Department of Pediatrics, University of Torino, Piazza Polonia 94, 10126, Torino, Italy.
Pediatr Nephrol. 2012 Mar;27(3):397-406. doi: 10.1007/s00467-011-2009-4. Epub 2011 Oct 21.
Beckwith-Wiedemann syndrome (BWS), an overgrowth disorder with several congenital abnormalities, encompasses nephrourological anomalies. The objective of the report is to analyze the latter and related genotype-phenotype correlations. The study was a retrospective review of nephrourological investigations and genotype in 67 BWS patients. Imaging and laboratory studies have been correlated with the molecular anomalies typical of BWS. Thirty-eight (56.7%) patients had a total of 61 nonmalignant nephrourological findings, including nephromegaly (n = 24), collecting system abnormalities (n = 14), cryptorchidism (n = 11), nephrolithiasis (n = 5), cysts (n = 5), and dysplasia (n = 1). Four patients had Wilms' tumor, all associated with renal hyperplasia. Renal findings were almost consistent in the BWS(IC1) group, with nephromegaly in all patients and collecting system abnormalities in half of them. BWS(UPD) and negative patients also had frequent anomalies (63.6% and 61.9% respectively), whereas only 36.0% of BWS(IC2) had renal findings (p = 0.003). Cryptorchidism was associated with abdominal wall defects (p < 0.001) appearing more frequently in BWS(IC2) (p = 0.028). Urinary tract infections were observed in 17.9% of patients, with two resulting in life-threatening sepsis. Hypercalciuria was present in 10% of cases. 55.5% of BWS patients have renal findings. Although variegate, these anomalies disclose a genotype-phenotype correlation.
贝-威二氏综合征(BWS)是一种伴有多种先天畸形的过度生长疾病,包括肾泌尿异常。本报告的目的是分析后者及其相关的基因型-表型相关性。本研究是对 67 例 BWS 患者的肾泌尿调查和基因型进行回顾性分析。影像学和实验室研究与 BWS 典型的分子异常相关。38 名(56.7%)患者共有 61 种非恶性肾泌尿异常,包括肾肥大(n = 24)、集合系统异常(n = 14)、隐睾(n = 11)、肾结石(n = 5)、囊肿(n = 5)和发育不良(n = 1)。4 例患者患有 Wilms 瘤,均与肾增生有关。BWS(IC1)组的肾异常几乎一致,所有患者均有肾肥大,半数患者有集合系统异常。BWS(UPD)和阴性患者也有频繁的异常(分别为 63.6%和 61.9%),而只有 36.0%的 BWS(IC2)有肾脏发现(p = 0.003)。隐睾与腹壁缺陷相关(p < 0.001),在 BWS(IC2)中更为常见(p = 0.028)。17.9%的患者发生尿路感染,其中 2 例导致危及生命的败血症。高钙尿症见于 10%的病例。55.5%的 BWS 患者有肾脏发现。尽管存在异质性,但这些异常揭示了一种基因型-表型相关性。