Maccora Ilaria, Orsini Sara Immacolata, Gallizzi Romina, Montin Davide, Cattalini Marco, La Torre Francesco, Spagnolo Alessandra, Diomeda Federico, Simonini Gabriele
Rheumatology Unit, ERN ReCONNET Center, Meyer Children's Hospital IRCCS, Viale Pieraccini 24, Florence 50139, Italy.
NeuroFARBA Department, University of Florence, Florence, Italy.
Ther Adv Musculoskelet Dis. 2024 Dec 6;16:1759720X241275822. doi: 10.1177/1759720X241275822. eCollection 2024.
Behçet disease (BD) is a rare disease in childhood and its uveitis may lead to blindness if not properly treated.
We aim to describe a cohort of paediatric BD patients with uveitis.
This is a multicentric retrospective study.
Six paediatric rheumatology units in Italy were involved including children with a diagnosis of paediatric BD according to the International Criteria for BD Criteria and/or to the International Study Group Criteria for BD, or Paediatric BD classification criteria if they had uveitis. Demographic, laboratory and clinical data were collected and followed up to March 2023. Ocular characteristics and treatment response were assessed according to Standardization Uveitis Nomenclature.
Among the 97 children with BD followed, 33 (34%) had uveitis (22 males, 66.7%). The median age at onset of BD and uveitis were, respectively, 11.5 years (2.5-17.1) and 11 years (3-17.3). Uveitis preceded BD diagnosis in 18 children (54.5%). Seventeen children had HLA B51 positivity (51.5%). Uveitis was bilateral in 25 (75.8%), and panuveitis in 16 (48.5%). All the patients received at least 1 systemic treatment for uveitis: 25 adalimumab, 2 tocilizumab, 1 abatacept, 3 infliximab, 4 azathioprine, 1 methotrexate and 1 corticosteroid. The remission was achieved with 30/35 treatments (85.7%) after a median time of 8 months (6-24). Six children had a relapse in therapy after the achievement of remission (20%). Fourteen patients stopped the therapy for persistent remission, but 5 relapsed (35.7%) after a median time of 9 months (range 1-48).
Uveitis in BD is a sight-threatening condition, and it is more frequently a panuveitis. Biologic treatments seem to be often required to control ocular inflammation.
白塞病(BD)在儿童中是一种罕见疾病,其葡萄膜炎若未得到恰当治疗可能导致失明。
我们旨在描述一组患有葡萄膜炎的儿童BD患者。
这是一项多中心回顾性研究。
意大利的六个儿科风湿病科参与其中,纳入了根据国际BD标准和/或国际BD研究组标准诊断为儿童BD的儿童,或者如果患有葡萄膜炎则根据儿科BD分类标准纳入的儿童。收集了人口统计学、实验室和临床数据,并随访至2023年3月。根据标准化葡萄膜炎命名法评估眼部特征和治疗反应。
在随访的97例BD儿童中,33例(34%)患有葡萄膜炎(22例男性,占66.7%)。BD和葡萄膜炎的发病中位年龄分别为11.5岁(2.5 - 17.1岁)和11岁(3 - 17.3岁)。18例儿童(54.5%)葡萄膜炎先于BD诊断。17例儿童HLA B51呈阳性(51.5%)。葡萄膜炎为双侧的有25例(75.