Obstructive Jaundice Caused by Pancreaticoduodenal Artery Aneurysms Associated with Celiac Axis Stenosis: Case Report and Review of the Literature Tiansheng Yin, Zhili Wan, Hongwei Chen, Xixian Mao, Yayang Yi, and Dewei Li, Chongqing, China
Pancreaticoduodenal artery aneurysm (PDA) is quite rare, which accounts for only approximate 2% of all visceral aneurysms. Besides, PDA is usually related to celiac axis stenosis (CAS) and prone to rupture. Advanced imaging examination can facilitate the disclosure of such peripancreatic masses, but most of them were seldom diagnosed until they rupture because of the nonspecific symptoms. Secondary to PDA, obstructive jaundice is however an extremely rare manifestation. A case of an 84-year-old man is reported here, who suffered from severe jaundice caused by a ruptured PDA associated with CAS. In addition, this review collects and organizes PDAs with jaundice by applying a MEDLINE search and discusses the pathogenesis and therapeutic options of these aneurysms leading to external compression over the bile duct. Consequently, the formation of PDA with obstructive jaundice is based on the specific anatomy of pancreaticoduodenal arcades. When there is a retroperitoneal mass around the head of the pancreas associated with unexpected jaundice, PDA should be considered, for which early aggressive therapy is required. The case report and literature review suggest that PDA associated with obstructive jaundice may be treated successfully by single transcatheter arterial embolization (TAE) without auxiliary biliary drainage, whether it ruptures or not.
Pancreaticoduodenal artery aneurysms (PDAs) are quite rare, which account for only 2% of all visceral artery aneurysms.1 Since Sutton and Lawton 2 firstly put forward the relation between CAS and PDA, approximate 63% of reported cases had been found to be related to CAS.3 More than 60% of PDAs are shown to be at rupture, and emergent treatments are required.4 Although advanced imaging examination facilitates the disclosure of such
Conflicts of Interest: The authors state that there are no conflicts of interests to declare. Department of Hepatobiliary Surgery, The First Affiliated Hospital of Chongqing Medical University, Chongqing, China. Correspondence to: Dewei Li, MD, PhD, Department of Hepatobiliary Surgery, The First Affiliated Hospital of Chongqing Medical University, No.1 Friendship Road, Yuzhong District, Chongqing 400000, China; E-mail: [email protected] Ann Vasc Surg 2015; 29: 1016.e1e1016.e6 http://dx.doi.org/10.1016/j.avsg.2014.12.011 Ó 2015 Elsevier Inc. All rights reserved. Manuscript received: July 19, 2014; manuscript accepted: December 14, 2014; published online: March 11, 2015.
peripancreatic masses, most of them were seldom diagnosed until they rupture because of the nonspecific symptoms. Secondary to PDA, Jaundice is however an extremely rare manifestation. A case of an 84-year-old man is reported here, who suffered from severe jaundice caused by a ruptured PDA associated with CAS. In addition, this review collects and organizes PDA with jaundice by adopting a MEDLINE search and discusses the pathogenesis and therapeutic options of these aneurysms leading to external compression over the bile duct.
CASE REPORT An 84-year-old man was admitted to the local hospital, who suffered from a sudden onset of abdominal pain in the right upper quadrant. There was no history of abdominal trauma, chronic pancreatitis, bile duct calculus, or alcohol abuse, but he had suffered from hypertension for 10 years. On admission, the blood pressure was 85/ 54 mm Hg, and an immediate ultrasound showed that 1016.e1
1016.e2 Case reports
there was a retroperitoneal mass. Subsequently, an urgent laparotomy was performed and the gigantic mass was found to be a retroperitoneal hematoma. The hematoma was located in the peripancreatic region without a clear limit out of the surrounding tissue, which had high surface tension with the tendency to rupture, but no bleeding focus was found. On account of the limitations in local technology and experience, further dissection around the mass was difficult and risky. Considering the patient safety, the surgeons set up a drainage tube and the operation was terminated. The advanced age patient was intubated during the laparotomy and extubated the next day. Postoperative vital signs stabilized without any vasoactive substance, but he was transferred to the hospital because of unexpected jaundice. Subsequent laboratory analysis revealed that a serum total bilirubin was at 24.4 mg/dL, direct bilirubin at 21.5 mg/dL, alanine transaminase at 60 m/L, and gamma glutamyl transpeptidase at 84 m/L, which indicated severe obstructive jaundice. Additionally, abdominal contrast-enhanced computed tomography (CT) and CT angiography showed that retroperitoneal hematoma was secondary to ruptured pancreaticoduodenal artery aneurysm with celiac axis stenosis (Fig. 1AeC). Angiography under local anesthesia confirmed the existence of PDA with a diameter of 2 cm, which is derived from the first branch of the superior mesenteric artery on the right side (Fig. 2AeC). In the meantime, the initial involvement of the celiac axis was obviously narrowed, which was compensated by vascular revascularization via the pancreaticoduodenal artery arcades. A guiding catheter was inserted to the branch of the superior mesenteric artery, and superselective arterial embolization was successful by placing 2 microcoils in the proximal portion of the aneurysm (Fig. 3A). However, it was infeasible to embolism the distal main vessel, for there was no definitive vessel that supplied blood to the aneurysm. Arteriography of SMA (Fig. 3B) shows that the aneurysm had disappeared, and selective angiography of the gastroduodenal artery via celiac axis confirmed similarly that there was no ‘‘back’’ bleeding from the distal pancreaticoduodenal artery branch (Fig. 3C). The patient was returned to intensive care unit, and the jaundice subsided quickly after endovascular treatment. There were no further signs of bleeding from the aneurysm during admission. Unfortunately, the patient was subjected to multidrug-resistant respiratory infection and died from breathing failure 26 days after embolization.
DISCUSSION As an uncommon vascular disease, PDA was first reported in 1895 by Ferguson.5 These aneurysms can be divided into the true aneurysms frequently related to celiac axis stenosis and the pseudoaneurysms whose underlying cause is the
Annals of Vascular Surgery
peripancreatic inflammation or trauma involving chronic pancreatitis and abdominal injury. According to Kallamadi et al.,6 abdominal pain is the most common initial symptom of PDA. However, it is difficult to establish an early diagnosis of PDA until it ruptures, which results in a mortality rate of 26%.3 Most of them usually rupture into the retroperitoneal space around the head of pancreas, which simulates the peripancreatic mass. In addition, whether to rupture is hardly dependent on the size of aneurysms (the median diameter is 22.2 mm in patients with rupture, 21.4 mm to the contrary), but dependent on the propensity for rupture itself.3,7e9 Both a gigantic aneurysm itself and a secondary retroperitoneal hematoma can produce oppression over surrounding tissues, which leads to obstructive jaundice uncommonly. In this regard, retroperitoneal mass around the head of the pancreas with jaundice can arouse the awareness of PDA, and early aggressive therapy is required. ‘‘jaundice’’, ‘‘iterus’’, ‘‘pancreaticoduodenal’’, ‘‘aneurysm’’ and ‘‘pseudoaneurysm’’ are regarded as key words and searched in the MEDLINE database by electronic and manual searches. As a result, totally 8 cases of PDA associated with jaundice are collected and organized in Table 1.9e15 Unexpected jaundice is based on the specific peripancreatic anatomy (Picture 1). The pancreaticoduodenal arcades are around the head of pancreas and formed by the branches of celiac axis and superior mesenteric artery (SMA), which supply corresponding tissues and organs. The anterior and posterior of the superior pancreaticoduodenal artery rooted in gastroduodenal artery are connected with the anterior and posterior of the inferior pancreaticoduodenal artery derived from SMA, respectively. There are several rami communicantes between the celiac axis and SMA, such as the Arc of Buhler and the Arc of Barkow.17e19 Because of CAS and provided by the common hepatic artery, the flow to the liver, spleen and stomach, spleen artery, and left gastric artery is compensated by the arcades that serve as a retrograde bypass pathway. As a result, the accompanied arteries dilate, which is supposed to be PDA. It usually gathers on the duodenal ampulla along with the pancreatic duct behind the head of the pancreas, whereas the distal common bile duct tends to be compressed by the unruptured PDA or the hematomas caused by rupture. The bulky mass exerts direct pressure over the common bile duct, the head of the pancreas, and duodenum, which results in obstruction. As in our case, a giant retroperitoneal hematoma oppresses the extrahepatic duct, which
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Case reports 1016.e3
Fig. 1. Contrast-enhanced abdominal CT scan shows a retroperitoneal hematoma accompanied with suspicious aneurysm (A). Stenosis of the celiac trunk (B). Vascular reconstruction of CT angiography (C, D).
leads to abundant biliary countercurrents into the hepatic sinusoid. On the basis of these factors, severe icterus occurs rapidly. Pancreaticoduodenal artery aneurysms often manifest as retroperitoneal masses around the head of the pancreas. Accompanied with unexpected jaundice, in particular with painless jaundice, PDAs are inclined to be confused with pancreatic head carcinoma. As described in this instance, the role of ultrasound is controversial because of its weakness in detecting such a retroperitoneal mass related to PDA, on account of the influence of intestinal gas, level of experience of the operator, and lack of discrimination. Instead, contrast-enhanced CT has become the golden standard, which qualitatively reveals this indeterminate lesion, especially the CT angiography and 3-dimensional reconstruction offer valuable first-hand information concerning the aneurysm. In our review, in addition to a few cases at early stage, most PDAs with jaundice are
discovered by contrast-enhanced CT. Moreover, intravascular angiography can confirm and locate the aneurysms, as well as judge and cure. In cases of severe jaundice, some patients undergo magnetic resonance cholangio-paneretography (MRCP) to visually delineate the biliary tree. In the early days, patients with PDA were subjected to surgical treatment, which was first reported in 1951 by Van Ouwerkerk.20 Nevertheless, approximately 70% of open surgery failed and even PDA could be detected, as in our case.3,21 Afterward, endovascular therapy became the preferred, and most effective method after the imaging techniques was available. This was supported by Mandel et al., who statistically analyzed that a success ratio of 79% and a mortality rate of 0% were linked to the procedure.22 To obtain the road map of pancreaticoduodenal arcades under digital subtraction angiography, selective catheterization and angiography of the superior mesenteric
1016.e4 Case reports
Annals of Vascular Surgery
Fig. 2. Selective superior mesenteric arteriography (A, B) and superselective injection (C) shows dilation of the pancreaticoduodenal arcade and a saccular aneurysm with retrograde filling of the celiac artery branches.
Fig. 3. Superselective arterial embolization in the proximal portion of the aneurysm (A). Arteriography of SMA ensures no contrast agent regurgitation around the aneurysm after embolization (B). Selective angiog-
artery are required. Meanwhile, appropriate coils can be placed in the lump body using a microcatheter or in both inflow and outflow of the aneurysm when fails. However, it also carries the risk of coil migration, ischemia of hepatic tissue, and failure of operation. Surgical technique is still widely considered as the definitive therapy for PDA, as the open approach is to expose and ligate the aneurysm directly. However, when these aneurysms rupture or hide in the parenchyma of the pancreas, the process of location and isolation can be difficult. Since Sutton and Lawton2 first put forward the significance of celiac axis stenosis in PDA, an increasing number of studies had supported this viewpoint and
raphy of the gastroduodenal artery via celiac axis confirms that there is no ‘‘back’’ bleeding from the distal PDA branch (C).
Picture 1. The peripancreatic anatomy.
more than half of all cases had been found to match this situation.3 CAS may be secondary to
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Case reports 1016.e5
Picture 2. Bile duct compressed by the unruptured aneurysm or the hematoma caused by rupture (A, B). Bile duct decompressed after ERCP (C), PTCD (D), surgical treatment (E), and TAE (F, G).
Table I. Reported cases of pancreaticoduodenal artery aneurysm with obstructive jaundice Author
Year
Age, sex
Presentation
CAS
Rupture
Treatment
Outcome
Sampsel et al.10
1952
68, Male
Painless jaundice
NA
Yes
Death
Hasselgren et al.11
1976
62, Female
NA
No
Scheflan et al.12
1977
56, Male
Pain under the right costal margin, jaundice Painless jaundice, pruritus
Yes
Yes
Kadir et al.13
1978
72, Male
Yes
No
Widjaja et al.14
1999
51, Male
Gastrointestinal bleeding, jaundice Epigastric pain, jaundice
Hepaticodochoenterostomy Aneurysmectomy Exploratory choledochotomy Aneurysmectomy Exploratory choledochotomy + T-tube drainage Untreated
NA
Yes
Colak et al.15
2009
57, Male
No
Yes
Wattze et al.16
2013
64, Female
Yes
No
Current report
2014
84, Male
Epigastric pain, jaundice, indigestion, weight loss Acute abdominal pain, biliary tract dilatation (14 mm; by ultrasound scan) Abdominal pain in the right upper quadrant, jaundice
Yes
Yes
PTCD Transcatheter arterial embolization Untreated
Laparotomy and injection of polymeric synthetic into the aneurysm Transcatheter arterial embolization
Survival
Survival
Death Survival
Death
Survival
Death
NA, not available.
atherosclerosis, pancreatitis, connective tissue disorders, polyarteritis nodosa, Takayasu disease, trauma, necrotizing arteritis, fibromuscular dysplasia, and so forth,23 whereas the most studied and relevant element is the compression by median arcuate ligament, which can be seen in 10e24% of patients.24 Various therapeutic methods are used in the management of obstructive jaundice resulting from aneurysms that involve PDA or retroperitoneal hematoma caused by rupture (Picture 2). Percutaneous transhepatic cholangial drainage (PTCD) is generally a safe and well-tolerated method with a few complications, such as weakness, pain, and biliary leak.25,26 It is an alternative remedy for mechanical jaundice even if
there is no way to decompress the common bile duct. In addition, endoscopic retrograde cholangiopancreatograph (ERCP) with biliary stent insertion is considered to be less invasive and providing a fair delineation of biliary anatomy, nevertheless, on the condition that the bile duct is compressed by an aneurysm, especially the one whose wall is very thin and fragile, and the stent has the potential to enter the aneurysm, which leads to massive hemorrhage.27 Although jaundice companied by aneurysm appears to be resolved simultaneously, surgical treatment is less recommended because of its difficulty and extensive damage. Retzlaff reported that in nearly 70% of cases, even the bleeding point was not identified at
1016.e6 Case reports
operation, just as described in our case.21,27 Sciurne et al. also emphasized the technique of PTCD combined with ERCP, called ‘‘rendezvous.’’28 In our case, we detect a notable outcome that jaundice gets improved after single transcatheter arterial embolization (TAE). Furthermore, similar situations are observed in some cases, as Donmez and Kuroiwa described.29,30 The bile duct is decompressed after effective endovascular therapy, which is due to the reduced volume of isolated aneurysm or the decreased surface tension of absorbed hematoma. Therefore, it is speculated that PDA associated with obstructive jaundice may be treated successfully by single TAE technique without any auxiliary biliary drainage, whether it ruptures or not. More research is needed. Pancreaticoduodenal Artery Aneurysms associated with jaundice are very rare. PDA is usually related to celiac axis stenosis, whose pathogenesis is based on the specific anatomy of pancreaticoduodenal arcades. When there is a retroperitoneal mass around the head of the pancreas associated with unexpected jaundice, PDA should be considered, for which early aggressive therapy is required. The case report and literature review suggest that the pancreaticoduodenal artery aneurysm associated with obstructive jaundice may be treated successfully by single TAE without auxiliary biliary drainage, whether it ruptures or not.
REFERENCES 1. Stanley JC, Wakefield TW, Graham LM, et al. Clinical importance and management of splanchnic artery aneurysms. J Vasc Surg 1886;3:836e40. 2. Sutton D, Lawton G. Coeliac stenosis or occlusion with aneurysm of the collateral supply. Clin Radiol 1973;24:49e53. 3. De Perrot M, Berney T, Deleaval J, et al. Management of true aneurysms of the pancreaticoduodenal arteries. Ann Surg 1999;229:416e20. 4. Moore E, Matthews MR, Minion DJ, et al. Surgical management of peripancreatic arterial aneurysms. J Vasc Surg 2004;40:247e53. 5. Ferguson F. Aneurysm of superior pancreaticoduodenal artery. Proc N Y Pathol Soc 1895;24:45e9. 6. Kallamadi R, Demoya MA, Kalva SP. Inferior pancreaticoduodenal artery aneurysms in association with celiac stenosis/occlusion. Semin Intervent Radiol 2009;26:215e23. 7. Bageacu S, Cuilleron M, Kaczmarek D, et al. True aneurysms of the pancreaticoduodenal artery: successful non-operative management. Surgery 2006;139:608e16. 8. Takao H, Doi I, Watanabe, et al. Natural history of true pancreaticoduodenal artery aneurysms. Br J Radiol 2010;83: 744e6. 9. Suzuki K, Kashimura H, Sato M, et al. Pancreaticoduodenal artery aneurysms associated with celiac axis stenosis due to compression by median arcuate ligament and celiac plexus. J Gastroenterol 1998;33:434e8.
Annals of Vascular Surgery
10. Sampsel JW, Barry FM, Steele HD. Aneurysm of an anomalous pancreaticoduodena1 artery. Arch Surg 1952;64:74e9. 11. Hasselgren PO, Rottier A. Aneurysm of the pancreaticoduodenal artery, a case report. Acta chir Scand 1976;142:543e4. 12. Scheflan M, Kadir S, Athanasoulis CA, et al. Pancreaticoduodenal artery aneurysm simulating carcinoma of the head of the pancreas. Arch Surg 1977;112:1201. 13. Kadir S, Athanasoulis CA, Yune HY, et al. Aneurysms of the pancreaticoduodenal arteries in association with celiac axis occlusion. Cardiovasc Radiol 1978;1:173e7. 14. Widjaja A, Rosenthal H, Bleck J, et al. Pitfall: a pseudo tumor within the left liver lobe presenting with abdominal pain, jaundice and severe weight loss. Ultraschall Med 1999;20:268e72. 15. Colak MC, Kocaturk H, Bayram E, et al. Inferior pancreaticoduodenal artery false aneurysm: a rare cause of gastrointestinal bleeding diagnosed by three-dimensional computed tomography. Singapore Med J 2009;50:346e9. 16. Wattez H, Lancelevee J, Perot C, et al. Compressive pancreaticoduodenal artery aneurysm associated with celiac artery stenosis. Ann Vasc Surg 2013;27:1187.e1e4. 17. Grabbe E, Bucheler E. B€ uhler’s anastomosis (author’s transl). Rofo 1980;132:541e6. 18. Bertelli E, Di Gregorio F, Mosca S, et al. The arterial blood supply of the pancreas: a review. V. The dorsal pancreatic artery. An anatomic review and a radiologic study. Surg Radiol Anat 1998;20:445e52. 19. Lin PH, Chaikof EL. Embryology, anatomy, and surgical exposure of the great abdominal vessels. Surg Clin North Am 2000;80:417e33. 20. Gangahar DM, Carveth SW, Reese HE, et al. True aneurysm of the pancreaticoduodenal artery: a case report and review of the literature. J Vasc Surg 1985;2:741e2. 21. Retzlaff JA, Hagedom AB, Bartholomew LG. Abdominal exploration for gastrointestinal bleeding of obscure origin. JAMA 1961;177:104e7. 22. Khan KN, Nakata K, Shima M, et al. Pancreatic tissue damage by transcatheter arterial embolization for hepatoma. Dig Dis Sci 1993;38:65e70. 23. Paty PS, Cordero JA Jr, Darling RC 3rd, et al. Aneurysms of the pancreaticoduodenal artery. J Vasc Surg 1996;23:710e3. 24. Lindner HH, Kemprud E. A clinicoanatomic study of the arcuate ligament of the diaphragm. Arch Surg 1971;103: 600e5. 25. Bednarek M, Budzynski P, Pozniczek M, et al. Percutaneous ultrasound-guided drainage of the biliary tree in palliative treatment of mechanical jaundice: 17 years of experience. Wideochir Inne Tech Malo Inwazyjne 2012;7:193e6. 26. Sheldon DM, Crawford M, Mihrshahi S, et al. Therapeutic considerations in obstructive jaundice due to hepatic artery aneurysm. HPB (Oxford) 2006;8:74. 27. Eckhauser FE, Stanley JC, Zelenock GB, et al. Gastroduodenal and pancreaticoduodenal artery aneurysms: acomplication of pancreatitis causing spontaneous gastrointestinal hemorrhage. Surgery 1980;88:335e44. 28. Sciurne C, Geraci G, Pisello F, et al. ‘‘Randez-vous’’ technique for palliation of neoplastic jaundice: personal experience. Ann Ital Chir 2004;75:634e47. 29. Donmez H, Men S, Dilli A, et al. Giant gastroduodenal artery pseudoaneurysm due to polyarteritis nodosa as a cause of obstructive jaundice: imaging findings and coil embolization results. Cardiovasc Intervent Radiol 2005;28:850e3. 30. Kuroiwa T, Yoshimitsu K, Honda H, et al. Re: Two-stage transcatheter arterial embolization of a large hepatic artery pseudoaneurysm causing obstructive jaundice. Cardiovasc Intervent Radiol 2002;25:76e7.
Pancreaticoduodenal artery aneurysm (PDA) is quite rare, which accounts for only approximate 2% of all visceral aneurysms. Besides, PDA is usually related to celiac axis stenosis (CAS) and prone to rupture. Advanced imaging examination can facilitate the disclosure of such peripancreatic masses, but most of them were seldom diagnosed until they rupture because of the nonspecific symptoms. Secondary to PDA, obstructive jaundice is however an extremely rare manifestation. A case of an 84-year-old man is reported here, who suffered from severe jaundice caused by a ruptured PDA associated with CAS. In addition, this review collects and organizes PDAs with jaundice by applying a MEDLINE search and discusses the pathogenesis and therapeutic options of these aneurysms leading to external compression over the bile duct. Consequently, the formation of PDA with obstructive jaundice is based on the specific anatomy of pancreaticoduodenal arcades. When there is a retroperitoneal mass around the head of the pancreas associated with unexpected jaundice, PDA should be considered, for which early aggressive therapy is required. The case report and literature review suggest that PDA associated with obstructive jaundice may be treated successfully by single transcatheter arterial embolization (TAE) without auxiliary biliary drainage, whether it ruptures or not.
Pancreaticoduodenal artery aneurysms (PDAs) are quite rare, which account for only 2% of all visceral artery aneurysms.1 Since Sutton and Lawton 2 firstly put forward the relation between CAS and PDA, approximate 63% of reported cases had been found to be related to CAS.3 More than 60% of PDAs are shown to be at rupture, and emergent treatments are required.4 Although advanced imaging examination facilitates the disclosure of such
Conflicts of Interest: The authors state that there are no conflicts of interests to declare. Department of Hepatobiliary Surgery, The First Affiliated Hospital of Chongqing Medical University, Chongqing, China. Correspondence to: Dewei Li, MD, PhD, Department of Hepatobiliary Surgery, The First Affiliated Hospital of Chongqing Medical University, No.1 Friendship Road, Yuzhong District, Chongqing 400000, China; E-mail: [email protected] Ann Vasc Surg 2015; 29: 1016.e1e1016.e6 http://dx.doi.org/10.1016/j.avsg.2014.12.011 Ó 2015 Elsevier Inc. All rights reserved. Manuscript received: July 19, 2014; manuscript accepted: December 14, 2014; published online: March 11, 2015.
peripancreatic masses, most of them were seldom diagnosed until they rupture because of the nonspecific symptoms. Secondary to PDA, Jaundice is however an extremely rare manifestation. A case of an 84-year-old man is reported here, who suffered from severe jaundice caused by a ruptured PDA associated with CAS. In addition, this review collects and organizes PDA with jaundice by adopting a MEDLINE search and discusses the pathogenesis and therapeutic options of these aneurysms leading to external compression over the bile duct.
CASE REPORT An 84-year-old man was admitted to the local hospital, who suffered from a sudden onset of abdominal pain in the right upper quadrant. There was no history of abdominal trauma, chronic pancreatitis, bile duct calculus, or alcohol abuse, but he had suffered from hypertension for 10 years. On admission, the blood pressure was 85/ 54 mm Hg, and an immediate ultrasound showed that 1016.e1
1016.e2 Case reports
there was a retroperitoneal mass. Subsequently, an urgent laparotomy was performed and the gigantic mass was found to be a retroperitoneal hematoma. The hematoma was located in the peripancreatic region without a clear limit out of the surrounding tissue, which had high surface tension with the tendency to rupture, but no bleeding focus was found. On account of the limitations in local technology and experience, further dissection around the mass was difficult and risky. Considering the patient safety, the surgeons set up a drainage tube and the operation was terminated. The advanced age patient was intubated during the laparotomy and extubated the next day. Postoperative vital signs stabilized without any vasoactive substance, but he was transferred to the hospital because of unexpected jaundice. Subsequent laboratory analysis revealed that a serum total bilirubin was at 24.4 mg/dL, direct bilirubin at 21.5 mg/dL, alanine transaminase at 60 m/L, and gamma glutamyl transpeptidase at 84 m/L, which indicated severe obstructive jaundice. Additionally, abdominal contrast-enhanced computed tomography (CT) and CT angiography showed that retroperitoneal hematoma was secondary to ruptured pancreaticoduodenal artery aneurysm with celiac axis stenosis (Fig. 1AeC). Angiography under local anesthesia confirmed the existence of PDA with a diameter of 2 cm, which is derived from the first branch of the superior mesenteric artery on the right side (Fig. 2AeC). In the meantime, the initial involvement of the celiac axis was obviously narrowed, which was compensated by vascular revascularization via the pancreaticoduodenal artery arcades. A guiding catheter was inserted to the branch of the superior mesenteric artery, and superselective arterial embolization was successful by placing 2 microcoils in the proximal portion of the aneurysm (Fig. 3A). However, it was infeasible to embolism the distal main vessel, for there was no definitive vessel that supplied blood to the aneurysm. Arteriography of SMA (Fig. 3B) shows that the aneurysm had disappeared, and selective angiography of the gastroduodenal artery via celiac axis confirmed similarly that there was no ‘‘back’’ bleeding from the distal pancreaticoduodenal artery branch (Fig. 3C). The patient was returned to intensive care unit, and the jaundice subsided quickly after endovascular treatment. There were no further signs of bleeding from the aneurysm during admission. Unfortunately, the patient was subjected to multidrug-resistant respiratory infection and died from breathing failure 26 days after embolization.
DISCUSSION As an uncommon vascular disease, PDA was first reported in 1895 by Ferguson.5 These aneurysms can be divided into the true aneurysms frequently related to celiac axis stenosis and the pseudoaneurysms whose underlying cause is the
Annals of Vascular Surgery
peripancreatic inflammation or trauma involving chronic pancreatitis and abdominal injury. According to Kallamadi et al.,6 abdominal pain is the most common initial symptom of PDA. However, it is difficult to establish an early diagnosis of PDA until it ruptures, which results in a mortality rate of 26%.3 Most of them usually rupture into the retroperitoneal space around the head of pancreas, which simulates the peripancreatic mass. In addition, whether to rupture is hardly dependent on the size of aneurysms (the median diameter is 22.2 mm in patients with rupture, 21.4 mm to the contrary), but dependent on the propensity for rupture itself.3,7e9 Both a gigantic aneurysm itself and a secondary retroperitoneal hematoma can produce oppression over surrounding tissues, which leads to obstructive jaundice uncommonly. In this regard, retroperitoneal mass around the head of the pancreas with jaundice can arouse the awareness of PDA, and early aggressive therapy is required. ‘‘jaundice’’, ‘‘iterus’’, ‘‘pancreaticoduodenal’’, ‘‘aneurysm’’ and ‘‘pseudoaneurysm’’ are regarded as key words and searched in the MEDLINE database by electronic and manual searches. As a result, totally 8 cases of PDA associated with jaundice are collected and organized in Table 1.9e15 Unexpected jaundice is based on the specific peripancreatic anatomy (Picture 1). The pancreaticoduodenal arcades are around the head of pancreas and formed by the branches of celiac axis and superior mesenteric artery (SMA), which supply corresponding tissues and organs. The anterior and posterior of the superior pancreaticoduodenal artery rooted in gastroduodenal artery are connected with the anterior and posterior of the inferior pancreaticoduodenal artery derived from SMA, respectively. There are several rami communicantes between the celiac axis and SMA, such as the Arc of Buhler and the Arc of Barkow.17e19 Because of CAS and provided by the common hepatic artery, the flow to the liver, spleen and stomach, spleen artery, and left gastric artery is compensated by the arcades that serve as a retrograde bypass pathway. As a result, the accompanied arteries dilate, which is supposed to be PDA. It usually gathers on the duodenal ampulla along with the pancreatic duct behind the head of the pancreas, whereas the distal common bile duct tends to be compressed by the unruptured PDA or the hematomas caused by rupture. The bulky mass exerts direct pressure over the common bile duct, the head of the pancreas, and duodenum, which results in obstruction. As in our case, a giant retroperitoneal hematoma oppresses the extrahepatic duct, which
Vol. 29, No. 5, July 2015
Case reports 1016.e3
Fig. 1. Contrast-enhanced abdominal CT scan shows a retroperitoneal hematoma accompanied with suspicious aneurysm (A). Stenosis of the celiac trunk (B). Vascular reconstruction of CT angiography (C, D).
leads to abundant biliary countercurrents into the hepatic sinusoid. On the basis of these factors, severe icterus occurs rapidly. Pancreaticoduodenal artery aneurysms often manifest as retroperitoneal masses around the head of the pancreas. Accompanied with unexpected jaundice, in particular with painless jaundice, PDAs are inclined to be confused with pancreatic head carcinoma. As described in this instance, the role of ultrasound is controversial because of its weakness in detecting such a retroperitoneal mass related to PDA, on account of the influence of intestinal gas, level of experience of the operator, and lack of discrimination. Instead, contrast-enhanced CT has become the golden standard, which qualitatively reveals this indeterminate lesion, especially the CT angiography and 3-dimensional reconstruction offer valuable first-hand information concerning the aneurysm. In our review, in addition to a few cases at early stage, most PDAs with jaundice are
discovered by contrast-enhanced CT. Moreover, intravascular angiography can confirm and locate the aneurysms, as well as judge and cure. In cases of severe jaundice, some patients undergo magnetic resonance cholangio-paneretography (MRCP) to visually delineate the biliary tree. In the early days, patients with PDA were subjected to surgical treatment, which was first reported in 1951 by Van Ouwerkerk.20 Nevertheless, approximately 70% of open surgery failed and even PDA could be detected, as in our case.3,21 Afterward, endovascular therapy became the preferred, and most effective method after the imaging techniques was available. This was supported by Mandel et al., who statistically analyzed that a success ratio of 79% and a mortality rate of 0% were linked to the procedure.22 To obtain the road map of pancreaticoduodenal arcades under digital subtraction angiography, selective catheterization and angiography of the superior mesenteric
1016.e4 Case reports
Annals of Vascular Surgery
Fig. 2. Selective superior mesenteric arteriography (A, B) and superselective injection (C) shows dilation of the pancreaticoduodenal arcade and a saccular aneurysm with retrograde filling of the celiac artery branches.
Fig. 3. Superselective arterial embolization in the proximal portion of the aneurysm (A). Arteriography of SMA ensures no contrast agent regurgitation around the aneurysm after embolization (B). Selective angiog-
artery are required. Meanwhile, appropriate coils can be placed in the lump body using a microcatheter or in both inflow and outflow of the aneurysm when fails. However, it also carries the risk of coil migration, ischemia of hepatic tissue, and failure of operation. Surgical technique is still widely considered as the definitive therapy for PDA, as the open approach is to expose and ligate the aneurysm directly. However, when these aneurysms rupture or hide in the parenchyma of the pancreas, the process of location and isolation can be difficult. Since Sutton and Lawton2 first put forward the significance of celiac axis stenosis in PDA, an increasing number of studies had supported this viewpoint and
raphy of the gastroduodenal artery via celiac axis confirms that there is no ‘‘back’’ bleeding from the distal PDA branch (C).
Picture 1. The peripancreatic anatomy.
more than half of all cases had been found to match this situation.3 CAS may be secondary to
Vol. 29, No. 5, July 2015
Case reports 1016.e5
Picture 2. Bile duct compressed by the unruptured aneurysm or the hematoma caused by rupture (A, B). Bile duct decompressed after ERCP (C), PTCD (D), surgical treatment (E), and TAE (F, G).
Table I. Reported cases of pancreaticoduodenal artery aneurysm with obstructive jaundice Author
Year
Age, sex
Presentation
CAS
Rupture
Treatment
Outcome
Sampsel et al.10
1952
68, Male
Painless jaundice
NA
Yes
Death
Hasselgren et al.11
1976
62, Female
NA
No
Scheflan et al.12
1977
56, Male
Pain under the right costal margin, jaundice Painless jaundice, pruritus
Yes
Yes
Kadir et al.13
1978
72, Male
Yes
No
Widjaja et al.14
1999
51, Male
Gastrointestinal bleeding, jaundice Epigastric pain, jaundice
Hepaticodochoenterostomy Aneurysmectomy Exploratory choledochotomy Aneurysmectomy Exploratory choledochotomy + T-tube drainage Untreated
NA
Yes
Colak et al.15
2009
57, Male
No
Yes
Wattze et al.16
2013
64, Female
Yes
No
Current report
2014
84, Male
Epigastric pain, jaundice, indigestion, weight loss Acute abdominal pain, biliary tract dilatation (14 mm; by ultrasound scan) Abdominal pain in the right upper quadrant, jaundice
Yes
Yes
PTCD Transcatheter arterial embolization Untreated
Laparotomy and injection of polymeric synthetic into the aneurysm Transcatheter arterial embolization
Survival
Survival
Death Survival
Death
Survival
Death
NA, not available.
atherosclerosis, pancreatitis, connective tissue disorders, polyarteritis nodosa, Takayasu disease, trauma, necrotizing arteritis, fibromuscular dysplasia, and so forth,23 whereas the most studied and relevant element is the compression by median arcuate ligament, which can be seen in 10e24% of patients.24 Various therapeutic methods are used in the management of obstructive jaundice resulting from aneurysms that involve PDA or retroperitoneal hematoma caused by rupture (Picture 2). Percutaneous transhepatic cholangial drainage (PTCD) is generally a safe and well-tolerated method with a few complications, such as weakness, pain, and biliary leak.25,26 It is an alternative remedy for mechanical jaundice even if
there is no way to decompress the common bile duct. In addition, endoscopic retrograde cholangiopancreatograph (ERCP) with biliary stent insertion is considered to be less invasive and providing a fair delineation of biliary anatomy, nevertheless, on the condition that the bile duct is compressed by an aneurysm, especially the one whose wall is very thin and fragile, and the stent has the potential to enter the aneurysm, which leads to massive hemorrhage.27 Although jaundice companied by aneurysm appears to be resolved simultaneously, surgical treatment is less recommended because of its difficulty and extensive damage. Retzlaff reported that in nearly 70% of cases, even the bleeding point was not identified at
1016.e6 Case reports
operation, just as described in our case.21,27 Sciurne et al. also emphasized the technique of PTCD combined with ERCP, called ‘‘rendezvous.’’28 In our case, we detect a notable outcome that jaundice gets improved after single transcatheter arterial embolization (TAE). Furthermore, similar situations are observed in some cases, as Donmez and Kuroiwa described.29,30 The bile duct is decompressed after effective endovascular therapy, which is due to the reduced volume of isolated aneurysm or the decreased surface tension of absorbed hematoma. Therefore, it is speculated that PDA associated with obstructive jaundice may be treated successfully by single TAE technique without any auxiliary biliary drainage, whether it ruptures or not. More research is needed. Pancreaticoduodenal Artery Aneurysms associated with jaundice are very rare. PDA is usually related to celiac axis stenosis, whose pathogenesis is based on the specific anatomy of pancreaticoduodenal arcades. When there is a retroperitoneal mass around the head of the pancreas associated with unexpected jaundice, PDA should be considered, for which early aggressive therapy is required. The case report and literature review suggest that the pancreaticoduodenal artery aneurysm associated with obstructive jaundice may be treated successfully by single TAE without auxiliary biliary drainage, whether it ruptures or not.
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Annals of Vascular Surgery
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