Wilcken B, Turner G
Arch Dis Child. 1978 Mar;53(3):242-5. doi: 10.1136/adc.53.3.242.
Homocystinuria was studied in 27 patients from 15 families in New South Wales. All 2 had biochemical findings consistent with cystathionine synthetase deficiency. One patient was ascertained by newborn screening, but the remaining index cases were detected because of symptoms: poor eyesight 6, mental retardation 3, thromboses 2, skeletal abnormalities 2, and urinary infection1. 9 patients, one-third of all cases, were mildly affected: either they had no features of the disease, or these did not occur until the late teens. Pyridoxine responsiveness was found in 8 sibships, and clinically there were two distinct kinds of response. For patients born in the decade 1960-69 the ascertainment rate for the total population was 1:58 000. The true incidence must be much higher. Our series indicates that homocystinuria occurs more frequently than has heretofore been thought, and that mild cases are common. It is likely that cases are often missed in current newborn screening programmes.
对新南威尔士州15个家庭的27名患者进行了高胱氨酸尿症研究。所有27名患者均有与胱硫醚合成酶缺乏相符的生化检查结果。1例患者通过新生儿筛查确诊,其余索引病例因出现以下症状而被发现:视力差6例、智力发育迟缓3例、血栓形成2例、骨骼异常2例、泌尿系统感染1例。9例患者(占所有病例的三分之一)症状较轻:要么没有该病的特征,要么直到青少年晚期才出现这些特征。在8个同胞组中发现了维生素B6反应性,临床上有两种不同的反应类型。对于1960 - 1969年这十年间出生的患者,总人口中的确诊率为1:58000。实际发病率肯定要高得多。我们的系列研究表明,高胱氨酸尿症的发生频率比以往认为的要高,而且轻症病例很常见。在当前的新生儿筛查项目中,病例很可能经常被漏诊。