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 Table of Contents  
CASE REPORT
Year : 2017  |  Volume : 5  |  Issue : 2  |  Page : 89-91

Single coronary artery and Takayasu's arteritis: An extremely rare association


Department of Interventional Cardiology, R. G. Kar Medical College and Hospital, Kolkata, West Bengal, India

Date of Web Publication20-Jun-2017

Correspondence Address:
K N Sudeep
Department of Interventional Cardiology, R. G. Kar Medical College and Hospital, 1, Khudiram Bose Sarani, Kolkata - 700 004, West Bengal
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/heartindia.heartindia_51_16

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  Abstract 

Single coronary artery is a rare congenital anomaly of the coronary artery. The association ranges from predominantly asymptomatic to myocardial infarction, syncope being an uncommon presentation of Takayasu's arteritis. Given a limited literature of single coronary artery, we have made an attempt in our case report to relate an uncommon association of single coronary artery and Takayasu's arteritis.

Keywords: Single coronary artery, syncope, Takayasu's arteritis


How to cite this article:
Majumder B, Sarker B, Sudeep K N, Bagchi PC, Ghosh S. Single coronary artery and Takayasu's arteritis: An extremely rare association. Heart India 2017;5:89-91

How to cite this URL:
Majumder B, Sarker B, Sudeep K N, Bagchi PC, Ghosh S. Single coronary artery and Takayasu's arteritis: An extremely rare association. Heart India [serial online] 2017 [cited 2021 Dec 2];5:89-91. Available from: https://www.heartindia.net/text.asp?2017/5/2/89/208556


  Introduction Top


Coronary artery anomalies being an uncommon entity with few cases which are incidentally detected. Associations with coronary artery anomalies are limited. Takayasu's arteritis presenting as syncope is not much reported. With a learning objective of extending our knowledge and adding to the literature of single coronary artery, we here report an extremely rare case of single coronary artery with Takayasu's arteritis presenting as syncope.


  Case Report Top


A 35-year-old male was referred to the department of cardiology for the management of recurrent episodes of syncope. He had recurrent episodes of syncope for 6 months. On detailed inquiry, he revealed episodes of syncope when doing heavy physical activity of the left upper limbs and some episodes on turning of neck on the right side. He would immediately regain consciousness without any postictal drowsiness. There was no history of head injury, chest pain, palpitation, ataxia, vertigo, and sudden onset of weakness. There was no history of fever, arthralgia, and joint pain.

He also complained of fatigue, numbness, and pain in bilateral upper extremities, more on the left side that worsened on exertion and resembled ischemic pain. There was no history of hypertension, diabetes, and smoking.

On physical examination, his bilateral upper extremity pulsations were absent, while lower limb pulses felt normal. His right carotid pulses were feeble with no audible bruits. A 12-lead electrocardiogram, chest X-ray, computed tomography (CT) scan, and 24 h Holter monitoring were normal. Transthoracic echo showed left ventricular hypertrophy and normal left ventricular systolic function.

Color Doppler imaging of both proximal common carotid arteries demonstrated long-segment, diffuse circumferential thickening of the vessel wall. Doppler waveform of both arteries showed increased flow velocity and turbulence and spectral broadening due to a narrow lumen. Internal carotid artery on both sides revealed dampened waveforms. Right vertebral artery showed circumferential thickening and reversal of flow with reduced velocity. Left vertebral artery was normal. Triphasic pattern in the right upper extremity was lost.

Coronary angiography demonstrated a single coronary artery originating from the right sinus of Valsalva. From the single trunk, an anomalous coronary trunk and right coronary artery (RCA) raised. The anomalous coronary trunk then coursed anteriorly and divided into left anterior descending (LAD) and left circumflex artery (LCX). LCX was nondominant with LAD ending short of apex [Figure 1]b.
Figure 1: (a) Magnetic resonance angiogram of the neck vessels showing thickening and stenotic neck vessels. (b) Coronary angiogram. Right anterior oblique view showing single ostium with a common trunk from which right coronary artery, left anterior descending, and left circumflex artery arise

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CT coronary angiography revealed that the left coronary ostium was absent, and a blind left aortic sinus of Valsalva was noted. In addition, no artery was noted to arise from the noncoronary sinus or ascending aorta. A single coronary artery was observed to be arising from the right aortic sinus of Valsalva, with a single ostium. All coronary arteries are seen to originate from a single trunk arising from the right sinus of Valsalva. Thereafter, RCA coursed through the right atrioventricular groove and divided into the right marginal artery, posterior descending artery, and posterior left ventricular branch in keeping with the course of anatomic RCA. There was mild segmental atherosclerosis in mid-LAD [Figure 2].
Figure 2: Computed tomography angiography showing course and division of single coronary artery

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Magnetic resonance angiography of the brain and neck vessels revealed arteritic disease involving the right common carotid and right vertebral arteries. The origin of brachiocephalic artery and left subclavian artery (SCA) was not well visualized. Brachiocephalic, left common carotid, and the left SCA showed diffuse wall thickening with significant luminal compromise, however the left vertebral artery was well observed [Figure 1]a.


  Discussion Top


A single coronary artery is a rare congenital anomaly of the coronary arteries. The incidence rate was found to be 0.019% in a large series of 126,595 coronary angiographies at Cleveland clinic.[1] Nearly 40% of the single coronary artery is associated with congenital heart disease such as Tetralogy of Fallot.[2]



Four aberrant pathways to reach its proper vascular territory were described. These pathways are designated as Type A (anterior to the right ventricular outflow tract), Type B (between the aorta and pulmonary trunk), Type C (cristal, coursing through the crista supraventricularis portion of the septum), and Type D (dorsal or posterior to aorta).[4]

Our case report had a unique association of aortoarteritis with subclavian steal syndrome and SCA. Although it was an incidental finding, other modes of presentation were sudden cardiac deaths (SCDs), myocardial infarction (MI), and syncope.[5] MI and SCDs were associated more when the artery courses between aorta and pulmonary artery.

Coronary arterial involvement in Takayasu's arteritis being ostial stenosis, care should be taken in coronary angiogram to avoid deep intubation, rapid and prolong dye injection, and preferably to use 4F and 5F catheters. Transient occlusion by dye injection, coronary spasm, and dampening may put whole of the myocardium under jeopardy leading to ventricular arrhythmias, fibrillation, and cardiac arrest. The detrimental impact of coherence of SCA with Takayasu's arteritis is with the occurrence of coronary artery disease leading to MI with left ventricular dysfunction, which further aggravates subclavian steal syndrome leading to profound cerebral ischemia.

Subclavian steal syndrome results from occlusion of the proximal SCA and with retrograde flow to the SCA from the vertebral artery. Takayasu's arteritis and coronary involvement are rare and coronary involvement occur in the form of stenosis and ostial lesion.[6] There are previously reported associations of single coronary artery and fistula to pulmonary artery.[7]

Takayasu's arteritis being a progressive disease and with a single coronary artery, our patient was fortunate as there was minor coronary artery disease, otherwise whole of the myocardium would have been jeopardized leading to adverse consequences.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
  References Top

1.
Yamanaka O, Hobbs RE. Coronary artery anomalies in 126,595 patients undergoing coronary arteriography. Cathet Cardiovasc Diagn 1990;21:28-40.  Back to cited text no. 1
    
2.
Antonellis J, Rabaouni A, Kostopoulos K, Margaris N, Kranidis A, Salahas A, et al. Single coronary artery from the right sinus of valsalva, associated with absence of left anterior descending and an ostium-secundum-type atrial septal defect: A rare combination. A case report. Angiology 1996;47:621-5.  Back to cited text no. 2
    
3.
Lipton MJ, Barry WH, Obrez I, Silverman JF, Wexler L. Isolated single coronary artery: Diagnosis, angiographic classification, and clinical significance. Radiology 1979;130:39-47.  Back to cited text no. 3
    
4.
Roberts WC, Shirani J. The four subtypes of anomalous origin of the left main coronary artery from the right aortic sinus (or from the right coronary artery). Am J Cardiol 1992;70:119-21.  Back to cited text no. 4
    
5.
Shirani J, Roberts WC. Solitary coronary ostium in the aorta in the absence of other major congenital cardiovascular anomalies. J Am Coll Cardiol 1993;21:137-43.  Back to cited text no. 5
    
6.
Tyagi S, Khan AA, Kaul UA, Arora R. Percutaneous transluminal angioplasty for stenosis of the aorta due to aortic arteritis in children. Pediatr Cardiol 1999;20:404-10.  Back to cited text no. 6
    
7.
Xie LJ, Zhang L, Xiao TT, Shen J. Coronary artery fistula between single right coronary artery and right pulmonary artery: A case report and literature review. BMC Cardiovasc Disord 2015;15:171.  Back to cited text no. 7
    


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  [Figure 1], [Figure 2]



 

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