四个小鼠过氧化物酶体ABC转运蛋白基因在组成型、诱导型和发育表达方面存在差异。
The four murine peroxisomal ABC-transporter genes differ in constitutive, inducible and developmental expression.
作者信息
Berger J, Albet S, Bentejac M, Netik A, Holzinger A, Roscher A A, Bugaut M, Forss-Petter S
机构信息
Brain Research Institute, University of Vienna, Austria.
出版信息
Eur J Biochem. 1999 Oct;265(2):719-27. doi: 10.1046/j.1432-1327.1999.00772.x.
Four ATP-binding cassette (ABC) half-transporters have been identified in mammalian peroxisomes: adrenoleukodystrophy protein (ALDP), adrenoleukodystrophy-related protein (ALDRP), 70-kDa peroxisomal membrane protein (PMP70) and PMP70-related protein (P70R). Inherited defects in ALDP cause the neurodegenerative disorder X-linked adrenoleukodystrophy (X-ALD). By comparative Northern blot analyses we found each of the four murine peroxisomal ABC transporter mRNA species at maximum abundance only in a few tissues, which differed for each family member. The four genes were also regulated differentially during mouse brain development: ALDP mRNA was most abundant in embryonic brain and gradually decreased during maturation; ALDRP and P70R mRNA accumulated in the early postnatal period; and the amount of PMP70 transcript increased slightly during the second and third postnatal week. The different expression patterns could explain why beta-oxidation is defective in X-ALD, although ALDRP and PMP70 can replace ALDP functionally in fibroblasts. Dietary fenofibrate had no effect on the ALD and P70R genes, but strongly increased expression of the ALDR and PMP70 genes in mouse liver. However, in P-glycoprotein Mdr1a-deficient mice fenofibrate treatment increased ALDR gene expression also in the brain, suggesting that the multidrug-transporter P-glycoprotein restricts entry of fenofibrate to the brain at the blood-brain barrier. Analysis of the promoter sequences revealed a cryptic nuclear hormone receptor response element of the DR+4 type in the ALDR promoter and a novel 18-bp sequence motif present only in the 5' flanking DNA of the ALDR and PMP70 genes. The mouse ALDR gene uses a single transcription start site but alternative polyadenylation sites. These data are of importance for the use of ALDP-deficient mice as a model in pharmacological gene therapy studies.
在哺乳动物过氧化物酶体中已鉴定出四种ATP结合盒(ABC)半转运蛋白:肾上腺脑白质营养不良蛋白(ALDP)、肾上腺脑白质营养不良相关蛋白(ALDRP)、70 kDa过氧化物酶体膜蛋白(PMP70)和PMP70相关蛋白(P70R)。ALDP的遗传性缺陷会导致神经退行性疾病X连锁肾上腺脑白质营养不良(X-ALD)。通过比较Northern印迹分析,我们发现四种小鼠过氧化物酶体ABC转运蛋白mRNA种类中的每一种仅在少数组织中以最大丰度存在,且每个家族成员的情况都不同。这四个基因在小鼠大脑发育过程中也受到不同的调控:ALDP mRNA在胚胎脑中最为丰富,并在成熟过程中逐渐减少;ALDRP和P70R mRNA在出生后早期积累;而PMP70转录本的量在出生后第二和第三周略有增加。尽管ALDRP和PMP70在成纤维细胞中可在功能上替代ALDP,但不同的表达模式可以解释为什么X-ALD中的β氧化存在缺陷。饮食中的非诺贝特对ALD和P70R基因没有影响,但能显著增加小鼠肝脏中ALDR和PMP70基因的表达。然而,在P-糖蛋白Mdr1a缺陷小鼠中,非诺贝特治疗也会增加脑中ALDR基因的表达,这表明多药转运蛋白P-糖蛋白在血脑屏障处限制了非诺贝特进入大脑。对启动子序列的分析揭示了ALDR启动子中一个DR+4型的隐蔽核激素受体反应元件以及仅存在于ALDR和PMP70基因5'侧翼DNA中的一个新的18 bp序列基序。小鼠ALDR基因使用单个转录起始位点,但有多个聚腺苷酸化位点。这些数据对于将ALDP缺陷小鼠用作药理基因治疗研究的模型具有重要意义。