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通过外显子组测序对中国婴儿巴特综合征样低钾碱中毒进行临床特征分析和诊断。

Clinical characterization and diagnosis of cystic fibrosis through exome sequencing in Chinese infants with Bartter-syndrome-like hypokalemia alkalosis.

机构信息

Department of Pediatrics, Tongji Hospital, Tongji Medical College, Huazhong University of Science & Technology, Wuhan, 430030, China.

出版信息

Front Med. 2018 Oct;12(5):550-558. doi: 10.1007/s11684-017-0567-y. Epub 2018 Mar 9.

Abstract

Cystic fibrosis (CF) is a fatal autosomal-recessive disease caused by mutations in the CF transmembrane conductance regulator (CFTR) gene. CF is characterized by recurrent pulmonary infection with obstructive pulmonary disease. CF is common in the Caucasian population but is rare in the Chinese population. The symptoms of early-stage CF are often untypical and may sometimes manifest as Bartter syndrome (BS)-like hypokalemic alkalosis. Therefore, the ability of doctors to differentiate CF from BS-like hypokalemic alkalosis in Chinese infants is a great challenge in the timely and accurate diagnosis of CF. In China, sporadic CF has not been diagnosed in children younger than three years of age to date. Three infants, who were initially admitted to our hospital over the period of June 2013 to September 2014 with BS-like hypokalemic alkalosis, were diagnosed with CF through exome sequencing and sweat chloride measurement. The compound heterozygous mutations of the CFTR gene were detected in two infants, and a homozygous missense mutation was found in one infant. Among the six identified mutations, two are novel point mutations (c.1526G > C and c.3062C > T) that are possibly pathogenic. The three infants are the youngest Chinese patients to have been diagnosed with sporadic CF at a very early stage. Follow-up examination showed that all of the cases remained symptom-free after early intervention, indicating the potential benefit of very early diagnosis and timely intervention in children with CF. Our results demonstrate the necessity of distinguishing CF from BS in Chinese infants with hypokalemic alkalosis and the significant diagnostic value of powerful exome sequencing for rare genetic diseases. Furthermore, our findings expand the CFTR mutation spectrum associated with CF.

摘要

囊性纤维化(CF)是一种由 CF 跨膜电导调节因子(CFTR)基因突变引起的致命常染色体隐性遗传病。CF 的特征是反复发生伴有阻塞性肺病的肺部感染。CF 在白种人群中很常见,但在中国人中很少见。早期 CF 的症状通常不典型,有时可能表现为 Bartter 综合征(BS)样低钾性碱中毒。因此,医生区分中国婴儿 CF 和 BS 样低钾性碱中毒的能力是及时准确诊断 CF 的巨大挑战。在中国,迄今为止尚未在 3 岁以下儿童中诊断出散发性 CF。2013 年 6 月至 2014 年 9 月期间,我院最初收治了 3 例具有 BS 样低钾性碱中毒的婴儿,通过外显子组测序和汗液氯测量,这 3 例患儿被诊断为 CF。在 2 例患儿中检测到 CFTR 基因突变的复合杂合子,在 1 例患儿中发现纯合错义突变。在鉴定的 6 个突变中,有 2 个是可能致病的新的点突变(c.1526G > C 和 c.3062C > T)。这 3 例患儿是中国最早在极早期确诊的散发性 CF 婴儿。随访检查显示,所有病例在早期干预后均无症状,表明对 CF 患儿进行早期诊断和及时干预具有潜在益处。我们的结果表明,在中国低钾性碱中毒的婴儿中,有必要将 CF 与 BS 区分开来,强大的外显子组测序对罕见遗传病具有重要的诊断价值。此外,我们的研究结果扩展了与 CF 相关的 CFTR 突变谱。

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