Butbul-Aviel Yonatan, Uziel Yosef, Hezkelo Nofar, Brik Riva, Amarilyo Gil
Department of Pediatrics B, Ruth Rappaport Children's Hospital, Rambam Medical Center, Haifa, Israel.
Pediatric Rheumatology Service, Ruth Rappaport Children's Hospital, Rambam Medical Center, Haifa, Israel.
Pediatr Rheumatol Online J. 2018 Feb 13;16(1):12. doi: 10.1186/s12969-018-0227-z.
Palindromic rheumatism is an idiopathic, periodic arthritis characterized by multiple, transient, recurring episodes. Palindromic rheumatism is well-characterized in adults, but has never been reported in pediatric populations. The aim of this study was to characterize the clinical features and outcomes of a series of pediatric patients with palindromic rheumatism.
We defined clinical criteria for palindromic rheumatism and reviewed all clinical visits in three Pediatric Rheumatology centers in Israel from 2006through 2015, to identify patients with the disease. We collected retrospective clinical and laboratory data on patients who fulfilled the criteria, and reviewed their medical records in order to determine the proportion of patients who had developed juvenile idiopathic arthritis.
Overall, 10 patients were identified. Their mean age at diagnosis was 8.3 ± 4.5 years and the average follow-up was 3.8 ± 2.7 years. The mean duration of attacks was 12.2 ± 8.4 days. The most frequently involved joints were knees. Patients tested positive for rheumatoid factor in 20% of cases. One patient developed polyarticular juvenile idiopathic arthritis after three years of follow-up, six patients (60%) continued to have attacks at their last follow-up and only three children (30%) achieved long-term remission.
Progression to juvenile idiopathic arthritis is rare amongst children with palindromic rheumatism and most patients continued to have attacks at their last follow-up. Longer follow-up periods are required to predict the long-term outcomes of pediatric patients with palindromic rheumatism.
回纹型风湿症是一种特发性、周期性关节炎，其特征为多次短暂复发发作。回纹型风湿症在成人中已有充分描述，但从未在儿科人群中报道过。本研究的目的是描述一系列回纹型风湿症儿科患者的临床特征和结局。
我们定义了回纹型风湿症的临床标准，并回顾了2006年至2015年以色列三个儿科风湿病中心的所有临床就诊记录，以确定患有该疾病的患者。我们收集了符合标准患者的回顾性临床和实验室数据，并查阅了他们的病历，以确定发展为幼年特发性关节炎的患者比例。
总共确定了10例患者。他们诊断时的平均年龄为8.3±4.5岁，平均随访时间为3.8±2.7年。发作的平均持续时间为12.2±8.4天。最常受累的关节是膝关节。20%的病例类风湿因子检测呈阳性。一名患者在随访三年后发展为多关节型幼年特发性关节炎，六名患者（60%）在最后一次随访时仍有发作，只有三名儿童（30%）实现了长期缓解。
在回纹型风湿症儿童中进展为幼年特发性关节炎的情况很少见，大多数患者在最后一次随访时仍有发作。需要更长的随访期来预测回纹型风湿症儿科患者的长期结局。
