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人类的苯丙酮尿症基因座位于12号染色体上。

The PKU locus in man is on chromosome 12.

作者信息

Lidksy A S, Robson K J, Thirumalachary C, Barker P E, Ruddle F H, Woo S L

出版信息

Am J Hum Genet. 1984 May;36(3):527-33.

Abstract

Classical phenylketonuria (PKU) is a typical example of inborn errors in metabolism and is characterized by a complete lack of the hepatic enzyme phenylalanine hydroxylase, which normally converts phenylalanine to tyrosine. The genetic disorder causes impairment of postnatal brain development, resulting in severe mental retardation in untreated children. The disease is transmitted as an autosomal recessive trait and has a collective prevalence of about one in 10,000 among Caucasians, so that 2% of the population are carriers of the PKU trait. We have recently reported the cloning of human phenylalanine hydroxylase cDNA and that the human chromosomal phenylalanine hydroxylase gene is encoded by a unique DNA sequence. Using the human phenylalanine hydroxylase cDNA clone to analyze a clonal human/mouse hybrid cell panel by Southern hybridization, the phenylalanine hydroxylase gene has been assigned to human chromosome 12. Since the hypothesis that classical PKU is caused by structural mutations in the phenylalanine hydroxylase gene itself rather than through some transregulatory mechanisms has recently been confirmed by gene mapping, the PKU locus in man is determined to be on chromosome 12.

摘要

经典型苯丙酮尿症(PKU)是先天性代谢缺陷的典型例子,其特征是肝脏中完全缺乏苯丙氨酸羟化酶,该酶通常将苯丙氨酸转化为酪氨酸。这种遗传性疾病会损害出生后脑的发育,导致未经治疗的儿童出现严重智力迟钝。该疾病以常染色体隐性性状遗传,在白种人中的总体患病率约为万分之一,因此2%的人群是PKU性状的携带者。我们最近报道了人类苯丙氨酸羟化酶cDNA的克隆,并且人类染色体苯丙氨酸羟化酶基因由独特的DNA序列编码。利用人类苯丙氨酸羟化酶cDNA克隆通过Southern杂交分析克隆的人/鼠杂交细胞系,已将苯丙氨酸羟化酶基因定位于人类第12号染色体。由于最近通过基因定位证实经典型PKU是由苯丙氨酸羟化酶基因本身的结构突变而非某些反式调节机制引起的这一假说,因此确定人类PKU基因座位于第12号染色体上。

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