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儿童期无荨麻疹性血管性水肿。

Angioedema without urticaria in childhood.

机构信息

Department of Pediatric Allergy and Asthma, Gazi University School of Medicine, Ankara, Turkey.

出版信息

Pediatr Allergy Immunol. 2013 Nov;24(7):685-90. doi: 10.1111/pai.12118. Epub 2013 Sep 12.

Abstract

BACKGROUND

There has been no separate study investigating angioedema without urticaria (Aw/oU) exclusively in children so far. The purpose of this study was to investigate the frequency, clinical presentation, etiology, management and follow-up of Aw/oU in children.

METHODS

This is a prospective study that included all consecutive patients with a history of Aw/oU referred to our clinic between January 2011 and May 2012. A standard diagnostic and therapeutic algorithm was applied to all patients.

RESULTS

The frequency of Aw/oU was found to be 1.6% during the study period. An etiological factor could be found in only 45 patients (49%). The causes of Aw/oU were infection (21%), allergy (14%), thyroid autoimmunity (TA)-related (8%) and nonsteroid anti-inflammatory drug hypersensitivity (6%), and idiopathic angioedema (51%). There was no hereditary type I, II or acquired type of angioedema or rare syndromes associated with Aw/oU. The median follow-up was 16 months (range: 12-30 months). Antihistamine prophylaxis was initiated at therapeutic doses in 20 patients with frequently recurrent angioedema due to idiopathic and euthyroid TA-related Aw/oU for 3 months. These patients responded to antihistamine prophylaxis for 3 months. Four patients relapsed after cessation of prophylaxis at the end of 3 months. Antihistamine prophylaxis was prolonged to 6 months in three patients and to 9 months in one patient.

CONCLUSIONS

Angioedema without urticaria in children is a rare condition and no etiology can be identified in half of them. Antihistamine treatment alone is sufficient, and prognosis is good in recurrent non hereditary cases in a short-term follow-up period.

摘要

背景

迄今为止,尚无单独研究专门调查儿童中无荨麻疹的血管性水肿(Aw/oU)。本研究的目的是调查 Aw/oU 在儿童中的频率、临床表现、病因、治疗和随访。

方法

这是一项前瞻性研究,纳入了 2011 年 1 月至 2012 年 5 月期间我院就诊的所有连续 Aw/oU 患者。对所有患者应用了标准的诊断和治疗方案。

结果

研究期间,Aw/oU 的频率为 1.6%。仅在 45 例患者(49%)中发现病因。Aw/oU 的病因是感染(21%)、过敏(14%)、甲状腺自身免疫(TA)相关(8%)、非甾体抗炎药过敏(6%)和特发性血管性水肿(51%)。无遗传性 1 型、2 型或获得性血管性水肿或与 Aw/oU 相关的罕见综合征。中位随访时间为 16 个月(范围:12-30 个月)。由于特发性和甲状腺功能正常的 TA 相关 Aw/oU 频繁复发,20 例患者开始接受抗组胺药预防治疗,剂量为治疗剂量,持续 3 个月。这些患者在 3 个月的抗组胺药预防治疗中得到缓解。3 个月后预防治疗结束时,4 例患者复发。有 3 例患者将抗组胺药预防治疗延长至 6 个月,1 例患者延长至 9 个月。

结论

儿童无荨麻疹性血管性水肿是一种罕见疾病,其中一半患者无法确定病因。单独使用抗组胺药治疗即可,在短期随访中,对于非遗传性复发性病例,预后良好。

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