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锚蛋白那不勒斯:一种与球形红细胞增多症相关的锚蛋白基因(ANK1)第16外显子的新发缺失移码突变。

Ankyrin Napoli: a de novo deletional frameshift mutation in exon 16 of ankyrin gene (ANK1) associated with spherocytosis.

作者信息

del Giudice E M, Hayette S, Bozon M, Perrotta S, Alloisio N, Vallier A, Iolascon A, Delaunay J, Morlé L

机构信息

Dipartimento di Pediatria, Seconda Università di Napoli, Italy.

出版信息

Br J Haematol. 1996 Jun;93(4):828-34. doi: 10.1046/j.1365-2141.1996.d01-1746.x.

Abstract

We report a case of apparently recessive hereditary spherocytosis in an Italian child. The proband exhibited a reduction of overall ankyrin in the red cell membrane. The parents were free of any haematological manifestations. The VNDR associated with the ankyrin gene (ANK1) were consistent with the following diplotypes: AC11/ AC14 (father), AC14/AC14 (mother) and AC11/AC14 (child). The cDNA of the patient disclosed the expression of the AC11 allele only. As a consequence, we put forward the hypothesis of a de novo inactivation affecting the ankyrin allele of maternal origin (AC14) and accounting for the disease. PCR amplification of exons, SSCP analysis and nucleotide sequencing disclosed a polymorphism: GAC --> AAC; Asp --> Asn in codon 328 of exon 10, and a one-nucleotide deletion : CTG --> CG in codon 573 of the exon 16. This frameshift mutation placed in phase the TGA triplet that normally overlaps codons 636 and 637. Termination of translation near the middle of ankyrin mRNA coding sequence resulted, presumably, in its premature degadation. The present allele has been designated allele Napoli.

摘要

我们报告了一例意大利儿童患明显隐性遗传性球形红细胞增多症的病例。先证者红细胞膜中锚蛋白总量减少。其父母无任何血液学表现。与锚蛋白基因(ANK1)相关的可变数目串联重复序列(VNDR)符合以下单倍型:AC11/AC14(父亲)、AC14/AC14(母亲)和AC11/AC14(患儿)。患者的cDNA仅显示AC11等位基因的表达。因此,我们提出一种假设,即母源(AC14)锚蛋白等位基因发生了新生失活,这是导致该疾病的原因。外显子的PCR扩增、单链构象多态性分析(SSCP)和核苷酸测序揭示了一种多态性:第10外显子328密码子处GAC→AAC;天冬氨酸→天冬酰胺,以及第16外显子573密码子处单核苷酸缺失:CTG→CG。这种移码突变使通常与636和637密码子重叠的TGA三联体处于同一相位。翻译在锚蛋白mRNA编码序列中部附近终止,可能导致其过早降解。目前该等位基因已被命名为那不勒斯等位基因。

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