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保加利亚急性肝卟啉病(AHP)患者中的七个新突变

Seven Novel Mutations in Bulgarian Patients with Acute Hepatic Porphyrias (AHP).

作者信息

Dragneva Sonya, Szyszka-Niagolov Monika, Ivanova Aneta, Mateva Lyudmila, Izumi Rumiko, Aoki Yoko, Matsubara Yoichi

机构信息

Clinic of Gastroenterology and Hepatology, University Hospital "Saint Ivan Rislki", Sofia, Bulgaria,

出版信息

JIMD Rep. 2014;16:57-64. doi: 10.1007/8904_2014_320. Epub 2014 Jul 6.

Abstract

Acute intermittent porphyria (AIP), variegate porphyria (VP), and hereditary coproporphyria (HCP) are caused by mutations in the hydroxymethylbilane synthase (HMBS), protoporphyrinogen oxidase (PPOX), and coproporphyrinogen oxidase (CPOX) genes, respectively. This study aimed to identify mutations in seven Bulgarian families with AIP, six with VP, and one with HCP. A total of 33 subjects, both symptomatic (n = 21) and asymptomatic (n = 12), were included in this study. The identification of mutations was performed by direct sequencing of all the coding exons of the corresponding enzymes in the probands. The available relatives were screened for the possible mutations. A total of six different mutations in HMBS were detected in all seven families with AIP, three of which were previously described: c.76C>T [p.R26C] in exon 3, c.287C>T [p.S96F] in exon 7, and c.445C>T [p.R149X] in exon 9. The following three novel HMBS mutations were found: c.345-2A>C in intron 7-8, c.279-280insAT in exon 7, and c.887delC in exon 15. A total of three different novel mutations were identified in the PPOX gene in the VP families: c.441-442delCA in exon 5, c.917T>C [p.L306P] in exon 9, and c.1252T>C [p.C418R] in exon 12. A novel nonsense mutation, c.364G>T [p.E122X], in exon 1 of the CPOX gene was identified in the HCP family. This study, which identified mutations in Bulgarian families with AHP for the first time, established seven novel mutation sites. Seven latent carriers were also diagnosed and, therefore, were able to receive crucial counseling to prevent attacks.

摘要

急性间歇性卟啉病(AIP)、混合型卟啉病(VP)和遗传性粪卟啉病(HCP)分别由羟甲基胆色素原合酶(HMBS)、原卟啉原氧化酶(PPOX)和粪卟啉原氧化酶(CPOX)基因突变引起。本研究旨在鉴定7个患AIP的保加利亚家族、6个患VP的家族和1个患HCP的家族中的突变。本研究共纳入33名受试者,包括有症状者(n = 21)和无症状者(n = 12)。通过对先证者相应酶的所有编码外显子进行直接测序来鉴定突变。对现有的亲属进行可能突变的筛查。在所有7个患AIP的家族中,共检测到HMBS的6种不同突变,其中3种先前已有描述:外显子3中的c.76C>T [p.R26C]、外显子7中的c.287C>T [p.S96F]和外显子9中的c.445C>T [p.R149X]。还发现了以下3种新的HMBS突变:内含子7 - 8中的c.345 - 2A>C、外显子7中的c.279 - 280insAT和外显子15中的c.887delC。在VP家族的PPOX基因中鉴定出3种不同的新突变:外显子5中的c.441 - 442delCA、外显子9中的c.917T>C [p.L306P]和外显子12中的c.1252T>C [p.C418R]。在HCP家族中鉴定出CPOX基因外显子1中的一种新的无义突变c.364G>T [p.E122X]。本研究首次鉴定了保加利亚患AHP家族中的突变,确定了7个新的突变位点。还诊断出7名潜在携带者,因此他们能够接受关键的咨询以预防发作。

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本文引用的文献

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6
HMBS mutations in Chinese patients with acute intermittent porphyria.
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8
Porphyria in Sweden.
Physiol Res. 2006;55 Suppl 2:S109-118. doi: 10.33549/physiolres.930000.55.S2.109.
9
Modern diagnosis and management of the porphyrias.
Br J Haematol. 2006 Nov;135(3):281-92. doi: 10.1111/j.1365-2141.2006.06289.x. Epub 2006 Sep 4.

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