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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.

DOI:10.1172/jci110944
PMID:6863546
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC370394/
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
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/628a/370394/77b3df7219bd/jcinvest00707-0378-a.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/628a/370394/77b3df7219bd/jcinvest00707-0378-a.jpg

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引用本文的文献

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Adenosine deaminase deficiency: a review.腺苷脱氨酶缺乏症:综述。
Orphanet J Rare Dis. 2018 Apr 24;13(1):65. doi: 10.1186/s13023-018-0807-5.
2
Characterization of normal and mutant adenosine deaminase messenger RNAs by translation and hybridization to a cDNA probe.通过翻译以及与互补DNA探针杂交对正常和突变腺苷脱氨酶信使核糖核酸进行特性分析。
Hum Genet. 1984;68(2):169-72. doi: 10.1007/BF00279309.
3
Sequence of human adenosine deaminase cDNA including the coding region and a small intron.包含编码区和一个小内含子的人腺苷脱氨酶cDNA序列。

本文引用的文献

1
Protein measurement with the Folin phenol reagent.使用福林酚试剂进行蛋白质测定。
J Biol Chem. 1951 Nov;193(1):265-75.
2
Human neutral alpha-glucosidase C: genetic polymorphism including a "null" allele.人中性α-葡萄糖苷酶C:包括“无效”等位基因在内的基因多态性。
Am J Hum Genet. 1980 Jul;32(4):497-507.
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Increased excretion of modified adenine nucleosides by children with adenosine deaminase deficiency.腺苷脱氨酶缺乏症患儿修饰腺嘌呤核苷排泄增加。
Nucleic Acids Res. 1984 Mar 12;12(5):2439-46. doi: 10.1093/nar/12.5.2439.
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Structure of adenosine deaminase mRNAs from normal and adenosine deaminase-deficient human cell lines.来自正常和腺苷脱氨酶缺陷型人类细胞系的腺苷脱氨酶mRNA的结构
Mol Cell Biol. 1984 Sep;4(9):1712-7. doi: 10.1128/mcb.4.9.1712-1717.1984.
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Cloning of cDNA sequences of human adenosine deaminase.人腺苷脱氨酶cDNA序列的克隆
Proc Natl Acad Sci U S A. 1983 Dec;80(24):7481-5. doi: 10.1073/pnas.80.24.7481.
6
Genetic heterogeneity in adenosine deaminase (ADA) deficiency: five different mutations in five new patients with partial ADA deficiency.腺苷脱氨酶(ADA)缺乏症中的遗传异质性:5例部分ADA缺乏症新患者中的5种不同突变
Am J Hum Genet. 1986 Jan;38(1):13-25.
7
Identification of a point mutation in the adenosine deaminase gene responsible for immunodeficiency.鉴定导致免疫缺陷的腺苷脱氨酶基因中的一个点突变。
J Clin Invest. 1985 Aug;76(2):894-7. doi: 10.1172/JCI112050.
8
Transient expression of human adenosine deaminase cDNAs: identification of a nonfunctional clone resulting from a single amino acid substitution.人腺苷脱氨酶cDNA的瞬时表达:鉴定由单个氨基酸取代导致的无功能克隆。
Mol Cell Biol. 1985 Apr;5(4):762-7. doi: 10.1128/mcb.5.4.762-767.1985.
9
Partial adenosine deaminase deficiency: another family from southern Africa.部分腺苷脱氨酶缺乏症:来自非洲南部的另一个家族。
Hum Genet. 1986 Nov;74(3):307-12. doi: 10.1007/BF00282554.
10
Identification of a point mutation resulting in a heat-labile adenosine deaminase (ADA) in two unrelated children with partial ADA deficiency.在两名患有部分腺苷脱氨酶(ADA)缺乏症的非亲缘儿童中鉴定出一种导致热不稳定腺苷脱氨酶(ADA)的点突变。
J Clin Invest. 1989 Feb;83(2):497-501. doi: 10.1172/JCI113909.
Pediatr Res. 1982 May;16(5):362-9. doi: 10.1203/00006450-198205000-00009.
4
Adenosine deaminase deficiency without immunodeficiency: clinical and metabolic studies.无免疫缺陷的腺苷脱氨酶缺乏症:临床与代谢研究
Pediatr Res. 1980 Jul;14(7):885-9. doi: 10.1203/00006450-198007000-00009.
5
Further data on the adenosine deaminase (ADA) polymprphism and a report of a new phenotype.腺苷脱氨酶(ADA)多态性的更多数据及一种新表型的报告。
Ann Hum Genet. 1969 May;32(4):361-7. doi: 10.1111/j.1469-1809.1969.tb00087.x.
6
The investigation of reactive sulphydryls in enzymes and their variants by starch gel electrophoresis. Studies on red cell adenosine deaminase.通过淀粉凝胶电泳研究酶及其变体中的反应性巯基。红细胞腺苷脱氨酶的研究。
Ann Hum Genet. 1969 Jul;33(1):81-7. doi: 10.1111/j.1469-1809.1969.tb01632.x.
7
Evidence for control of several different tissue-specific isozymes of adenosine deaminase by a single genetic locus.单一基因座控制腺苷脱氨酶几种不同组织特异性同工酶的证据。
Nat New Biol. 1973 Dec 19;246(155):200-2. doi: 10.1038/newbio246200a0.
8
Assignment of a gene for adenosine deaminase to human chromosome 20.将腺苷脱氨酶基因定位于人类第20号染色体。
Hum Hered. 1974;24(1):1-11. doi: 10.1159/000152631.
9
Red-blood-cell adenosine deaminase deficiency in a "healthy" Kung individual.一名“健康”的昆族人红细胞腺苷脱氨酶缺乏症
Lancet. 1973 Sep 29;2(7831):736. doi: 10.1016/s0140-6736(73)92568-3.
10
Adenosine-deaminase deficiency in two patients with severely impaired cellular immunity.两名细胞免疫严重受损患者的腺苷脱氨酶缺乏症。
Lancet. 1972 Nov 18;2(7786):1067-9. doi: 10.1016/s0140-6736(72)92345-8.