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High‐resolution chromosome analysis showed the karyotype 46,XX,del(1)(p13.3 p22.3) in a female infant with an extreme tetralogy of Fallot and multiple congenital anomalies. The patient showed characteristic features: upper and lower eyelids connected to each other by a string‐like epithelium, low hairline, epicanthal folds, saddle nose with a broad, flat root, micrognathia, short neck, high‐arched palate, prominent xiphisternum, wide‐spaced nipples, bilateral pes equinovarus, fifth toes that overlapped the fourth toes bilaterally, a deep fissure between the first and second toes bilaterally, and abnormal flexions of fingers and toes. Growth and psychomotor retardation were also noted. Cardiac catheterization revealed an extreme tetralogy of Fallot complicated by a patent ductus arteriosus. Ventricular tachycardia and ventricular premature beats developed during the neonatal period and did not respond well to anti‐arrhythmic drugs. She died of the anoxia caused by closure of the patent ductus arteriosus when she was 7 months old.

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Dipyridamole has been widely used in Japan to treat patients with a coronary aneurysm resulting from Kawasaki disease, but its effect in these patients has not been established. In the present study we assessed the effect of dipyridamole on the coronary arteries of patients with a history of Kawasaki disease by measuring the diameter of the coronary arteries and by quantifying the disappearance time of contrast medium (runoff time) on coronary angiography. Intravenous injection of dipyridamole increased the diameter of nondilated arteries by 7.9%. Its effect on the diameter of dilated coronary arteries (coronary aneurysm) was less than 3% (p < 0.01). Runoff time of dilated coronary arteries was significantly (p < 0.01) greater than that of nondilated coronary arteries. Dipyridamole accelerated runoff time not only in nondilated coronary arteries (p < 0.01) but also in coronary arteries with various degrees of dilatation (p < 0.01). We conclude that dipyridamole increases blood flow in coronary arteries without dilating the proximal aneurysm in children with a history of Kawasaki disease.

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