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尿卟啉原脱羧酶:一个剪接位点突变导致多个迟发性皮肤卟啉症家族中外显子6缺失。

Uroporphyrinogen decarboxylase: a splice site mutation causes the deletion of exon 6 in multiple families with porphyria cutanea tarda.

作者信息

Garey J R, Harrison L M, Franklin K F, Metcalf K M, Radisky E S, Kushner J P

机构信息

Division of Hematology, University of Utah School of Medicine, Salt Lake City 84132.

出版信息

J Clin Invest. 1990 Nov;86(5):1416-22. doi: 10.1172/JCI114856.

Abstract

Uroporphyrinogen decarboxylase (URO-D) is a cytosolic heme-biosynthetic enzyme that converts uroporphyrinogen to coproporphyrinogen. Defects at the uroporphyrinogen decarboxylase locus cause the human genetic disease familial porphyria cutanea tarda. A splice site mutation has been found in a pedigree with familial porphyria cutanea tarda that causes exon 6 to be deleted from the mRNA. The intron/exon junctions on either side of exon 6 fall between codons, so the resulting protein is shorter than the normal protein, missing only the amino acids coded by exon 6. The shortened protein lacks catalytic activity, is rapidly degraded when exposed to human lymphocyte lysates, and is not detectable by Western blot analysis in lymphocyte lysates derived from affected individuals. The mutation was detected in five of 22 unrelated familial porphyria cutanea tarda pedigrees tested, so it appears to be common. This is the first splice site mutation to be found at the URO-D locus, and the first mutation that causes familial porphyria cutanea tarda to be found in more than one pedigree.

摘要

尿卟啉原脱羧酶(URO-D)是一种胞质血红素生物合成酶,可将尿卟啉原转化为粪卟啉原。尿卟啉原脱羧酶基因座的缺陷会导致人类遗传性疾病迟发性皮肤卟啉症。在一个迟发性皮肤卟啉症家系中发现了一个剪接位点突变,该突变导致外显子6从mRNA中缺失。外显子6两侧的内含子/外显子连接位于密码子之间,因此产生的蛋白质比正常蛋白质短,仅缺失由外显子6编码的氨基酸。缩短的蛋白质缺乏催化活性,暴露于人类淋巴细胞裂解物时会迅速降解,并且在来自受影响个体的淋巴细胞裂解物中通过蛋白质印迹分析无法检测到。在所检测的22个无关的迟发性皮肤卟啉症家系中有5个检测到该突变,因此它似乎很常见。这是在URO-D基因座发现的第一个剪接位点突变,也是在多个家系中发现的导致迟发性皮肤卟啉症的第一个突变。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/7902/296884/ececa92e5f8d/jcinvest00077-0045-a.jpg

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