一名囊性纤维化患者的复发性症状性尿路结石。

IF 2.6 3区 医学 Q1 PEDIATRICS
Pediatric Nephrology Pub Date : 2024-12-01 Epub Date: 2024-06-25 DOI:10.1007/s00467-024-06433-2
Sibel Yel, Ismail Dursun, Mehmet Köse, Aslıhan Kiraz, Muammer Hakan Poyrazoglu, Munis Dündar
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引用次数: 0

摘要

一名 6 个月大的女孩曾被诊断患有囊性纤维化(CF),因肾结石入院。她的父母是一级表亲。患者接受了内窥镜结石治疗。尽管患者没有结石家族史,也接受了枸橼酸钾治疗,但在随访期间,患者反复出现肾结石和非典型尿路感染。基本检查均正常。由于是近亲结婚,且肾结石出现较早,因此考虑了胱氨酸尿症和高草酸尿症等代谢性病因。由于胱氨酸水平正常,胱氨酸尿症被排除。由于 CF 患者存在吸收性(继发性)高草酸尿症,因此尿草酸排泄量较高。该患者有早期结石负担,且有治疗史和近亲结婚史,因此我们对其进行了基因检测。基因检测发现,AGXT 基因第 1 外显子存在一个错义同源变异。患者被诊断为原发性高草酸尿症 1 型。在同一个孩子身上同时发现了两种危及生命的罕见遗传病。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Recurrent symptomatic urolithiasis in a patient with cystic fibrosis.

A 6-month-old girl, previously diagnosed with cystic fibrosis (CF), was admitted to hospital for nephrolithiasis. Her parents were first-degree cousins. The patient underwent endoscopic stone management. Despite no family history of stones and medical treatment with potassium citrate, the patient developed recurrent renal stones and atypical urinary tract infections during follow-up. Basic investigations were all normal. Due to consanguinity and early presentation of nephrolithiasis, metabolic causes such as cystinuria and hyperoxaluria were considered. Cystinuria was excluded due to normal cystine levels. High urinary oxalate excretion was found as expected due to absorptive (secondary) hyperoxaluria in CF patients. An early stone burden in the patient with a history of medical treatment and consanguinity led us to perform a genetic testing. Genetic testing revealed a missense homozygous variant in exon 1 of the AGXT gene. The patient was diagnosed with primary hyperoxaluria type 1. Two rare life-threatening genetic diseases were found together in the same child.

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来源期刊
Pediatric Nephrology
Pediatric Nephrology 医学-泌尿学与肾脏学
CiteScore
4.70
自引率
20.00%
发文量
465
审稿时长
1 months
期刊介绍: International Pediatric Nephrology Association Pediatric Nephrology publishes original clinical research related to acute and chronic diseases that affect renal function, blood pressure, and fluid and electrolyte disorders in children. Studies may involve medical, surgical, nutritional, physiologic, biochemical, genetic, pathologic or immunologic aspects of disease, imaging techniques or consequences of acute or chronic kidney disease. There are 12 issues per year that contain Editorial Commentaries, Reviews, Educational Reviews, Original Articles, Brief Reports, Rapid Communications, Clinical Quizzes, and Letters to the Editors.
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