Ann Vasc Dis Vol. 7, No. 3; 2014; pp 312–315 ©2014 Annals of Vascular Diseases
Online Month July 30, 2014 doi:10.3400/avd.cr.14-00040
Case Report
Pancreaticoduodenal Artery Aneurysm Formation with Superior Mesenteric Artery Stenosis Tadashi Kitaoka, MD, Juno Deguchi, MD, PhD, Chiaki Kamiya, MD, Jun Suzuki, MD, PhD, and Osamu Sato, MD, PhD Celiac stenosis or occlusion is attributed partly to increase blood flow at pancreatic arcade from the superior mesenteric artery (SMA) system and may play a causal role in true aneurysm of pancreaticoduodenal artery (PDAA) formation. However, despite possible increased blood flow in the pancreatic arcades like celiac stenosis, PDAAs with a stenotic SMA are extremely rare, with only three cases have been reported in the literature. We report a case of PDAA with SMA stenosis and review the literature. Keywords: pancreaticoduodenal artery aneurysm, superior mesenteric artery, true aneurysm
Introduction Pancreaticoduodenal artery aneurysm (PDAA) is a rare condition, that accounts for only 2% of splanchnic aneurysms.1) PDAAs are classified as either true or false aneurysms. While false PDAA results from surrounding inflammation such as pancreatitis or trauma, true PDAA is degenerative or related to atherosclerosis and remains elusive in origin.1) Sutton, et al. reported that true PDAA is associated with celiac stenosis or occlusion, and that 80% of patients with true PDAA had celiac stenosis.2) Celiac stenosis or occlusion, which is a result of atherosclerosis, fibromuscular hyperplasia or compression by the median arcuate ligament may induce hemodynamic changes in the pancreatic arcade. Increased blood flow in the pancreatic arcades is speculated to induce some of true PDAAs. However, despite possible increased blood flow in the pancreatic arcades, PDAAs with a narrowed Department of Vascular Surgery, Saitama Medical Center, Saitama Medical School, Kawagoe, Saitama, Japan Received: March 18, 2014; Accepted: May 10, 2014 Corresponding author: Juno Deguchi, MD. Department of Vascular Surgery, Saitama Medical Center, Saitama Medical School, 1981 Kamoda, Kawagoe, Saitama 385-8550, Japan Tel: +81-49-228-3400, Fax: +81-49-228-3467 E-mail: [email protected]
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superior mesenteric artery (SMA) are rare, with only three cases have been reported in the literature, to the best of our knowledge.3–6) The present report describes a case of PDAA with stenosis at the origin of the SMA.
Case Report A 73-year-old woman was admitted to our hospital for detailed examination of a right abdominal mass detected during medical checkup. She had no symptoms such as pain, poor appetite and weight loss. The physical findings were not applicable except pulsatile abdominal mass at the right subcostal region. She suffered from chronic hepatitis C infection, which she contracted from a blood transfusion given during the delivery of one of her children. Abdominal computed tomography (CT) using contrast medium revealed an aneurysm 2-cm in diameter in the anterior superior pancreaticoduodenal artery (Fig. 1A). Blood testing showed the following, white blood cells 3500/µl, red blood cells 361 × 104/µl, platelets 62000/µl, activated partial thromboplastin time 32.7 s, prothrombin time 13.2 s, AST 89 U/L, ALT 96 U/L, serum amylase 131 U/L. Selective abdominal angiography from the celiac axis revealed excessive development of the pancreatic Annals of Vascular Diseases Vol. 7, No. 3 (2014)
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10:17:07
Pancreaticoduodenal Artery Aneurysm with SMA Stenosis (A)
CHA GDA SAPDA
SMA Ao
(B)
CHA GDA
SMA
SAPDA
Ao SMA
Fig. 1 Images of computed tomography (CT) (A) and angiography (B) revealed a 2 cm aneurysm in the superior anterior pancreaticoduodenal artery. (A) Left panel shows cross-sectional CT image of an aneurysm (white arrowhead). Right panel indicates a three-dimensional reconstructed CT image of an aneurysm (black arrowhead) with surrounding arteries. Note a thick pancreatic arcade and the normal orifice of the celiac axis. (B) Left panel reveals a selective angiogram from the celiac axis showing a developed pancreatic arcade with blood flow from the celiac to SMA area. Left panel indicates an angiogram showing a narrowed orifice of the superior mesenteric artery. Ao: aorta; CHA: common hepatic artery; GDA: gastroduodenal artery; SMA: superior mesenteric artery; SAPDA: superior anterior pancreaticoduodenal artery.
arcade and an aneurysm of 2 cm in diameter in the anterior superior pancreaticoduodenal artery without celiac stenosis. It also showed blood flow circulated smoothly from the celiac to the SMA. A selective angiogram from the SMA showed a narrowed orifice of the SMA and contrast medium did not perfuse into the distal SMA, which indicated that blood flow of the distal SMA comes from the celiac artery (Fig. 1B). The patient underwent open surgery because aneurysm resection might decrease mesenteric circulation and need potential bypass to the SMA. An abdominal transverse incision was made under general anesthesia. She had liver cirrhosis and a hemorrhagic tendency on gross findings. After proximal and distal artery control was obtained, the aneurysm was completely Annals of Vascular Diseases Vol. 7, No. 3 (2014)
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isolated and resected under systemic heparinization. During clamping of the pancreaticoduodenal artery, pulsation of the SMA had ceased, but resumed after declamping. The pulsation of the aneurysm disappeared when clamping of the pancreaticoduodenal artery was made at the celiac axis side, which indicated that direction of blood flow was from celiac axis to the SMA. Histological specimens revealed a saccular aneurysm with layers of destroyed media and stretched adventitia. Except formation of retroperitoneal hematoma around the ascending colon, which occurred due to traction and extravasation from small vessels during the operation, her postoperative course was uneventful. Postoperative CT displayed patent pancreaticoduodenal artery without aneurysm (Fig. 2). She 313
2014/09/11
10:17:09
Kitaoka T, et al. Table 1 A list showed reference related to pancreaticoduodenal artery aneurysm with narrowed superior mesenteric artery
Ichinokawa Takao
76F 80M
Ikoma Kitaoka
71M 73F
Location
Size (mm)
Rupture
CA stenosis
Tx to aneurysm
Tx to SMA stenosis
Anterior- superior Inferior Posterior Inferior Anterior- superior
30 11 10 20 20
no no no no no
no no no no no
Resection no no Coil packing Resection
no no no no no
Ref 3 6 6 5
CA: celiac axix; Tx: treatment: SMA: superior mesenteric artery
Fig. 2 P ostoperative three-dimensional computed tomography image of the celiac and the superior mesenteric artery (SMA). Note that an aneurysm (black arrowhead) was excised without arterial stenosis.
was discharged from the hospital on the 13th postoperative day.
Discussion The hemodynamic stress of pancreaticoduodenal arcades induced by a narrowed or occlusive celiac trunk might lead to the development of true PDAAs.7) Mano, et al. reported that patients of true PDAA with occlusive celiac trunk had increased blood flow in the pancreaticoduodenal arcades and that the direction of the flow was from the SMA to the hepatic artery. The present patient with no history of pancreatitis or trauma had a true PDAA, which was confirmed by histologic specimens. She did not have narrowed celiac trunk but had stenosis of the SMA, the main blood flow of which was supplied via the pancreaticoduodenal arcade from the celiac axis. The cause of SMA stenosis in the present patient was not identified; however, based on the findings from CT and angiography, it can be assumed that her pancreaticoduodenal arcades had increased blood flow. The SMA stenosis may also induce hemodynamic stress 314
05_14_CR_Deguchi_00040.indd
in the pancreaticoduodenal arcades and lead to develop a true PDAA. However, despite the fact that chronic mesenteric ischemia is not so rare, there were only three cases of patients with PADDs and SMA stenosis or occlusion have been published (Table 1). Moreover, Kimura, et al. reported a case of PDAA with both celiac and SMA stenosis.4) It remains elusive whether there is direct association between hemodynamics of pancreaticoduodenal arcades and development of a tree PDAA. Brocker et al. reviewed 93 cases of true PDAAs with celiac stenosis or occlusion and reported both that 52% were ruptured at the time of presentation and that aneurysm size did not correlate with rupture.8) These results suggest that PDAAs should be treated at the time of diagnosis. Treatment options include surgery (e.g., ligation, resection, or pancreaticoduodenectomy) and endovascular embolization. On the other hand, the optimal treatment for coexiting stenosis or occlusion of trunk arteries remains unclear. Brocker, et al. also did not observe PDAA recurrence secondary to residual celiac stenosis, on the other hand, 21% of the patients underwent treatment of their celiac stenosis with a corresponding mortality rate of 7.6%. We reviewed four cases of patients with PDAA and SMA stenosis, and only one of those patients underwent treatment of coexisting trunk stenosis. The present patient had liver cirrhosis and underwent resection of an aneurysm without treatment of the narrowed SMA. She had good nutritional status and did not abdominal pain or diarrhea. We plan on following her and performing endovascular treatment for SMA stenosis if necessary in the future. In conclusion, this report described a rare case of a patient with pancreaticoduodenal artery aneurysm and stenosis of the SMA. This disease entity remains poorly defined and requires additional investigation to determine the optimal treatment strategy. Annals of Vascular Diseases Vol. 7, No. 3 (2014)
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10:17:09
Pancreaticoduodenal Artery Aneurysm with SMA Stenosis
Disclosure Statement Each author of this article has no conflict of interest such as employment, leadership position, advisory role, stock ownership, patent royalties or honoraria. Each author has no sponsor that provided financial support for this study.
References 1) Rockman CB, Maldonado TS. Splanchnic Artery Aneurysms. Philadelphia: Saunders, 2005. 2) Sutton D, Lawton G. Coeliac stenosis or occlusion with aneurysm of the collateral supply. Clin Radiol 1973; 24: 49-53. 3) Ichinokawa M, Iwai K, Matsumura Y, et al. A case of the anterior superior pancreaticoduodenal artery aneurysm with complete occusion of the superior mesenteric artery. Jpn J Gastroenterol Surg 2006; 39: 1678-82. (in Japanese)
Annals of Vascular Diseases Vol. 7, No. 3 (2014)
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4) Kimura C, Adachi H, Yamaguchi A, et al. A case of an unruptured inferior pancreaticoduodenal artery aneurysm with celiac aretery occlusion and superior mesenteric aretery stenosis. Jpn J Vasc Surg 2009; 18: 691-4. (in Japanese) 5) Ikoma A, Nakai M, Sato M, et al. Inferior pancreaticoduodenal artery aneurysm treated with coil packing and stent placement. World J Radiol 2012; 4: 387-90. 6) Takao H, Doi I, Watanabe T, et al. Natural history of true pancreaticoduodenal artery aneurysms. Br J Radiol 2010; 83: 744-6. 7) Mano Y, Takehara Y, Sakaguchi T, et al. Hemodynamic assessment of celiaco-mesenteric anastomosis in patients with pancreaticoduodenal artery aneurysm concomitant with celiac artery occlusion using flow-sensitive fourdimensional magnetic resonance imaging. Eur J Vasc Endovasc Surg 2013; 46: 321-8. 8) Brocker JA, Maher JL, Smith RW. True pancreaticoduodenal aneurysms with celiac stenosis or occlusion. Am J Surg 2012; 204: 762-8.
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Online Month July 30, 2014 doi:10.3400/avd.cr.14-00040
Case Report
Pancreaticoduodenal Artery Aneurysm Formation with Superior Mesenteric Artery Stenosis Tadashi Kitaoka, MD, Juno Deguchi, MD, PhD, Chiaki Kamiya, MD, Jun Suzuki, MD, PhD, and Osamu Sato, MD, PhD Celiac stenosis or occlusion is attributed partly to increase blood flow at pancreatic arcade from the superior mesenteric artery (SMA) system and may play a causal role in true aneurysm of pancreaticoduodenal artery (PDAA) formation. However, despite possible increased blood flow in the pancreatic arcades like celiac stenosis, PDAAs with a stenotic SMA are extremely rare, with only three cases have been reported in the literature. We report a case of PDAA with SMA stenosis and review the literature. Keywords: pancreaticoduodenal artery aneurysm, superior mesenteric artery, true aneurysm
Introduction Pancreaticoduodenal artery aneurysm (PDAA) is a rare condition, that accounts for only 2% of splanchnic aneurysms.1) PDAAs are classified as either true or false aneurysms. While false PDAA results from surrounding inflammation such as pancreatitis or trauma, true PDAA is degenerative or related to atherosclerosis and remains elusive in origin.1) Sutton, et al. reported that true PDAA is associated with celiac stenosis or occlusion, and that 80% of patients with true PDAA had celiac stenosis.2) Celiac stenosis or occlusion, which is a result of atherosclerosis, fibromuscular hyperplasia or compression by the median arcuate ligament may induce hemodynamic changes in the pancreatic arcade. Increased blood flow in the pancreatic arcades is speculated to induce some of true PDAAs. However, despite possible increased blood flow in the pancreatic arcades, PDAAs with a narrowed Department of Vascular Surgery, Saitama Medical Center, Saitama Medical School, Kawagoe, Saitama, Japan Received: March 18, 2014; Accepted: May 10, 2014 Corresponding author: Juno Deguchi, MD. Department of Vascular Surgery, Saitama Medical Center, Saitama Medical School, 1981 Kamoda, Kawagoe, Saitama 385-8550, Japan Tel: +81-49-228-3400, Fax: +81-49-228-3467 E-mail: [email protected]
312
05_14_CR_Deguchi_00040.indd
superior mesenteric artery (SMA) are rare, with only three cases have been reported in the literature, to the best of our knowledge.3–6) The present report describes a case of PDAA with stenosis at the origin of the SMA.
Case Report A 73-year-old woman was admitted to our hospital for detailed examination of a right abdominal mass detected during medical checkup. She had no symptoms such as pain, poor appetite and weight loss. The physical findings were not applicable except pulsatile abdominal mass at the right subcostal region. She suffered from chronic hepatitis C infection, which she contracted from a blood transfusion given during the delivery of one of her children. Abdominal computed tomography (CT) using contrast medium revealed an aneurysm 2-cm in diameter in the anterior superior pancreaticoduodenal artery (Fig. 1A). Blood testing showed the following, white blood cells 3500/µl, red blood cells 361 × 104/µl, platelets 62000/µl, activated partial thromboplastin time 32.7 s, prothrombin time 13.2 s, AST 89 U/L, ALT 96 U/L, serum amylase 131 U/L. Selective abdominal angiography from the celiac axis revealed excessive development of the pancreatic Annals of Vascular Diseases Vol. 7, No. 3 (2014)
312
2014/09/11
10:17:07
Pancreaticoduodenal Artery Aneurysm with SMA Stenosis (A)
CHA GDA SAPDA
SMA Ao
(B)
CHA GDA
SMA
SAPDA
Ao SMA
Fig. 1 Images of computed tomography (CT) (A) and angiography (B) revealed a 2 cm aneurysm in the superior anterior pancreaticoduodenal artery. (A) Left panel shows cross-sectional CT image of an aneurysm (white arrowhead). Right panel indicates a three-dimensional reconstructed CT image of an aneurysm (black arrowhead) with surrounding arteries. Note a thick pancreatic arcade and the normal orifice of the celiac axis. (B) Left panel reveals a selective angiogram from the celiac axis showing a developed pancreatic arcade with blood flow from the celiac to SMA area. Left panel indicates an angiogram showing a narrowed orifice of the superior mesenteric artery. Ao: aorta; CHA: common hepatic artery; GDA: gastroduodenal artery; SMA: superior mesenteric artery; SAPDA: superior anterior pancreaticoduodenal artery.
arcade and an aneurysm of 2 cm in diameter in the anterior superior pancreaticoduodenal artery without celiac stenosis. It also showed blood flow circulated smoothly from the celiac to the SMA. A selective angiogram from the SMA showed a narrowed orifice of the SMA and contrast medium did not perfuse into the distal SMA, which indicated that blood flow of the distal SMA comes from the celiac artery (Fig. 1B). The patient underwent open surgery because aneurysm resection might decrease mesenteric circulation and need potential bypass to the SMA. An abdominal transverse incision was made under general anesthesia. She had liver cirrhosis and a hemorrhagic tendency on gross findings. After proximal and distal artery control was obtained, the aneurysm was completely Annals of Vascular Diseases Vol. 7, No. 3 (2014)
05_14_CR_Deguchi_00040.indd
313
isolated and resected under systemic heparinization. During clamping of the pancreaticoduodenal artery, pulsation of the SMA had ceased, but resumed after declamping. The pulsation of the aneurysm disappeared when clamping of the pancreaticoduodenal artery was made at the celiac axis side, which indicated that direction of blood flow was from celiac axis to the SMA. Histological specimens revealed a saccular aneurysm with layers of destroyed media and stretched adventitia. Except formation of retroperitoneal hematoma around the ascending colon, which occurred due to traction and extravasation from small vessels during the operation, her postoperative course was uneventful. Postoperative CT displayed patent pancreaticoduodenal artery without aneurysm (Fig. 2). She 313
2014/09/11
10:17:09
Kitaoka T, et al. Table 1 A list showed reference related to pancreaticoduodenal artery aneurysm with narrowed superior mesenteric artery
Ichinokawa Takao
76F 80M
Ikoma Kitaoka
71M 73F
Location
Size (mm)
Rupture
CA stenosis
Tx to aneurysm
Tx to SMA stenosis
Anterior- superior Inferior Posterior Inferior Anterior- superior
30 11 10 20 20
no no no no no
no no no no no
Resection no no Coil packing Resection
no no no no no
Ref 3 6 6 5
CA: celiac axix; Tx: treatment: SMA: superior mesenteric artery
Fig. 2 P ostoperative three-dimensional computed tomography image of the celiac and the superior mesenteric artery (SMA). Note that an aneurysm (black arrowhead) was excised without arterial stenosis.
was discharged from the hospital on the 13th postoperative day.
Discussion The hemodynamic stress of pancreaticoduodenal arcades induced by a narrowed or occlusive celiac trunk might lead to the development of true PDAAs.7) Mano, et al. reported that patients of true PDAA with occlusive celiac trunk had increased blood flow in the pancreaticoduodenal arcades and that the direction of the flow was from the SMA to the hepatic artery. The present patient with no history of pancreatitis or trauma had a true PDAA, which was confirmed by histologic specimens. She did not have narrowed celiac trunk but had stenosis of the SMA, the main blood flow of which was supplied via the pancreaticoduodenal arcade from the celiac axis. The cause of SMA stenosis in the present patient was not identified; however, based on the findings from CT and angiography, it can be assumed that her pancreaticoduodenal arcades had increased blood flow. The SMA stenosis may also induce hemodynamic stress 314
05_14_CR_Deguchi_00040.indd
in the pancreaticoduodenal arcades and lead to develop a true PDAA. However, despite the fact that chronic mesenteric ischemia is not so rare, there were only three cases of patients with PADDs and SMA stenosis or occlusion have been published (Table 1). Moreover, Kimura, et al. reported a case of PDAA with both celiac and SMA stenosis.4) It remains elusive whether there is direct association between hemodynamics of pancreaticoduodenal arcades and development of a tree PDAA. Brocker et al. reviewed 93 cases of true PDAAs with celiac stenosis or occlusion and reported both that 52% were ruptured at the time of presentation and that aneurysm size did not correlate with rupture.8) These results suggest that PDAAs should be treated at the time of diagnosis. Treatment options include surgery (e.g., ligation, resection, or pancreaticoduodenectomy) and endovascular embolization. On the other hand, the optimal treatment for coexiting stenosis or occlusion of trunk arteries remains unclear. Brocker, et al. also did not observe PDAA recurrence secondary to residual celiac stenosis, on the other hand, 21% of the patients underwent treatment of their celiac stenosis with a corresponding mortality rate of 7.6%. We reviewed four cases of patients with PDAA and SMA stenosis, and only one of those patients underwent treatment of coexisting trunk stenosis. The present patient had liver cirrhosis and underwent resection of an aneurysm without treatment of the narrowed SMA. She had good nutritional status and did not abdominal pain or diarrhea. We plan on following her and performing endovascular treatment for SMA stenosis if necessary in the future. In conclusion, this report described a rare case of a patient with pancreaticoduodenal artery aneurysm and stenosis of the SMA. This disease entity remains poorly defined and requires additional investigation to determine the optimal treatment strategy. Annals of Vascular Diseases Vol. 7, No. 3 (2014)
314
2014/09/11
10:17:09
Pancreaticoduodenal Artery Aneurysm with SMA Stenosis
Disclosure Statement Each author of this article has no conflict of interest such as employment, leadership position, advisory role, stock ownership, patent royalties or honoraria. Each author has no sponsor that provided financial support for this study.
References 1) Rockman CB, Maldonado TS. Splanchnic Artery Aneurysms. Philadelphia: Saunders, 2005. 2) Sutton D, Lawton G. Coeliac stenosis or occlusion with aneurysm of the collateral supply. Clin Radiol 1973; 24: 49-53. 3) Ichinokawa M, Iwai K, Matsumura Y, et al. A case of the anterior superior pancreaticoduodenal artery aneurysm with complete occusion of the superior mesenteric artery. Jpn J Gastroenterol Surg 2006; 39: 1678-82. (in Japanese)
Annals of Vascular Diseases Vol. 7, No. 3 (2014)
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4) Kimura C, Adachi H, Yamaguchi A, et al. A case of an unruptured inferior pancreaticoduodenal artery aneurysm with celiac aretery occlusion and superior mesenteric aretery stenosis. Jpn J Vasc Surg 2009; 18: 691-4. (in Japanese) 5) Ikoma A, Nakai M, Sato M, et al. Inferior pancreaticoduodenal artery aneurysm treated with coil packing and stent placement. World J Radiol 2012; 4: 387-90. 6) Takao H, Doi I, Watanabe T, et al. Natural history of true pancreaticoduodenal artery aneurysms. Br J Radiol 2010; 83: 744-6. 7) Mano Y, Takehara Y, Sakaguchi T, et al. Hemodynamic assessment of celiaco-mesenteric anastomosis in patients with pancreaticoduodenal artery aneurysm concomitant with celiac artery occlusion using flow-sensitive fourdimensional magnetic resonance imaging. Eur J Vasc Endovasc Surg 2013; 46: 321-8. 8) Brocker JA, Maher JL, Smith RW. True pancreaticoduodenal aneurysms with celiac stenosis or occlusion. Am J Surg 2012; 204: 762-8.
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