Pinapala A, Garg M, Kamath N, Iyengar A
Department of Pediatric Nephrology, St. John's Medical College Hospital, Bengaluru, Karnataka, India.
Indian J Nephrol. 2017 May-Jun;27(3):222-224. doi: 10.4103/0971-4065.202831.
Primary hyperoxaluria (PH) has heterogeneous renal manifestations in infants and children. This often leads to delay in diagnosis. In the past 3 years, genetic samples were sent for seven children with a clinical diagnosis of PH. Their medical records were reviewed for clinical presentation and outcomes. Of the seven children, three were males. The median age of presentation was 4.9 years with the youngest presenting at 3 months of age. Nephrolithiasis, the most common presentation was associated with renal dysfunction in two children. Two children with no significant history presented in end-stage renal disease (ESRD). The sibling of one of the children in ESRD, with a history of consanguinity in parents, was screened for asymptomatic nephrolithiasis. Bilateral multiple renal calculi were found in majority of children followed by echogenic kidneys on ultrasound examination. Genetic analysis suggested PH Type 1 in five children and type 2 in two children. The mutations detected in our cohort were different from the previously reported common mutations. There was no obvious genotype-phenotype correlation noticed. Three children in ESRD are on maintenance dialysis. Nephrolithiasis being a common presentation of PH needs prompt evaluation. Mutations are generally population specific, and whole gene sequence analysis is critical in diagnosis.
原发性高草酸尿症(PH)在婴幼儿和儿童中具有异质性的肾脏表现。这常常导致诊断延迟。在过去3年中,我们对7例临床诊断为PH的儿童进行了基因样本检测。我们回顾了他们的病历,以了解临床表现和治疗结果。这7名儿童中,3名是男性。发病的中位年龄为4.9岁,最小的在3个月大时发病。肾结石是最常见的表现,在2名儿童中与肾功能不全有关。2名无明显病史的儿童以终末期肾病(ESRD)就诊。ESRD患儿之一的同胞,其父母有近亲结婚史,接受了无症状肾结石筛查。大多数儿童在超声检查中发现双侧多发性肾结石,其次是肾回声增强。基因分析表明,5名儿童为1型PH,2名儿童为2型PH。我们队列中检测到的突变与先前报道的常见突变不同。未发现明显的基因型-表型相关性。3名ESRD患儿正在接受维持性透析。肾结石作为PH的常见表现需要及时评估。突变通常具有人群特异性,全基因序列分析对诊断至关重要。