Pasini Agneza Marija, Gagro Alenka, Roić Goran, Vrdoljak Ozren, Lujić Lucija, Žutelija-Fattorini Matija
Children's Hospital Zagreb, Zagreb, Croatia
Children's Hospital Zagreb, Zagreb, Croatia.
Pediatrics. 2017 Feb;139(2). doi: 10.1542/peds.2016-1279. Epub 2017 Jan 12.
We report, to the best of our knowledge, the first case of a child with typical ataxia telangiectasia (A-T) who developed juvenile idiopathic arthritis (JIA). The patient was a 15-year-old boy with A-T who presented with noninfectious polyarthritis. A-T is a rare, autosomal recessive disorder characterized by cerebellar atrophy, oculocutaneous telangiectasia, immunodeficiency, radiosensitivity, and predisposition to cancer. The gene responsible for A-T is the A-T mutated (ATM) gene. Clinical manifestations of the disorder are the result of lacking ATM protein, which is involved in DNA repair, apoptosis, various checkpoints in the cell cycle, gene regulation, translation, initiation, and telomere maintenance. There are a few articles that describe deficiency of the DNA repair enzyme, ATM, in rheumatoid arthritis, but the connection between the absence of ATM protein and JIA has not been presented or studied yet. JIA is a heterogeneous group of diseases characterized by arthritis of unknown origin with onset before the age of 16 years. It is the most common childhood chronic rheumatic disease and causes significant disability. Because immunodeficiency can be part of A-T, infectious arthritis can occur, but chronic autoimmune arthritis in these patients is rare. We report a rare case of a 15-year-old boy with A-T and JIA. This case shows a possible relationship between altered function of ATM protein and the pathogenesis of JIA.
据我们所知,我们报告了首例患有典型共济失调毛细血管扩张症(A-T)并发展为幼年特发性关节炎(JIA)的儿童病例。该患者是一名患有A-T的15岁男孩,表现为非感染性多关节炎。A-T是一种罕见的常染色体隐性疾病,其特征为小脑萎缩、眼皮肤毛细血管扩张、免疫缺陷、放射敏感性以及易患癌症。导致A-T的基因是A-T突变(ATM)基因。该疾病的临床表现是由于缺乏ATM蛋白所致,ATM蛋白参与DNA修复、细胞凋亡、细胞周期的各种检查点、基因调控、翻译起始以及端粒维持。有几篇文章描述了类风湿性关节炎中DNA修复酶ATM的缺乏,但ATM蛋白缺失与JIA之间的联系尚未被提出或研究。JIA是一组异质性疾病,其特征为16岁之前发病的不明原因关节炎。它是儿童期最常见的慢性风湿性疾病,会导致严重残疾。由于免疫缺陷可能是A-T的一部分,因此可能会发生感染性关节炎,但这些患者中的慢性自身免疫性关节炎很少见。我们报告了一例15岁患有A-T和JIA的男孩的罕见病例。该病例显示了ATM蛋白功能改变与JIA发病机制之间可能存在的关系。