Kouri Anne M, Andreoli Sharon P
Department of Pediatric Nephrology, Indiana University School of Medicine, 699 Riley Hospital Drive, Rm 230, Indianapolis, IN, 46202, USA.
Pediatr Nephrol. 2017 Mar;32(3):449-455. doi: 10.1007/s00467-016-3490-6. Epub 2016 Sep 27.
Antineutrophil cytoplasmic antibody (ANCA)-associated vasculitis is a small- and medium-sized vasculitis classically seen in adult patients, with peak onset near the fifth to seventh decade of life. There is little data on ANCA-associated vasculitis in pediatric patients, and most studies have limited follow-up.
This is a retrospective chart review of 22 patients with ANCA-positive glomerulonephritis in a single institution from 1991 to 2013.
Of the 22 patients, eight (36 %) required renal replacement therapy (RRT) at diagnosis; four of these patients recovered sufficient renal function to initially discontinue dialysis. Five patients (23 %) were treated with plasmapheresis at presentation. The median time from presentation until first clinical or serologic relapse was 1.7 ± 1.2 years. After a median follow-up of 5.8 years, just over half of our patients had chronic kidney disease (CKD) stages 1-3 (55 %). Seven (32 %) patients progressed to end-stage renal disease (ESRD) and eventually required kidney transplant.
ANCA-associated glomerulonephritis is a rare disorder in children. Presentation and outcomes vary significantly among patients. More research is required to follow these patients who are diagnosed in childhood to further characterize the long-term outcome of the disease.
抗中性粒细胞胞浆抗体(ANCA)相关血管炎是一种主要见于成年患者的中小血管炎,发病高峰在50至70岁左右。关于儿童患者ANCA相关血管炎的数据很少,且大多数研究的随访时间有限。
这是一项对1991年至2013年在单一机构确诊的22例ANCA阳性肾小球肾炎患者的回顾性病历审查。
22例患者中,8例(36%)在诊断时需要肾脏替代治疗(RRT);其中4例患者肾功能恢复到足以最初停止透析。5例患者(23%)在就诊时接受了血浆置换治疗。从就诊到首次临床或血清学复发的中位时间为1.7±1.2年。中位随访5.8年后,略超过一半的患者处于慢性肾脏病(CKD)1至3期(55%)。7例(32%)患者进展至终末期肾病(ESRD),最终需要进行肾移植。
ANCA相关肾小球肾炎在儿童中是一种罕见疾病。患者的表现和预后差异很大。需要更多研究来跟踪这些童年确诊的患者,以进一步明确该疾病的长期预后。