Infliximab in infantile Takayasu arteritis: a case report and literature review
10.3760/cma.j.cn112140-20200719-00736
- VernacularTitle:英夫利昔单抗治疗婴儿多发性大动脉炎一例并文献复习
- Author:
Ming LI
1
;
Xuefeng SUN
;
Yingjie XU
;
Jun HOU
;
Dan ZHANG
;
Ying CHI
;
Jia ZHU
;
Min KANG
;
Shengnan LI
;
Gaixiu SU
;
Zhixuan ZHOU
;
Jianming LAI
Author Information
1. 首都儿科研究所附属儿童医院风湿免疫科，北京 100020
- Keywords:
Arteritis;
Infant;
Antibodies, monoclonal
- From:
Chinese Journal of Pediatrics
2020;58(12):1001-1005
- CountryChina
- Language:Chinese
-
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 (22.6×10 9/L), thrombocytosis (858×10 9/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.
