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无明显潜在病因的婴儿假性巴特综合征:一例报告

Pseudo-Bartter syndrome in an infant without obvious underlying conditions: A case report.

作者信息

Toyoda Junya, Adachi Masanori, Ochi Ayako, Okada Yuki, Honda Aiko, Mizuno Katsumi, Nozu Kandai

机构信息

Department of Pediatrics, Showa University School of Medicine, Tokyo, Japan.

Department of Pediatrics, Kobe University Graduate School of Medicine, Kobe, Japan.

出版信息

Clin Pediatr Endocrinol. 2023;32(3):155-160. doi: 10.1297/cpe.2022-0069. Epub 2023 Mar 19.

DOI:10.1297/cpe.2022-0069
PMID:37362166
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC10288294/
Abstract

Pseudo-Bartter syndrome (PBS) develops owing to renal or extrarenal chloride loss, leading to hypokalemic alkalosis. Whereas most adult cases result from diuretic/laxative abuse, many infantile cases occur secondary to cystic fibrosis. Rarely, infantile PBS is caused by renal salt loss with anomalies of the kidney/urinary tract or genetic disorders, such as Dent disease. Here, we report the case of a 10-mo-old girl with a one-month history of decreased formula intake and 5.6% body weight loss. She showed typical laboratory findings as PBS, including hypokalemia (2.7 mEq/L) and high levels of bicarbonate (32.7 mEq/L) with a plasma renin activity of 399 ng/mL/h. With minimum supplementation of potassium and sodium, an improvement in body mass index, from -1.13 SD to +0.52 SD, with complete resolution of laboratory data was obtained in approximately one month. No causative mutations were identified in candidate genes for Bartter-Gitelman syndrome. Due to profound hypochloruria (< 15 mEq/L), PBS of renal origin was unlikely. In addition, extrarenal chloride loss did not seem to be the case, because the patient never manifested gastrointestinal symptoms. Therefore, we speculate that a temporary decrease in chloride intake, coupled with the putative genetic/epigenetic disadvantage of chloride retention, such as a subtle renal leak, may be responsible for the PBS in our patient.

摘要

假性巴特综合征(PBS)是由于肾脏或肾外氯丢失导致低钾性碱中毒。大多数成人病例是由利尿剂/泻药滥用引起的,而许多婴儿病例继发于囊性纤维化。婴儿期PBS很少由肾脏盐丢失伴肾脏/泌尿道异常或遗传性疾病(如丹特病)引起。在此,我们报告一例10个月大的女孩,有1个月配方奶摄入量减少及体重减轻5.6%的病史。她表现出PBS典型的实验室检查结果,包括低钾血症(2.7 mEq/L)、高碳酸氢盐水平(32.7 mEq/L),血浆肾素活性为399 ng/mL/h。通过最低限度的钾和钠补充,大约1个月内体重指数从-1.13标准差改善到+0.52标准差,实验室数据完全恢复正常。在巴特-吉特曼综合征候选基因中未发现致病突变。由于严重的低氯尿症(<15 mEq/L),不太可能是肾源性PBS。此外,肾外氯丢失似乎也不成立,因为该患者从未表现出胃肠道症状。因此,我们推测氯摄入量的暂时减少,加上氯潴留的假定遗传/表观遗传缺陷,如轻微的肾渗漏,可能是我们患者发生PBS的原因。

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本文引用的文献

1
Bartter-Like Syndrome as the Initial Presentation of Dent Disease 1: A Case Report.巴特综合征样表现作为1型丹特病的首发症状:一例报告
Front Pediatr. 2021 Sep 28;9:725251. doi: 10.3389/fped.2021.725251. eCollection 2021.
2
Adrenal steroids reference ranges in infancy determined by LC-MS/MS.用 LC-MS/MS 测定婴儿期肾上腺类固醇的参考范围。
Pediatr Res. 2022 Jul;92(1):265-274. doi: 10.1038/s41390-021-01739-5. Epub 2021 Sep 23.
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Diagnosis and management of Bartter syndrome: executive summary of the consensus and recommendations from the European Rare Kidney Disease Reference Network Working Group for Tubular Disorders.巴特综合征的诊断与管理:欧洲罕见肾脏病参考网络肾小管疾病工作组的共识与建议执行摘要
Kidney Int. 2021 Feb;99(2):324-335. doi: 10.1016/j.kint.2020.10.035.
4
A neonate with intrauterine growth restriction and pseudo-Bartter syndrome due to severe maternal eating disorder: A case report.一名因母亲严重饮食失调导致宫内生长受限和假性巴特综合征的新生儿:病例报告。
Clin Case Rep. 2020 Aug 6;8(12):2541-2544. doi: 10.1002/ccr3.3223. eCollection 2020 Dec.
5
Dietary Chloride Deficiency Syndrome: Pathophysiology, History, and Systematic Literature Review.膳食氯缺乏综合征:病理生理学、历史及系统文献综述
Nutrients. 2020 Nov 9;12(11):3436. doi: 10.3390/nu12113436.
6
Cystic Fibrosis Presenting as Pseudo-Bartter Syndrome: An Important Diagnosis that is Missed!囊性纤维化表现为假性巴特综合征:一个被忽视的重要诊断!
Indian J Pediatr. 2020 Sep;87(9):726-732. doi: 10.1007/s12098-020-03342-8. Epub 2020 Jun 5.
7
The hypokalemia mystery: distinguishing Gitelman and Bartter syndromes from 'pseudo-Bartter syndrome'.低钾血症之谜:区分吉特曼综合征和巴特综合征与“假性巴特综合征”。
Nephrol Dial Transplant. 2021 Dec 31;37(1):29-30. doi: 10.1093/ndt/gfaa100.
8
Inherited salt-losing tubulopathy: An old condition but a new category of tubulopathy.遗传性失盐性肾小管病:一种古老的疾病,但却是一种新的肾小管病类别。
Pediatr Int. 2020 Apr;62(4):428-437. doi: 10.1111/ped.14089. Epub 2020 Apr 13.
9
Mimicry and well known genetic friends: molecular diagnosis in an Iranian cohort of suspected Bartter syndrome and proposition of an algorithm for clinical differential diagnosis.拟态和知名遗传伙伴:疑似 Bartter 综合征伊朗队列的分子诊断及临床鉴别诊断算法的提出。
Orphanet J Rare Dis. 2019 Feb 13;14(1):41. doi: 10.1186/s13023-018-0981-5.
10
Congenital chloride diarrhea needs to be distinguished from Bartter and Gitelman syndrome.先天性氯性腹泻需要与巴特综合征和 Gitelman 综合征相鉴别。
J Hum Genet. 2018 Jul;63(8):887-892. doi: 10.1038/s10038-018-0470-7. Epub 2018 May 30.