Department of Pediatrics, Kayseri City Hospital, Kayseri, Turkey.
Department of Pediatric Nephrology, Kayseri City Hospital, Kayseri, Turkey.
Eur J Pediatr. 2021 Dec;180(12):3477-3482. doi: 10.1007/s00431-021-04144-6. Epub 2021 Jun 6.
Neurofibromatosis type 1 (NF1) is an autosomal-dominant neurocutaneous syndrome affecting various parts of the body, including the renovascular and urinary systems. We evaluated the renovascular, urinary, glomerular, and tubular functions of children with NF1. We compared blood pressures, urinary findings, and renal glomerular and tubular functions in children with NF1 with those of a healthy age- and gender-matched control group. We evaluated 46 NF1 patients and 33 healthy controls. The mean ages of the NF1 group (female/male: 20/26) and the control group (female/male: 15/18) were 10.1 ± 4.6 and 10.6 ± 4.3 years respectively. Six NF1 patients were hypertensive. The mean blood pressures of the NF1 group were significantly higher than those of the control group. Renal artery stenosis was detected in one NF1 patient. Urinary tract anomalies were evident in 21.7% of NF1 but only 9% of control subjects. The mean estimated glomerular filtration rate (eGFR) of the NF1 group was significantly lower than that of the control group. Six NF1 patients evidenced eGFRs < 90 mL/min. In the NF1 group, tubular phosphorus reabsorption was significantly lower and uric acid excretion significantly higher than in the control group.Conclusion: Hypertension, urinary tract anomalies, and impaired renal function were more common in NF1 patients than healthy controls. Regular blood pressure measurements and evaluation of urinary tract and kidney function are essential for NF1 patients. What is Known: • NF1 is most commonly associated with systemic hypertension due to renal artery vasculopathy and the development of a pheochromocytoma. • Hydronephrosis and bladder involvement have been documented in NF1. What is New: • Renal glomerular and tubular functions may be affected in NF1.
神经纤维瘤病 1 型(NF1)是一种常染色体显性遗传性神经皮肤综合征,影响身体的各个部位,包括肾血管和泌尿系统。我们评估了 NF1 患儿的肾血管、泌尿系统、肾小球和肾小管功能。我们比较了 NF1 患儿与健康年龄和性别匹配对照组的血压、尿液结果以及肾肾小球和肾小管功能。我们评估了 46 名 NF1 患者和 33 名健康对照组。NF1 组(女性/男性:20/26)和对照组(女性/男性:15/18)的平均年龄分别为 10.1 ± 4.6 岁和 10.6 ± 4.3 岁。6 名 NF1 患者患有高血压。NF1 组的平均血压明显高于对照组。1 名 NF1 患者检测到肾动脉狭窄。NF1 中有 21.7%存在尿路异常,而对照组仅有 9%。NF1 组的平均估计肾小球滤过率(eGFR)明显低于对照组。6 名 NF1 患者的 eGFR<90 mL/min。在 NF1 组中,磷重吸收率明显降低,尿酸排泄明显升高。结论:与健康对照组相比,NF1 患者更常见高血压、尿路异常和肾功能受损。NF1 患者需要定期测量血压,并评估尿路和肾功能。已知:• NF1 最常与肾动脉血管病变和嗜铬细胞瘤的发展引起的全身高血压相关。• NF1 中已记录到肾积水和膀胱受累。新发现:• NF1 可能会影响肾肾小球和肾小管功能。