Bland-White-Garland Syndrome: Not just a Pediatric Coronary Anomaly?
| |
|
Case Report. A 66-year-old female presented with new-onset angina and heart failure. Electrocardiography demonstrated chronic atria fibrillation and left axis deviation. Echocardiography demonstrated mild global left ventricular hypokinesia without regional wall motion abnormalities, severe left
|  | | Figure 1. Right coronary angiography demonstrating collateral circulation to the left coronary system that arises from the pulmonary artery. |
atrial enlargement, mild mitral regurgitation and mild pulmonary hypertension. Adenosine sestamibi demonstrated a reversible medium-sized anterior and anterolateral perfusion defect. At right heart catheterization, there was mild pulmonary hypertension (systolic: 32 mmHg, diastolic: 11 mmHg, mean 22 mmHg). Coronary angiography revealed a large right-dominant coronary artery that filled the entire left coronary circulation via collaterals. The left main coronary artery arose from the pulmonary trunk (Figure 1). The patient was referred for coronary artery bypass surgery, but has deferred undergoing surgical intervention.
The Bland, White and Garland syndrome is a rare congenital coronary anomaly that accounts for approximately 0.5% of congenital coronary abnormalities1 and was first described at autopsy in 1933 in a 3.5-month-old boy who presented with attacks of dyspnea, pallor and profuse sweating.2 Survival into late adulthood is extremely rare and probably depends on the patient’s ability to rapidly develop a collateral arterial circulation.3–9 Surgical reimplantation of the left coronary artery into the ascending aorta is the preferred treatment.10,11 |
References
1. Perloff J. Anomalous origin of the left coronary artery from the pulmonary trunk. In: The Clinical Recognition of Congenital Heart Disease (4th edition) JK P (ed). . Philadelphia: WB Saunders Co, 1994, pp. 546–561.
2. Bland E, White P, Garland J. Congenital anomalies of the coronary arteries: Report of an unusual case associated with cardiac hypertrophy. Am Heart J 1933;8:787–801.
3. Wesselhoeft H, Fawcett J, Johnson A. Anomalous origin of the left coronary artery from the pulmonary trunk. Its clinical spectrum, pathology and pathophysiology based on a review of 140 cases with seven further cases. Circulation 1968;38:403.
4. Purut C, Sabiston D. Origin of the left coronary artery from the pulmonary artery in older adults. J Thorac Cardiovasc Surg 1991;102:566.
5. Mascioli G, Turelli A, Niccoli L, et al. [Anomalous origin of the left coronary artery from the pulmonary artery. A rare case diagnosed in adulthood.] G Ital Cardiol 1993;23:87–93.
6. Arsan S, Naseri E, Keser N. An adult case of Bland White Garland syndrome with huge right coronary aneurysm. Ann Thorac Surg 1999;68:1832.
7. Nightingale AK, Burrell CJ, Marshall AJ. Anomalous origin of the left coronary artery from the pulmonary artery: Natural history and normal pregnancies. Heart 1998;80:629–631.
8. Maeder M, Vogt PR, Ammann P, et al. Bland-White-Garland syndrome in a 39-year-old mother. Ann Thorac Surg 2004;78:1451.
9. Barbetakis N, Efstathiou A, Efstathiou N, et al. A long-term survivor of Bland-White-Garland syndrome with systemic collateral supply: A case report and review of the literature. BMC Surg 2005;5:23.
10. Backer C, Stout M, Zales V. Anomalous origin of the left coronary artery. A twenty-year review of surgical management. J Thorac Cardiovasc Surg 1992;103:1049–1058.
11. Alexi-Meskishvili V BF, Weng Y, Lange PE, Hetzer R. Anomalous origin of the left coronary artery from the pulmonary artery in adults. J Card Surg 1995;10(4 Pt 1):309–315.
|
| The Journal of Invasive Cardiology - ISSN: 1042-3931 - Volume 19 - Issue 1 - January 2007 - Pages: E9 - E9 | |
|
 Create a Successful Vena Cava Filter Practice
Accredited CD
This activity is supported by an educational grant from Cook Incorporated and has been designed for Interventional Cardiologists, Vascular Surgeons, Fellows and Interventional Cardiovascular Nurses and Technologists.
|
| |
|
|
|
|
 Achieving Optimal Outcomes in Carotid Stenting: Lessons Learned from Recent Clinical Trials
Complimentary Accredited ON DEMAND Webcast
Topics
1. EVA-3S & Space-Bumps in the road
2. CAPTURE 3500-Lesion morphology & Predictors for Stroke
3. CAPTURE II vs. EXACT 1500-Does open or Closed Cell Stent design really matter?
This activity has been developed for Interventional Cardiologists, Vascular Surgeons, Interventional Radiologists, Neurologists, Interventional Nurses and Technologists with an interest in the diagnosis and treatment of peripheral artery disease. |
|
 Anticoagulation Techniques for Peripheral Vascular Interventions
Complimentary Accredited ON DEMAND Webcast
This activity has been developed for Interventional Cardiologists, Vascular Surgeons, Interventional Radiologists, Podiatric Physicians, Endovascular Allied Professionals, Endocrinologists, Wound Care Specialists, Directors of the Wound Care Clinic, and Primary Care Physicians, Pharmacists, Nurses and Technologists.
|
|
|
|
|
|
March 2007 Supplement
|
On-Demand Webcast
|
Archived Webcast
|
|
|
|
|
|
© 2008 HMP Communications | All Rights Reserved
83 General Warren Blvd, Suite 100 | Malvern, PA 19355 | 800.237.7285
|
|
| | |
Presented By: 
Release Date: December 8, 2006