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北非的Sp alpha I/65遗传性椭圆形红细胞增多症。

Sp alpha I/65 hereditary elliptocytosis in North Africa.

作者信息

Alloisio N, Guetarni D, Morlé L, Pothier B, Ducluzeau M T, Soun A, Colonna P, Clerc M, Philippe N, Delaunay J

出版信息

Am J Hematol. 1986 Oct;23(2):113-22. doi: 10.1002/ajh.2830230205.

Abstract

The Sp alpha I/65 variant of the spectrin has been recently described in black people with hereditary elliptocytosis (HE). The present study reports on a similar Sp alpha I/65 variant in nine North African persons belonging to four unrelated families. The abnormality was associated with a variable degree of elliptocytosis. In one case, red cell morphology was normal. In the nine carriers of the biochemical abnormality, the spectrin dimer self-association was defective. The association constant was reduced: 0.65 to 1.7 X 10(5) M-1 (controls: 4.6 +/- 0.5 X 10(5) mM-1 (n = 21)); in six cases, there was a higher level of spectrin dimer in the low ionic strength extract at 4 degrees C: 13.0 to 19.7% (controls: 6.4 +/- 2.1% (n = 7)). Limited tryptic digests of spectrin from the nine persons revealed a decrease of the 80,000-dalton alpha-1 domain, and the concomitant appearance of a peptide with a molecular weight of 65,000 daltons and an isoelectric point ranging from 5.0 to 5.1. There was a correlation between the proportion of the 65,000-dalton fragments, the defect of spectrin self-association, and the extent of morphological alteration. This is the first large series concerning a spectrin abnormality in non-black persons. In North Africa, cases of HE that are not due to a protein 4.1 defect have turned out so far to be associated with the Sp alpha I/65 variant.

摘要

最近在患有遗传性椭圆形红细胞增多症(HE)的黑人中发现了血影蛋白的SpαI/65变体。本研究报告了在四个无亲缘关系的家庭中的九名北非人中存在类似的SpαI/65变体。这种异常与不同程度的椭圆形红细胞增多症相关。在一个病例中,红细胞形态正常。在这九名生化异常携带者中,血影蛋白二聚体的自我缔合存在缺陷。缔合常数降低:为0.65至1.7×10⁵M⁻¹(对照组:4.6±0.5×10⁵mM⁻¹(n = 21));在六个病例中,4℃低离子强度提取物中的血影蛋白二聚体水平较高:为13.0%至19.7%(对照组:6.4±2.1%(n = 7))。对这九个人的血影蛋白进行有限的胰蛋白酶消化后发现,80,000道尔顿的α-1结构域减少,同时出现了一种分子量为65,000道尔顿、等电点在5.0至5.1之间的肽段。65,000道尔顿片段的比例、血影蛋白自我缔合的缺陷与形态改变的程度之间存在相关性。这是关于非黑人血影蛋白异常的首个大型系列研究。在北非,迄今为止发现并非由蛋白4.1缺陷引起的HE病例与SpαI/65变体相关。

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