Toyoda Junya, Adachi Masanori, Ochi Ayako, Okada Yuki, Honda Aiko, Mizuno Katsumi, Nozu Kandai
Department of Pediatrics, Showa University School of Medicine, Tokyo, Japan.
Department of Pediatrics, Kobe University Graduate School of Medicine, Kobe, Japan.
Clin Pediatr Endocrinol. 2023;32(3):155-160. doi: 10.1297/cpe.2022-0069. Epub 2023 Mar 19.
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.
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。