Priyadarshini Sukanya, Sinha Aditi, Jana Manisha, Tandon Radhika, Sikka Kapil, Mathur Vijay Prakash, Bhatt Girish Chandra, Yadav Menka, Meena Jitendra Kumar, Khandelwal Priyanka, Hari Pankaj, Bagga Arvind
Division of Nephrology, Department of Pediatrics, All India Institute of Medical Sciences, New Delhi, 110029, India.
Department of Radiodiagnosis, All India Institute of Medical Sciences, New Delhi, India.
Pediatr Nephrol. 2025 Apr 15. doi: 10.1007/s00467-025-06747-9.
This study investigates the etiology, outcomes, and genotype-phenotype correlations in patients with renal tubular acidosis (RTA) at a tertiary care center in New Delhi.
This cross-sectional study included children and young adults with RTA who underwent clinical, biochemical, radiological and/or genetic evaluations between July 2020 and December 2024. We report clinical phenotype, anthropometry, metabolic control and progression to chronic kidney disease (CKD) in relation to genotype of distal RTA.
Of 135 patients enrolled, 69 had distal RTA. The yield of genetic testing was 72% in distal RTA and 88.7% in Fanconi syndrome. Variants in SLC4A1 (42.4%) and ATP6V1B1 (28.8%) were the most common etiologies of distal RTA. Compared to other etiologies, patients with SLC4A1 variants were older at symptom onset (P = 0.008). Hematological abnormalities were more frequent in patients with biallelic compared to heterozygous SLC4A1 variants (50% vs. 12.5%; P = 0.18). Nephrocalcinosis and metabolic control were similarly prevalent across genetic categories of distal RTA. Sensorineural hearing loss was more common with ATP6V1B1 than with ATP6V0A4 variants (61.5% vs. 22.2%, P = 0.099) and did not vary by metabolic control. At median follow-up of 5-years, 74.1% of patients with distal RTA had short stature, 74.6% had poor metabolic control and 2.9% had progressed to CKD G3-5.
This study outlines the genetic etiology and phenotype of distal RTA in south Asia. Over short-term follow-up, poor metabolic control and severe stunting were common, while CKD was uncommon.
本研究在新德里的一家三级医疗中心调查肾小管酸中毒(RTA)患者的病因、结局以及基因型-表型相关性。
这项横断面研究纳入了2020年7月至2024年12月期间接受临床、生化、放射学和/或基因评估的RTA儿童和青年。我们报告了与远端RTA基因型相关的临床表型、人体测量学、代谢控制情况以及慢性肾脏病(CKD)的进展情况。
在纳入的135例患者中,69例患有远端RTA。远端RTA的基因检测阳性率为72%,范可尼综合征为88.7%。SLC4A1(42.4%)和ATP6V1B1(28.8%)的变异是远端RTA最常见的病因。与其他病因相比,SLC4A1变异患者症状出现时年龄更大(P = 0.008)。与杂合子SLC4A1变异患者相比,双等位基因SLC4A1变异患者血液学异常更常见(50% 对12.5%;P = 0.18)。远端RTA的不同基因类别中,肾钙质沉着症和代谢控制情况同样普遍。与ATP6V0A4变异相比,ATP6V1B1变异导致的感音神经性听力损失更常见(61.5% 对22.2%,P = 0.099),且与代谢控制无关。在中位随访5年时,74.1%的远端RTA患者身材矮小,74.6%的患者代谢控制不佳,2.9%的患者进展为CKD G3-5期。
本研究概述了南亚地区远端RTA的遗传病因和表型。在短期随访中,代谢控制不佳和严重发育迟缓很常见,而CKD并不常见。