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儿科人群中的囊性纤维化管理——从临床特征到个性化治疗

Cystic fibrosis management in pediatric population-from clinical features to personalized therapy.

作者信息

Azoicai Alice Nicoleta, Lupu Ancuta, Trandafir Laura Mihaela, Alexoae Monica Mihaela, Alecsa Mirabela, Starcea Iuliana Magdalena, Cuciureanu Magdalena, Knieling Anton, Salaru Delia Lidia, Hanganu Elena, Mocanu Adriana, Lupu Vasile Valeriu, Ioniuc Ileana

机构信息

Pediatrics, "Grigore T. Popa" University of Medicine and Pharmacy, Iasi, Romania.

Faculty of Medicine, "Grigore T. Popa" University of Medicine and Pharmacy, Iasi, Romania.

出版信息

Front Pediatr. 2024 May 10;12:1393193. doi: 10.3389/fped.2024.1393193. eCollection 2024.

DOI:10.3389/fped.2024.1393193
PMID:38798310
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC11116730/
Abstract

Cystic fibrosis (CF) is an autosomal recessive disease caused by mutations of the gene encoding the cystic fibrosis transmembrane conductance regulator (CFTR). In 1949, it's been identified as a monogenic disease and was thought to primarily affect individuals of Northern European descent. It was the most prevalent autosomal recessive disease that shortens life. With the availability of multiple testing methodologies nowadays, there is a chance to create novel and enhanced treatment options. Even in the absence of a high sweat chloride test (SCT) result, the discovery of two causal mutations is diagnostic for cystic fibrosis (CF). For a CF diagnosis, however, at least two positive E sweat chloride tests are still required. In order to achieve early and active intervention to manage cystic fibrosis (CF) and its comorbidities, treatment regimens for pediatric patients should be evaluated, improved, and closely monitored. New developments in the treatment of cystic fibrosis (CF) have led to the development of medications derived from molecules that target the pathogenetic pathway of the illness. These options are very efficient and allow pediatric patients to receive individualized care. However, in order to better direct patient care and enhance patient outcomes, it is crucial to research uncommon CF mutations, which can provide crucial information about the prognosis of the disease and the relationships between genotype and phenotype. To ensure the success of creating novel, safer, and more efficient treatment approaches, a deeper understanding of the pathogeny of the illness is required. In the age of customized medicine, genetic research will be essential to improving patient care and quality of life for those with uncommon mutations.

摘要

囊性纤维化(CF)是一种常染色体隐性疾病,由编码囊性纤维化跨膜传导调节因子(CFTR)的基因突变引起。1949年,它被确定为一种单基因疾病,当时认为主要影响北欧血统的个体。它是缩短寿命的最常见的常染色体隐性疾病。如今有了多种检测方法,就有机会创造新的和更好的治疗方案。即使没有高汗液氯化物测试(SCT)结果,发现两个致病突变也可诊断为囊性纤维化(CF)。然而,对于CF诊断,仍至少需要两次汗液氯化物测试呈阳性。为了实现对囊性纤维化(CF)及其合并症的早期积极干预,应评估、改进并密切监测儿科患者的治疗方案。囊性纤维化(CF)治疗的新进展导致了从针对该疾病致病途径的分子衍生而来的药物的开发。这些选择非常有效,使儿科患者能够接受个性化护理。然而,为了更好地指导患者护理并改善患者预后,研究罕见的CF突变至关重要,这可以提供有关疾病预后以及基因型与表型之间关系的关键信息。为确保创造新的、更安全和更有效的治疗方法取得成功,需要更深入地了解该疾病的发病机制。在个性化医疗时代,基因研究对于改善罕见突变患者的护理和生活质量至关重要。

相似文献

1
Cystic fibrosis management in pediatric population-from clinical features to personalized therapy.儿科人群中的囊性纤维化管理——从临床特征到个性化治疗
Front Pediatr. 2024 May 10;12:1393193. doi: 10.3389/fped.2024.1393193. eCollection 2024.
2
[Chinese experts consensus statement: diagnosis and treatment of cystic fibrosis (2023)].[中国专家共识声明:囊性纤维化的诊断与治疗(2023年)]
Zhonghua Jie He He Hu Xi Za Zhi. 2023 Apr 12;46(4):352-372. doi: 10.3760/cma.j.cn112147-20221214-00971.
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CFTR genotype and clinical outcomes of adult patients carried as cystic fibrosis disease.囊性纤维化病携带成年患者的 CFTR 基因型与临床结局
Gene. 2014 May 1;540(2):183-90. doi: 10.1016/j.gene.2014.02.040. Epub 2014 Feb 26.
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Uncommon clinical presentation of cystic fibrosis in a patient homozygous for a rare CFTR mutation: a case report.罕见 CFTR 突变纯合子患者囊性纤维化的不常见临床表现:病例报告。
BMC Pediatr. 2020 Feb 27;20(1):90. doi: 10.1186/s12887-020-1980-y.
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The diagnosis of cystic fibrosis.囊性纤维化的诊断。
Presse Med. 2017 Jun;46(6 Pt 2):e97-e108. doi: 10.1016/j.lpm.2017.04.010. Epub 2017 May 31.
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Outcomes of children with cystic fibrosis screen positive, inconclusive diagnosis/CFTR related metabolic syndrome.囊性纤维化筛查呈阳性、诊断不确定/与CFTR相关代谢综合征患儿的预后
Front Pediatr. 2023 Mar 9;11:1127659. doi: 10.3389/fped.2023.1127659. eCollection 2023.
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Personalized medicine in CF: from modulator development to therapy for cystic fibrosis patients with rare CFTR mutations.囊性纤维化个体化医学:从调节剂开发到治疗罕见 CFTR 突变的囊性纤维化患者。
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State of the Art on Approved Cystic Fibrosis Transmembrane Conductance Regulator () Modulators and Triple-Combination Therapy.已批准的囊性纤维化跨膜传导调节因子(CFTR)调节剂及三联组合疗法的最新进展。
Pharmaceuticals (Basel). 2021 Sep 15;14(9):928. doi: 10.3390/ph14090928.
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Ataluren and similar compounds (specific therapies for premature termination codon class I mutations) for cystic fibrosis.依伐卡托(Ataluren)及类似化合物(针对 I 类提前终止密码子突变的特异性治疗药物)治疗囊性纤维化。
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Applying Cystic Fibrosis Transmembrane Conductance Regulator Genetics and CFTR2 Data to Facilitate Diagnoses.应用囊性纤维化跨膜传导调节因子遗传学和CFTR2数据辅助诊断。
J Pediatr. 2017 Feb;181S:S27-S32.e1. doi: 10.1016/j.jpeds.2016.09.063.

本文引用的文献

1
Antibiotic Resistance in Patients with Cystic Fibrosis: Past, Present, and Future.囊性纤维化患者的抗生素耐药性:过去、现在与未来
Antibiotics (Basel). 2023 Jan 20;12(2):217. doi: 10.3390/antibiotics12020217.
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Managing cystic fibrosis in children aged 6-11yrs: a critical review of elexacaftor/tezacaftor/ivacaftor combination therapy.6至11岁儿童囊性纤维化的管理:依列卡福/替扎卡福/依伐卡托联合疗法的批判性综述
Expert Rev Respir Med. 2023 Feb;17(2):97-108. doi: 10.1080/17476348.2023.2179989. Epub 2023 Feb 26.
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Future therapies for cystic fibrosis.
囊性纤维化的未来疗法。
Nat Commun. 2023 Feb 8;14(1):693. doi: 10.1038/s41467-023-36244-2.
4
Effectiveness and Safety of Beta-Lactam Antibiotics with and without Therapeutic Drug Monitoring in Patients with Pseudomonas aeruginosa Pneumonia or Bloodstream Infection.β-内酰胺类抗生素在铜绿假单胞菌性肺炎或菌血症患者中进行治疗药物监测与不进行治疗药物监测的有效性和安全性。
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Antibiotherapy in Children with Cystic Fibrosis-An Extensive Review.囊性纤维化患儿的抗生素治疗——全面综述
Children (Basel). 2022 Aug 20;9(8):1258. doi: 10.3390/children9081258.
6
Antimicrobial peptides for tackling cystic fibrosis related bacterial infections: A review.抗微生物肽治疗囊性纤维化相关细菌感染:综述。
Microbiol Res. 2022 Oct;263:127152. doi: 10.1016/j.micres.2022.127152. Epub 2022 Jul 30.
7
Mucus-targeting therapies of defective mucus clearance for cystic fibrosis: A short review.针对囊性纤维化黏液清除缺陷的黏液靶向治疗:简短综述。
Curr Opin Pharmacol. 2022 Aug;65:102248. doi: 10.1016/j.coph.2022.102248. Epub 2022 Jun 8.
8
TMEM16A (ANO1) as a therapeutic target in cystic fibrosis.TMEM16A(ANO1)作为囊性纤维化的治疗靶点。
Curr Opin Pharmacol. 2022 Jun;64:102206. doi: 10.1016/j.coph.2022.102206. Epub 2022 Mar 29.
9
Prevalence of antibiotic resistance of Pseudomonas aeruginosa in cystic fibrosis infection: A systematic review and meta-analysis.囊性纤维化感染中铜绿假单胞菌的抗生素耐药性患病率:一项系统评价和荟萃分析。
Microb Pathog. 2022 Apr;165:105461. doi: 10.1016/j.micpath.2022.105461. Epub 2022 Feb 28.
10
Cystic fibrosis-related liver disease: Clinical presentations, diagnostic and monitoring approaches in the era of CFTR modulator therapies.囊性纤维化相关性肝病:CFTR 调节剂治疗时代的临床特征、诊断和监测方法。
J Hepatol. 2022 Feb;76(2):420-434. doi: 10.1016/j.jhep.2021.09.042. Epub 2021 Oct 20.