无明显基础疾病的婴儿伪巴特综合征1例报告。

IF 1 Q4 ENDOCRINOLOGY & METABOLISM Clinical Pediatric Endocrinology Pub Date : 2023-01-01 DOI:10.1297/cpe.2022-0069
Junya Toyoda, Masanori Adachi, Ayako Ochi, Yuki Okada, Aiko Honda, Katsumi Mizuno, Kandai Nozu
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引用次数: 0

摘要

伪巴特尔综合征(PBS)的发展是由于肾脏或肾外氯流失,导致低钾性碱中毒。虽然大多数成人病例是由于滥用利尿剂/泻药,但许多婴儿病例是继发于囊性纤维化。很少,婴儿PBS是由肾/尿路异常或遗传性疾病(如Dent病)引起的肾盐丢失引起的。在这里,我们报告了一个10个月大的女孩,一个月的配方奶粉摄入量减少,体重下降5.6%。她表现出典型的实验室检查结果,包括低钾血症(2.7 mEq/L)和高碳酸氢盐水平(32.7 mEq/L),血浆肾素活性为399 ng/mL/h。在少量补充钾和钠的情况下,体重指数从-1.13 SD改善到+0.52 SD,在大约一个月内获得了完全分辨率的实验室数据。未发现Bartter-Gitelman综合征候选基因的致病突变。由于重度次氯尿(< 15 mEq/L),不太可能有肾源性PBS。此外,由于患者从未表现出胃肠道症状,因此似乎并未出现肝外氯流失。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,如细微的肾渗漏,可能是我们患者PBS的原因。
本文章由计算机程序翻译,如有差异,请以英文原文为准。

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

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.

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来源期刊
Clinical Pediatric Endocrinology
Clinical Pediatric Endocrinology ENDOCRINOLOGY & METABOLISM-
CiteScore
2.40
自引率
7.10%
发文量
34
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