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 Table of Contents  
CASE REPORT
Year : 2019  |  Volume : 29  |  Issue : 1  |  Page : 23-25

Dual coronary-pulmonary fistula firstly found at routine doppler echocardiogram


1 Cardiology Department, University of Foggia, Foggia, Italy
2 Cardiology Department, Ospedali Riuniti, Bari, Italy
3 GVM Care and Research, Bari, Italy

Date of Web Publication20-Mar-2019

Correspondence Address:
Natale Daniele Brunetti
University of Foggia, Foggia
Italy
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jcecho.jcecho_47_18

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  Abstract 


Congenital coronary-pulmonary fistulas (CPFs) are defined as an abnormal direct communication between one or more coronary arteries, with a cardiac or thoracic structure bypassing the capillary network. We report the case of a 73-year-old male, with a history of hypertension, asymptomatic for angina and dyspnea, who was referred for routine clinical control. Echocardiogram unexpectedly showed the presence of diastolic flow from the pulmonary trunk in parasternal short-axis view. Pulsed-wave Doppler confirmed the presence of diastolic flow and displayed the typical coronary flow pattern. Coronary angiography hence showed the presence of dual CPFs originating from the second segment of the left anterior descending coronary and the right coronary arteries. Careful routine Doppler echocardiograph examination may raise the suspicion of CPF in case of otherwise unexplained symptoms and may represent a simple, easy, repeatable tool for the first suspected diagnosis and follow-up of CPFs.

Keywords: Coronary anomaly, coronary fistula, doppler echocardiogram


How to cite this article:
Casavecchia G, Zicchino S, Gravina M, Martone A, Cuculo A, Macarini L, Di Biase M, Brunetti ND. Dual coronary-pulmonary fistula firstly found at routine doppler echocardiogram. J Cardiovasc Echography 2019;29:23-5

How to cite this URL:
Casavecchia G, Zicchino S, Gravina M, Martone A, Cuculo A, Macarini L, Di Biase M, Brunetti ND. Dual coronary-pulmonary fistula firstly found at routine doppler echocardiogram. J Cardiovasc Echography [serial online] 2019 [cited 2019 Aug 26];29:23-5. Available from: http://www.jcecho.org/text.asp?2019/29/1/23/254592




  Introduction Top


Congenital coronary-pulmonary fistulas (CPFs) are defined as an abnormal direct communication between one or more coronary arteries, with a cardiac or thoracic structure bypassing the capillary network.[1] CPFs represent rare anomalies with an incidence variable in echocardiographic (0.06–0.2%),[2],[3] necropsy (14%),[3] and angiographic studies (0.1%–0.67%).[4],[5],[6],[7],[8],[9] Despite the described secondary forms (following chest trauma, complications of left main trunk procedures, or CABG), congenital forms represent the majority. Dyspnea and chest pain may be a frequent complaint in adults with CPFs, while, in the pediatric age group, the majority of cases are silent.[10] The most common coronary artery of origin for CPFs is the left main/left anterior descending, followed by the right coronary artery. CPF most commonly terminates in the main pulmonary artery.[1] The majority of CPFs are single (unilateral) communications,[11] but bilateral (dual) fistulas[12],[13] and multilateral, triple,[14] and quadruple[15] fistulas have been previously described.


  Case Report Top


We report the case of a 73-year-old male, with a history of hypertension, asymptomatic for angina and dyspnea, who was referred for routine clinical control. Physical examination was normal, and electrocardiogram showed a sinus rhythm with left bundle branch block. Echocardiographic examination revealed normal left and right ventricular dimension, but a moderate left ventricular systolic dysfunction (ejection fraction 40%). Unexpectedly, parasternal short-axis view showed the presence of diastolic flow from the pulmonary trunk upward [Figure 1]a. The ostia of the left and right coronary arteries were therefore carefully examined to assess their exact origin from the aorta or pulmonary artery. Pulsed-wave Doppler confirmed the presence of diastolic flow and displayed the typical coronary flow pattern [Figure 1]b. Therefore, a CPF was suspected. The patient was thus sent to cathlab for coronary angiography that showed the presence of dual CPFs originating from the second segment of the left anterior descending coronary [Figure 1]c and the right coronary arteries [Figure 1]d. Clinical and echocardiographic follow-up showed progressive reduction of systolic function, however, with a left ventricular ejection fraction >50% and effort dyspnea.
Figure 1: (a) Diastolic flow from the pulmonary trunk directed upward. (b) Pulsed-wave Doppler confirming typical coronary flow pattern. (c) Coronary-pulmonary fistula from the mid segment of the left anterior descending coronary artery (arrow). (d) Coronary-pulmonary fistula from the right coronary artery (arrow)

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  Discussion Top


Although the occurrence of congenital CPFs is rare, such anomalies could be the cause of different clinical conditions such as chest pain, exertion dyspnea, palpitations, and angina. Echocardiogram is an important tool for the detection of CPFs; when compared with coronary angio or angio computed tomography (CT), it is much simpler, easier, less expensive, safer, repeatable, and accurate. Occasionally, echocardiography could be the first-line method for the diagnosis of suspected congenital coronary artery fistulas.[16]

Echocardiography can show the presence of dilated coronary arteries and ventricular chambers and a variety of associated congenital and acquired heart defects.[17] In a case series from Vitarelli et al.,[18] transthoracic echocardiography was suggestive for the presence of coronary artery fistulas in 33% of cases and transesophageal echocardiography confirmed the diagnosis in all patients. Of course, the diagnosis of CPF must be always confirmed by coronary angiography or coronary CT that may not only eventually confirm the presence of CPFs, but also more clearly define the whole fistula characteristics (origin, pathway, and outflow) and identify possible complications. Echocardiography, ideal for first-line assessment, should be included within a multimodality imaging approach, especially in the differential diagnosis of CPFs, a potential challenging task.


  Conclusions Top


Careful routine Doppler echocardiograph examination may raise the suspicion of CPF in case of otherwise unexplained symptoms and may represent a simple, easy, repeatable tool for the first suspected diagnosis and follow-up of CPFs.

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.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Said SA, Nijhuis RL, Akker JW, Takechi M, Slart RH, Bos JS, et al. Unilateral and multilateral congenital coronary-pulmonary fistulas in adults: Clinical presentation, diagnostic modalities, and management with a brief review of the literature. Clin Cardiol 2014;37:536-45.  Back to cited text no. 1
    
2.
Sherwood MC, Rockenmacher S, Colan SD, Geva T. Prognostic significance of clinically silent coronary artery fistulas. Am J Cardiol 1999;83:407-11.  Back to cited text no. 2
    
3.
Hsieh KS, Huang TC, Lee CL. Coronary artery fistulas in neonates, infants, and children: Clinical findings and outcome. Pediatr Cardiol 2002;23:415-9.  Back to cited text no. 3
    
4.
Angelini P. Coronary artery anomalies – Current clinical issues: Definitions, classification, incidence, clinical relevance, and treatment guidelines. Tex Heart Inst J 2002;29:271-8.  Back to cited text no. 4
    
5.
Gillebert C, Van Hoof R, Van de Werf F, Piessens J, De Geest H. Coronary artery fistulas in an adult population. Eur Heart J 1986;7:437-43.  Back to cited text no. 5
    
6.
Vavuranakis M, Bush CA, Boudoulas H. Coronary artery fistulas in adults: Incidence, angiographic characteristics, natural history. Cathet Cardiovasc Diagn 1995;35:116-20.  Back to cited text no. 6
    
7.
Bhandari S, Kanojia A, Kasliwal RR, Kler TS, Seth A, Trehan N, et al. Coronary artery fistulae without audible murmur in adults. Cardiovasc Intervent Radiol 1993;16:219-23.  Back to cited text no. 7
    
8.
Said SA, el Gamal MI, van der Werf T. Coronary arteriovenous fistulas: Collective review and management of six new cases – Changing etiology, presentation, and treatment strategy. Clin Cardiol 1997;20:748-52.  Back to cited text no. 8
    
9.
Chiu CZ, Shyu KG, Cheng JJ, Lin SC, Lee SH, Hung HF, et al. Angiographic and clinical manifestations of coronary fistulas in Chinese people: 15-year experience. Circ J 2008;72:1242-8.  Back to cited text no. 9
    
10.
Said SA, Lam J, van der Werf T. Solitary coronary artery fistulas: A congenital anomaly in children and adults. A contemporary review. Congenit Heart Dis 2006;1:63-76.  Back to cited text no. 10
    
11.
Mullasari AS, Umesan CV, Kumar KJ. Transcatheter closure of coronary artery to pulmonary artery fistula using covered stents. Heart 2002;87:60.  Back to cited text no. 11
    
12.
Cheon WS, Kim EJ, Kim SH, Choi YJ, Rhim CY. Bilateral coronary artery fistulas communicating with main pulmonary artery and left ventricle: Case report. Angiology 2007;58:118-21.  Back to cited text no. 12
    
13.
Takeuchi N, Takada M, Nishibori Y, Maruyama T. A case report of coronary arteriovenous fistulas with an unruptured coronary artery aneurysm successfully treated by surgery. Case Rep Cardiol 2012;2012:314685.  Back to cited text no. 13
    
14.
Gundogdu F, Arslan S, Buyukkaya E, Kantarci M. Coronary artery fistula in a patient with coronary artery disease: Evaluation by coronary angiography and multidetector computed tomography. Int J Cardiovasc Imaging 2007;23:299-302.  Back to cited text no. 14
    
15.
Fujii H, Tsutsumi Y, Ohashi H, Kawai T, Iino K, Onaka M, et al. Surgical treatment of multiple coronary artery fistulas with an associated small saccular aneurysm – A case report. J Card Surg 2006;21:493-5.  Back to cited text no. 15
    
16.
Xie M, Li L, Cheng TO, Sun Z, Wang X, Lv Q, et al. Coronary artery fistula: Comparison of diagnostic accuracy by echocardiography versus coronary arteriography and surgery in 63 patients studied between 2002 and 2012 in a single medical center in China. Int J Cardiol 2014;176:470-7.  Back to cited text no. 16
    
17.
Said SA. Current characteristics of congenital coronary artery fistulas in adults: A decade of global experience. World J Cardiol 2011;3:267-77.  Back to cited text no. 17
    
18.
Vitarelli A, De Curtis G, Conde Y, Colantonio M, Di Benedetto G, Pecce P, et al. Assessment of congenital coronary artery fistulas by transesophageal color Doppler echocardiography. Am J Med 2002;113:127-33.  Back to cited text no. 18
    


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