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DOI:10.2214/AJR.05.0281
AJR 2006; 187:1463-1469
© American Roentgen Ray Society

Clinical Observations

Segmental Arterial Mediolysis: CTA Findings at Presentation and Follow-Up

Maren Michael1, Urs Widmer2, Simon Wildermuth1, Andre Barghorn3, Stefan Duewell4 and Thomas Pfammatter1

1 Institute of Diagnostic Radiology, University Hospital of Zurich, Raemistrasse 100, CH-8091 Zurich, Switzerland.
2 Department of Medicine, University Hospital of Zurich, Zurich, Switzerland.
3 Department of Pathology, University Hospital of Zurich, Zurich, Switzerland.
4 Institute of Radiology, Kantonsspital, Frauenfeld, Switzerland.

Received February 18, 2005; accepted after revision June 24, 2005.

 
Address correspondence to T. Pfammatter (thomas.pfammatter{at}usz.ch).


Abstract
Top
 Abstract
Introduction
 Materials and Methods
 Results
 Discussion
 References
 
OBJECTIVE. Segmental arterial mediolysis is a rare noninflammatory vascular disease of the abdominal splanchnic arteries. The purpose of our study was to retrospectively describe the CT angiography (CTA) findings of this disease and the evolution of those findings over time in five patients.

CONCLUSION. Comparison of CTA and digital subtraction angiography suggests that CTA is useful to diagnose symptomatic segmental arterial mediolysis. Midterm CTA follow-up (median, 3 years) indicates that segmental arterial mediolysis lesions may resolve or remain unchanged.

Keywords: cardiovascular disease • coil embolization • CT angiography • segmental arterial mediolysis


Introduction
Top
 Abstract
 Introduction
Materials and Methods
 Results
 Discussion
 References
 
Segmental arterial mediolysis is a rare nonarteriosclerotic, noninflammatory vascular disease of unknown origin that involves the visceral arteries of the abdomen. Lesions typically occur in a skip pattern within the large abdominal arteries and have no predilection for bifurcations. Segmental arterial mediolysis primarily affects the outer layer of the media, leading to smooth muscle cell vacuolar degeneration. The disruption of vacuoles and concomitant loss of their fluid contents ultimately results in disruption of the media, intramural hemorrhage, and periadventitial fibrin deposition. Gaps may be filled with fibrin, thrombi, or granulation tissue and can lead to saccular aneurysms, dissecting aneurysms, or thrombosis. The intima is spared from these lytic changes, and there is minimal inflammation.

This particular group of findings was first described as segmental mediolytic arteritis in 1976 by Slavin and Gonzalez-Vitale [1] and has subsequently become known as segmental arterial mediolysis [2]. To our knowledge, 30 cases of segmental arterial mediolysis have been described in the literature, with most patients presenting with intraabdominal hemorrhage [1, 3-6]. Bowel infarction caused by arterial occlusion either by thrombus or dissection has been less frequent [2]. The typical digital subtraction angiography (DSA) features of the disease were first described by Heritz et al. [3], who found a pattern of focal aneurysms, beading, and narrowing of the splanchnic and renal arteries with an otherwise normal vascular appearance.

Current segmental arterial mediolysis management is limited to surgical or interventional treatment of symptoms related to ruptured aneurysms or thrombosed arterial segments. The natural course of segmental arterial mediolysis is unknown. Indeed, follow-up ranging between a few months and 2 years has been reported in only four cases [4, 5, 7, 8]. Currently, little is known about the use of CT angiography (CTA) as a less-invasive imaging alternative for the diagnosis and follow-up of segmental arterial mediolysis. In this retrospective study, we investigated the potential role of CTA and compared the results with DSA and surgical pathologic findings, whenever obtained.


Materials and Methods
Top
 Abstract
 Introduction
 Materials and Methods
Results
 Discussion
 References
 
Patients
We included all cases of segmental arterial mediolysis diagnosed at our hospital over a period of 6.5 years (July 1998 through December 2004). Four cases were drawn from the files of the department of radiology, and one additional case was found in the database of the pathology department. The study group consisted of five patients (four men and one woman) who were between 47 and 75 years old (median age, 57 years) at the time of segmental arterial mediolysis diagnosis. In all patients, the leading symptom of segmental arterial mediolysis was abdominal pain.

Imaging
Patients had undergone at least one CTA within 1 month of segmental arterial mediolysis diagnosis. Because CT technology evolved over the study period, the generation of scanners was not uniform. The first patient had undergone initial scanning with a single-detector helical CT scanner (Somatom Plus 4, Siemens Medical Solutions), the next three patients with a 4-MDCT scanner (Somatom Plus 4 Volume Zoom, Siemens Medical Solutions), and the last patient with a 16-MDCT scanner (Sensation 16, Siemens Medical Solutions). Follow-up CTA was performed for a median of 3 years (range, 0.75-4 years). Follow-up CTA of the abdomen was performed on the 16-MDCT system. Scanning was performed after administration of 120 mL of nonionic IV contrast agent at an injection rate of 3.0 mL/s with bolus tracking performed in the abdominal aorta. The scanning parameters were as follows: collimation, 16 x 0.75 mm; table feed, 12 mm/rotation; tube potential, 100 kV; and tube current, 225 mAs. Axial images were reconstructed using a medium-soft kernel (B30f) optimized for postprocessing. Multiplanar reconstruction images, maximum intensity projections, and 3D projections (volume-rendered images) were generated using a radiologic workstation (Leonardo, Siemens Medical Solutions).


Figure 1
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  		Fig. 1A —75-year-old man with segmental arterial mediolysis (case 1). Contrast-enhanced CT image shows mesenteric hematoma (arrows).

 


Figure 2
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  		Fig. 1B —75-year-old man with segmental arterial mediolysis (case 1). Digital subtraction angiography (DSA) image of superior mesenteric artery shows typical string-of-beads appearance and small aneurysms of middle colic artery (arrow).

 


Figure 3
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  		Fig. 1C —75-year-old man with segmental arterial mediolysis (case 1). DSA image of same artery 4 years later shows normal artery.

 
DSA with standard selective injections of the celiac trunk, the superior mesenteric artery (SMA), and the renal arteries had been performed within a week of the initial CTA in four of the five patients, all using the same unit (Integris 5000, Philips Medical Systems). In one patient (case 4), no DSA had ever been performed. Follow-up DSA was performed just once (case 1) because unexpected CTA changes needed confirmation by the current gold standard. The DSA images of interest had been stored on hard copies at the time of their acquisition and were all available for review. The DSA and CTA images were reviewed in consensus by two experienced vascular radiologists.


Figure 4
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  		Fig. 2A —46-year-old man with segmental arterial mediolysis (case 2). Contrast-enhanced axial CT image shows aneurysm of celiac trunk (arrow) (2 cm in diameter) and proper hepatic artery (3.5 cm in diameter) (arrowhead). In addition, there is segmental kidney infarction.

 


Figure 5
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  		Fig. 2B —46-year-old man with segmental arterial mediolysis (case 2). Thin-slice maximum-intensity-projection image from CT angiography at 3-year follow-up shows that aneurysm of proper hepatic artery was successfully obliterated after coil embolization. Size of aneurysm of celiac trunk is unchanged.

 
Case Descriptions
Case 1—A 75-year-old man had been admitted for Guillain-Barré syndrome. His neurologic condition improved with IV immunoglobulin therapy. On hospital day 10, he complained of abdominal pain and minor rectal blood loss. CT revealed a mesenteric hematoma (Fig. 1A). CTA and DSA (Fig. 1B) showed a characteristic pattern of caliber irregularities of the splanchnic arteries, suggesting segmental arterial mediolysis. Laboratory screening for vasculitis (C-reactive protein, erythrocyte sedimentation rate, antinuclear antibodies, antineutrophil cytoplasmic antibodies, and rheumatoid factor) was negative. The patient was given RBC transfusions and improved without additional therapy. The patient's remaining clinical course was unremarkable, and CTA follow-up 4 years later showed complete resolution of the previous vascular abnormalities. Because of this unexpected course, confirmatory DSA was also performed (Fig. 1C).

Case 2—A 46-year-old man with no risk factors for pancreatitis presented with abdominal pain, tarry stools, and slightly elevated pancreatic enzymes. CTA and DSA images depicted an aneurysm of the proper hepatic artery (3.5 cm in diameter) and smaller aneurysms in the gastroduodenal artery, celiac trunk, and left renal artery (Fig. 2A). The pancreas itself was unremarkable. The portal system was unaffected by the disease process. Laboratory screening for vasculitis was negative. Mild acute pancreatitis was interpreted as a consequence of hemobilia and not as the cause of the aneurysm, and segmental arterial mediolysis was diagnosed. Transarterial coil embolization of the proper hepatic artery proximally and distally to the aneurysm was performed without complications. The patient remained well at clinical follow-up 3 years after the embolization. The aneurysms of the celiac trunk and the renal artery were identical in size (Fig. 2B), and no additional lesions were found.

Case 3—A 73-year-old man was admitted with a 3-day history of lower abdominal pain. CT showed a mesenteric hematoma. CTA images depicted an aneurysm (maximum diameter, 8 mm) of the middle colic artery and a string-of-beads appearance of the SMA (Fig. 3A). DSA images revealed an additional small aneurysm of the gastroduodenal artery (Fig. 3B). Segmental arterial mediolysis was suggested based on the angiographic findings. A hemicolectomy was performed because of the marked hemoglobin drop (8.8 g/dL; normal, 14.4-17.5 g/dL). All other laboratory findings were normal. Histologic examination of the middle colic artery revealed typical features of segmental arterial mediolysis. CTA follow-up was performed after 3 years and detected no new lesions (Fig. 3C).


Figure 6
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  		Fig. 3A —73-year-old man with segmental arterial mediolysis (case 3). Surface shaded display 3D reconstruction image from CT angiography of superior mesenteric artery (SMA) shows small aneurysm of middle colic artery (arrowhead) and caliber irregularities of SMA (arrows).

 

Figure 7
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  		Fig. 3B —73-year-old man with segmental arterial mediolysis (case 3). Digital subtraction angiography image shows aneurysm of middle colic artery (arrowhead), string-of-beads appearance of SMA (small arrows), and small aneurysm of gastroduodenal artery (large arrow).

 

Figure 8
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  		Fig. 3C —73-year-old man with segmental arterial mediolysis (case 3). At follow-up 3 years after diagnosis and hemicolectomy (including resection of middle colic artery aneurysm), surface shaded display 3D reconstruction image from CT angiography shows persisting string-of-beads appearance of SMA (small arrows) and small aneurysm of gastroduodenal artery (large arrow).

 
Case 4—A 57-year-old woman with end-stage invasive ductal breast cancer had diffuse pain, including abdominal, and received palliative care until her death at our institution. The postmortem examination surprisingly revealed segmental arterial mediolysis in addition to disseminated cancer. The tubular aneurysm of the middle colic artery and Riolan's arch can be seen in the gross specimen (Fig. 4). A contrast-enhanced MDCT scan obtained 1 month before the patient's death revealed pulmonary and bone metastases. Even at retrospective review with additional postprocessing of the data, no abdominal vascular abnormalities could be found.


Figure 9
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  		Fig. 4 —57-year-old woman with segmental arterial mediolysis (case 4). Gross postmortem specimen of abdominal aorta shows superior and inferior mesenteric artery. Note tubular ectasia of middle colic artery and Riolan's arch (arrowheads).

 
Case 5—A 46-year-old man presented with abdominal pain. CTA (Fig. 5A) and DSA (Fig. 5B) images revealed occlusion of the middle colic artery and jejunal branches of the SMA, tubular aneurysms of the jejunal branches, ectasia of the right renal artery, and a right renal infarction. There were no CT findings of acute enteric or colonic ischemia. Segmental arterial mediolysis was suggested after ruling out vasculitis by laboratory screening. Therapy consisted solely of aspirin because no thrombolytic agents could be used due to concerns of hemorrhagic complications. Follow-up 9 months after the initial onset of the disease showed only slight caliber irregularities of the SMA with unchanged occluded branches (Fig. 5C) and completely normal morphology of the renal arteries.


Figure 10
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  		Fig. 5A —46-year-old man with segmental arterial mediolysis (case 5). Digital subtraction angiography (A) and surface shaded display 3D reconstruction image of CT angiography (B) of superior mesenteric artery (SMA) show occlusion of middle colic artery and several jejunal branches. One jejunal branch has tubular aneurysm (arrow).

 

Figure 11
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  		Fig. 5B —46-year-old man with segmental arterial mediolysis (case 5). Digital subtraction angiography (A) and surface shaded display 3D reconstruction image of CT angiography (B) of superior mesenteric artery (SMA) show occlusion of middle colic artery and several jejunal branches. One jejunal branch has tubular aneurysm (arrow).

 

Figure 12
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  		Fig. 5C —46-year-old man with segmental arterial mediolysis (case 5). Thin-slice maximum-intensity-projection image from CT angiography at follow-up 9 months after diagnosis shows regression of tubular aneurysm of jejunal branch (arrow) and only slight caliber irregularities of SMA.

 


Results
Top
 Abstract
 Introduction
 Materials and Methods
 Results
Discussion
 References
 
Clinical data and the radiologic patterns of segmental arterial mediolysis at initial CTA and DSA are summarized in Table 1. The leading symptom at the time of segmental arterial mediolysis diagnosis was abdominal pain, followed by lower gastrointestinal blood loss. The SMA or its branches were affected in all five patients. Additional segmental arterial mediolysis changes were noted in the celiac artery or its branches in three patients and in a renal artery in two patients. DSA and CTA images obtained in the same patient within 1 week were available in four instances. Both DSA and CTA identified the characteristic findings of segmental arterial mediolysis in four cases. In one patient, alterations of the gastroduodenal artery were detected on DSA but not CTA. Nevertheless, CTA was thought to provide enough evidence of segmental arterial mediolysis.


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  		TABLE 1: Clinical Data and Radiologic Findings at Initial Diagnosis

 

Although our segmental arterial mediolysis patients had a rather sudden disease onset, at least four of the five patients had a benign course (Table 2). The cause of death of one patient (case 4), in whom segmental arterial mediolysis was diagnosed at postmortem examination, was most likely related to her metastatic breast cancer and can therefore be viewed separately. The fact that the abdominal CTA performed 1 month before her death did not show any arterial changes supports the hypothesis that mediolysis is an acute process. The findings on follow-up CTA (median, 3 years; range, 0.75-4 years) showed unchanged arterial lesions in two patients, partially regressed lesions in one patient, and completely resolved lesions in another patient. No new lesions were seen in any of the cases.


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  		TABLE 2: Therapy, Histology, Clinical Outcome, and CTA Follow-Up

 


Discussion
Top
 Abstract
 Introduction
 Materials and Methods
 Results
 Discussion
References
 
Little clinical information about segmental arterial mediolysis is available because of the rarity of the disease. The incidence of segmental arterial mediolysis is most likely underestimated because angiography is not part of the routine workup for patients with abdominal pain. The causes for this acute, self-limiting, and noninflammatory visceral arteriopathy are not yet known. Because some angiographic findings are similar to those seen with fibromuscular dysplasia, segmental arterial mediolysis is considered by some authors to be a variant of that disease [6, 9]. However, fibromuscular dysplasia occurs mainly in young to middle-aged women, and the pattern of arterial involvement is different. Fibromuscular dysplasia affects mostly renal and carotid arteries, whereas segmental arterial mediolysis involves predominantly the visceral arteries. On the other hand, fibrodysplasia may also show segmental arterial narrowing due to intimal, medial, or adventitial fibroplasias [10] and small aneurysms. The cause of fibromuscular dysplasia is also unknown.

Various forms of vasculitis must be considered in the differential diagnosis of segmental arterial mediolysis. Systemic inflammation with inflammatory destruction of the wall of the mesenteric arteries is seen in polyarteritis nodosa, Takayasu's arteritis, Behçet's syndrome, and Henoch-Schönlein purpura [11]. Additional clinical features, such as aphthous stomatitis in Behçet's syndrome, and laboratory findings, such as elevated nonspecific parameters of systemic inflammation and specific autoantibodies, can be helpful in differentiating patients with these diseases from patients with segmental arterial mediolysis.

Mycotic aneurysms arise after infectious destruction of the arterial wall and show a preference for bifurcations, whereas segmental arterial mediolysis-related aneurysms have a random distribution.

Congenital vascular conditions must also be excluded. Neurofibromatosis type 1 first involves larger vessels, which are surrounded by neurofibromatous tissue and lead to stenosis (mostly coarctation of the aorta and ostial renal artery stenosis) and aneurysm formation [12]. In Ehlers-Danlos syndrome type IV (EDS IV), the vascular symptoms are related to dissection or rupture of the aorta and its branches. Most EDS IV patients will have at least one complication by the age of 40 years [13] and are therefore younger than the segmental arterial mediolysis population.

There is also little known about the progression and outcome of segmental arterial mediolysis. Until now, only four cases of segmental arterial mediolysis with a maximum follow-up time of 2 years have been published [4, 5, 7, 8]. This report presents four consecutive patients with a median clinical and CTA follow-up of 3 years. We had the opportunity of comparing visceral and renal catheter angiography with CTA at presentation in these patients and think that CTA was sufficient for making a radiologic diagnosis of segmental arterial mediolysis. Therefore, CTA seems a useful, less-invasive follow-up technique for these patients.

After a dramatic onset requiring hemicolectomy or transarterial embolization of bleeding aneurysms in half of these patients, the clinical course of this disease was benign despite the absence of any specific treatment. Indeed, the previously reported high mortality rate of this condition, thought to be 50% [4], was not seen in our series. We even saw a patient in whom the vascular changes completely vanished. To our knowledge, this surprising finding has not yet been described. In the other three patients, who also became asymptomatic within weeks after the initial acute event, CTA follow-up did not show any disease progression. The patient in whom the diagnosis of segmental arterial mediolysis was made postmortem was considered separately.

The factors involved in lesion resolution are not known. So far, there is no animal model for segmental arterial mediolysis that would allow the quantification of potential apoptosis of various cell types in affected visceral arteries. According to our limited experience, a conservative approach with CTA follow-up appears most appropriate in patients without initial bleeding complications or with aneurysms of small size because no disease progression was seen on follow-up over several years in this segmental arterial mediolysis patient series.

Histology is the gold standard of segmental arterial mediolysis diagnosis, but unfortunately vascular tissue is only available in patients undergoing surgery for resection of vessels altered by segmental arterial mediolysis or in postmortem cases. Incomplete histologic documentation is the main limitation of this report. Histology was available in just two of the five patients of our small series. However, in our opinion, the diagnosis of segmental arterial mediolysis should be made using DSA or CTA and based on the characteristic pattern of arterial involvement and morphologic changes after excluding vasculitis by clinical and laboratory findings. Radiologists should be aware of segmental arterial mediolysis because they may be the only ones to suggest the diagnosis, optimally treat it by interventional means if needed, and safely follow-up most of the cases.

We conclude that CTA appears to be a useful tool for the diagnosis and follow-up of patients with segmental arterial mediolysis.


References
Top
 Abstract
 Introduction
 Materials and Methods
 Results
 Discussion
 References
 

  1. Slavin RE, Gonzalez-Vitale JC. Segmental me diolytic arteritis: a clinical pathologic study. Lab In vest1976; 35:23 -29
  2. Slavin RE, Cafferty L, Cartwright J Jr. Segmental mediolytic arteritis: a clinicopathologic and ultrastructural study of two cases. Am J Surg Pathol 1989;13 : 558-568[Medline]
  3. Heritz DM, Butany J, Johnston KW, Sniderman KW. Intraabdominal hemorrhage as a result of seg mental mediolytic arteritis of an omental artery: case report. J Vasc Surg 1990;12 : 561-565[CrossRef][Medline]
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  5. Juvonen T, Niemela O, Reinila A, Nissinen J, Kair aluoma MI. Spontaneous intraabdominal haemor rhage caused by segmental mediolytic arteritis in a patient with systemic lupus erythematosus: an un derestimated entity of autoimmune origin? Eur J Vasc Surg1994; 8:96 -100[CrossRef][Medline]
  6. Chan RJ, Goodman TA, Aretz TH, Lie JT. Segmen tal mediolytic arteriopathy of the splenic and he patic arteries mimicking systemic necrotizing vas culitis. Arthritis Rheum1998; 41:935 -938[CrossRef][Medline]
  7. Ryan JM, Suhocki PV, Smith TP. Coil embolization of segmental arterial mediolysis of the hepatic ar tery. J Vasc Interv Radiol 2000; 11:865 -868[Medline]
  8. Soulen MC, Cohen DL, Itkin M, Townsend RR, Roberts DA. Segmental arterial mediolysis: angio plasty of bilateral renal artery stenoses with 2-year imaging follow-up. J Vasc Interv Radiol2004; 15:763 -767[Medline]
  9. Lie JT. Segmental mediolytic arteritis: not an arteri tis but a variant of arterial fibromuscular dysplasia. Arch Pathol Lab Med 1992; 116:238 -241[Medline]
  10. Begelman SM, Olin JW. Fibromuscular dysplasia. Curr Opin Rheumatol 2000; 12:41 -47[CrossRef][Medline]
  11. Geboes K, Dalle I. Vasculitis and the gastro intestinal tract. Acta Gastroenterol Belg 2002;65 : 204-212[Medline]
  12. Booth C, Preston R, Clark G, Reidy J. Management of renal vascular disease in neurofibromatosis type 1 and the role of percutaneous transluminal angioplasty. Nephrol Dial Transplant2002; 17:1235 -1240[Abstract/Free Full Text]
  13. Pepin M, Schwarze U, Superti-Furga A, Byers PH. Clinical and genetic features of Ehlers-Danlos syn drome type IV, the vascular type. N Engl J Med 2000;342 : 673-680[Abstract/Free Full Text]

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