Rodriguez Mier Noelia, Antoons Virginie, Cuyx Senne, Santo Ramalho Anabela, Boon Mieke, Proesmans Marijke, Mekahli Djalila, Vermeulen François
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.
J Cyst Fibros. 2025 Mar;24(2):401-403. doi: 10.1016/j.jcf.2024.10.007. Epub 2024 Oct 31.
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.
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。