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部分腺苷脱氨酶缺乏症中的遗传异质性。

Genetic heterogeneity in partial adenosine deaminase deficiency.

作者信息

Hirschhorn R, Martiniuk F, Roegner-Maniscalco V, Ellenbogen A, Perignon J L, Jenkins T

出版信息

J Clin Invest. 1983 Jun;71(6):1887-92. doi: 10.1172/jci110944.

Abstract

Inherited deficiency of the enzyme adenosine deaminase (ADA) results in a syndrome of severe combined immunodeficiency (SCID). Children with ADA- -SCID lack ADA in all cells and tissues. In contrast, a "partial" deficiency of ADA has been described in six immunologically normal children from four different "families." These children lack ADA in their erythrocytes but retain variable amounts of activity in their lymphoid cells. We have examined ADA activity in lymphoid line cells from four of these children, who are unrelated, for evidence of genetic heterogeneity. One child, who is Caucasian, has an enzyme with increased electrophoretic mobility, a diminished isoelectric point (pI 4.8 vs. Nl = 4.9) and very low activity (2.3 vs. Nl = 82.9 +/- 12.9 nmol/mg protein per min); as a second child has an enzyme with normal electrophoretic mobility but increased isoelectric point (pI = 5.0), markedly diminished heat stability at 56 degrees C (t1/2 = 4.2' vs. Nl = 40') and low activity (12.1); a third has an enzyme with only diminished heat stability (t1/2 = 6.5'), no detectable abnormality in charge and almost normal activity (41.9); while the fourth exhibits only diminished ADA activity (25.0) with no striking qualitative abnormalities. Thus, we have found evidence for three different mutations at the structural locus for ADA in three of these individuals, (a) an acidic, low activity heat stable mutation (b) a basic, somewhat higher activity, heat labile mutation, and (c) a relatively normal activity heat labile mutation. In the fourth, there is as yet no compelling evidence for a mutation at the structural locus for ADA and a mutation at a regulatory locus cannot be excluded.

摘要

腺苷脱氨酶(ADA)的遗传性缺乏会导致严重联合免疫缺陷综合征(SCID)。患有ADA - SCID的儿童在所有细胞和组织中都缺乏ADA。相比之下,在来自四个不同“家族”的六名免疫功能正常的儿童中描述了ADA的“部分”缺乏。这些儿童的红细胞中缺乏ADA,但淋巴细胞中保留了不同程度的活性。我们检查了其中四名无亲缘关系儿童的淋巴系细胞中的ADA活性,以寻找遗传异质性的证据。一名白人儿童的酶具有增加的电泳迁移率、降低的等电点(pI 4.8 vs. Nl = 4.9)和非常低的活性(2.3 vs. Nl = 82.9 +/- 12.9 nmol/mg蛋白质每分钟);第二名儿童的酶具有正常的电泳迁移率但等电点增加(pI = 5.0),在56摄氏度时热稳定性明显降低(t1/2 = 4.2分钟vs. Nl = 40分钟)且活性较低(12.1);第三名儿童的酶仅热稳定性降低(t1/2 = 6.5分钟),电荷无明显异常且活性几乎正常(41.9);而第四名儿童仅表现出ADA活性降低(25.0),无明显的定性异常。因此,我们在其中三名个体中发现了ADA结构基因座上三种不同突变的证据,(a)一种酸性、低活性、热稳定的突变,(b)一种碱性、活性稍高、热不稳定的突变,以及(c)一种活性相对正常、热不稳定的突变。在第四名儿童中,尚无令人信服的证据表明ADA结构基因座发生突变,也不能排除调控基因座发生突变的可能性。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/628a/370394/77b3df7219bd/jcinvest00707-0378-a.jpg

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