Legal Medicine 17 (2015) 388–390

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Case Report

Rupture of massive coronary artery aneurysm resulting in cardiac tamponade Takeshi Kondo a,⇑, Motonori Takahashi a, Kanako Nakagawa a, Azumi Kuse a, Mai Morichika a, Makoto Sakurada a, Migiwa Asano b, Yasuhiro Ueno a a b

Division of Legal Medicine, Department of Community Medicine and Social Health Science, Kobe University Graduate School of Medicine, Kobe, Japan Department of Legal Medicine, Ehime University Graduate School of Medicine, Ehime, Japan

a r t i c l e

i n f o

Article history: Received 17 March 2015 Received in revised form 15 May 2015 Accepted 18 May 2015 Available online 27 May 2015 Keywords: Coronary artery aneurysm Rupture Cardiac tamponade

a b s t r a c t Coronary artery aneurysm is a fairly uncommon clinical entity, which is defined by a characteristic dilatation that exceeds 1.5 times the width of normal adjacent coronary artery segments. In the present report, we describe a case of rupture of a massive coronary artery aneurysm. A man in his 40 s was found dead in his bed. The pericardial cavity contained 270 mL of blood with 428.2 g of coagulation. Two true aneurysms of the right coronary artery were identified. A proximal aneurysm, adjacent to the right auricle, had ruptured on the right. A distal unruptured aneurysm was identified 5.1 cm distal to the proximal ruptured aneurysm. Atherosclerosis of the coronary arteries and aorta was severe. The heart weighed 799.1 g and showed concentric ventricular hypertrophy, myocardial thinning, and patchy fibrosis. Histological analysis showed that both aneurysms were purely atherosclerotic true aneurysms without considerable inflammation. The cause of death was determined as cardiac tamponade due to rupture of a giant coronary atherosclerotic aneurysm. Ó 2015 Elsevier Ireland Ltd. All rights reserved.

1. Introduction

2. Case report

Coronary artery aneurysm is a relatively uncommon clinical entity, which is defined by a characteristic dilatation that exceeds 1.5 times the width of normal adjacent coronary artery segments [1]. Complications from coronary aneurysms include thrombosis, embolization, and ischemia. Rupture occurs in only a small number of cases. Coronary artery aneurysms are rarely large enough to be called giant coronary artery aneurysms. There is no universally accepted definition of giant coronary artery aneurysm. Coronary artery aneurysms with diameters of greater than 20, 40, and 50 mm, and quadruple the reference-vessel diameter have been proposed as definitive in the medical literature. Published data on giant coronary artery aneurysms show a prevalence of 0.02– 0.2% (the prevalence of a giant coronary artery aneurysm P50 mm in diameter is 0.02%) [2]. To date, there are several autopsy cases associated with this particular coronary condition [3,4]. In the present report, we describe a case of rupture of a giant coronary artery aneurysm.

A man in his 40 s was found dead in his bed. His past medical history included diabetes, chronic renal failure, and malignant hypertension. The deceased had undergone renal transplantation 15 years previously and surgical repair of a ruptured abdominal aortic aneurysm 10 years previously. The case was subject to medico-legal investigation because the individual was relatively young and was found dead, with no witnesses. An autopsy was performed 24-h postmortem. The deceased was 182 cm tall and weighed 90 kg. The deceased had no injuries on the body, including on the chest. The pericardial cavity contained 270 mL of blood with 428.2 g of coagulation. Two true saccular aneurysms of the right coronary artery were identified (Fig. 1A and B). The proximal aneurysm (7.2  5.1  4.2 cm), adjacent to the right auricle, had ruptured on the right. The rupture site was 3.5  0.7 cm (Fig. 1C). A distal unruptured aneurysm (4.5  2.8  2.4 cm) was identified 5.1 cm distal to the proximal ruptured aneurysm. A fresh and organized mural thrombus was adhered to the inner surface of both aneurysms (Fig. 1D). Fistula formation was not found. Histological analysis revealed that both aneurysms were purely atherosclerotic true aneurysms with no features/signs of inflammation or vasculitis (Fig. 2). Although the left anterior descending and left circumflex branches did not show

⇑ Corresponding author at: Division of Legal Medicine, Department of Community Medicine and Social Healthcare Science, Kobe University Graduate School of Medicine, 7-5-1 Kusunoki-cho, Chuo-ku, Kobe 650-0017, Japan. Tel.: +81 78 382 5582; fax: +81 78 382 5599. E-mail address: [email protected] (T. Kondo). http://dx.doi.org/10.1016/j.legalmed.2015.05.006 1344-6223/Ó 2015 Elsevier Ireland Ltd. All rights reserved.

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distal aneurysm

distal aneurysm

A

proximal aneurysm (ruptured)

B

proximal aneurysm (ruptured)

D

C

Ruptured site

Fig. 1. Two true aneurysms of the right coronary artery were identified (A and B). The proximal aneurysm (7.2  5.1  4.2 cm), adjacent to the right auricle, had ruptured on the right. The rupture site was 3.5  0.7 cm (C). The distal unruptured aneurysm (4.5  2.8  2.4 cm) was identified 5.1 cm distal to the proximal ruptured aneurysm. A fresh and organized mural thrombus was found to be adhered to the inner surface of both aneurysms (D).

proximal aneurysm (ruptured)

A

proximal aneurysm (ruptured)

B

Ruptured site

C

proximal aneurysm (ruptured)

D distal aneurysm

Fig. 2. Histological analysis showed that both aneurysms (the proximal ruptured aneurysm: A, B and C and the distal aneurysm: D) were purely atherosclerotic true aneurysms with no features/signs of inflammation or vasculitis. Elastic fibers were destroyed (B inset). Hematoxylin and eosin stain. (A) 100, (B) 20, (C) 40, and (D) 40. EVG stain. (B) inset 200.

any aneurysmal changes, the left anterior descending branch showed mild dilatation. The heart weighed 799.1 g and showed concentric ventricular hypertrophy. Thinning of the anterior wall of the myocardium and patchy fibrosis of the posterior and lateral walls were identified, suggesting an old myocardial infarction. Histopathologically, acute myocardial necrosis was not identified.

Severe atherosclerosis of the coronary arteries and aorta was documented. However, we had no access to information on the origin of the abdominal aortic aneurysm nor to the results of the pathological examination of the specimen (aortic aneurysm). Cerebral arteries showed moderate atherosclerosis and renal arteries showed severe atherosclerosis. We excluded the presence of any persistent inflammation or vasculitis by histopathologic

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examination of blood vessels from different locations in the body. The lungs showed focal congestion. No ethanol was detected in blood or urine. Drug screening results using Instant-View M-1™ (Alfa Scientific Designs Inc., Poway, CA, USA) were negative. The cause of death was determined as cardiac tamponade due to rupture of a giant coronary atherosclerotic aneurysm. 3. Discussion Aneurysmal coronary artery disease is characterized by abnormal dilatation of a localized or diffuse segment of the coronary artery. The incidence of coronary artery aneurysm has been reported as 0.15–4.9% [5]. Aneurysms are associated with atherosclerosis, Kawasaki disease, Takayasu disease, syphilis, Marfan syndrome, Ehlers–Danlos syndrome, polyarteritis nodosa, or trauma [3]. The most common cause is atherosclerotic coronary artery disease, which accounts for 50–90% of cases [6]. Coronary artery aneurysm is classified as fusiform or saccular. Saccular aneurysms have transverse diameters greater than their longitudinal extension, whereas fusiform aneurysms have a dominant longitudinal dilatation parallel to the vessel axis [7]. Histological features of atherosclerotic aneurysm include hyalinization, lipid deposition, disruption of the intima and media, calcification, fibrosis, eosinophilic debris, neovascularization, cholesterin crystals, intramural hemorrhage, foreign-body giant-cell reactions, and extensive destruction of musculoelastic elements [4,8,9]. The natural history of atherosclerotic aneurysmal coronary artery disease is largely unknown because most reports in the literature include small numbers of patients with short-term follow-ups [10]. The pathogenesis of coronary artery aneurysm involves underlying destruction of the vessel media. This thinning of the media, together with increased wall stress, causes progressive dilatation of the segment of the coronary artery and manifests as diffuse coronary ectasia, as well as localized ectatic segments, in angiographic assessments. Coronary ectasia and coronary aneurysm may represent either end of the spectrum of a common pathological process [11]. Although most patients with coronary artery aneurysms are asymptomatic, manifestations of myocardial ischemia may occur. Altered blood flow in aneurysmal segments is thought to predispose to thrombosis or embolism and subsequent ischemia or infarction. Rupture is a rare event; however, once it happens, it can be lethal because of the consequent cardiac tamponade [6]. The left descending branch has maximum flow, and shows atherosclerosis more frequently than the two other major coronary branches [12,13]. However, the most common site for a coronary aneurysm is the right coronary artery, followed by the left descending and left circumflex branches. Left main coronary artery aneurysms are rare [14]. Multiple aneurysms of the right coronary artery have been reported [15].

Previously reported sudden death cases are associated with luminal thrombosis [3,4]. In this case, fresh and organized mural thrombus was also noted, but the direct cause of death was due to aneurysmal rupture. In our case, systemic atherosclerosis was severe and the deceased also had a past medical history of renal transplantation and abdominal aortic aneurysm. Previous studies have suggested that there is a relationship between coronary artery aneurysm (or dissection) and renal transplantation [16]. Long-term immunosuppressant therapy after renal transplantation can lead to deterioration of systemic atherosclerosis [16]. Although we have no detailed medical information on our case, immunosuppressant therapy may have deteriorated systemic atherosclerosis. Forensic pathologists should perform a complete autopsy to determine the cause of death. Furthermore, pathological examinations of vascular lesions should be performed. References [1] Syed M, Lesch M. Coronary artery aneurysm: a review. Prog Cardiovasc Dis 1997;40:77–84. [2] Crawley PD, Mahlow WJ, Huntsinger DR, Afiniwala S, Wortham DC. Giant coronary artery aneurysms: review and update. Tex Heart Inst J 2014;41:603–8. [3] Erbersdobler A, Tsokos M. Sudden, unexpected death resulting from a giant coronary artery aneurysm. Forensic Sci Med Pathol 2006;2:199–201. [4] Cohle SD. Sudden unexpected death due to atherosclerotic coronary artery aneurysm. Am J Forensic Med Pathol 1985;6:153–7. [5] Daneshvar DA, Czak S, Patil A, Wasserman PG, Coplan NL, Garratt KN. Spontaneous rupture of a left main coronary artery aneurysm. Circ Cardiovasc Interv 2012;5:e63–5. [6] Kimura S, Miyamoto K, Ueno Y. Cardiac tamponade due to spontaneous rupture of large coronary artery aneurysm. Asian Cardiovasc Thorac Ann 2006;14:422–4. [7] Maehara A, Mintz GS, Ahmed JM, Fuchs S, Castagna MT, Pichard AD, et al. An intravascular ultrasound classification of angiographic coronary artery aneurysms. Am J Cardiol 2001;88:365–70. [8] Nichols L, Lagana S, Parwani A. Coronary artery aneurysm: a review and hypothesis regarding etiology. Arch Pathol Lab Med 2008;132:823–8. [9] Vranckx P, Pirot L, Benit E. Giant left main coronary artery aneurysm in association with severe atherosclerotic coronary disease. Cathet Cardiovasc Diagn 1997;42(1):54–7. [10] Cacucci M, Catanoso A, Valentini P, Lodi Rizzini A, Agricola P, Pedrinazzi C, et al. Right coronary artery aneurysm: percutaneous treatment with graftcoated stent during the acute phase of myocardial infarction. Int J Cardiol 2009;131:e56–8. [11] Chia HM, Tan KH, Jackson G. Non-atherosclerotic coronary artery aneurysms: two case reports. Heart 1997;78:613–6. [12] White NK, Edwards JE, Dry TJ. The relationship of the degree of coronary atherosclerosis with age, in men. Circulation 1950;1:645–54. [13] Ackerman RF, Dry TJ, Edwards JE. Relationship of various factors to the degree of coronary atherosclerosis in women. Circulation 1950;1:1345–54. [14] Imren Y, Zor H, Koksal P, Halit V. Giant aneurysm of left anterior descending coronary artery treated by surgical off-pump therapy. Thorac Cardiovasc Surg 2006;54:349–52. [15] Daoud AS, Pankin D, Tulgan H, Florentin RA. Aneurysms of the coronary artery. Report of ten cases and review of literature. Am J Cardiol 1963;11:228–37. [16] Tsimikas S, Giordano FJ, Tarazi RY, Beyer RW. Spontaneous coronary artery dissection in patients with renal transplantation. J Invasive Cardiol 1999;11:316–21.

Rupture of massive coronary artery aneurysm resulting in cardiac tamponade.

Coronary artery aneurysm is a fairly uncommon clinical entity, which is defined by a characteristic dilatation that exceeds 1.5 times the width of nor...
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