La Bella Saverio, Vivarelli Marina, Di Ludovico Armando, Di Donato Giulia, Chiarelli Francesco, Breda Luciana
Department of Pediatrics, University "G. D'Annunzio" of Chieti-Pescara, Chieti, Italy.
Division of Nephrology, Laboratory of Nephrology, Bambino Gesù Children's Hospital IRCCS, Rome, Italy.
Pediatr Nephrol. 2024 Mar;39(3):711-721. doi: 10.1007/s00467-023-06135-1. Epub 2023 Aug 28.
Approximately 1% of all patients with Sjögren's syndrome (SS) are children. Unlike the adult form, in which sicca syndrome is the main presentation, in children, the most common clinical finding is recurrent enlargement of the salivary glands. In pediatric SS, extraglandular manifestations represent a significant feature and, among these, kidney manifestations are relevant. Kidney involvement is observed in 5-20.5% of children with SS, most frequently tubulointerstitial nephritis. This injury can lead to serious phenotypes, including distal kidney tubular acidosis with the development of severe hypokalemia, which can lead to ECG abnormalities, weakness, and hypokalemic periodic paralysis. Kidney implications in pediatric SS also include nephrolithiasis, nephrocalcinosis, and various types of glomerular damage, which often require immunosuppressive therapies. Laboratory findings are usually comparable to adults, including hyperglobulinemia and high rates of antinuclear antibodies (ANA, 63.6-96.2%), and anti-Ro/SSA (36.4-84.6%). The current classification criteria for SS are inaccurate for the pediatric population, and more specific criteria are needed to improve the diagnostic rate. Due to the rarity of the disease, strong recommendations for treatment are lacking, and several therapeutic strategies have been reported, mostly based on glucocorticoids and disease-modifying antirheumatic drugs, with different outcomes. The aim of this paper is to provide an overview of the kidney implications of pediatric SS based on the latest evidence of the medical literature.
约1%的干燥综合征(SS)患者为儿童。与以干燥综合征为主要表现的成人形式不同,儿童最常见的临床发现是唾液腺反复肿大。在儿童SS中,腺外表现是一个显著特征,其中肾脏表现较为重要。5 - 20.5%的儿童SS患者存在肾脏受累,最常见的是肾小管间质性肾炎。这种损伤可导致严重的表型,包括远端肾小管酸中毒并伴有严重低钾血症的发生,进而可导致心电图异常、肌无力和低钾性周期性麻痹。儿童SS的肾脏影响还包括肾结石、肾钙质沉着症以及各种类型的肾小球损害,这些情况通常需要免疫抑制治疗。实验室检查结果通常与成人相似,包括高球蛋白血症以及高比例的抗核抗体(ANA,63.6 - 96.2%)和抗Ro/SSA(36.4 - 84.6%)。目前SS的分类标准对儿童群体并不准确,需要更具体的标准来提高诊断率。由于该疾病罕见,缺乏强有力的治疗推荐,并且已经报道了几种治疗策略,大多基于糖皮质激素和改善病情的抗风湿药物,但疗效各异。本文旨在根据医学文献的最新证据,对儿童SS的肾脏影响进行概述。