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대한내과학회지: 제 88 권 제 5 호 2015 http://dx.doi.org/10.3904/kjm.2015.88.5.560 무증상의 빈혈로 발현된 상장간막동맥 거짓동맥류 파열 1예 아주대학교 의과대학 소화기내과학교실 김주성·진우람·이길호·황수현·이연경·임기현·임선교 Rupture of a Superior Mesenteric Artery Pseudoaneurysm Presenting with Asymptomatic Anemia Joo-Sung Kim, U-Ram Jin, Gil-Ho Lee, SuHyun Hwang, YeonKyung Lee, Kihyun Lim, and Sun Gyo Lim Department of Gastroenterology, Ajou University School of Medicine, Suwon, Korea Anemia is a common cause of referrals to gastroenterologists. Only a small number of anemia cases result from vascular abnormalities. Visceral artery aneurysms and pseudoaneurysms are rare forms of vascular disease that have significant potential for rupture, resulting in potentially life-threatening hemorrhaging. We present the case of a 70-year-old female patient with a pseudoaneurysm of the superior mesenteric artery complicated with rupture, who had no abdominal pain and only anemia. (Korean J Med 2015;88:560-563) Keywords: Mesenteric artery, Superior; Pseudoaneurysm; Anemia INTRODUCTION with confirmed anemia, unless there is a history of significant overt non-gastrointestinal blood loss. If no obvious lesions are Blood loss from the gastrointestinal tract is the most common detected by esophagogastroduodenoscopy and colonoscopy, eval- cause of anemia in adult men and postmenopausal women. uation of the small-bowel by video capsule endoscopy or en- Other causes of anemia include intestinal malabsorption, as in teroscopy should also be considered in patients with symptoms celiac disease, gastrectomy, or gastric atrophy through achlorhy- suggestive of small bowel disease. However, in some cases, the dria, and Helicobacter pylori infection. A small subset of ane- causes of anemia are not detected by an endoscopic examination mia is caused by vascular abnormalities. alone. An endoscopic evaluation should be considered in patients Visceral artery aneurysms and pseudoaneurysms are vascular Received: 2014. 7. 15 Revised: 2014. 8. 18 Accepted: 2014. 10. 25 Correspondence to Sun Gyo Lim, M.D. Department of Gastroenterology, Ajou University School of Medicine, 164 World cup-ro, Yeongtong-gu, Suwon 443-380, Korea Tel: +82-31-219-6939, Fax: +82-31-219-5999, E-mail: [email protected] * Conflict of Interest: The authors disclose no potential conflicts of interest. Copyright ⓒ 2015 The Korean Association of Internal Medicine This is an Open Access article distributed under the terms of the Creative Commons Attribution - 560 - Non-Commercial License (http://creativecommons.org/licenses/by-nc/3.0/) which permits unrestricted noncommercial use, distribution, and reproduction in any medium, provided the original work is properly cited. - Joo-Sung Kim, et al. Anemia by rupture of SMA pseudoaneurysm - 3 abnormalities that cannot be detected readily by routine endo- 186,000/mm and prothrombin time was 15.1 s (international nor- scopic examinations. In addition, diagnosis of vascular diseases is malized ratio [INR] = 1.43). On routine chemistry, total pro- more difficult because patients do not have predisposing symp- tein/albumin was 5.2/2.4 g/dL, AST/ALT was 8/5 IU/L, total bi- toms in most cases. Thus, the condition of patients with these lirubin was 0.2 mg/dL, and alkaline phosphatase was 55 IU/L. vascular abnormalities can quickly become life-threatening due Due to the severe anemia, packed red blood cells (RBCs) to the sudden rupture of an aneurysm or pseudoaneurysm [1,2]. were transfused. After the transfusion, hemoglobin was elevated We report a case of a 70-year-old female with a ruptured to 11.4 g/dL. However, it decreased to 7.6 g/dL after 15 h. pseudoaneurysm of the superior mesenteric artery (SMA), which To investigate the possibility of gastrointestinal bleeding, was diagnosed with an abdominal contrast-enhanced computed esophagogastroduodenoscopy and sigmoidoscopy were performed. tomography (CT) scan and treated by angiographic emboliza- However, we found nothing that would suggest gastrointestinal tion. bleeding. In addition, a contrast-enhanced CT scan was performed to find the cause of the bleeding. The scan revealed an -1.3 cm CASE REPORT pseudoaneurysm of the SMA, in addition to a hematoma and A 70-year-old female patient was admitted to the hospital hemoperitoneum due to the ruptured pseudoaneurysm (Fig. 1). with mild dizziness. The patient had hypertension. She regularly Angiography for SMA was performed with access to the right took a calcium channel blocker and an angiotensin receptor femoral artery. A pseudoaneurysm was found in a branch of the blocker. Her blood pressure was known to be well-controlled SMA (Fig. 2). according to her family, although exact information about her The main feeding vessel was selected by microcatheter, and blood pressure before admission was not provided. She had no a selective embolization with glue was performed at a feeding specific familial medical history, or any trauma or surgical vessel and the pseudoaneurysm. After embolization, the blood history. The patient did not have any symptoms to suggest gas- supply to the pseudoaneurysm was blocked, which was con- trointestinal bleeding. firmed angiographically (Fig. 3). On physical examination, her blood pressure was 123/67 The patient’s blood pressure was 123/67 mmHg when she mmHg and heart rate was 69/min. Body temperature was 36.6℃. came to the hospital. During admission, her blood pressure was The mental status of the patient was alert and her conjunctivas were pale. Lung sounds were clear and heartbeat was regular. Bowel sound was normoactive. There was no direct or rebound abdominal tenderness. A Levine tube was inserted into the patient’s stomach and irrigation with normal saline was performed. The color of saline after recovery through the Levin tube was clear. On digital rectal examination, no mass lesion was palpable and the color of the stool was normal. The hydrogen peroxide (H2O2) response of the patient’s stool was normal. A fecal occult blood test was not performed. On a complete blood count, the white blood cell (WBC) was 3 8,600/mm (neutrophils 77.5%, lymphocytes 17.1%, monocytes 4.5%, eosinophils 0.5%), and hemoglobin and hematocrit were 3.7 g/dL and 11.5%, respectively. The platelet count was Figure 1. Initial CT finding of superior mesenteric artery pseudoaneurysm. Initial CT scan shows a -1.3 cm pseudoaneurysm at the superior mesenteric artery branch (arrow) and hemoperitoneum in the abdominopelvic cavity (due to the pseudoaneurysmal rupture). CT, computed tomography. - 561 - - 대한내과학회지: 제 88 권 제 5 호 통권 제 657 호 2015 - Figure 2. Angiographic finding of the superior mesenteric artery. Selective angiogram shows a pseudoaneurysm of superior mesenteric artery branch (arrow). kept stable. After the embolization, the level of hemoglobin was above 10.0 g/dL without additional bleeding signs. At 7 days after embolization, the patient was discharged. The patient was followed-up at 2 weeks and 6 weeks after Figure 3. Angiographic findings after transcatheter embolization. After embolization of superior mesenteric artery branch, the pseudoaneurysm was no longer seen on the angiography. discharge. No bleeding event or abdominal symptom suggestive pseudoaneurysms are diverse, and include surgical injuries, ab- of bowel ischemia occurred. The level of hemoglobin was main- dominal trauma, chronic pancreatitis, atherosclerosis, medial ne- tained above 10.0 g/dL. However, a follow-up CT scan could crosis, collagen vascular disease, arteritis, vascular dissections, not be performed, because the patient did not revisit our clinic. and infection [4]. Most patients are asymptomatic prior to rup- We had a telephone interview with her family 12 weeks after ture, and diffuse abdominal pain may be the only symptom of discharge; no additional bleeding event had occurred. the ruptured pseudoaneurysm. It is important that more than 50% of visceral artery aneurysms present acutely with rupture and show a mortality rate of about 10-25% [3]. Our patient DISCUSSION showed severe anemia with no evidence of overt gastrointestinal An aneurysm is defined as pathological dilation of a segment bleeding or abdominal pain. Vital signs were stable during ad- of a blood vessel. Visceral artery aneurysms include both true mission, although the SMA pseudoaneurysm had already rup- aneurysms and pseudoaneurysms. In true aneurysms, all three tured. Thus, it was difficult to diagnose initially. In addition, layers of the arterial wall are intact, whereas pseudoaneurysms there was no predisposing factor for SMA pseudoaneurysm in lack a complete arterial wall. Visceral artery aneurysm is a rare our patient. We assume that atherosclerosis or medial degener- disease; the incidence in postmortem studies range from 0.01% ation of the artery could have caused our patient’s SMA pseu- to 0.2% [3]. The incidence of visceral artery pseudoaneurysms doaneurysm. is even rarer, accounting for about 5% of all visceral artery A CT scan can be an excellent method in the differential di- aneurysms [4]. SMA aneurysms or pseudoaneurysms are the agnosis of SMA pseudoaneurysms. Specifically, a CT scan third most common visceral artery aneurysm (splenic and celiac should be considered in patients with unidentifiable causes of artery aneurysms occur more frequently). The causes of SMA anemia after other diagnostic methods fail to identify a cause. - 562 - - 김주성 외 6인. 상장간막동맥의 거짓동맥류 파열 - Angiography is considered the gold standard for the diagnosis SMA pseudoaneurysmal rupture can occur even in patients of aneurysms. Other associated vascular anomalies can also be without risk factors or symptoms, such as abdominal pain. Thus, revealed by angiography [5]. Endovascular techniques can be patients need to be evaluated with abdominal CT scans or an- used to treat most visceral artery aneurysms regardless of the giography, even if asymptomatic anemia is the only evidence of clinical presentation, etiology, and location of the aneurysm. an SMA pseudoaneurysmal rupture. Endovascular treatment is also indicated for ruptured visceral artery aneurysms in hemodynamically stable patients, especially 중심 단어: 상장간막 동맥; 거짓동맥류; 빈혈 when the aneurysm is poorly accessible for surgical treatment or when operative risks are high [6]. REFERENCES Recently, many studies on stent insertion in SMA pseudoaneurysms have been reported. McGraw et al. [7] reported the successful use of an autogenous vein-covered stent for management of an SMA pseudoaneurysm. Gandini et al. [8] reported an emergency endovascular treatment of giant SMA pseudoaneurysm using a polytetrafluoroethylene-covered nitinol stent. There is no definite superiority between embolization and stent insertion. We decided to treat the superior mesenteric artery pseudoaneurysm with an endovascular technique. However, embolization is feasible only in the case of small pseudoaneurysms, because treatment of large ones requires the use of large amounts of embolizing material for the complete occlusion of the affected vessel [8]. In addition, embolization is not believed to be appropriate in a case of a wide-neck pseudoaneurysm [9]. The main limitations to endovascular treatment are the availability of facilities for emergency treatment and the risk of failure due to difficulty in achieving selective catheterization [6]. Open surgery for SMA pseudoaneurysms is often rendered difficult by the underlying cause of the psuedoaneurysm (such as pancreatitis), anesthesia in an unstable patient, or by adhesions, which increase the risk of failure [10]. In our case, an abdominal contrast-enhanced CT scan revealed a ruptured SMA pseudoaneurysm and hemoperitoneum. Angiography definitively diagnosed the disease and the patient was treated with transcatheter embolization. Subsequently, no bleeding from the ruptured pseudoaneurysm was observed and no procedure-related complications were apparent. Following the procedure, the patient stabilized and we concluded that the de- 1. Huang YK, Hsieh HC, Tsai FC, Chang SH, Lu MS, Ko PJ. Visceral artery aneurysm: risk factor analysis and therapeutic opinion. Eur J Vasc Endovasc Surg 2007;33:293301. 2. Røkke O, Søndenaa K, Amundsen S, Bjerke-Larssen T, Jensen D. The diagnosis and management of splanchnic artery aneurysms. Scand J Gastroenterol 1996;31:737-743. 3. Carr SC, Mahvi DM, Hoch JR, Archer CW, Turnipseed WD. Visceral artery aneurysm rupture. J Vasc Surg 2001;33:806811. 4. Gandini R, Pipitone V, Konda D, et al. Endovascular treatment of a giant superior mesenteric artery pseudoaneurysm using a nitinol stent-graft. Cardiovasc Intervent Radiol 2005;28:102-106. 5. Waslen T, Wallace K, Burbridge B, Kwauk S. Pseudoaneurysm secondary to pancreatitis presenting as GI bleeding. Abdom Imaging 1998;23:318-321. 6. Sessa C, Tinelli G, Porcu P, Aubert A, Thony F, Magne JL. Treatment of visceral artery aneurysms: description of a retrospective series of 42 aneurysms in 34 patients. Ann Vasc Surg 2004;18:695-703. 7. McGraw JK, Patzik SB, Gale SS, Dodd JT, Boorstein JM. Autogenous vein-covered stent for the endovascular management of a superior mesenteric artery pseudoaneurysm. J Vasc Interv Radiol 1998;9:779-782. 8. Gandini R, Pipitone V, Konda D, et al. Endovascular treatment of a giant superior mesenteric artery pseudoaneurysm using a nitinol stent-graft. Cardiovasc Intervent Radiol 2005;28:102-106. 9. Cowan S, Kahn MB, Bonn J, et al. Superior mesenteric artery pseudoaneurysm successfully treated with polytetrafluoroethylene covered stent. J Vasc Surg 2002;35:805-807. 10. Tulsyan N, Kashyap VS, Greenberg RK, et al. The endovascular management of visceral artery aneurysms and pseudoaneurysms. J Vasc Surg 2007;45:276-283. creased hemoglobin and hematocrit were due to the ruptured SMA pseudoaneurysm and not gastrointestinal bleeding. - 563 -