Journal Pre-proof Successful Conservative Management of Inferior Mesenteric Artery Aneurysm with Arteriovenous Fistula: A Case Report Shingo Kunioka, Hiroto Kitahara, Noriyuki Yuasa, Miri Fujita, Norifumi Otani, Hiroyuki Kamiya PII:
S0890-5096(19)30922-7
DOI:
https://doi.org/10.1016/j.avsg.2019.10.067
Reference:
AVSG 4729
To appear in:
Annals of Vascular Surgery
Received Date: 27 November 2018 Revised Date:
5 September 2019
Accepted Date: 2 October 2019
Please cite this article as: Kunioka S, Kitahara H, Yuasa N, Fujita M, Otani N, Kamiya H, Successful Conservative Management of Inferior Mesenteric Artery Aneurysm with Arteriovenous Fistula: A Case Report, Annals of Vascular Surgery (2019), doi: https://doi.org/10.1016/j.avsg.2019.10.067. This is a PDF file of an article that has undergone enhancements after acceptance, such as the addition of a cover page and metadata, and formatting for readability, but it is not yet the definitive version of record. This version will undergo additional copyediting, typesetting and review before it is published in its final form, but we are providing this version to give early visibility of the article. Please note that, during the production process, errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain. © 2019 Elsevier Inc. All rights reserved.
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Successful Conservative Management of Inferior Mesenteric Artery Aneurysm
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with Arteriovenous Fistula: A Case Report
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Shingo Kunioka1, Hiroto Kitahara2, Noriyuki Yuasa3, Miri Fujita4, Norifumi Otani5,
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Hiroyuki Kamiya2
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Japan
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2
Department of Cardiac Surgery, Asahikawa Medical University, Hokkaido, Japan
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3
Department of Radiology, Steel Memorial Muroran Hospital, Hokkaido, Japan
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Department of Pathology, Steel Memorial Muroran Hospital, Hokkaido, Japan
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Department of Cardiovascular Surgery, Sapporo Teishinkai Hospital, Hokkaido, Japan
Department of Cardiovascular Surgery, Steel Memorial Muroran Hospital, Hokkaido,
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Corresponding author: Hiroto Kitahara, MD
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Department of Cardiac Surgery, Asahikawa Medical University, Midorigaoka Higashi
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2-1-1-1, Asahikawa 078-8510, Japan
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Tel: +81-166-68-2490; Fax: +81-166-68-2499; E-mail:
[email protected]
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Abstract
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Inferior mesenteric artery (IMA) aneurysm is a rare occurrence, accounting for
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1% of all visceral artery aneurysms and is often found incidentally. Surgical resection
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and endovascular intervention have been first-line treatments because IMA aneurysms
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have a relatively high risk of life-threatening rupture. Herein, we report the case of a
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57-year-old man having a large IMA aneurysm with an arteriovenous fistula that was
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treated conservatively. The IMA aneurysm was incidentally found using computed
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tomography (CT) and was connected to the splenic vein through the abnormally dilated
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tortuous vessels of an arteriovenous fistula. Surgical resection was planned initially;
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however, preoperative follow-up CT revealed that the aneurysm had shrunk with the
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growth of an intraluminal thrombus. Subsequently, the condition was conservatively
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managed with serial CT follow-up. Two years after the first visit, the aneurysm had
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shrunk and been completely replaced with a thrombus.
36 37 38 39 40 41 42 43 44 45 46
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Introduction
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We rarely encounter aneurysms, particularly inferior mesenteric artery (IMA)
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aneurysms, in abdominal visceral arteries. Traditionally, distribution of aneurysms in
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visceral arteries involves the splenic artery (60%), hepatic artery (20%), superior
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mesenteric artery (6%), celiac artery (4%), gastric and gastroepiploic arteries (4%),
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jejunal and ileocolic arteries (3%), pancreatic duodenal and pancreatic arteries (2%),
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gastroduodenal artery (1.5%), and IMA (<1%).1 Edogawa et al. reviewed 54 IMA
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aneurysm case reports worldwide and recommended aneurysm repair if the aneurysm’s
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size exceeded 2 cm at the proximal or 1 cm at the distal IMA, due to increased risk of
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rupture.2 However, because of IMA aneurysms’ rarity, definitive surgical indication does
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not yet exist. We experienced an extremely rare case of a large IMA aneurysm with an
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arteriovenous fistula (AVF), which was successfully managed conservatively with serial
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computed tomography (CT) follow-up.
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Case Report
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A 57-year-old man with a history of ulcerative colitis (UC) but no history of
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abdominal surgery or trauma was referred to our institution. He had been treated with
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mesalazine (1500 mg/day), and the UC was relatively under control. During imaging
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work-up, abdominal ultrasonography incidentally found a bulky mass in the pelvis.
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Contrast-enhanced CT revealed an aneurysm 50 mm × 43 mm in size in the terminal
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IMA, with abnormal tortuous vessels with tumor-like expansions (69 mm) arising from
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the aneurysm and flowing into the splenic vein as AVFs (Figure 1). Initially, we
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considered surgical resection for the IMA aneurysm; however, preoperative follow-up
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CT showed that the aneurysm had shrunk with intraluminal thrombus (ILT) growth. The
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man did not have any AVF-related symptoms, such as abdominal pain, gastrointestinal
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bleeding, colonic ischemia, or portal hypertension. Serial short-interval follow-up CT
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found the aneurysm and dilated vein shrinking were gradually being replaced with a
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thrombus (Figure 2). After 2 years, the aneurysm had shrunk to 48 mm × 31 mm and
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completely changed into a thrombus, entirely enclosing the AVF. The treatment course
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was uneventful and without any abdominal symptoms, and the latest follow-up CT (5
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years after the first visit) showed no significant changes (Figure 3).
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Discussion
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We experienced an extremely rare case of IMA aneurysm with an AVF that was
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managed conservatively without any intervention. To our knowledge, there has been no
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case report on the spontaneous enclosure of an inferior mesenteric AVF and constructive
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regression of an IMA aneurysm.
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IMA aneurysms are rare, accounting for < 1 % of all visceral artery
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aneurysms.1 They were originally reported by Duke et al., and were treated with
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surgical vascular reconstruction consisting of bilateral aorto-femoral bypass and a left
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profunda femoris artery angioplasty; additionally, the IMA was anastomosed into the
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left limb of a bifurcated graft. Edogawa et al. investigated 22 patients with IMA
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aneurysm, and found out that atherosclerotic disease was the most common cause of
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IMA aneurysm.
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They are often asymptomatic and discovered incidentally with evaluation for
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other intra-abdominal pathologies. The main etiology of IMA aneurysm is
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atherosclerotic disease; other reported causes include mycosis, polyarteritis nodosa,
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dissecting hematoma, Takayasu disease, and tuberculosis.2 In our case, the IMA
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aneurysm was found incidentally during UC work-up. There are a few studies
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investigating UC and related vascular disease. Imamura et al. reported a case of
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coexisting arteritis in a patient with UC. 4 Zancoli et al. reported that inflammatory
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bowel disease such as UC might affect endothelial function and arterial stiffness and
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might cause vascular disease.5 Although there has been no report regarding the
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relationship between UC and IMA aneurysm, regression of UC might have contributed
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to the IMA aneurysm or AVF in our case.
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The true prevalence of IMA aneurysm remains unknown. The risk of IMA
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aneurysm rupture, a life-threatening complication in visceral artery aneurysms, has been
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reported to be 20 % - 50 %.1 Edogawa et al. suggested that IMA aneurysm size greater
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than 2 cm at the proximal or 1 cm at the distal IMA should be treated surgically,
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considering the risk of rupture.2 As the size of the IMA aneurysm in our case was
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greater than 5 cm, we planned surgical intervention, however, serial follow-up CT
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showed continuous growth of an ILT concomitant with the shrinking of the aneurysm
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incidentally, which changed our management plan.
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The effect of an ILT on aneurysm rupture risk is controversial. Haller et al.
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reported that ILTs were associated with early rupture of abdominal aortic aneurysms6. In
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contrast, Thubrikar et al. suggested that an ILT might prevent aneurysm rupture through
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diminishing wall strain, even though the thrombus allows transmission of luminal
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pressure to the aneurysm wall.7 Klausner et al. reviewed cases of asymptomatic renal
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artery aneurysms treated conservatively for a mean follow-up of 36 ± 9 months with no
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late complications or mortality.8 In our case, serial follow-up CT showed continuous
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growth of an ILT concomitant with the shrinking of the aneurysm. ILTs in IMA
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aneurysms may be associated with spontaneous recovery of the aneurysms, in which
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case patients could be managed with imaging follow-up only, however, further studies
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will be needed to detect predictors of low rupture risk and spontaneous recovery.
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Coexisting AVFs might also play an important role in the spontaneous recovery
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of IMA aneurysms. An AVF is an abnormal communication or passage between an
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artery and a vein. An inferior mesenteric AVF acts as a left-to-right shunt causing the
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pathophysiologic alteration of bowel function and manifesting with various symptoms
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such as abdominal pain, abdominal mass with thrill, upper or lower gastrointestinal
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bleeding, portal hypertension, ischemic colitis, and cardiac failure.9-11 A previous
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mini-review of literature reported 17 congenital and 13 acquired inferior mesenteric
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AVF cases; the mean age of patients was 58 years, and 67.7% of the patients were
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male.11 Inferior mesenteric AVFs can have congenital, acquired (iatrogenic or traumatic),
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or idiopathic etiology.9 In our case, the cause of the inferior mesenteric AVF might have
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been a spontaneous rupture of a preexisting IMA aneurysm into the inferior mesenteric
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vein as previously reported.12 Koen et al. reported the conservative management of
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persistent aortocaval fistulas after endovascular aortic repair, suggesting that different
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pressure dynamics caused by the AVF directly communicating with the low-pressure
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venous system lowered the pressure in the aneurysmal sac and enhanced its
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remodeling.13 In the present case, coexisting AVF might allow the IMA aneurysm to
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drain in a relatively low-pressure venous system, resulting in spontaneous recovery of
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the aneurysm and enclosing of the AVF itself, more studies were needed to investigate
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this phenomenon.
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The optimal management of IMA aneurysm is controversial. Shanley et al.
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reviewed various treatments options for IMA aneurysm including aneurysmectomy,
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bypass/revascularization, ligation, aneurysmorrhaphy and embolization.14 Jesus-Silva
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et al. reported a successful management of giant IMA aneurysm with embolization.15 In
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this case, IMA catheterization was performed with Simmons II catheter, followed by
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implantation of two Interlock-18 pushable microcoils (Boston Scientific, Natrick, MA,
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USA) in a small dilation before its out flow branch and five Interlock-35 pushable coils
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in the aneurysm lumen. In our case, the aneurysm existed distal of the IMA,
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embolization might be one of the appropriate treatments if the aneurysms are enlarged
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in the follow up period.
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Conclusion
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We experienced an extremely rare case of IMA aneurysm with an AVF that was
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successfully managed conservatively. Large IMA aneurysms should be treated with
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surgical or endovascular intervention; however, conservative management may be a
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treatment option in select cases of coexisting AVFs and ILTs.
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Figure Legends
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Figure 1. Contrast-enhanced computed tomography findings. Three-dimensional
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reconstruction shows an inferior mesenteric artery aneurysm (white arrowhead) (A) and
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dilated vein (white arrow) (B). Coronal plane (C).
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Figure 2. Serial follow-up computed tomography findings. Inferior mesenteric artery
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aneurysm (white arrowhead) and dilated vein (white arrow). The diameter of the
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inferior mesenteric artery aneurysm had been continuously shrinking: first visit, 50 mm
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× 43 mm (A); second visit, 48 mm × 41 mm (B); and 2 years after the first visit, 48 mm
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× 31 mm (C).
168 169
Figure 3. Computed tomography findings 5 years after the first visit. Three-dimensional
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reconstruction shows a normal inferior mesenteric artery and vein (A). The IMA
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aneurysm (white arrowhead) and dilated vein (white arrow) completely became a
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thrombus (B).
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References
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425-42. 2. Edogawa S, Shibuya T, Kurose K, et al. Inferior mesenteric artery aneurysm: case report and literature review. Ann Vasc Dis. 2013; 6: 98-101. 3. Duke LJ, Lamberth WC Jr, Wright CB. Inferior mesenteric artery aneurysm: Case report and discussion. Surgery.1979;85:385-387.
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4. Imamura R, Hayashi K, Sada KE, et al. Hemoptysis originating from the bronchial
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artery in Takayasu arteritis with ulcerative colitis. Intern Med. 2018. [Epub ahead of
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print]. doi: 10.2169/internalmedicine.1463-18.
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5. Zancoli L, Rastelli S, Inserra G, et al. Arterial structure and function in inflammatory bowel disease. World J Gastroenterol. 2015; 28: 11304-11.
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6. Haller SJ, Crawford JD, Courchaine KM, et al. Intraluminal thrombus is associated
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