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阿达木单抗治疗儿童难治性葡萄膜炎。

Adalimumab therapy for refractory childhood uveitis.

作者信息

Bravo-Ljubetic Luciano, Peralta-Calvo Jesús, Noval Susana, Pastora-Salvador Natalia, Abelairas-Gómez José, Merino Rosa

机构信息

Service of Ophthalmology, Instituto de Investigación Hospital Universitario La Paz (IdiPaz), Hospital Universitario La Paz, Madrid, Spain.

出版信息

J AAPOS. 2013 Oct;17(5):456-9. doi: 10.1016/j.jaapos.2013.06.009.

Abstract

PURPOSE

To report the results of adalimumab therapy in a cohort of children with refractory noninfectious uveitis.

METHODS

The medical records of patients diagnosed with uveitis and treated with biweekly adalimumab injections for a period of at least 3 months at the University Hospital of La Paz from 2007-2012 were retrospectively reviewed. Improvement in inflammatory activity was graded according to grading schema of the Standardization of Uveitis Nomenclature Working Group.

RESULTS

A total of 15 patients participated in the study (12 girls; mean patient age, 12 years). Diagnoses included juvenile idiopathic arthritis in 10 patients, idiopathic uveitis in 4, and familial juvenile systemic granulomatosis or Blau syndrome in 1. Mean follow-up was 32 months (median, 36; range, 15-58 months). Improvement in inflammatory activity was initially observed in 12 (86%) of 14 children, with a mean time to achieve response of 6 weeks (median, 4; range, 1-18). Treatment was effective in 9 patients (60%), mildly effective in 2 (13%), ineffective in 2 (13%), and resulted in worsening in 2 (13%). In the juvenile idiopathic arthritis patients, response was effective in 6 cases (60%), mildly effective in 2 (20%), and ineffective in 2 (20%). Adalimumab therapy was discontinued in 4 patients.

CONCLUSIONS

Adalimumab was effective in most patients in the initial control of acute inflammatory activity in children with refractory uveitis, although therapy appears to become less effective in the long term.

摘要

目的

报告阿达木单抗治疗难治性非感染性葡萄膜炎患儿队列的结果。

方法

回顾性分析2007年至2012年在拉巴斯大学医院被诊断为葡萄膜炎并接受每两周一次阿达木单抗注射治疗至少3个月的患者的病历。根据葡萄膜炎命名标准化工作组的分级方案对炎症活动的改善情况进行分级。

结果

共有15例患者参与研究(12例女孩;平均患者年龄12岁)。诊断包括10例幼年特发性关节炎、4例特发性葡萄膜炎和1例家族性幼年系统性肉芽肿病或布劳综合征。平均随访32个月(中位数36个月;范围15 - 58个月)。14例儿童中有12例(86%)最初观察到炎症活动改善,达到反应的平均时间为6周(中位数4周;范围1 - 18周)。治疗对9例患者(60%)有效,2例(13%)轻度有效,2例(13%)无效,2例(13%)病情恶化。在幼年特发性关节炎患者中,6例(60%)反应有效,2例(20%)轻度有效,2例(20%)无效。4例患者停用阿达木单抗治疗。

结论

阿达木单抗对大多数难治性葡萄膜炎患儿在急性炎症活动的初始控制中有效,尽管从长期来看治疗效果似乎会降低。

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