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Characteristics of Congenital Coronary Artery Fistulas Complicated with Infective Endocarditis: Analysis of 25 Reported Cases

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Characteristics of Congenital Coronary Artery Fistulas Complicated with Infective Endocarditis: Analysis of 25 Reported Cases 1 REVIEW ARTICLE Characteristics of Congenital Coronary Artery Fistulas Complicated with Infective Endocarditis: Analysis of 25 Reported Cases Salah AM Said, MD, PhD Department of Cardiology, Hospital Group Twente, Hengelo, The Netherlands ABSTRACT Congenital coronary artery fistulas (CAFs) are infrequent congenital coronary artery anomalies. Complications such as left-to-right shunt, congestive heart failure, myocardial infarction, pericardial effusion, aneurysm formation, rupture, hemopericardium, pulmonary hypertension, infective endocarditis (IE), syncope, stroke, and sudden death may occur with a variable low frequency. To describe the clinical characteristics of patients with CAFs complicated by IE. A search was conducted through PubMed using the terms “CAFs” and “IE.” Papers with a full description of the fistula character- istics and detailed data regarding bacterial endocarditis were included for evaluation. In the overall group of reviewed sub- jects (n 5 25, 9 females), the mean patient age was 42.5 years (range: 16 and 87). The right coronary artery (RCA) and left coronary artery (LCA) contributed equally to fistula formation. Terminations into the right heart side occurred in 19 (76%) fistulas. The majority of the fistulas (92%) were unilateral. The cultured microorganism was Streptococcus in 14 (56%) and Staphylococcus in 4 (16%) of the reviewed subjects. Echocardiographic single or multiple valvular regurgita- tion was found in 8 (32%) of the reviewed subjects. Small and large intracardiac vegetations were detected in 18 patients (72%). Antibiotic therapy was initiated in 20 (80%) subjects and 16 fistulas were treated surgically. During surgery, spon- taneous closure of the fistula was observed in one patient. Percutaneous therapeutic embolization (PTE) was successfully performed in two subjects. CAFs complicated by IE may affect all age groups with a slight male preponderance. Unilateral fistulas, either arising from the right or left coronary artery, are predominant, draining mainly into the right heart side. It is emphasized that antibiotic prophylaxis is strongly advised for pediatric and adult patients with congenital CAFs. Key Words. Congenital Coronary Artery Fistulas; Congenital Anomaly; Infective Endocarditis; Fistula Charac- teristics; Valvular Heart Disease; Surgical Closure Introduction CAFs patients with IE. Valvular15 and nonvalvu- lar4,9 infection and vegetation may occur. A litera- ongenital coronary artery fistulas (CAFs) are ture review describing the clinical characteristics associated with a risk of complications such as C of congenital CAFs complicated by IE in 25 infective endocarditis (IE), rupture, and sudden reported cases is presented. death. Congenital CAFs have been associated with 1 2 This review aimed to summarize the clinical IE in children and adults. The fistulas may origi- characteristics of patients and features of the CAFs nate from the right coronary artery (RCA) or left complicated by IE. coronary artery (LCA) and may terminate into any right or left cardiac chamber or any thoracic venous or arterial vessel. IE may involve unilateral3 as well Methods 4 as bilateral fistulas. The pathogenic microorgan- A search was conducted through PubMed using 5,6 isms may invade the semilunar or atrioventricu- the terms “CAFs” and “IE.” For a paper to be 3,7,8 lar valvular structures. included, it had to contain a full and detailed 4,9–12 Both Streptococci and to a lesser extent description of the fistula accompanied by data Staphylococci13,14 species have been isolated from regarding IE. This resulted in a total of 60 papers. VC 2016 Wiley Periodicals, Inc. Congenit Heart Dis. 2016;00:00–00 2 Said Twenty-four were not included because of irrele- was found in two, another eight were caused by vance and 15 were excluded due to incompleteness Streptococcus infection, HACEK was responsible for and a lack of detail. Reference lists were scrutinized the infection in one, by S. aureus in four and the for relevant publications, resulting in the identifica- microorganism was not reported in three. Echocar- tion of an additional two papers. Therefore, 23 diographic evaluation revealed single or multiple papers were eligible for analysis and evaluation. valvular involvement, including regurgitation in Other congenital coronary artery anomalies were eight of the reviewed subjects. Valvular vegetations excluded. The following items for evaluation of were demonstrated in 12 subjects (single n 5 9and coronary artery fistulas complicated by IE were multiple n 5 3 producing 15 valvular lesions) stipulated: age, gender, involved microorganism, including involvement of the aortic (AV n 5 4), diagnostic modalities (echocardiography, coronary mitral (MV n 5 7), tricuspid (TV n 5 3), and pul- angiography, and cardiac catheterization), fistula monary valve (PV n 5 1) (Table 2). characteristics (origin, termination, number of Among the reviewed subjects, 20 received anti- donor vessels “unilateral, bilateral or multilateral,” biotic therapy and 16 were treated surgically (6 and mode of termination [cameral “CCFs” vs. isolated and 10 combined with coronary artery vascular “CVFs”], details regarding IE (blood bypass grafting (n 5 4) and/or valvular correction culture when available, vegetation, valvular, and (n 5 10)). Six subjects received antibiotic therapy nonvalvular involvement) and surgical or nonsur- alone. gical management. Percutaneous therapeutic embolization (PTE) was successfully performed in two subjects. During Statistical Analysis surgery, spontaneous closure of the fistula by Values were expressed as means, averages, and thrombus and debris was observed in one patient. percentages. Aortic valve replacement was performed in four patients and four underwent mitral valve replace- Results ment. Furthermore, mitral valve repair in one and Overall, 25 (9 females and 16 males) reviewed sub- combined with tricuspid valve plasty in another jects were included in the analysis. The mean patient. patient age was 42.5 years (range: 16 and 87). Clini- cal presentations included fever (15 subjects), Laboratory Data: Echocardiographic/Angiographic/ fatigue (5 subjects), bacterial endocarditis (4 sub- Hemodynamic/Microbiologic/Septic Embolization and jects), congestive heart failure (2 subjects), weight Associated Coronary Artery Disease loss and nocturnal sweats (2 subjects each), hemopt- Echocardiographic Data ysis (1 subject), and stroke (1 subject) (Table 1). Valvular vegetations were demonstrated in 12 sub- There was an equal distribution of the RCA jects (single n 5 9 and multiple n 5 3valvular (n 5 14) and the LCA (n 5 13) in the fistula forma- lesions) including involvement of the aortic (AV tion. There were 23 (92%) unilateral fistulas, with n 5 4), mitral (MV n 5 7), tricuspid (TV n 5 3), bilateral fistulas being found in only two patients. and pulmonary valve (PV n 5 1). The size of vege- None of the subjects presented multilateral fistulas. tation was variable (mean 6.9–13 mm, range 3 3 6 Terminations into the left heart side were found in to 12 3 18 mm). Echocardiographic evaluation six patients and into the right heart side in 19 revealed single or multiple valvular involvement, (76%) patients. Regarding mode of termination, 14 including (mild, moderate, and severe) regurgita- patients (56%) had coronary-cameral fistulas and tion in eight of the reviewed subjects. None of the 11 possessed coronary-vascular fistulas (CVFs). reviewed subjects had preexistent significant valvu- The cultured microorganism was Streptococcus in 14 lar disease. subjects (viridans n 5 7, mitis n 5 1, pneumoniae n 5 1, and unspecified n 5 5), Staphylococcus aureus Angiographic Data: (Fistula Characteristics: in four, negative culture in two, HACEK (Haemo- Origin/Pathway/Termination/Diameter) philus, Aggregatibacter, Cardiobacterium, Eikenella, There was an equal distribution of the RCA and Kingella) in one and not reported in four of the (n 5 14) and LCA (n 13) in the fistula formation. reviewed subjects. The unilateral fistulas accounted for 92% of cases Small and large vegetations, which were con- (23 patients) and the bilateral fistulas were found in firmed echocardiographically and/or surgically, only two patients. None of the subjects presented occurred in 18 patients. Of those, sterile culture multilateral fistulas. Terminations into the left Congenit Heart Dis. 2016;00:00–00 Congenital Coronary Artery Fistulas and Infective Endocarditis Table 1. IE in Reviewed Subjects with Congenital Coronary Artery Fistulas Pathogenic Pharmacological and Case/Age/ Year/Reference/ Microorganism/ Vegetations/ Fistula Characteristics Nonpharmacological Gender Clinical Presentation Septic Embolization Involved Valves L-R Shunt Interventions 35F 1964(17)/intermittent fever, dysp- Streptococcus viridans None Cx ! PLSVC AB/SL nea, fatigue 31M 1992(41)/intermittent fever after Streptococcus viridans None RCA ! LV Dilated RCA AB (4 weeks) dental procedure 17F 1993(45)/intermittent fever, hemopt- Streptococcus viridans Vegetations on the edges of TV RCA ! CS Aneurysm RCA AB ysis/weight loss Septic pulmonary embolism 41M 1995(11)/fatigue Streptococcus viridans None RCA ! CS (1.4:1) AB/CABG/SL 72F 1995(11)/fever, nocturnal sweats, Streptococcus viridans None Aneurysm RCA AB/CABG/SL weight loss Cx ! CS (1.2:1) Aneurysm Cx 30M 1998(35)/nocturnal sweats Streptococcus viridans None RCA ! LV AB/SL 61M 1999(49)/fever, nocturnal sweats, Streptococcus Vegetation Cx ! CS (25%) AB general malaise 31M 2004(14)/fever, myalgias, neck stiff- Staphylococcus
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