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Palmquist-Gomes P, Ruiz-Villalba A, Guadix JA, Romero JP, Bessiéres B, MacGrogan D, Conejo L, Ortiz A, Picazo B, Houyel L, Gómez-Cabrero D, Meilhac SM, de la Pompa JL, Pérez-Pomares JM. Origin of congenital coronary arterio-ventricular fistulae from anomalous epicardial and myocardial development. Exp Mol Med 2023; 55:228-239. [PMID: 36653444 PMCID: PMC9898521 DOI: 10.1038/s12276-022-00913-x] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/27/2022] [Revised: 10/27/2022] [Accepted: 11/01/2022] [Indexed: 01/19/2023] Open
Abstract
Coronary Artery Fistulae (CAFs) are cardiac congenital anomalies consisting of an abnormal communication of a coronary artery with either a cardiac chamber or another cardiac vessel. In humans, these congenital anomalies can lead to complications such as myocardial hypertrophy, endocarditis, heart dilatation, and failure. Unfortunately, despite their clinical relevance, the aetiology of CAFs remains unknown. In this work, we have used two different species (mouse and avian embryos) to experimentally model CAFs morphogenesis. Both conditional Itga4 (alpha 4 integrin) epicardial deletion in mice and cryocauterisation of chick embryonic hearts disrupted epicardial development and ventricular wall growth, two essential events in coronary embryogenesis. Our results suggest that myocardial discontinuities in the embryonic ventricular wall promote the early contact of the endocardium with epicardial-derived coronary progenitors at the cardiac surface, leading to ventricular endocardial extrusion, precocious differentiation of coronary smooth muscle cells, and the formation of pouch-like aberrant coronary-like structures in direct connection with the ventricular lumen. The structure of these CAF-like anomalies was compared with histopathological data from a human CAF. Our results provide relevant information for the early diagnosis of these congenital anomalies and the molecular mechanisms that regulate their embryogenesis.
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Affiliation(s)
- P. Palmquist-Gomes
- grid.10215.370000 0001 2298 7828Department of Animal Biology, Faculty of Sciences, University of Málaga, 29071 Málaga, Spain ,grid.10215.370000 0001 2298 7828IBIMA-Plataforma BIONAND (Junta de Andalucía, Universidad de Málaga), 29590 Campanillas (Málaga), Málaga, Spain ,Université de Paris, Imagine-Institut Pasteur, Unit of Heart Morphogenesis, INSERM UMR1163, 75015 Paris, France
| | - A. Ruiz-Villalba
- grid.10215.370000 0001 2298 7828Department of Animal Biology, Faculty of Sciences, University of Málaga, 29071 Málaga, Spain ,grid.10215.370000 0001 2298 7828IBIMA-Plataforma BIONAND (Junta de Andalucía, Universidad de Málaga), 29590 Campanillas (Málaga), Málaga, Spain
| | - J. A. Guadix
- grid.10215.370000 0001 2298 7828Department of Animal Biology, Faculty of Sciences, University of Málaga, 29071 Málaga, Spain ,grid.10215.370000 0001 2298 7828IBIMA-Plataforma BIONAND (Junta de Andalucía, Universidad de Málaga), 29590 Campanillas (Málaga), Málaga, Spain
| | - J. P. Romero
- grid.5924.a0000000419370271Advanced Genomics Laboratory, Program of Hemato-Oncology, CIMA, University of Navarra, Pamplona, 31008 Spain
| | - B. Bessiéres
- Université de Paris, M3C-Necker Enfants malades, AP-HP, 75015 Paris, France
| | - D. MacGrogan
- grid.413448.e0000 0000 9314 1427Intercellular Signalling in Cardiovascular Development and Disease Laboratory, National Centre of Cardiovascular Research-Instituto de Salud Carlos III, 28029 Madrid, Spain ,grid.510932.cCIBER de Enfermedades Cardiovasculares, 28029 Madrid, Spain
| | - L. Conejo
- grid.411457.2Hospital Materno-Infantil de Málaga, Instituto Malagueño de Biomedicina (IBIMA), 29080 Málaga, Spain
| | - A. Ortiz
- grid.411457.2Hospital Materno-Infantil de Málaga, Instituto Malagueño de Biomedicina (IBIMA), 29080 Málaga, Spain
| | - B. Picazo
- grid.411457.2Hospital Materno-Infantil de Málaga, Instituto Malagueño de Biomedicina (IBIMA), 29080 Málaga, Spain
| | - L. Houyel
- Université de Paris, M3C-Necker Enfants malades, AP-HP, 75015 Paris, France
| | - D. Gómez-Cabrero
- Traslational Bioinformatics Unit, Navarrabiomed, Complejo Hospitalario de Navarra (CHN), Universidad Pública de Navarra (UPNA), IdiSNA, 31008 Pamplona, Spain ,grid.45672.320000 0001 1926 5090Biological and Environmental Sciences and Engineering Division, King Abdullah University of Science and Technology, 23955 Thuwal, Saudi Arabia
| | - S. M. Meilhac
- Université de Paris, Imagine-Institut Pasteur, Unit of Heart Morphogenesis, INSERM UMR1163, 75015 Paris, France
| | - J. L. de la Pompa
- grid.413448.e0000 0000 9314 1427Intercellular Signalling in Cardiovascular Development and Disease Laboratory, National Centre of Cardiovascular Research-Instituto de Salud Carlos III, 28029 Madrid, Spain ,grid.510932.cCIBER de Enfermedades Cardiovasculares, 28029 Madrid, Spain
| | - J. M. Pérez-Pomares
- grid.10215.370000 0001 2298 7828Department of Animal Biology, Faculty of Sciences, University of Málaga, 29071 Málaga, Spain ,grid.10215.370000 0001 2298 7828IBIMA-Plataforma BIONAND (Junta de Andalucía, Universidad de Málaga), 29590 Campanillas (Málaga), Málaga, Spain ,grid.510932.cCIBER de Enfermedades Cardiovasculares, 28029 Madrid, Spain
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Affiliation(s)
| | - Gary Randall Green
- Department of Cardiothoracic Surgery SUNY Upstate Medical University Syracuse New York
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3
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Coronary artery fistula: a review. Cardiovasc Pathol 2015; 24:141-8. [DOI: 10.1016/j.carpath.2014.01.010] [Citation(s) in RCA: 58] [Impact Index Per Article: 5.8] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 11/25/2013] [Revised: 01/31/2014] [Accepted: 01/31/2014] [Indexed: 11/19/2022] Open
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Said SAM, Schiphorst RHM, Derksen R, Wagenaar L. Coronary-cameral fistulas in adults (first of two parts). World J Cardiol 2013; 5:329-336. [PMID: 24109496 PMCID: PMC3783985 DOI: 10.4330/wjc.v5.i9.329] [Citation(s) in RCA: 15] [Impact Index Per Article: 1.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 05/24/2013] [Revised: 07/05/2013] [Accepted: 08/29/2013] [Indexed: 02/06/2023] Open
Abstract
This is a case series and review of the literature adding 11 new cases. Coronary-cameral fistulas (CCFs) are infrequent anomalies which are in general co-incidentally found during diagnostic coronary angiography (CAG). To delineate the characteristics of congenital and acquired CCFs in adults, we performed a PubMed search for papers dealing with congenital or acquired CCFs in adults. Publications on coronary-vascular fistulas or paediatric subjects were not included. From the world literature, a total of 243 adult patients were identified who had congenital (65%) or acquired (35%) CCFs. In this review, which is part one of a two-part series on CCFs, we describe and discuss the congenital fistulas, give an overview on the published literature and report details of our own series of 11 patients with MMFs and solitary macro CCFs. Of the congenital group, 85% were small or large solitary macro CCFs (cut-off 1.5 mm) and 15% were coronary artery-ventricular multiple micro-fistulas (MMFs). Apical hypertrophic cardiomyopathy was reported in some of the reviewed subjects with MMFs (3/24 = 13%) but not was seen in our own series. Conservative medical management was generally the treatment of choice in congenital MMFs; prophylactic implantable cardioverter defibrillators (ICD) were implanted in 2/24 (8%) of subjects, especially when extensive micro-fistulisations were involved. None of the patients of our own series required an ICD, as the MMFs were of limited size. Congenital or acquired CCFs in adults are infrequent anomalies having a wide spectrum of clinical presentation may varies from asymptomatic to severely devastating states requiring different treatment modalities.
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Lilli A, Chioccioli M, Del Meglio J, Menichetti F, Magnacca M, Tessa C, Salvatori L, Casolo G. Coronary microfistulae associated with non compacted myocardium: A rare cause of myocardial ischemia unraveled by a multimodality imaging approach. Int J Cardiol 2012; 159:e16-7. [DOI: 10.1016/j.ijcard.2011.11.009] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 11/03/2011] [Accepted: 11/04/2011] [Indexed: 10/28/2022]
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Caliskan K, Balk AHHM, Wykrzykowska JJ, van Geuns RJ, Serruys PW. How should I treat an unusual referral for heart transplantation? EUROINTERVENTION 2010; 5:861-5. [PMID: 20142204 DOI: 10.4244/eijv5i7a144] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 12/29/2022]
Abstract
BACKGROUND A 55 years old man was referred for cardiac transplantation because of intractable angina and fatigue. INVESTIGATION Physical examination, laboratory test, echocardiography, exercise ECG, MRI and coronary arteriography. DIAGNOSIS Multiple coronary artery fistulae. MANAGEMENT Beta-blockers, angiotensin-converting enzyme inhibitor ICD.
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7
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Weaver JC, McCrohon JA, Rees D, Prasan AM. Multivessel coronary-ventricular fistulae and ischaemia on cardiac MRI. Int J Cardiol 2010; 139:e36-8. [DOI: 10.1016/j.ijcard.2008.11.008] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 05/02/2008] [Accepted: 11/01/2008] [Indexed: 11/28/2022]
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Jang SN, Her SH, Do KR, Kim JS, Yoon HJ, Lee JM, Jin SW. A case of congenital bilateral coronary-to-right ventricle fistula coexisting with variant angina. Korean J Intern Med 2008; 23:216-8. [PMID: 19119260 PMCID: PMC2687685 DOI: 10.3904/kjim.2008.23.4.216] [Citation(s) in RCA: 6] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 02/08/2023] Open
Abstract
A coronary arteriovenous (AV) fistula consists of a communication between a coronary artery and a cardiac chamber, a great artery or the vena cava. It is the most common anomaly that can affect coronary perfusion. Yet bilateral involvement of a coronary fistula, constitutes an uncommon subgroup of coronary AV fistulas. We herein report on a case of bilateral coronary AV fistula that was coexistent with variant angina originating from the distal right ventricular branch of the right coronary artery and the distal septal branch of the left anterior descending artery, and the latter drained into the right ventricle.
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Affiliation(s)
- Se-Na Jang
- Department of Internal Medicine, The Catholic University of Korea College of Medicine, Seoul, Korea
| | - Sung-Ho Her
- Department of Internal Medicine, The Catholic University of Korea College of Medicine, Seoul, Korea
| | - Kyong-Rock Do
- Department of Internal Medicine, The Catholic University of Korea College of Medicine, Seoul, Korea
| | - Joon-Sung Kim
- Department of Internal Medicine, The Catholic University of Korea College of Medicine, Seoul, Korea
| | - Hee-Jeong Yoon
- Department of Internal Medicine, The Catholic University of Korea College of Medicine, Seoul, Korea
| | - Jong-Min Lee
- Department of Internal Medicine, The Catholic University of Korea College of Medicine, Seoul, Korea
| | - Seung-Won Jin
- Department of Internal Medicine, The Catholic University of Korea College of Medicine, Seoul, Korea
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9
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Abstract
In normal human hearts the left ventricle (LV) has up to 3 prominent trabeculations and is, thus, less trabeculated than the right ventricle. Rarely, more than 3 prominent trabeculations can be found at autopsy and by various imaging techniques in the LV. For this abnormality, different synonyms are used such as spongy myocardium, LV noncompaction, and LV hypertrabeculation (LVHT). In this review it is stated that: (1) LVHT has a higher prevalence than previously thought and the prevalence of LVHT seems to increase with the improvement of cardiac imaging; (2) because LVHT is most frequently diagnosed primarily by echocardiography, echocardiographers should be aware and trained to recognize this abnormality; (3) LVHT is frequently associated with other cardiac and extracardiac, particularly neuromuscular, disorders; (4) there are indications that the cause of LVHT is usually a genetic one and quite heterogeneous; and (5) controversies exist about diagnostic criteria, nomenclature, prognosis, origin, pathogenesis, and the necessity to classify LVHT as a distinct entity and cardiomyopathy by the World Health Organization.
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10
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Julius B, Igual M, Jiang Z, Turina M, Kiowski W, Hess O. Coronary steal phenomenon after repeated endomyocardial biopsies in cardiac transplant recipients? Radiography (Lond) 2000. [DOI: 10.1053/radi.2000.0273] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/11/2022]
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11
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Wolf A, Rockson SG. Myocardial ischemia and infarction due to multiple coronary-cameral fistulae: two case reports and review of the literature. CATHETERIZATION AND CARDIOVASCULAR DIAGNOSIS 1998; 43:179-83. [PMID: 9488552 DOI: 10.1002/(sici)1097-0304(199802)43:2<179::aid-ccd15>3.0.co;2-j] [Citation(s) in RCA: 22] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Subscribe] [Scholar Register] [Indexed: 02/06/2023]
Abstract
The functional significance of coronary-cameral fistulae, and the effect of these arterial anomalies upon effective coronary blood flow, continue to be debated. Two cases of coronary cameral fistulae, each of which illustrates the likelihood of an ischemic substrate, are herein presented, along with a review of the relevant literature regarding this disorder.
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Affiliation(s)
- A Wolf
- Division of Cardiology, Moffett Hospital, University of California, San Francisco, USA
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12
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Stierle U, Giannitsis E, Sheikhzadeh A, Potratz J. Myocardial ischemia in generalized coronary artery-left ventricular microfistulae. Int J Cardiol 1998; 63:47-52. [PMID: 9482144 DOI: 10.1016/s0167-5273(97)00280-5] [Citation(s) in RCA: 58] [Impact Index Per Article: 2.1] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 02/06/2023]
Abstract
Generalized (multiple) arterio-systemic fistulae are fistulae arising from all three major coronary arteries and drain into the left ventricle are rare and the clinical and hemodynamic sequelae are incompletely understood. This communication is based on the clinical and hemodynamic data of a series of patients (eight cases out of 7262 consecutive patients) incidentally identified at coronary angiography combined with data from cases previously reported in literature. The aim was to assess the role of generalized coronary artery fistulae as a non-atherosclerotic cause of myocardial ischemia by means of a coronary sinus lactate study. Coronary sinus lactate study demonstrated myocardial ischemia in 6/7 patients. Mean arterio-coronary venous lactate difference decreased from 0.31+/-0.18 mmol/l (lactate extraction ratio, LER, 29.4+/-13.9%) at rest to 0.04+/-0.13 mmol/l (LER -4.0+/-13.3%) at peak exercise. Five minutes after cessation of pacing, lactate difference increased to 0.22+/-0.21 mmol/l (LER -20.7+/- 13.2%). At peak pacing stress, 4/7 patients showed frank lactate production, and two patients presented with a reduced cardiac lactate extraction rate also indicating myocardial ischemia metabolically. In the present study, we demonstrated a possible role of a coronary steal mechanism due to microfistulae pathways in the pathogenesis of myocardial ischemia in patients with generalized coronary artery-left ventricular microfistulae.
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Affiliation(s)
- U Stierle
- Curschmann Klinik, Department of Cardiology (Internal Medicine II) Medical University, Lübeck, Germany
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13
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Nawa S, Miyachi Y, Toshino N, Shiba T, Hayashi K, Tamesue K, Yamamoto H, Shimizu N. Three major coronary artery-to-left ventricular shunts: report of three cases and review of literature. Cardiovasc Intervent Radiol 1997; 20:300-4. [PMID: 9211778 DOI: 10.1007/s002709900155] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 02/04/2023]
Abstract
Among the congenital coronary artery fistulas, diffuse fistulation into the left ventricular chamber, usually expressed in terms of a coronary artery-left ventricular shunt, is not as rare today as was previously thought. However, the origin of such a shunt from all three major coronary arteries is rare. This paper reports three cases of such an occurrence and presents the clinical features and management of this rare anomaly by analyzing 31 cases, including 28 from the literature.
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Affiliation(s)
- S Nawa
- Division of Surgery, Okayama Red Cross General Hospital, 65-1 Aoe, Okayama 700, Japan
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14
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Steiner I, Hrubecký J, Pleskot J, Koks̆tejn Z. Persistence of spongy myocardium with embryonic blood supply in an adult. Cardiovasc Pathol 1996; 5:47-53. [DOI: 10.1016/1054-8807(95)00058-5] [Citation(s) in RCA: 14] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 09/26/1994] [Accepted: 06/14/1995] [Indexed: 10/27/2022] Open
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15
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Abstract
A medico-legal autopsy conducted on a young Chinese man, a victim of sudden unexpected death, with a vague and unconfirmed history of congenital cardiovascular disease, revealed the presence of a congenital right coronary artery (arteriosystemic) fistula communicating with the markedly hypertrophied left ventricle, which showed microscopic evidence of myocardial ischaemia. The pathology of this rare condition is described and briefly reviewed.
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Affiliation(s)
- G Lau
- Department of Forensic Medicine, Institute of Science and Forensic Medicine, Singapore
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16
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Bartorelli AL, Pepi M, Sganzerla P, Montorsi P, Loaldi A, Ravagnani P, Fabbiocchi F. Syncope with cardiac arrest as the first manifestation of two congenital left coronary artery-to-main pulmonary artery fistulae. Am Heart J 1994; 127:207-9. [PMID: 8273742 DOI: 10.1016/0002-8703(94)90527-4] [Citation(s) in RCA: 6] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 01/29/2023]
Affiliation(s)
- A L Bartorelli
- Istituto di Cardiologia, Università degli Studi, Fondazione I. Monzino, IRCCS, Milan, Italy
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17
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Kinugawa T, Kazuhide O, Sawada Y, Hoshio A, Miyakoda H, Kotake H, Mashiba H, Kasahara T, Endo S. Case report: a case of multiple coronary artery to left ventricular communications. Am J Med Sci 1993; 306:306-8. [PMID: 8238085 DOI: 10.1097/00000441-199311000-00006] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 01/29/2023]
Abstract
A 39-year-old man with anginal pain had multiple coronary artery to left ventricular communications. His electrocardiogram showed evidence of left ventricular hypertrophy, and an echocardiogram revealed a dilated left ventricle. A coronary angiogram revealed multiple coronary artery to left ventricular fistulae involving three major coronary arteries with no evidence of atherosclerotic lesions. Only 17 cases of such fistulous communications involving three major coronary arteries have been reported in the literature. It is suggested that the fistulous communications to the left ventricle was a cause of his angina pectoris, probably because of the coronary steal phenomenon.
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Affiliation(s)
- T Kinugawa
- First Department of Internal Medicine, Tottori University, Yonago, Japan
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18
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Kawakami K, Shimada T, Yamada S, Murakami R, Morioka S, Moriyama K. The detection of myocardial ischemia by thallium-201 myocardial scintigraphy in patients with multiple coronary arterioventricular connections. Clin Cardiol 1991; 14:975-80. [PMID: 1841023 DOI: 10.1002/clc.4960141208] [Citation(s) in RCA: 6] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 12/29/2022] Open
Abstract
The question of whether myocardial ischemia could be induced in 5 patients with multiple coronary arterioventricular connections by thallium-201 (201Tl) exercise stress myocardial scintigraphy was investigated. Both ST-T changes on ECG and transient myocardial perfusion defects in myocardial scintigrams were observed in 2 patients (40%). In previous reports, all multiple coronary arterioventricular connections, which were shown in angiograms, have been regarded as either Thebesian veins or embryonic sinusoids. However, it is unlikely that Thebesian veins cause myocardial ischemia judging from anatomy. If the vessels cause myocardial ischemia, they should be regarded as multiple coronary arterioventricular fistula. Angiography itself cannot differentiate multiple coronary arterioventricular fistula from Thebesian veins or remnants of embryonic sinusoids. Exercise stress myocardial scintigraphy has a high potential to detect myocardial ischemia due to intracoronary steal. Exercise stress myocardial scintigraphy was used to demonstrate myocardial ischemia in multiple coronary arterioventricular connections. It is concluded that exercise stress myocardial scintigraphy is a reliable test to differentiate multiple coronary arterioventricular fistula from Thebesian veins or remnants of embryonic sinusoids in clinical practice.
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Affiliation(s)
- K Kawakami
- Fourth Department of Internal Medicine, Shimane Medical University, Izumo, Japan
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19
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Black IW, Loo CK, Allan RM. Multiple coronary artery-left ventricular fistulae: clinical, angiographic, and pathologic findings. CATHETERIZATION AND CARDIOVASCULAR DIAGNOSIS 1991; 23:133-5. [PMID: 2070401 DOI: 10.1002/ccd.1810230216] [Citation(s) in RCA: 29] [Impact Index Per Article: 0.9] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Subscribe] [Scholar Register] [Indexed: 12/30/2022]
Abstract
Multiple fistulae between all 3 major coronary arteries and the left ventricle were found at cardiac catheterization in a 69-year-old woman presenting with typical angina. Subsequent necropsy showed abnormal intramyocardial vessels communicating with the left ventricular lumen. This is the first report to document clinical, angiographic, and pathologic findings in this anomaly.
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Affiliation(s)
- I W Black
- Department of Cardiovascular Medicine, Prince Henry Hospital, Little Bay, Sydney, Australia
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20
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Doorey AJ, Sullivan KL, Levin DC. Successful percutaneous closure of a complex coronary-to-pulmonary artery fistula using a detachable balloon: benefits of intra-procedural physiologic and angiographic assessment. CATHETERIZATION AND CARDIOVASCULAR DIAGNOSIS 1991; 23:23-7. [PMID: 1863956 DOI: 10.1002/ccd.1810230107] [Citation(s) in RCA: 10] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Subscribe] [Scholar Register] [Indexed: 12/29/2022]
Abstract
A 48-yr-old woman presented with a loud continuous precordial murmur and symptoms of fatigue. Color-flow doppler imaging and nuclear magnetic resonance imaging failed to show the cause of the murmur. Diagnostic catheterization showed a large left anterior descending coronary artery to pulmonary artery fistula with impaired left ventricular wall motion. Two detachable balloons were deployed in the fistula with complete abolition of flow in the main fistula channel. A small parallel channel of the fistula, previously not appreciated due to vessel overlap, remained patent but had trivial flow as assessed by green-dye and oximetric techniques. The patient had immediate resolution of her symptoms and return of normal ventricular wall motion.
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Affiliation(s)
- A J Doorey
- Department of Medicine, Jefferson Medical College, Thomas Jefferson University, Philadelphia, Pennsylvania
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21
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Oshiro K, Shimabukuro M, Nakada Y, Chibana T, Yoshida H, Nagamine F, Sunagawa R, Gushiken M, Murakami K, Mimura G. Multiple coronary LV fistulas: demonstration of coronary steal phenomenon by stress thallium scintigraphy and exercise hemodynamics. Am Heart J 1990; 120:217-9. [PMID: 2360509 DOI: 10.1016/0002-8703(90)90185-z] [Citation(s) in RCA: 26] [Impact Index Per Article: 0.7] [Reference Citation Analysis] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 12/31/2022]
Affiliation(s)
- K Oshiro
- Second Department of Internal Medicine, School of Medicine, Faculty of Medicine, University of the Ryukyus, Okinawa, Japan
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Abstract
In a fetus with ventricular extrasystoles a congenital aneurysm of the left ventricle was diagnosed prenatally. At 32 weeks of gestation, echocardiography showed a large apical left ventricular aneurysm with a thin, hypokinetic wall. Congestive heart failure did not occur. Prenatal and postnatal examinations did not detect the aetiology of the aneurysm, but excluded the majority of possible causes. The 2-year-old child is now asymptomatic and normally developed. Neither medication nor surgical treatment have been necessary, except for antithrombotic prophylaxis with low-dose aspirin.
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Affiliation(s)
- U Gembruch
- Department of Prenatal Diagnosis and Therapy, University of Bonn, F.R.G
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23
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Fitchett DH, Forbes C, Guerraty AJ. Repeated endomyocardial biopsy causing coronary arterial-right ventricular fistula after cardiac transplantation. Am J Cardiol 1988; 62:829-31. [PMID: 3048074 DOI: 10.1016/0002-9149(88)91237-4] [Citation(s) in RCA: 18] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 02/06/2023]
Affiliation(s)
- D H Fitchett
- Department of Medicine, Royal Victoria Hospital, McGill University, Montreal, Canada
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24
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Duckworth F, Mukharji J, Vetrovec GW. Diffuse coronary artery to left ventricular communications: an unusual cause of demonstrable ischemia. CATHETERIZATION AND CARDIOVASCULAR DIAGNOSIS 1987; 13:133-7. [PMID: 3581166 DOI: 10.1002/ccd.1810130212] [Citation(s) in RCA: 17] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Subscribe] [Scholar Register] [Indexed: 01/06/2023]
Abstract
Diffuse communications between both the left and right coronary arteries and the left ventricle were found in a 46-year-old man presenting with typical angina pectoris. Symptoms were reproducible on treadmill exercise and ST segment depression, and redistribution septal defects were documented on stress Thallium scintigraphy. Antianginal drugs were effective in treating the patient's symptoms. Only 13 patients with similar anatomy have been previously described. This report is the first to document reproducible objective evidence of ischemia in such patients. The literature is reviewed and possible mechanisms of ischemia and its treatment discussed.
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Case records of the Massachusetts General Hospital. Weekly clinicopathological exercises. Case 49-1986. A 41-year-old woman with atrial tachyarrhythmias and a continuous cardiac murmur. N Engl J Med 1986; 315:1533-43. [PMID: 3785309 DOI: 10.1056/nejm198612113152407] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 01/07/2023]
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Origuchi H, Shimokawa H, Sugihara M, Sagara T, Kikuchi Y. Demonstration of exercise-induced myocardial ischemia in a case of multiple coronary arterioventricular fistulas. Am Heart J 1986; 112:1096-7. [PMID: 3776808 DOI: 10.1016/0002-8703(86)90329-7] [Citation(s) in RCA: 4] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 01/07/2023]
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Knippel M, Ravizza P, Gullace G, Bana G, Savoia M, Locatelli V, Colombi P. An unusual case of congenital double coronary arteriovenous fistula. Chest 1982; 81:382-4. [PMID: 7056118 DOI: 10.1378/chest.81.3.382] [Citation(s) in RCA: 5] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 01/23/2023] Open
Abstract
A double coronary arteriovenous fistula (CAVF) was observed in a ten-year-old girl. A large, tortuous, and elongated right coronary artery, communicating with the right ventricle, was detected by two-dimensional echocardiography and confirmed at angiography. To our knowledge, no echocardiographic recognition of the origin and termination of a CAVF has been reported previously. Moreover, another fistula, connecting the left anterior descending coronary artery with the main pulmonary artery was also detected by selective left coronary arteriography. The anatomy of double CAVF was confirmed on surgery. A right CAVF draining into the right ventricle and a left CAVF draining into the main pulmonary artery seemed to be a very unusual combination.
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Abstract
Congenital fistulas are the most common of the coronary arterial malformations and with the widespread use of selective coronary arteriography are being recognized with increasing frequency. Twenty-eight patients with congenital coronary fistulas have been evaluated at the Duke University Medical Center between 1960 and 1981. An additional 258 patients have previously been reported in the literature, making a total of 286 available for review. The right coronary artery is most commonly involved, and the fistulous communication is most often to the right ventricle, right atrium or pulmonary artery. Slightly more than half of the patients with coronary fistulas are symptomatic at the time the diagnosis is made. Surgical correction is strongly recommended to prevent the development of congestive heart failure, angina, subacute bacterial endocarditis, myocardial infarction, and pulmonary hypertension, as well as coronary aneurysm formation, with subsequent rupture or embolization. There were no operative or late deaths in the patients who underwent operations. Moreover, there have been no recurrent fistulas during a mean follow-up period of ten years. The risks of operative correction appear to be considerably less than the potential for development of serious and potentially fatal complications, even in asymptomatic patients.
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Patel RG, Freedom RM, Moes CA, Bloom KR, Olley PM, Williams WG, Trusler GA, Rowe RD. Right ventricular volume determinations in 18 patients with pulmonary atresia and intact ventricular septum. Analysis of factors influencing right ventricular growth. Circulation 1980; 61:428-40. [PMID: 7351069 DOI: 10.1161/01.cir.61.2.428] [Citation(s) in RCA: 80] [Impact Index Per Article: 1.8] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 01/24/2023]
Abstract
Right ventricular growth was assessed angiocardiographically in 18 patients with pulmonary atresia, intact ventricular septum, and hypoplastic and hypertensive right ventricle. A variety of surgical procedures were performed. In only 12 patients (66.7%) was right ventricular-pulmonary artery continuity achieved (group 1). Nine of these 12 patients persisted with systemic or suprasystemic right ventricular pressures. Among the six patients in whom right ventricular-pulmonary artery continuity was not achieved (group 2), all maintained suprasystemic right ventricular pressures. Right ventricular growth was assessed in groups 1 and 2. The patients were also subdivided according to the qualitative degree of tricuspid regurgitation as determined angiocardiographically on right ventricular cineangiocardiograms at the preoperative catheter study. Right ventricular growth to normal levels as evidenced by change in right ventricular end-diastolic volume was rarely observed in group 2 patients. Among the four patients with severe tricuspid regurgitation and a large tricuspid valve, right ventricular growth to normal levels was achieved whether they were in group 1 or group 2. Right ventricular growth is thus predicated on numerous morphologic factors in these patients. However, reconstitution of right ventricular-pulmonary artery continuity and a nonobstructive tricuspid valve are probably two of the more important factors.
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Liberthson RR, Sagar K, Berkoben JP, Weintraub RM, Levine FH. Congenital coronary arteriovenous fistula. Report of 13 patients, review of the literature and delineation of management. Circulation 1979; 59:849-54. [PMID: 428095 DOI: 10.1161/01.cir.59.5.849] [Citation(s) in RCA: 440] [Impact Index Per Article: 9.6] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 12/15/2022]
Abstract
Thirteen new patients and 174 patients previously reported with coronary arteriovenous fistula (CAVF) were reviewed to delineate the course and management of CAVF and to clarify the role of surgical ligation in the young asymptomatic patient. Patients were grouped according to age: 99 patients (four new and 95 reported) were less than 20 years old and 88 (nine new and 79 reported) were greater than or equal to 20 years old. Of those under 20 years of age, 19% had preoperative symptoms or CAVF-related complications, including congestive heart failure (CHF) in 6%, subacute bacterial endocarditis in 3% and death in one patient. Seventy-six patients younger than 20 years old had CAVF ligation with only one significant complication. In contrast, 63% of the older group and all of our nine older patients had preoperative symptoms or complications, including CHF in 19%, SBE in 4%, myocardial infarction (MI) in 9%, death in 14% and fistula rupture in one patient. Of the 43 ligated older patients, 23% had surgical complications, including MI in three and death in three. Mean pulmonic-to-systemic flow in the entire group was 1.6:1 and did not differ significantly in those with or without symptoms or complications. One of our patients and one previously reported had spontaneous CAVF closure. In summary, early elective ligation of CAVF is indicated in all patients because of the high incidence of late symptoms and complications and the increased morbidity and mortality associated with ligation in older patients.
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Vogelbach KH, Edmiston WA, Stenson RE. Coronary artery-left ventricular communications: a report of two cases and review of the literature. CATHETERIZATION AND CARDIOVASCULAR DIAGNOSIS 1979; 5:159-67. [PMID: 487420 DOI: 10.1002/ccd.1810050210] [Citation(s) in RCA: 16] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Subscribe] [Scholar Register] [Indexed: 12/15/2022]
Abstract
Two patients with chest pain had angiographically-demonstrated communications between the three coronary arteries and the left ventricular chamber. Communications between coronary arteries and the left ventricle are unusual and communications between all three coronary arteries and the left ventricle are rare. These anomalies are, however, commonly associated with symptoms of chest pain. The presence of left ventricular hypertrophy and a widened pulse pressure may suggest a greater hemodynamic effect of the shunt flow than often suspected angiographically.
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