 Partensky [17] 1984 Foster [18] 1985 Gangahar [19] 1985 Ambrosetti [20] 1987
ANNAISOF VASCULAR SURGERY
MI42 Mi62
Nonruptured Ruptured
20 15 10
NA NA 5 4
I
.
Died
Died
Favorable
Favorable
Favorable
Died Favorable
,
‘Spontaneous thrombosis of aneurysm
cases, were associated with celiac axis stenosis (group I). In the I 1 other cases (group II), the causes of aneurysm were classic, including atheroma, angiodysplasia, and Takayasu’s disease. Patients’ ages at the time of discovery ranged from 21 to 69 years (mean 51 years) for group I and from 46 to 75 years (mean 53 years) for group 11. The male/female sex ratio was close to 1 : 1 within the two groups of 34 patients. The cause of celiac axis stenosis was not always clear. According to arteriographic or operative findings, arcuate ligament compression was the cause in two of our cases and in five others [5,9,15,16,20]. Other mechanisms such as atheroma and agenesis of the celiac axis, have been mentioned [I 1,171.
Revascularization of the hepatic artery through the gastroduodenal artery and development of anastomoses between the pancreaticoduodenal arteries are features that have always been underscored. The diameter of the aneurysm was evaluated in 23 cases, ranging from 4 to 70 mm (mean = 25 mm) without any difference being noted between the two groups. In 18 cases, (12 in group I , 6 in group 11), the aneurysm was discovered before any complication occurred, either by chance or during work-up for abdominal pain, which was occasionally accompanied with epigastric bruit upon auscultation. Aneurysmal calcifications on plain abdominal films combined with ultrasound and CT scans contributed to
VOLUME4 NO 6 - 1990
PANCREATICODUODENAL AKTER Y ANEUR YSMS
543
TABLE 11.-Reported cases of pancreaticoduodenalartery aneurysms nonassociated with celiac axis stenosis (Group II)
Author Murase [21] 1973 Spanos [22] 1974 Harris [23] 1975 Allegaert [24] (1976) Keehan [25] 1978 Noyes [26] 1979 Colluzza [27] 1981 Eyskens [3] 1982 Harbin [28] 1983 Thevenet [261 1983 Hubens [31] 1984
Sexlage MI48
Aneurysm status Nonruptured
MI58
Ruptured into peritoneal cavity Nonruptured
Aneurysm diameter (mm) 30
Management Resection
Outcome Favorable
10
Exclusion
Favorable
NA*
Exclusion
Favorable
NA
Exclusion
Favorable
NA
Resection
Favorable
Fl49
Ruptured into peritoneal cavity Ruptured into retroperitoneum Nonruptured
NA
Resection
FavorabIe
MI54
Nonruptured
NA
Exclusion
Favorable
Fl52
15
Resection
Favorable
Fl63
Ruptured into peritoneal cavity Nonruptured
20
Ernbolization
Favorable
F146
Nonruptured
NA
No surgery
NA
F147
Ruptured into peritoneal cavity
NA
Resection
Favorable
Fl54 MI45 MI75
*Not available
correct diagnoses. Arteriograms were obtained either as part of initial evaluation or to further delineate an anomaly found during other investigations. In the 16 other cases (1 1 in group I, 5 in group ll), rupture was the initial clinical manifestation. The consequence was hemoperitoneum, anterior or posterior peripancreatic or retroperitoneal hematoma, or intraductal hemorrhage, according to whether the aneurysm was anterior, posterior or intrapancreatic. Emergency clinical, sonographic and CT scan findings were easily diagnostic of hemorrhage and its localization. Arteriograms were used to identify the aneurysm and the eventual stenosis of the celiac axis. In four cases, arteriograms were obtained after initial laparotomy during which the aneurysm had been missed [6-8,19]. In three of these cases, the lesions were cured by reoperation [6,7,19]. Therapeutic decisions varied according to whether the celiac axis was stenotic or not, and whether the aneurysm had ruptured or not (Tables 111 and IV). The technique employed to treat the aneurysm was either resection or exclusion. These procedures were difficult to perform when the aneurysm was located behind the pancreas, impinged on the pancreas, or had already ruptured. Emergency pancreaticoduodenectomy was required to stop hemorrhaging in three instances in this group of patients. As this procedure interrupted the pancreaticoduodenal anastomoses, hepatic artery revascularization was necessary in two instances, requiring either division of the arcuate ligament or a bypass procedure [7,201.
In group I, after resection or exclusion performed in 14 cases, restoration of the hepatic artery was deemed necessary in four instances, either by reconstruction of the peripancreatic arterial network [16,19], aortohepatic bypass [12], or by division of the arcuate ligament (our Patient No. 2). In two cases of nonruptured aneurysms, 10 and 20 mm in diameter, the arcuate ligament was simply divided or the celiac artery was revascularized with complete disappearance of lesions on postoperative repeat arteriograms [6,91. In four instances, the patient was not operated on. The reasons for therapeutic abstention included
TABLE Ill.-Management of pancreaticoduodenal artery aneurysms associated with celiac artery stenosis (Group I)
Manaaement No surgery Colectorny Section of median arcuate ligament Revascularization of celiac axis Resection or exclusion Pancreatoduodenectomy Pancreatoduodenectorny with revascularization Resection or exclusion with revascularization (
). Perioperative mortality
Ruptured Nonruptured aneurvsms aneurvsms
2
2
544
PANCREAI’ICODUODENAL ARTERY ANEURYSMS
TABLE IV.-Management of pancreaticoduodenal artery aneurysms associated with celiac artery stenosis (Group II*)
ANNALS OF VASCULARSURGERY
cular reconstruction. Although results of surgery have generally been satisfactory, embolization could be an alternative to surgery in the poor risk Ruptured Nonruptured patient or when the aneurysm is easily accessible by Manaaement aneurysms aneurvsms catheter [28,32]. No surgery For aneurysms associated with stenosis of the 1 Embolization 1 celiac axis, exclusion or resection have been emResection or exclusion 5 4 ployed. In three cases, however, these procedures ‘There was no perioperativemortality in this group of patients failed and pancreatoduodenectomy was necessary to stop the bleeding. The effects of these procedures on the celiac circulation must be evaluated by rapidly lethal, sudden vascular collapse due to measurement of the aortohepatic arterial pressure intraperitoneal rupture [ 111, refusal of surgery [ 161, gradient. Revascularization did not appear to be poor risk patient (case I), and deliberate abstention necessary in 9 of 14 cases of exclusion or resection and in one of three pancreatoduodenectomies. in a patient with a 13 mm diameter lesion. Four patients died after surgical treatment, all in Good tolerance could be explained by multiple group I and all having been operated on for compli- collateral supply. In the absence of complications, when the aneucated lesions. No complications due to ischemia in the celiac axis vascular bed were noted. The aneu- rysm is small or difficult to approach, treatment of rysm never recurred when the celiac axis was not the celiac artery stenosis alone appears to be an attractive alternative. This proved to be successful reconstructed. for Mora and Proud [6,9]. In addition to local treatment of the aneurysm, complementary revascularization of the celiac terDISCUSSION ritory can be considered to preclude recurrence by The association of pancreaticoduodenal artery reestablishing normal circulation. To this intent, an aneurysm with celiac stenosis is not coincidental. aortohepatic bypass or the division of the arcuate According to Sutton IS], the dilation of the arterial ligament should be preferred to reconstruction of walls leading to aneurysmal formation is the result the aneurysmal arterial segment left in place. of increased flow through these arteries to vascularize the celiac axis bed through the superior mesenteric artery. Atheroma may be a promoting CONCLUSION factor [3]. The reports of Mora [6] and Proud [9] are The study of aneurysms of pancreaticoduodenal remarkable demonstrations of this theory because the aneurysms disappeared on follow-up arterio- arteries shows that stenosis of the celiac axis is a grams after they revascularized the celiac axis. The determining but not exclusive factor in their pathoprecise incidence of this association with regard to genesis. This association should always be searched all celiac axis stenoses is not known. Thevenet [30] for, even in the emergency. setting, by preoperative reported one case in a series of 35. These aneu- arteriography whenever feasible. Treatment should rysms may be due to other causes, such as ather- strive to cure the aneurysm and restore normal oma and angiodysplasia, as mentioned in other blood flow to the celiac territory. studies [ 1,3,31]. Irrespective of their origin, the clinical features of aneurysms of the pancreaticoduodenal arteries are REFERENCES similar. In the absence of complications, symptoms I . STANLEY JC. Splanchnic artery aneurysms. In: RUTHare not very characteristic. In half of cases, rupture ERFORD RB (ed). Vascular Surgery. Philadelphia, W B is the initial manifestation and leads to severe 1977: pp. 673-684. hemorrhage. Arteriography is essential to the iden- 2. Saunders. ECKHAUSER FE, STANLEY JC. ZELENOCK GB, et al. tification and location of the lesion, as well as to the Gastroduodenal and pancreaticoduodenal artery aneurysms: safety of treatment. Arteriography is the only way a complication of pancreatitis causing spontaneous gastrointestinal hemorrhage. Surgery 1980;88:335-344. of identifying stenosis of the celiac axis. Surgical intervention can then be planned before a compli- 3. EYSKENS E. L’anevrysme des arttres pancrtaticoduodh a l e s : prksentation d’un cas personnel, revue et analyse de cation arises because even small aneurysms less la littkrature. Chirurgie 1982;/08:734-743. than one centimeter in diameter can rupture [3]. It 4. LERUT JP, GIANELLO PP, OTTE JB, et al. Les anevrysmes des branches du tronc coeliaque. J Chir (Paris) 1985;/22:93-100. must be remembered that rupture is a serious com5 . SUTTON D. LAWTON G. Celiac stenosis or occlusion with plication since death occurred in 4 of 16 cases aneurysm of the collateral supply. C/in Radio/ 197324:4%53. reported. 6. MORA JD. OBST D. Celiac axis artery stenosis with aneuAneurysms without stenosis of the celiac axis are rysmal calcification of the collateral supply. Ausr Radio1 best treated by exclusion or resection without vas1976;20:252.
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