Archives of Academic Emergency Medicine. 2019; 7 (1): e25 CA S E RE P O RT Hepatic Artery Aneurysms as a Rare but Important Cause of Abdominal Pain; a Case Series Hamidreza Haghighatkhah1, Morteza Sanei Taheri1, Seyed Mohammadhadi Kharazi1, Maryam Zamini1, Sahar Rabani Khorasgani1∗, Zahra Jahangiri Zarkani1 1. Radiology Department, Shohadaye Tajrish Hospital, Shahid Beheshti University of Medical Sciences, Tehran, Iran. Received: February 2019; Accepted: March 2019; Published online: 6 April 2019 Abstract: Hepatic artery aneurysm (HAA) is the common visceral aneurysms with the highest reported rate of rupture. The clinical manifestations depending on the size of the aneurysm include epigastric pain, obstruction of biliary tract, rupture and death. Imaging modalities like computed tomography (CT) scan and CT-angiography have a valuable role in the early detection of HHAs. Complications and selecting appropriate treatments depending on the size and location of the aneurysms. This article aimed to report clinical presentation, imaging finding and treatment of some patients presenting with HAAs to emergency department. Keywords: Aneurysm; hepatic artery; abdominal pain; abdomen, acute; angiography Cite this article as: Haghighatkhah H, Sanei Taheri M, Kharazi S M, Zamini M, Rabani Khorasgani S, Jahangiri Zarkani Z. Hepatic Artery Aneurysms as a Rare but Important Cause of Abdominal Pain; a Case Series. Arch Acad Emerg Med. 2019; 7 (1): e25. 1. Introduction Hepatic artery aneurysm (HAA) is a rare disease (0.002%– 0.4%) but is a clinically important phenomenon (1). HAAs are traditionally the second most common visceral aneurysms with an incidence of 20% and have the highest (44%) re- ported rate of rupture (2). The clinical manifestations de- pending on the size of the aneurysm include epigastric pain, obstruction of biliary tract, rupture and death. Imag- ing modalities like computed tomography (CT) and CT- angiography have a valuable role in the early detection of HHA, its complications, and selecting appropriate treat- ments depending on the size and location of the aneurysms. In this case series, we reported 5 HAA cases with different eti- ologies, presentations and treatments. 2. Case presentations: 2.1. Case 1 The first case was a 50-year-old man with acute-onset epigas- tric and right upper abdominal pain. Abdominal ultrasonog- raphy demonstrated free abdominal fluid with internal clots. CT angiography was performed, which revealed a ruptured ∗Corresponding Author: Sahar Rabani Khorasgani; Department of radiol- ogy, Shohadaye Tajrish Hospital, Tajrish Square, Tehran, Iran. Email: ra- bani1369@yahoo.com Tel: 009822721155 Figure 1: A) Axial contrast-enhanced CT (CECT) scan demonstrate contrast filled saccular out-pouching at the level of the left hepatic artery (black arrow). Hemoperitoneum with areas of higher attenu- ation indicates acute active bleeding is present. Peripheral segmen- tal liver hypo-densities due to de-vascularization and vasospasm are also evident. B) Axial CECT through the celiac trunk level shows left gastric artery aneurysm with intraluminal thrombosis. C) Coro- nal Maximum Intensity Projection (MIP) reformation demonstrates concomitant aneurysm of hepatic artery (long arrow) and left gastric artery (short arrow). D) A volume-rendered 3D CT angiography im- age displays a small saccular left gastric artery aneurysm. proper HAA with hemo-peritoneum in perihepatic space, This open-access article distributed under the terms of the Creative Commons Attribution NonCommercial 3.0 License (CC BY-NC 3.0). Downloaded from: http://journals.sbmu.ac.ir/aaem H. Haghighatkhah et al. 2 para-colic gutters, and massive abdominopelvic hematoma. A simultaneous visceral aneurysm was also detected at the origin of the left gastric artery. The exploratory laparotomy revealed 2.5 liters of hemo-peritoneum, a ruptured aneurys- mal sac in proximal of the left branch of proper hepatic artery with surrounding clots and intact liver parenchyma. Ligation in the proximal and distal parts of artery was done (Figure 1). 2.2. Case 2 The second patient was an 11-year-old boy with history of falling and blunt trauma to the flank and formation of liver hematoma. Three weeks later, he was referred to emergency department with the chief complaint of abdominal pain. Af- ter proving pseudo-HAA in contrast-enhanced computed to- mography (CECT) scan, the patient was referred for catheter base angiography and treated with coiled embolization (Fig- ure 2). Figure 2: E, F) Axial contrast-enhanced CT (CECT) scan in ar- terial phase reveals out-pouching pseudo-aneurysm in hepatic artery branch along with intra parenchymal and sub-capsular liver hematoma (arrow). G) Selective digital subtraction angiography (DSA) from hepatic artery shows large pseudo-aneurysm in right hepatic artery. The patient underwent coil embolization for treat- ment of aneurysm. H) Aneurysm disappears after embolization. 2.3. Case 3 The patient was a 66-year-old male with epigastric pain and nausea for 10 days. Past medical history revealed smok- ing with hypertension. CT angiography showed saccular aneurysm in proper hepatic artery. The patient underwent surgical repair. A 70 × 70 mm aneurysmal lesion at the origin of proper hepatic artery was found in lesser sac and gastro- hepatic ligament. After attaining control of proper hepatic artery, end-to-end bypass graft was done in hepatic artery us- ing saphenous vein. After 12t h post-operative day hospital- ization, the patient referred to the hospital a week later with severe abdominal pain and vomiting. In CT scan, there was evidence of acute necrotizing pancreatitis, along with collec- tion in liver. The patient was successfully managed with con- servative treatment and discharged after two weeks without major complication (Figure 3). Figure 3: Figure 3: I, J) Axial computed tomography (CT) an- giogram showing spontaneous saccular aneurysm in proper hep- atic artery with peripheral thrombosis and internal diameter of 44 mm, and evidence of hematoma (white arrow) with compression to second part of duodenum and the head of pancreas. K) Volume- rendered 3D CT angiography image displays aneurysm from com- mon hepatic artery (red arrow) and hematoma (black arrow) below it. L) The patient was referred one week after surgical repairing fol- lowing abdominal pain. Contrast-enhanced CT (CECT) scan find- ing showed necrotizing center with severe edema around the head of pancreas with extension to porta-hepatis, peri-portal, and ante- rior sub-hepatic space suggestive of acute necrotizing pancreatitis. 2.4. Case 4 The patient was a 4-year-old boy with history of blunt trauma to his right flank. One month later, the patient admitted with melena, hematemesis, and epigastric pain. Color Doppler ul- trasonography, CECT scan, and magnetic resonance imaging (MRI) showed HAA. The patient was treated successfully with coiled embolization (Figure 4). 2.5. Case 5 The patient was a 24-year-old male with history of gunshot traumatization and surgery due to hepatic artery aneurysm and liver hematoma. He was referred due to re-bleeding and large liver-infected hematoma. The patient underwent suc- cessful coil embolization similar to case 2. 3. Discussion True HAAs are mostly due to degenerative or dysplastic change of the extrahepatic vessels. Their main cause is atherosclerosis (3), although vasculitis have also been re- ported (4). Pseudo-HAAs, accounting for approximately 20% of all HAAs (5), can be as intrahepatic or extrahepatic. Ap- proximately 80% of aneurysms of the hepatic artery are ex- trahepatic which are mostly spontaneous, usually due to im- This open-access article distributed under the terms of the Creative Commons Attribution NonCommercial 3.0 License (CC BY-NC 3.0). Downloaded from: http://journals.sbmu.ac.ir/aaem 3 Archives of Academic Emergency Medicine. 2019; 7 (1): e25 Figure 4: A) Ultrasound showed a hyper echo heterogeneous ill- defined lesion (arrow) in the right lobe of liver measuring 30*40 mm in favor of hematoma. B) Axial contrast-enhanced CT scan corre- lated with ultrasound finding (arrow). The patient underwent con- servative treatment. C, D, E) One month later, ultrasound revealed a 23*23 mm hyper echo lesion with central 14*14 mm hypo echo vascular area in segment 8 of liver (an aneurysmal lesion). F) Axial T2W MRI showed a round hyper signal lesion (arrow), the same sig- nal with aorta. G, H) Selective digital subtraction angiography (DSA) from right hepatic artery shows pseudo-aneurysm in right hepatic artery. The patient underwent coil embolization for treatment of aneurysm (arrow) and aneurysm disappeared after embolization (ar- row). munosuppression, biloma, and biliary tract infection (2, 6). The causes of intrahepatic pseudo-aneurysms are usually iatrogenic (liver transplantation, cholecystectomy, etc.), al- though they still have a potential for severe hemorrhage (6). The clinical manifestations tend to be non-specific, depend- ing on the size of the aneurysm. Although small HAAs are of- ten asymptomatic, the natural history is for progressive en- largement with increasing risk of rupture and death. HAAs may cause right upper quadrant and epigastric pain or ob- struction of biliary tract. The classic triad of Quinke’s, i.e., obstructive jaundice, abdominal pain and hemobilia, is seen in 30% of the patients (7). Inflammation associated with sep- Figure 5: I, J, K) Axial contrast enhanced computed tomography (CECT) scan in arterial phase and coronal reformatted images from liver show pseudo-aneurysm in right hepatic artery (arrow) with in- fected liver hematoma containing gas density and also sub-capsular hematoma. tic emboli may erode the arterial wall and then prompt HAA rupture, causing hemobilia (8). The presence of a common channel for both pancreatic and biliary ducts with no ac- cessory duct drainage is predisposed to the development of acute pancreatitis due to the blockage of the pancreatic duct by blood clot (9). Multiple diagnosis tests are used, such as abdominal ultra- sound, CT, CT angiography, MRI, endoscopy and angiog- raphy. Angiography is a therapeutic modality of choice in splanchnic aneurysms through embolization (10). The sen- sitivity and specificity of multi-detector computed tomo- graphic angiography to diagnose aneurysm of the hepatic ar- teries was 100% (11). The size and location of the aneurysm, patient age and comorbidities have critical role in determin- ing the specific approach (12). Trans-arterial embolization (TAE) has a high rate of success for all causes of HAA (13). For intrahepatic aneurysms, embolization is the accepted treatment (3). Surgical treatment of ruptured HAAs can be allocated when the patient is in an unstable condition, the aneurysm is extrahepatic or larger than 2 cm, and endovas- cular intervention fails (1). Lee et al. reported an incidental large HAA and the patient was undergone open surgery for aneurysmectomy and the proper hepatic artery was anasto- mosed with gastrodeodenal artery (14). This open-access article distributed under the terms of the Creative Commons Attribution NonCommercial 3.0 License (CC BY-NC 3.0). Downloaded from: http://journals.sbmu.ac.ir/aaem H. Haghighatkhah et al. 4 4. Discussion Visceral aneurysms such as hepatic artery aneurysm should be considered as a rare but important differential diagno- sis of acute abdominal pain. All emergency physicians and surgeons should kept this diagnosis in their mind to prevent from its life threatening complications. 5. Ethical statement All patients were followed up with a detailed description of the purpose of the research and written consent was ob- tained. Patient names were not included in the study and coding was used to classify the information. The benefits of participation in the plan were to undertake an important step in managing patients with similar disease. The cost of partic- ipation in the scheme was free. 6. Appendix 6.1. Acknowledgements None. 6.2. Authors contribution The author met the standard criteria for authorship based on the recommendations of the international committee of medical journal editors. Authors ORCIDs Hamidreza Haghighatkhah: 0000-0001-5411-0609 Morteza Sanei Taheri: 0000-0002-8881-9058 Sahar Rabani Khorasgani: 0000-0001-7927-6429 6.3. Conflict of interest None. 6.4. Funding No fund has been received. References 1. Altaca G. Ruptured aneurysm of replaced left hepatic artery as a cause of haemorrhagic shock: a challenge of diagnosis and treatment. Interactive cardiovascular and thoracic surgery. 2011;14(2):220-2. 2. Arneson MA, Smith RS. Ruptured hepatic artery aneurysm: case report and review of literature. An- nals of vascular surgery. 2005;19(4):540-5. 3. Dolapci M, Ersoz S, Kama NA. Hepatic artery aneurysm. Annals of vascular surgery. 2003;17(2):214-6. 4. Huisman M, van den Bosch MA, Mooiweer E, Mole- naar IQ, van Herwaarden JA. Endovascular treatment of a patient with an aneurysm of the proper hepatic artery and a duodenal fistula. Journal of vascular surgery. 2011;53(3):814-7. 5. Bennett J, Fay D, Krysztopik R. Right hepatic artery false aneurysm secondary to acalculous cholecystitis. Case Reports. 2010;2010:bcr0820103258. 6. Jeng K-S, Huang C-C, Lin C-K, Lin C-C, Liang C-C, Chung C-S, et al., editors. Early detection of a hepatic artery pseudoaneurysm after liver transplantation is the deter- minant of survival. Transplantation proceedings; 2016: Elsevier. 7. Turkvatan A, Okten R, Kelahmet E, Ozdemir E, Olcer T. Hepatic artery aneurysm: imaging findings. J Ankara Univ Fac Med. 2005;58:73-5. 8. Yu YH, Sohn JH, Kim TY, Jeong JY, Han DS, Jeon YC, et al. Hepatic artery pseudoaneurysm caused by acute id- iopathic pancreatitis. World Journal of Gastroenterology: WJG. 2012;18(18):2291. 9. Mee AS, Turner JA, Gow NM. Recurrent acute pancreatitis due to haemobilia from a hepatic artery aneurysm. Post- graduate medical journal. 1983;59(695):590-2. 10. BERNAL ADPA, LOURES P, CALLE JCO, CUNHA B, COR- DOBA JC. Right hepatic artery aneurysm. Revista do Colegio Brasileiro de Cirurgioes. 2016;43:401-3. 11. Kayahan Ulu EM, Coskun M, Ozbek O, Tutar NU, Ozturk A, Aytekin C, et al. Accuracy of Multidetector Computed Tomographic Angiography for Detecting Hepatic Artery Complications After Liver Transplantation. Transplanta- tion Proceedings. 2007;39(10):3239-44. 12. Saeed H, Buxey KN, Milne CPE, Cox G. Quinke’s triad: haemobilia secondary to hepatic artery aneurysm. ANZ journal of surgery. 2017. 13. Alrajraji M, Nawawi A, Jamjoom R, Qari Y, Aljiffry M. De- layed hemobilia due to hepatic artery pseudo-aneurysm: a pitfall of laparoscopic cholecystectomy. BMC surgery. 2016;16(1):59. 14. Lee D, Chung BH, Heo SH, Park YJ, Kim DI. Case Report of a Large Common Hepatic Artery Aneurysm. Ann Vasc Surg. 2018;52:316.e11-.e13. This open-access article distributed under the terms of the Creative Commons Attribution NonCommercial 3.0 License (CC BY-NC 3.0). Downloaded from: http://journals.sbmu.ac.ir/aaem Introduction Case presentations: Discussion Discussion Ethical statement Appendix References