J Korean Soc Echocardiogr.  1994 Jul;2(1):96-103. 10.4250/jkse.1994.2.1.96.

A Case of Bland-White-Garland Syndrome

Affiliations
  • 1Department of Internal Medicine, Han Yang University, Seoul, Korea.

Abstract

The congenital anomalous origin of the left coronary artery arising from the pulmonary artery, or the Bland-White-Garland syndrome, is uncommon but frequently lethal lesion of both children and adults. In several series, it has a frequency of 0.26-0.46% of all congenital cardiac defects. The mortality rate among infants and children without operation has been eighty to ninety percent. Survival to teen-age and adult has been infrequent ; review of the literature regarding this anomaly in Korean disclosed only 3 cases in infants and children and 2 cases in adults. In a 45-year-old male with palpitation and effort angina, the anomalous origin of the left coronary artery from the pulmonary artery was diagnosed by echocardiogram and coronary arteriography.


MeSH Terms

Adult
Angiography
Bland White Garland Syndrome*
Child
Coronary Vessels
Humans
Infant
Male
Middle Aged
Mortality
Pulmonary Artery

Figure

  • Fig. 1. Electrocardiography shows atrial fibrillation and ST change in lead I. aVL.

  • Fig. 2. Chest X ray shows cardiomegaly.

  • Fig. 3. Treadmil test; (a) resting, (b) shows ST change in Bruce protocol stage II.

  • Fig. 4. Short axis view at base; anomalous origin of left coronary artery from pulmonary artery(RVOT: right ventricular outflow tract. AO: aorta. FV: pulmonary valve).

  • Fig. 5. Short axis view.at base; Color Doppler shows reversed flow from left coronary artery to pulmonary artery(RVOT: right ventricular outflow tract. AO: aorta. PV: pulmonary valve. Arrow: toward flow)

  • Fig. 6. Short axis view at base; Enlarged right coronary artery(RVOT: right ventricular outflow tract, AO: aorta, LA: left atrium).

  • Fig. 7. Parasternal long axis view; Color Doppler showing diastolic flow in right coronary artery(Arrow; toward flow).

  • Fig. 8. Parasternal long axis view. Large right coronary artery(RV: right ventricle. LV: left ventricle, AO: aorta. AOV: aortic valve. LA: left atrium.

  • Fig. 9. Right coronary angiogram in shallow LAO view; enlarged, tortous coronary A.


Reference

References

1). Ogden JA. Congenital anomalies of the coronary arteries. Am J Cardiol. 25:474. 1970.
Article
2). Abott ME. Congenital Heart disease. Osler's Modern Medicine. IV. New York: Lea and Febiger;1908. ch 9.
3). Bland EF, White PD, Garland J. Congenital anomalies of coronary arteries: report of an unusual case associated with cardiac hypertrophy. Am Heart. 8:787. 1933.
4). Wesselhoeft H, Fawcett JS, Johnson AL. Anomalous origin of the left coronary artery from the pulmonary trunk. Circulation. 38:403–425. 1968.
Article
5). Modie DS, Fyfe D, Gill CC, Cook SA, Lytle BW, Tylor PC, et al. Anomalous origin of the left coronary artery from the pulmonary artery (Bland-White-Garland syndrome) in adult patients: Long-term follow-up after surgery. Am Heart J. 106:381–388. 1983.
6). Gutgesell HP, Pinsky WW, DePuey EG. Thallium-201 myocardial perfusion imageing in infants and children: Value in distingushing anomalous left coronary artery from congestive cardiomyopathy. Circulation. 38:403–425. 1968.
7). Kakou Guikahue M, Sidi D, Kachaner J, Villaine E, Cohen L, Piechaud JF, et al. Anomalous left coronary artery arising from the pulmonary artery in infancy: Is early operation better ? Br Heart J. 60:522–526. 1988.
8). Askenazi J, Nadas AS. Anomalous left coronary arteries originating from the pulmonary artery: report on 15 cases. Circulation. 51:976. 1975.
9). Nora JJ, and McNamara DG. Anomalies of the coronary arteries and coronary artery fistular. Waston H, editor. Pediatric cardiology. The C.V. Mosby Co.;1968. p. 295.
10). ��� ��: � ¥�££��oJ­. 27(3):277–281. 1985.
11). ·�·��¥����°°§. 20(1):85–88. 1984.
12). ��������£���. 7(1):107–114. 1990.
13). Brooks H. Two cases of the abnormal coronary artery of the heart arising from the pulmonary artery, J Anat Physiol. 20:26. 1886.
14). Wilson CL, Dlabal PW, Holeyfield RW, Akins CW, Knauf DG. Anomalous origin of left coronary artery from the pulmonary artery: Case report and review of literature concerning teenagers and adults. J Thorac Cardiovasc Surg. 73:887–893. 1977.
15). Ogden J. The origin of the coronary arteries. Circulation. 38:150. 1968; (Suppl. 6).
16). Edwards JE. Direction of blood flow in coronary arteries arising from the pulmonary trunk. Circulation. 29:163. 1974.
17). �·��: Bland-White-Garland ���������. 10(2):1993.
Full Text Links
  • JKSE
Actions
Cited
CITED
export Copy
Close
Share
  • Twitter
  • Facebook
Similar articles
Copyright © 2024 by Korean Association of Medical Journal Editors. All rights reserved.     E-mail: koreamed@kamje.or.kr