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  • Title: [Infliximab in infantile Takayasu arteritis: a case report and literature review].
    Author: Li M, Sun XF, Xu YJ, Hou J, Zhang D, Chi Y, Zhu J, Kang M, Li SN, Su GX, Zhou ZX, Lai JM.
    Journal: Zhonghua Er Ke Za Zhi; 2020 Dec 02; 58(12):1001-1005. PubMed ID: 33256323.
    Abstract:
    Objective: To analyze the clinical characteristics of infantile Takayasu arteritis and the efficacy of infliximab (IFX). Methods: Clinical manifestations, laboratory investigations and infliximab intervention of a case with infantile Takayasu arteritis, who was admitted to Department of Rheumatism and Immunology, Children's Hospital, Capital Institute of Pediatrics in January 2018, were reviewed and analyzed. The related literature published from the beginning to March 2020 were retrieved from CNKI, Wanfang, SinoMed and PubMed with the keywords of"Takayasu arteritis","Infant" in both Chinese and English. Results: This case was a 70-day-old boy admitted due to recurrent fever for 20 days. On admission, his blood pressure were 104/90, 95/59, 125/80, and 152/125 mmHg (1 mmHg=0.133 kPa) in the right arm, left arm, right leg, and left leg, respectively. The complete blood cell count showed leukocytosis (22.6×109/L), thrombocytosis (858×109/L) and mild anemia (80 g/L). He also had elevated erythrocyte sedimentation rate (119 mm/1h), serum ferritin (598 μg/L) and C-reactive protein (112 mg/L). Computed tomographic angiography (CTA) showed narrowing of the thoracic and abdominal aorta, with thickening and heterogenous enhancement of the vessel wall. Coronary artery ultrasound detected dilatation and wall thickening of the bilateral coronary arteries, and uneven dilatation of the middle segment of the right coronary artery, showing bead-like change. Vessel wall thickening was also found in the other main arteries, including both femoral arteries, axillary arteries, carotid arteries, and subclavian arteries, and both superficial femoral arteries were slightly narrowed in the distal segments. The diagnosis of TA was confirmed, and the boy was treated with infliximab monotherapy (5 mg/(kg·every time), a total of 13 times). Then his body temperature and all inflammatory markers were normalized, and the vascular pathology was resolved according to the radiography. No side effects such as allergy or infection were noted during the treatment. During the 2 years and 6 months of follow-up, the boy maintained normal growth and development. Literature review found 8 related articles, and one of them was in Chinese but had limited information. In the other 7 papers, a total of 7 infants with TA were reported. The most common symptom was fever (5 cases), and inflammatory markers usually elevated, and the most common affected artery was abdominal aorta (6 cases). Most cases were treated with glucocorticoid. Conclusions: TA is a rare disease in infants, usually presents with fever and increased inflammatory markers. At the early stage, infliximab monotherapy could effectively control the symptoms and ensure normal growth and development. 目的： 总结婴儿期起病的多发性大动脉炎（TA）的临床特点及英夫利昔单抗（IFX）治疗效果。 方法： 分析2018年1月首都儿科研究所附属儿童医院风湿免疫科收治的1例小年龄TA患儿的病例特点及IFX单药治疗效果。以“多发性大动脉炎”“婴儿”为检索词，检索建库至2020年3月中国知网、万方数据库、中国生物医学文献数据库及Pubmed数据库并进行文献复习。 结果： 患儿 男，70日龄，因反复发热20 d入院。四肢血压增高（右上肢104/90 mmHg，左上肢95/59 mmHg， 右下肢125/80 mmHg， 左下肢152/125 mmHg，1 mmHg=0.133 kPa）。外周血白细胞（22.6×109/L）及血小板（858×109/L）增高，轻度贫血（血红蛋白 80 g/L），红细胞沉降率（119 mm/1 h）、C反应蛋白（112 mg/L）、血清铁蛋白（598 μg/L）均增高，CT血管成像示胸主动脉、腹主动脉管壁明显增厚，不均匀强化，管腔狭窄。冠状动脉彩超声示双侧冠状动脉扩张，管壁增粗毛糙，右冠状动脉中段扩张不均匀，呈串珠样改变。血管超声示双侧股动脉、股浅动脉管壁毛糙不均匀增厚，双侧股浅动脉远端多处轻度狭窄，双侧腋动脉管壁毛糙不均匀增厚，双侧颈总动脉管壁毛糙增厚，双侧锁骨下动脉管壁毛糙增厚，管壁结构不清晰。确诊TA后应用IFX单药治疗［5 mg/（kg·次），共13次］后病情缓解，体温及炎性指标恢复正常，血管影像学恢复正常，治疗期间患儿未出现严重过敏反应及感染。随访2年6个月，患儿身高、体重等生长发育指标均达正常标准。文献检索中文文献1篇，无详细临床资料，英文文献7篇，共7例婴儿期发病的TA患儿，最常见临床表现为发热（5例），炎性指标多增高，最常见受累动脉为腹主动脉（6例），多接受激素治疗。 结论： TA于婴儿期发病罕见，发热可为其主要表现，炎性指标多增高。疾病早期应用IFX单药治疗可迅速控制病情，不影响儿童正常生长发育。.
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