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一名意大利患者被偶然发现携带纯合子p.M348K突变且无症状,这证实了囊性纤维化的多样性。

Incidental Finding of a Homozygous p.M348K Asymptomatic Italian Patient Confirms the Many Faces of Cystic Fibrosis.

作者信息

Molinario Rossana, Palumbo Sara, Concolino Paola, Rocchetti Sandro, Rizza Roberta, Scaglione Giovanni Luca, Minucci Angelo, Capoluongo Ettore

机构信息

Laboratory of Clinical Molecular and Personalized Diagnostics, Department of Laboratory Medicine, University Hospital "A. Gemelli", 8 Largo A. Gemelli, 00168 Rome, Italy.

出版信息

Case Rep Genet. 2015;2015:289627. doi: 10.1155/2015/289627. Epub 2015 Apr 1.

DOI:10.1155/2015/289627
PMID:25922769
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC4397493/
Abstract

Cystic fibrosis (CF; OMIM number 219700) is an autosomal recessive disease caused by mutations in the CFTR (cystic fibrosis transmembrane conductance regulator) gene, which results in abnormal viscous mucoid secretions in multiple organs and whose main clinical features are pancreatic insufficiency, chronic endobronchial infection, and male infertility. We report the case of a 47-year-old apparently normal male resulting in homozygosity for the mutation p.M348K from nonconsanguineous parents. The proband was screened using a standard panel of 70 different tested on NanoChip 400 platform. The massive parallel pyrosequencing on 454 JS machine allowed the second level analysis. The patient was firstly screened with two different platforms available in our laboratory, obtaining an ambiguous signal for the p.R347P mutation. For this reason we decided to clarify the discordant result of CFTR status by Next Generation Sequencing (NGS) using 454 Junior instrument. The patient is resulted no carrier of the p.R347P mutation, but NGS highlighted a homozygous substitution from T>A at position 1043 in the coding region, causing an amino acid substitution from methionine to lysine (p.M348K). Casual finding of p.M348K homozygote mutation in an individual, without any feature of classical or nonclassical CF form, allowed us to confirm that p.M348K is a benign rare polymorphism.

摘要

囊性纤维化(CF;OMIM编号219700)是一种常染色体隐性疾病,由CFTR(囊性纤维化跨膜传导调节因子)基因突变引起,该突变导致多个器官出现异常黏稠的黏液性分泌物,其主要临床特征为胰腺功能不全、慢性支气管感染和男性不育。我们报告了一例47岁的男性病例,其非近亲父母均为p.M348K突变的纯合子,但该男性外表正常。先证者使用在NanoChip 400平台上进行的70种不同检测的标准检测板进行筛查。在454 JS机器上进行的大规模平行焦磷酸测序允许进行二级分析。该患者首先在我们实验室可用的两个不同平台上进行筛查,对于p.R347P突变获得了模糊信号。因此,我们决定使用454 Junior仪器通过下一代测序(NGS)来澄清CFTR状态的不一致结果。结果显示该患者不是p.R347P突变的携带者,但NGS突出显示在编码区第1043位有一个从T到A的纯合子替代,导致氨基酸从甲硫氨酸替代为赖氨酸(p.M348K)。在一个没有任何经典或非经典CF形式特征的个体中偶然发现p.M348K纯合子突变,使我们能够确认p.M348K是一种良性罕见多态性。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/8183/4397493/846bf7dea9c9/CRIG2015-289627.001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/8183/4397493/846bf7dea9c9/CRIG2015-289627.001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/8183/4397493/846bf7dea9c9/CRIG2015-289627.001.jpg

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本文引用的文献

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Next generation diagnostics of cystic fibrosis and CFTR-related disorders by targeted multiplex high-coverage resequencing of CFTR.通过靶向多重高覆盖 CFTR 重测序对囊性纤维化和 CFTR 相关疾病进行下一代诊断。
J Med Genet. 2013 Jul;50(7):455-62. doi: 10.1136/jmedgenet-2013-101602. Epub 2013 May 17.
2
CFTR2: How will it help care?囊性纤维化跨膜传导调节因子 2:它将如何帮助治疗?
Paediatr Respir Rev. 2013 May;14 Suppl 1:2-5. doi: 10.1016/j.prrv.2013.01.006. Epub 2013 Mar 1.
3
Homozygous CFTR mutation M348K in a boy with respiratory symptoms and failure to thrive. Disease-causing mutation or benign alteration?
男孩有呼吸道症状和生长发育迟缓,携带 CFTR 基因纯合突变 M348K。致病突变还是良性变异?
Eur J Pediatr. 2012 Jul;171(7):1039-46. doi: 10.1007/s00431-012-1672-1. Epub 2012 Jan 25.
4
Atypical CF and CF related diseases.非典型囊性纤维化及囊性纤维化相关疾病
Paediatr Respir Rev. 2006;7 Suppl 1:S144-6. doi: 10.1016/j.prrv.2006.04.219. Epub 2006 Jun 5.
5
Segregation analysis in cystic fibrosis at-risk family demonstrates that the M348K CFTR mutation is a rare innocuous polymorphism.对囊性纤维化高危家庭进行的分离分析表明,M348K囊性纤维化跨膜传导调节因子突变是一种罕见的无害多态性。
Prenat Diagn. 2004 Dec 15;24(12):981-3. doi: 10.1002/pd.1058.
6
Prenatal screening for cystic fibrosis: past, present and future.囊性纤维化的产前筛查:过去、现在与未来。
Expert Rev Mol Diagn. 2004 Jan;4(1):49-62. doi: 10.1586/14737159.4.1.49.
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Cystic fibrosis: a worldwide analysis of CFTR mutations--correlation with incidence data and application to screening.囊性纤维化:CFTR 突变的全球分析——与发病率数据的相关性及在筛查中的应用
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Cystic fibrosis.囊性纤维化
Am J Respir Crit Care Med. 1996 Nov;154(5):1229-56. doi: 10.1164/ajrccm.154.5.8912731.
9
Description of a symptomless cystic fibrosis L346P/M348K compound heterozygous Cypriot individual.
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