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 Table of Contents  
CASE REPORT
Year : 2020  |  Volume : 6  |  Issue : 1  |  Page : 81-83

Whole coronary circulation originating from single ostia – Rare case report


1 Department of Cardiology, Apex Heart Institute, Ahmedabad, Gujarat, India
2 Department of Cardiology, Shri Siddhivinayak Hospital, Paonta Sahib, Himachal Pradesh, India

Date of Submission13-Nov-2019
Date of Decision21-Jan-2020
Date of Acceptance03-Mar-2020
Date of Web Publication17-Apr-2020

Correspondence Address:
Sunil Gurmukhani
Apex Heart Institute, Mondeal Business Park, SG Highway, Ahmedabad - 380 059, Gujarat
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jpcs.jpcs_69_19

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  Abstract 


The prevalence of coronary artery anomalies is approximately 1% in individuals undergoing angiography. Most of the anomalies are benign, but some can lead to myocardial infarction, cardiomyopathy, and sudden cardiac death. It is very rare to have an entire coronary circulation that arises from the right coronary cusp. We present the case of a 55-year-old female who presented with the complaints of chest pain on exertion with positive stress test. An invasive angiogram revealed all the three coronary arteries originating from the right coronary cusp.

Keywords: Anomalous origin of the coronary artery, coronary anomalies, single coronary artery


How to cite this article:
Gurmukhani S, Agrawal V, Mittal S, Tiwari P, Shah S, Patel T. Whole coronary circulation originating from single ostia – Rare case report. J Pract Cardiovasc Sci 2020;6:81-3

How to cite this URL:
Gurmukhani S, Agrawal V, Mittal S, Tiwari P, Shah S, Patel T. Whole coronary circulation originating from single ostia – Rare case report. J Pract Cardiovasc Sci [serial online] 2020 [cited 2020 May 30];6:81-3. Available from: http://www.j-pcs.org/text.asp?2020/6/1/81/282810




  Introduction Top


Incidence of coronary artery anomalies (CAAs) in the general population is approximately 1%.[1] Exact etiology, pathophysiology, and clinical implication are best known to no body. Most variations are benign; however, some may lead to myocardial ischemia and/or sudden cardiac arrest.[2] Among all coronary anomalies, anomalous origin of the coronary artery from the opposite sinus has been associated with sudden cardiac death (SCD) in young population.[3] Proposed mechanism is the compression of the coronary artery from two great vessel during the systole, particularly during strenuous exercise. Coronary anomaly with a single ostium is an extremely rare anomaly among all coronary anomalies.[4] Here, we describe an extremely rare coronary anomaly in which whole coronary circulation is arising from the single trunk (right main coronary stem) from the right coronary cusp.


  Case Report Top


A 55-year-old female with no coronary artery disease (CAD) risk factor presented with complaints of Class II angina. On examination, vital signs were normal. Electrocardiogram showed mild ST depression in the precordial leads. Echocardiographic examination showed normal left ventricular systolic function with no regional wall motion abnormalities. In view of her symptoms, we planned stress exercise test which revealed significant ST depression during peak exercise. Positive stress test compelled us to do coronary angiogram for her. During angiography, we were unable to engage the left system in its usual position with multiple diagnostic catheter with different curves. When we engage the right coronary system, we found all major coronary arteries were arising from the single common trunk from right coronary cusp [Figure 1], [Figure 2], [Figure 3], [Figure 4] and Videos 1-4 ]. We called this common trunk as the right main coronary artery. Left coronary cusp was bald in nonselective angiogram [Figure 5]. Coronary angiogram did not reveal any significant obstructive lesion in any of the coronary artery. Probable cause of her angina and positive stress test would be anomalous course of coronary arteries.
Figure 1: Single common trunk from right coronary cusp is giving rise to all three coronary arteries in left anterior oblique (LAO) - cranial view.

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Figure 2: Single common trunk from right coronary cusp is giving rise to all three coronary arteries in left anterior oblique (LAO) view.

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Figure 3: Single common trunk from right coronary cusp is giving rise to all three coronary arteries in right anterior oblique (RAO) view.

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Figure 4: Single common trunk from right coronary cusp is giving rise to all three coronary arteries in antero-posterior (AP)- cranial view.

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Figure 5: Bald left coronary cusp on nonselective.

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  Discussion Top


Congenital coronary anomalies are fascinating entity in cardiology. CAAs are found in approximately in 1% of the general population.[1] It may be broadly classified as abnormalities of origin, course, destination, and number of arteries. It may present clinically with chest pain, dyspnea, arrhythmias, syncope, and SCD or asymptomatic incidental finding. CAA are involved with approximately 12% of sports-related SCD as compared to 1.2% of nonsports-related deaths.[5] CAAs are a persistent challenge in cardiology, as etiology, pathophysiology, mechanism, clinical implication, and management are unanswered questions. The correct description of various types of coronary anomaly and their clinical significance for every individual patient is very important. On the other hand, with the increasing use of invasive and noninvasive imaging to rule out coronary artery atherosclerotic disease in middle aged and older population, we are facing the increased recognition of asymptomatic CAAs. Noninvasive anatomic imaging is complementary to invasive imaging and helps to further delineate the high-risk anatomic features.

Among all coronary anomalies, coronary artery origin from the opposite sinus is one which has clinical implication. There are many reports, suggesting the association of this anomaly with SCD. However, this entity can be cause of angina in nonobstructive coronary arteries. Even coronary interventions if required are also technically challenging in the presence of coronary anomaly. Specific guidelines for the diagnosis, classification, risk stratification, and management of such anomalies are lacking in the literature.

Here, we have described the rarest type of coronary anomaly where all the three major coronary arteries are arising from the single common trunk from the right coronary cusp in a female patient with positive stress test. Our patient did not have any obstructive CAD, but still had symptoms on exercise. There are no general guidelines on how to manage this particular CAA; however, due to the association of SCD with anomalous artery and exercise, patients are generally advised to avoid the strenuous activity. Clinical significance and management of this case are not known; potential treatment options are medical management and percutaneous intervention of surgery. Evidence and guidelines about treatment of different types of coronary anomalies is lacking. Further studies are desirable for this type of coronary anomalies.


  Conclusion Top


Coronary anomalies are extremely rare congenital heart disease, and single coronary stem gives rise to whole coronary circulation is rarest. The clinical implication and management in such kind of cases are not well described in the literature. Strong data and robust registry on coronary anomalies with a long follow-up is desirable to understand this entity. In our patient with classical angina and positive stress test warrants some treatment. However, which form of treatment is the best in such cases is still dilemma.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Ethics clearance

As per local institutinal Ethical committee.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Angelini P, Velasco JA, Flamm S. Coronary anomalies: Incidence, pathophysiology, and clinical relevance. Circulation 2002;105:2449-54.  Back to cited text no. 1
    
2.
Ogden JA. Congenital anomalies of the coronary arteries. Am J Cardiol 1970;25:474-9.  Back to cited text no. 2
    
3.
Villa AD, Sammut E, Nair A, Rajani R, Bonamini R, Chiribiri A. Coronary artery anomalies overview: The normal and the abnormal. World J Radiol 2016;8:537-55.  Back to cited text no. 3
    
4.
Patel KB, Gupta H, Nath H, Aqel RA, Zoghbi GJ, Soto B, et al. Origin of all three major coronary arteries from the right sinus of valsalva: Clinical, angiographic, and magnetic resonance imaging findings and incidence in a select referral population. Catheter Cardiovasc Interv 2007;69:711-8.  Back to cited text no. 4
    
5.
Modi H, Ericssossi B, Trivedi D. Anomalous origin of all three coronary arteries from right sinus of valsalva in a patient with angina pectoris. J Invasive Cardiol 2011;23:E240-2.  Back to cited text no. 5
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5]



 

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