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通过多重连接依赖探针扩增(MLPA)分析鉴定到可变型卟啉症患者的部分原卟啉原氧化酶(PPOX)基因缺失,这些缺失是由不同的 Alu 介导机制引起的。

Partial protoporphyrinogen oxidase (PPOX) gene deletions, due to different Alu-mediated mechanisms, identified by MLPA analysis in patients with variegate porphyria.

机构信息

Centre for Inherited Metabolic Diseases, Karolinska University Hospital, Stockholm, Sweden.

出版信息

Orphanet J Rare Dis. 2013 Jan 16;8:13. doi: 10.1186/1750-1172-8-13.

DOI:10.1186/1750-1172-8-13
PMID:23324528
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC3554555/
Abstract

Variegate porphyria (VP) is an autosomal dominantly inherited hepatic porphyria. The genetic defect in the PPOX gene leads to a partial defect of protoporphyrinogen oxidase, the penultimate enzyme of heme biosynthesis. Affected individuals can develop cutaneous symptoms in sun-exposed areas of the skin and/or neuropsychiatric acute attacks. The identification of the genetic defect in VP families is of crucial importance to detect the carrier status which allows counseling to prevent potentially life threatening neurovisceral attacks, usually triggered by factors such as certain drugs, alcohol or fasting.In a total of 31 Swedish VP families sequence analysis had identified a genetic defect in 26. In the remaining five families an extended genetic investigation was necessary. After the development of a synthetic probe set, MLPA analysis to screen for single exon deletions/duplications was performed.We describe here, for the first time, two partial deletions within the PPOX gene detected by MLPA analysis. One deletion affects exon 5 and 6 (c.339-197_616+320del1099) and has been identified in four families, most probably after a founder effect. The other extends from exon 5 to exon 9 (c.339-350_987+229del2609) and was found in one family. We show that both deletions are mediated by Alu repeats.Our findings emphasize the usefulness of MLPA analysis as a complement to PPOX gene sequencing analysis for comprehensive genetic diagnostics in patients with VP.

摘要

变异性卟啉症(VP)是一种常染色体显性遗传性肝卟啉症。PPOX 基因的遗传缺陷导致原卟啉原氧化酶的部分缺陷,原卟啉原氧化酶是血红素生物合成的倒数第二酶。受影响的个体可能会在暴露于阳光的皮肤区域出现皮肤症状和/或神经精神急性发作。在 VP 家族中鉴定遗传缺陷对于检测携带者状态至关重要,这可以进行咨询以预防潜在的危及生命的神经内脏发作,这些发作通常由某些药物、酒精或禁食等因素引发。在总共 31 个瑞典 VP 家族中,序列分析在 26 个家族中确定了遗传缺陷。在其余的 5 个家族中,需要进行扩展的遗传调查。在开发了一套合成探针后,进行 MLPA 分析以筛选单外显子缺失/重复。我们在此首次描述了 MLPA 分析检测到的 PPOX 基因内的两个部分缺失。一个缺失影响外显子 5 和 6(c.339-197_616+320del1099),已在四个家族中发现,很可能是在一个创始人效应之后。另一个缺失从外显子 5 延伸到外显子 9(c.339-350_987+229del2609),在一个家族中发现。我们表明,这两种缺失都是由 Alu 重复介导的。我们的研究结果强调了 MLPA 分析作为 PPOX 基因测序分析的补充在 VP 患者全面遗传诊断中的有用性。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/ec51/3554555/9af98bb95d33/1750-1172-8-13-3.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/ec51/3554555/afe757b19f25/1750-1172-8-13-1.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/ec51/3554555/d01ca4e14a31/1750-1172-8-13-2.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/ec51/3554555/9af98bb95d33/1750-1172-8-13-3.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/ec51/3554555/afe757b19f25/1750-1172-8-13-1.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/ec51/3554555/d01ca4e14a31/1750-1172-8-13-2.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/ec51/3554555/9af98bb95d33/1750-1172-8-13-3.jpg

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