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两名肝红细胞生成性卟啉症患者中人类尿卟啉原脱羧酶基因座新突变(R292G)及缺失的特征分析

Characterization of a new mutation (R292G) and a deletion at the human uroporphyrinogen decarboxylase locus in two patients with hepatoerythropoietic porphyria.

作者信息

de Verneuil H, Bourgeois F, de Rooij F, Siersema P D, Wilson J H, Grandchamp B, Nordmann Y

机构信息

Laboratoire de Génétique Moléculaire, Faculté Xavier Bichat, Paris, France.

出版信息

Hum Genet. 1992 Jul;89(5):548-52. doi: 10.1007/BF00219182.

Abstract

A deficiency in the activity of uroporphyrinogen decarboxylase (UROD), the fifth enzyme of the haem biosynthetic pathway, is found in familial porphyria cutanea tarda (F-PCT) and hepatoerythropoietic porphyria (HEP). A new mutation (R292G) and a deletion have been found in a pedigree with two HEP patients (two sisters). The R292G mutation was not detected in 13 unrelated affected patients with F-PCT, so it appears to be uncommon. The possibility that the arginine 292 may participate at the active site of the enzyme is discussed. A summary of the 7 mutations/deletions found in the UROD gene with their frequency is presented.

摘要

家族性迟发性皮肤卟啉症(F-PCT)和肝红细胞生成性卟啉症(HEP)中,可发现血红素生物合成途径的第五种酶——尿卟啉原脱羧酶(UROD)的活性存在缺陷。在一个有两名HEP患者(两姐妹)的家系中发现了一种新的突变(R292G)和一处缺失。在13名无亲缘关系的F-PCT患者中未检测到R292G突变,因此该突变似乎并不常见。本文讨论了精氨酸292可能参与该酶活性位点的可能性。还列出了在UROD基因中发现的7种突变/缺失及其频率的总结。

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