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假性巴特综合征:一种与囊性纤维化跨膜传导调节因子相关的疾病?

Pseudo-Bartter syndrome: A CFTR-related disorder?

作者信息

Rodriguez Mier Noelia, Antoons Virginie, Cuyx Senne, Santo Ramalho Anabela, Boon Mieke, Proesmans Marijke, Mekahli Djalila, Vermeulen François

机构信息

Department of Pediatrics, Pediatric Pulmonology, University Hospital of Leuven, Leuven, Belgium; Department of Development and Regeneration, Woman and Child Unit, CF Research Lab, KU Leuven, Leuven, Belgium.

Department of Pediatrics, Pediatric Nephrology, University Hospital of Leuven, Leuven, Belgium.

出版信息

J Cyst Fibros. 2025 Mar;24(2):401-403. doi: 10.1016/j.jcf.2024.10.007. Epub 2024 Oct 31.

Abstract

This case report presents a 14-month-old boy with a history of cystic fibrosis (CF) carrier status, diagnosed following a positive newborn screening for CF (CF-NBS), who developed symptoms suggestive of Pseudo-Bartter syndrome (PBS). Despite initial evaluations not meeting CF diagnostic criteria, subsequent investigations revealed an intermediate sweat chloride concentration, a second CFTR mutation, and CFTR dysfunction through rectal organoid morphology analysis (ROMA) consistent with CFTR-related disorder (CFTR-RD). This case raises important considerations regarding the diagnosis and management of CFTR-RD. PBS can be considered as a rare presentation of CFTR-RD and can occur in children with sweat chloride below the CF range. Functional testing of CFTR by ROMA enabled a more accurate diagnosis. Despite the negative work-up after CF-NBS, this infant developed CFTR-RD, but this should not be considered as a screen failure. Follow-up of children with CFTR-RD at a CF centre is preferred, because of the risk of developing CF.

摘要

本病例报告介绍了一名14个月大的男孩,其有囊性纤维化(CF)携带者状态史,在新生儿CF筛查(CF-NBS)呈阳性后被诊断出来,该男孩出现了提示假性巴特综合征(PBS)的症状。尽管初始评估未达到CF诊断标准,但后续调查显示出汗氯化物浓度处于中间值,存在第二个CFTR突变,并且通过直肠类器官形态分析(ROMA)发现CFTR功能障碍,这与CFTR相关疾病(CFTR-RD)一致。该病例引发了关于CFTR-RD诊断和管理的重要思考。PBS可被视为CFTR-RD的一种罕见表现,可发生在出汗氯化物低于CF范围的儿童中。通过ROMA对CFTR进行功能测试能够实现更准确的诊断。尽管CF-NBS后的检查结果为阴性,但该婴儿仍患上了CFTR-RD,但这不应被视为筛查失败。由于存在发展为CF的风险,CFTR-RD患儿最好在CF中心进行随访。

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