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Journal of Acute Medicine 5 (2015) 77e79 www.e-jacme.com Case Report Rare cause of abdominal pain and : Rupture of a sinus of Valsalva aneurysm

Yi-Ming Weng a,b,e, Tai-Yi Hsu c,e, Cheng-Wei Chan b,d,*, Jih-Chang Chen a,b

a Department of Emergency Medicine, Chang Gung Memorial Hospital, Taoyuan, Taiwan, ROC b College of Medicine, Chang Gung University, Taoyuan, Taiwan, ROC c Department of Emergency Medicine, China Medical University Hospital, Taichung, Taiwan, ROC d Department of Emergency Medicine, Chang Gung Memorial Hospital and Chang Gung University College of Medicine, Keelung, Taiwan, ROC

Received 18 April 2014; revised 12 May 2015; accepted 18 June 2015 Available online 9 September 2015

Abstract

Cardiogenic shock is defined by tissue hypoperfusion due to heart failure. The manifestations of acute right ventricular (RV) failure include a distended jugular and hypotension, but not rales. The causes of isolated acute RV failure include RV infarction, , , congenital disease, and valvular heart disease. Rupture of a sinus of Valsalva aneurysm (SVA) is rarely seen in emergency departments (EDs). We report a 46 year-old male who suffered from SVA rupture triggering acute RV failure and . Copyright © 2015, Taiwan Society of Emergency Medicine. Published by Elsevier Taiwan LLC. All rights reserved.

Keywords: Cardiogenic shock; Acute right ventricular failure; Rupture of a sinus of Valsalva aneurysm

1. Introduction 2. Case report

Cardiogenic shock is defined by tissue hypoperfusion due A 46-year-old male presented with acute abdominal pain 3 to heart failure. Acute right ventricular (RV) failure triggers hours in duration. He had a medical history of hypertension and cardiogenic shock by decreasing both preload and septal shift. hyperlipidemia, which were being controlled by medication. He Patients with acute RV failure may present with distended complained of moderate pain in the epigastric area and described jugular and hypotension, but not rales. The causes of it as dull in character. He denied any radiation pain. We found no isolated acute RV failure include RV infarction, pulmonary chest/abdominal trauma or any aggravating factor. His symp- embolism, cardiac tamponade, congenital disease, and toms included cold sweating, chest tightness, shortness of breath, valvular heart disease. Rupture of a sinus of Valsalva aneu- nausea, and vomiting. No bloody vomitus or tarry stool was rysm (SVA) is rarely seen in emergency department. We report mentioned. At the emergency department, the patient appeared a case of SVA rupture triggering acute RV failure and acutely ill and pale in color. Initial vital signs included a body cardiogenic shock. After emergency surgical repair, our pa- temperature of 34C, a rate of 65 beats/min, and a blood tient was discharged without sequelae. pressure of 81/39 mmHg. Physical examination was unremark- able, except for a distended external jugular vein in the sitting position, a loud continuous diastolic over the lower * Corresponding author. Department of Emergency Medicine, Chang Gung sternal border, and cold and clammy skin. Breathing sounds were Memorial Hospital, Keelung, Number 222, Maijin Road, Anle District, clear and symmetrical in both lungs. The abdomen was soft, with Keelung City 204, Taiwan, ROC. mild tenderness over the peri-umbilical area but no peritoneal E-mail address: [email protected] (C.-W. Chan). e These authors contributed equally to this manuscript. signs. Bedside ultrasonography was negative for ascites and

http://dx.doi.org/10.1016/j.jacme.2015.06.002 2211-5587/Copyright © 2015, Taiwan Society of Emergency Medicine. Published by Elsevier Taiwan LLC. All rights reserved. 78 Y.-M. Weng et al. / Journal of Acute Medicine 5 (2015) 77e79 pericardiac fluid. revealed junctional rhythm without an ST-T change. The plain film showed a poorly expanded lung and cardiomegaly (Figure 1). Our initial suspi- cion was acute RV failure. To rule out pulmonary embolism, mechanical obstruction of the RVoutflow, and any other anom- aly, we ordered a computed tomographic (CT) scan. This revealed contrast pooling over the RV and superior and inferior vena cava. Retrograde flow into the RV via an aortic root was evident (Figures 2 and 3). We found no pulmonary embolism, mechanical lesion, or anomaly. Further two-dimensional trans- thoracic echocardiography revealed an SVA with a left-to-right

Figure 3. Shunt from the aortic root to the right ventricle (arrow), which is possible evidence of sinus of Valsalva aneurysm rupture.

shunt from the right coronary sinus into the RV. Rupture of an SVA was suspected. An emergency operation using a patch repair was performed immediately. Our patient made an un- eventful recovery and was discharged 9 days after the surgery.

3. Discussion

Figure 1. Chest x-ray revealing cardiomegaly. Our case presentation emphasizes that differential diagnosis of shock is critical for appropriate management. First, the finding of cold and clammy skin rendered a diagnosis of unlikely. Also, neither neurogenic nor anaphylactic shock was considered, based on the present illness and medical history. Second, septic shock was ruled out because of the rapidly progressive course, lack of fever, and focus of infection. Third, rather than hypovolemic shock, we classified our case as cardiogenic shock because of the pres- ence of a distended jugular vein without obvious volume loss. Furthermore, clear breathing sounds upon chest indicated an isolated RV failure. Although a retrograde flow was suspected following CT scan examination, we ordered two-dimensional transthoracic echocardiography because we considered this tool appropriate to aid diagnosis. Additionally, the loud diastolic heart murmur over the lower sternal border suggested valvular heart disease or an intracardiac shunt. Our report confirms previous suggestions that transthoracic echocardiography1e3 is a useful and reliable tool when employed to confirm SVA rupture. Other useful imaging modalities include transesophageal echocardiography,2e4 CT angiography,5 and magnetic resonance imaging. SVA is a dilatation caused by separation of the aortic media Figure 2. Contrast regurgitation to the superior and inferior vena cava, evi- and annulus fibrosus.6 Acute rupture of the SVA is fatal. Most dence of poor heart pump function. Y.-M. Weng et al. / Journal of Acute Medicine 5 (2015) 77e79 79 ruptured SVAs occur in patients <40 years of age1,2,4,6 and References predominate in males.4,6 The right coronary sinus2,6 is affected most frequently, followed by the noncoronary sinus.7 Ruptures 1. Guenther F, von Zur Muhlen C, Lohrmann J, Bode C, Geibel A. Rupture of of an SVA into the right ventricle are most common.2,6 When an aneurysm of the noncoronary sinus of Valsalva into the right atrium. Eur 7 J Echocardiogr. 2008;9:186e187. an SVA ruptures, the patient may complain of ,   1,6 1 1 6 2. Vincelj J, Starcevic B, Sokol I, Sutlic Z. Rupture of a right sinus of valsalva dyspnea, , tinnitus, orthopnea, upper abdom- aneurysm into the right ventricle during vaginal delivery: a case report. 6,8 inal discomfort, and edema of the lower extremities. Echocardiography. 2005;22:844e846. Abdominal pain as the initial presentation of SVA rupture is 3. Bouvier E, Porte JM, Laperche T, et al. Rupture of a sinus of Valsalva rare; only one similar case has been reported previously.9 The aneurysm in the left atrium. Arch Mal Coeur Vaiss. 2001;94:1409e1412.  í cause of abdominal pain may be nonocclusive ischemia of the 4. Sanchez ME, Garc a-Palmieri MR, Quintana CS, Kareh J. Heart failure in rupture of a sinus of valsalva aneurysm. Am J Med Sci. 2006;331:100e102. small bowel attributed to hypoperfusion caused, in turn, by 5. Kantarci M, Doganay S, Gundogdu F, Unlu Y. A case with noncoronary 10 acute heart-pump dysfunction. Other findings on physical sinus of Valsalva aneurysm: multidetector computed tomography findings. examination include , wide variations in pulse Heart Surg Forum. 2008;11:E372e374. pressure, bounding carotid and peripheral , pulmonary 6. Galicia-Tornell MM, Marín-Solís B, Mercado-Astorga O, Espinoza- í í crackles,4 a prominent continuous precordial murmur8 with a Anguiano S, Mart nez-Mart nez M, Villalpando-Mendoza E. Sinus of 4,8 6 Valsalva aneurysm with rupture. Case report and literature review. Cir Cir. thrill, hepatomegaly, upper abdominal tenderness, severe 2009;77:441e445. 6 6,8 kidney insufficiency, and congestive hepatic insufficiency. 7. Turgeon AF, Dagenais F, Poirier P, Bauset R, Mathieu P. Myocardial The standard management for a ruptured SVA is early surgi- infarction in a young woman secondary to a rupture of a noncoronary sinus cal intervention.1,2,6 Nonsurgical transcatheter-mediated of Valsalva aneurysm without coronary disease. Can J Cardiol. e closure of the rupture is another option.11 2003;19:723 724. 8. Amoah AG, Frimpong-Boateng K, Baddoo H. Rupture of sinus of Valsalva This report reminds clinicians that rupture of an SVA is a aneurysm: case report. East Afr Med J. 2000;77:627e628. rare etiology of acute RV failure. An understanding of the 9. Sharkey A, Cooper G. An unusual and life-threatening cause of epigastric pathophysiology of the disease entity helps emergency pain presenting to the general surgical team: a case report. Ann R Coll Surg department physicians recognize specific manifestations of the Engl. 2010;92:W26e27. condition. Prompt specialist attention with early intervention 10. Baumgartner F, Velez M, Zulim R, State D. Abdominal pain, colomegaly, and jaundice from heart failure related to mitral stenosis. J Natl Med saves lives. Assoc. 1989;81:1091e1093. 11. Cui W, van Bergen AH, Patel D, Javois AJ, Roberson DA. Trans- Conflicts of interest catheter closure of ruptured sinus of Valsalva aneurysm and secundum atrial septal defect with limited inferior rim. Echocardiography. e All authors have no conflicts of interest to declare. 2008;25:208 213.