Legal Medicine xxx (2014) xxx–xxx

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Case report

An autopsy case of a ruptured pseudoaneurysm of the ascending aorta complicated by previous cardiac surgery for ventricular septal defect Motonori Takahashi a,⇑, Takeshi Kondo a, Mai Morichika a, Kanako Nakagawa a, Azumi Kuse 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

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Article history: Received 17 March 2014 Received in revised form 1 May 2014 Accepted 3 May 2014 Available online xxxx Keywords: Aortic cannulation Pseudoaneurysm Cardiac surgery Ventricular septal defect

a b s t r a c t A man in his thirties was found dead in bed. He had undergone repair of a ventricular septal defect in his infancy and had a 2-month history of antemortem chest pain. On autopsy, a ruptured saccular aneurysm of the ascending aorta was identified, and the right thoracic cavity was found to contain coagulated blood. The oval ostium of the aneurysm was smoothly endothelialized and a black suture was found near its edge. Histological analysis revealed a defect of the intimal and medial layers in the wall of the aneurysm and hypertensive changes in several organs. The cause of death was presumed to be the rupture of a pseudoaneurysm at the aortic cannulation site after a long postoperative period. In the present case, preexisting hypertension was suspected as the cause of the formation and rupture of the pseudoaneurysm. In case of unexpected death, it is important to examine the past medical history even after a long postoperative period. Ó 2014 Elsevier Ireland Ltd. All rights reserved.

1. Introduction

2.2. Autopsy findings

Postoperative pseudoaneurysm of the ascending aorta has been reported in the field of cardiac surgery [1–13]. Though the rupture of a postoperative aortic pseudoaneurysm can be fatal, no medicolegal autopsy cases have been previously reported. This may be because making the postmortem diagnosis of a cardiac surgery complication is difficult when an aneurysm ruptures after a long postoperative period. The following is an autopsy case of sudden death due to the rupture of an aortic pseudoaneurysm related to cardiac surgery.

Autopsy was performed 15 h postmortem. The deceased was 163 cm in height and 56 kg in weight without arachnodactyly. He had a linear surgical scar on the midline of the anterior chest attributed to cardiac surgery. A ruptured saccular aneurysm (12.8  10.5 cm) of the ascending aorta was identified (Fig. 1), and the right thoracic cavity contained 1120 ml of blood with 1756 g of coagulation. The oval ostium between the aneurysm and the native ascending aorta was smoothly endothelialized, and a black suture thread was identified along the inner surface of its border (Fig. 2). Upon closer examination the aneurysm was found to contain a mural thrombus. The heart weighed 403 g and showed left ventricular hypertrophy. The defect of the ventricular septum was completely repaired with a patch and was endothelialized. Histological analysis revealed a disruption of the aortic media caused by the emplaced suture at the ostium of the aneurysm (Fig. 3). The immediate vicinity of the suture was surrounded by necrotic material and mild fibrosis. Evidence in support of inflammation was mild but several foreign body giant cells were detected running parallel with the suture. The aneurysm wall was constructed of fibrous granulation tissue infiltrated by siderophages and lymphocytes. Fresh hemorrhage was found at the rupture site. This aneurysm was diagnosed as a pseudoaneurysm because of the

2. Case report 2.1. Clinical history A man in his thirties was found dead in bed. He had undergone repair of a ventricular septal defect in his infancy; however, we could not access his medical records including the surgical notes. He had no history of periodical medical checks for at least several years. For two months prior to death, he had experienced anterior chest pain and numbness in the distal portion of the upper limbs. ⇑ Corresponding author. Tel.: +81 78 382 5582; fax: +81 78 382 5599. E-mail address: [email protected] (M. Takahashi). http://dx.doi.org/10.1016/j.legalmed.2014.05.003 1344-6223/Ó 2014 Elsevier Ireland Ltd. All rights reserved.

Please cite this article in press as: Takahashi M et al. An autopsy case of a ruptured pseudoaneurysm of the ascending aorta complicated by previous cardiac surgery for ventricular septal defect. Leg Med (2014), http://dx.doi.org/10.1016/j.legalmed.2014.05.003

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Fig. 1. Gross appearance of the ruptured pseudoaortic aneurysm. A saccular aneurysm of the ascending aorta was found to be ruptured in its lower right side (arrow).

Fig. 2. Macroscopic appearance of the ostium between the aneurysm and the native aorta. The oval ostium (arrow) between the aneurysm and the native ascending aorta was smoothly endothelialized (a). When the aneurysm was dissected, a black suture thread (arrowhead) was identified close to the edge of the ostium (arrow). Insert: low magnification image (b).

Please cite this article in press as: Takahashi M et al. An autopsy case of a ruptured pseudoaneurysm of the ascending aorta complicated by previous cardiac surgery for ventricular septal defect. Leg Med (2014), http://dx.doi.org/10.1016/j.legalmed.2014.05.003

M. Takahashi et al. / Legal Medicine xxx (2014) xxx–xxx

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Fig. 3. Histological analysis of the aneurysm ostium. Histological examination with Elastica van Gieson stain revealed discontinuance of the medial elastic fibers (arrow) adjacent to the sutures (asterisk) (a). High magnification image with hematoxylin–eosin stain showed disruption of the media (arrow). Sutures were surrounded by eosinophilic necrotic tissue (b).

lack of intimal and medial layers in its wall. The remaining segments of the aorta were intact and maintained an otherwise unremarkable three-layered structure; medionecrosis was not found and no other significant pathological changes were detected in the adventitia except for a congestion of the vasa vasorum. The kidneys showed hypertensive changes such as hyaline arteriolosclerosis and mild glomerulosclerosis; arteriosclerosis was also found in the spleen. All organs presented with venous congestion to limited degree. The cause of death was diagnosed as the rupture of a pseudoaortic aneurysm related to previous cardiac surgery.

3. Discussion A pseudoaneurysm is a pathologic arterial dilatation lined by adventitia [14]; it is differentiated from a true aneurysm, in which all three vascular layers remain intact. Postoperative pseudoaneurysm of the ascending aorta is an unusual complication of thoracic surgery. It occurs at sites where the aortic wall is disrupted, such as

aortic cannulation sites [1–8], suture lines [1–4,9–11], and other surgical invasions of the aorta [3,12,13]. During cardiac surgery, direct aortic cannulation for the access of blood returning from cardiopulmonary bypass is preferred because of its technical simplicity and lower complication rate compared with femoral artery cannulation [15]. Although we could not access the medical records, the aortic cannulation site was presumed to be the origin of the aortic pseudoaneurysm in the present case, because the deceased had undergone cardiac surgery, the aneurysm originated in the ascending aorta, and a black suture was identified beside the ostium of the aneurysm. Hypertension appears to be related to aneurysm formation and rupture [8,16,17]. The autopsy findings in this case (cardiac hypertrophy, arteriosclerosis, and glomerulosclerosis) suggest antemortem hypertension, which was suspected as a cause of the aneurysmal enlargement. Infectious states, such as mediastinitis, sepsis, and endocarditis, are recognized as potential causes of pseudoaneurysms [1–3]. Although we did not perform bacterial or mycotic culture of the aneurysm, there were no macroscopic or histological findings of inflammation. Tissue fragility of the aorta

Please cite this article in press as: Takahashi M et al. An autopsy case of a ruptured pseudoaneurysm of the ascending aorta complicated by previous cardiac surgery for ventricular septal defect. Leg Med (2014), http://dx.doi.org/10.1016/j.legalmed.2014.05.003

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M. Takahashi et al. / Legal Medicine xxx (2014) xxx–xxx

complicated by aortic dissection and Marfan syndrome is another risk factor for postoperative pseudoaneurysm [2]. In the present case, the aorta was not dissected, and histological examination failed to reveal medionecrosis of the aorta. The patient did not meet the diagnostic criteria for Marfan syndrome [18]. On autopsy, it was not possible to identify when the pseudoaneurysm began to enlarge. Hemosiderin in an aneurysm wall indicates intrawall hemorrhage of at least several weeks’ duration. The 2-month history of antemortem chest pain in this case may have been caused by expansion of the aneurysm [17,19]. These data suggest the possibility that this patient’s aneurysm may have progressively enlarged for several weeks or months prior to death. References [1] Atik FA, Navia JL, Svensson LG, Vega PR, Feng J, Brizzio ME, et al. Surgical treatment of pseudoaneurysm of the thoracic aorta. J Thorac Cardiovasc Surg 2006;132:379–85. [2] Malvindi PG, van Putte BP, Heijmen RH, Schepens MA, Morshuis WJ. Reoperations for aortic false aneurysms after cardiac surgery. Ann Thorac Surg 2010;90:1437–43. [3] Sullivan KL, Steiner RM, Smullens SN, Griska L, Meister SG. Pseudoaneurysm of the ascending aorta following cardiac surgery. Chest 1988;93:138–43. [4] Razzouk A, Gundry S, Wang N, Heyner R, Sciolaro C, Van Arsdell G, et al. Pseudoaneurysms of the aorta after cardiac surgery or chest trauma. Am Surg 1993;59:818–23. [5] Flick WF, Hallermann FJ, Feldt RH, Danielson GK. Aneurysm of aortic cannulation site. Successful repair by means of peripheral cannulation, profound hypothermia, and circulatory arrest. J Thorac Cardiovasc Surg 1971;61:419–23. [6] Branchini B, Zingone B, Vaccari M. Ascending aortic false aneurysm following cannulation for perfusion. Thorax 1976;31:234–7.

[7] Yoshida K, Ohshima H, Murakami F, Matsuura A, Hibi M, Kawamura M. Nonmycotic pseudoaneurysm in the ascending aorta following cardiac surgery. Jpn J Thorac Cardiovasc Surg 1999;47:295–7. [8] Sabri MN, Henry D, Wechsler AS, DiSciascio G, Vetrovec GW. Late complications involving the ascending aorta after cardiac surgery: recognition and management. Am Heart J 1991;121:1779–83. [9] Chevalier P, Moncada E, Kirkorian G, Touboul P. Acquired aortopulmonary fistula in pseudoaneurysm of the aorta six years after a Bentall operation. J Thorac Cardiovasc Surg 1995;110:1143–4. [10] Linz PE, Wallace RB, Baker WP. Long-term follow-up and resection of a postoperative false aortic aneurysm. Ann Thorac Surg 1993;55:758–9. [11] Mohammadi S, Bonnet N, Leprince P, Kolsi M, Rama A, Pavie A, et al. Reoperation for false aneurysm of the ascending aorta after its prosthetic replacement: surgical strategy. Ann Thorac Surg 2005;79:147–52. [12] Mazzei V, Benvenuto D, Gagliardi M, Guarracini S, Di Mauro M. Thyrocervical trunk pseudoaneurysm following central venous catheterization. J Card Surg 2011;26:617–8. [13] Stassano P, De Amicis V, Gagliardi C, Di Lello F, Spampinato N. False aneurysm from the aortic vent site. J Cardiovasc Surg (Torino) 1982;23:401–2. [14] Mark AC, Joseph L. Disease of the Aorta. In: Loscalzo Joseph, editor. Harrison’s Cardiovascular Medicine. New York: McGraw-Hill; 2010. p. 445–53. [15] Gerbode F, Kerth WJ, Kovacs G, Sanchez PA, Hill JD. Cannulation of the ascending aorta for perfusion during cardiopulmonary bypass. A new technique and analysis of results. J Cardiovasc Surg (Torino) 1968;9:293–6. [16] Bickerstaff LK, Pairolero PC, Hollier LH, Melton LJ, Van Peenen HJ, Cherry KJ, et al. Thoracic aortic aneurysms: a population-based study. Surgery 1982; 92:1103–8. [17] Spittell Jr JA. Hypertension and arterial aneurysm. J Am Coll Cardiol 1983; 1:533–40. [18] Loeys BL, Dietz HC, Braverman AC, Callewaert BL, De Backer J, Devereux RB, et al. The revised Ghent nosology for the Marfan syndrome. J Med Genet 2010; 47:476–85. [19] Katsumata T, Moorjani N, Vaccari G, Westaby S. Mediastinal false aneurysm after thoracic aortic surgery. Ann Thorac Surg 2000;70:547–52.

Please cite this article in press as: Takahashi M et al. An autopsy case of a ruptured pseudoaneurysm of the ascending aorta complicated by previous cardiac surgery for ventricular septal defect. Leg Med (2014), http://dx.doi.org/10.1016/j.legalmed.2014.05.003

An autopsy case of a ruptured pseudoaneurysm of the ascending aorta complicated by previous cardiac surgery for ventricular septal defect.

A man in his thirties was found dead in bed. He had undergone repair of a ventricular septal defect in his infancy and had a 2-month history of antemo...
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