评估丙戊酸治疗发绀性呼吸暂停发作的有效性：一项前瞻性初步研究。

Evaluation of the effectiveness of valproic acid for treating cyanotic breath holding spells: A Pilot prospective study.

机构信息

Department of Neurology and Psychiatry, Assiut University Hospital , Assiut, Egypt.

Department of Neuropsychiatry, Al Azher University , Assiut, Egypt.

出版信息

Expert Rev Clin Pharmacol. 2020 Nov;13(11):1263-1270. doi: 10.1080/17512433.2020.1828059. Epub 2020 Nov 1.

DOI:10.1080/17512433.2020.1828059

PMID:32969724

Abstract

BACKGROUND

Cyanotic breath-holding spells (CBHS) are self-limited conditions among younger children. Frequent spells cause parents' fear and anxiety. Seizures, brain damage and sudden death have been rarely reported with BHS. Some reported spells' frequency reduction with iron or piracetam. We evaluated the effectiveness of valproic acid (VPA) to treat CBHS and predictors of improvement.

METHODS

Participants were 90 children with CBHS (≥4/week) (age: 1.6±0.4yrs). They were treated with VPA (5 mg/kg/d). Follow-ups occurred after 3-≥6 months. Autonomic nervous system functions were evaluated.

RESULTS

The majority (74.4%) had daily spells and 19% had ≥2 spells/d. Crying or anger provoked spells. Postural hypotension was found in 46.7%. They had normal electroencephalography and QT, QTc interval or QTd or QTcd and heart rate. Compared to controls, postural fall in systolic (>20mmHg) and diastolic (>10mmHg) blood pressures and mean arterial pressure (>10mmHg) were observed in 46.7%, 74.4% and 72.2% and miosis observed with 0.125% pilocarpine in 28.9% (P=0.001). Spells' frequency reduction (P=0.001) occurred within 3 months with VPA. The independent prdictors for spell' frequency reduction were reduction of anger and crying [OR=4.52(95%CI=2.35-6.04), P =0.01].

CONCLUSION

VPA therapy reduces CBHS' frequency. Mood improvement is a suggestive effective mechanism.

CLINICAL TRIAL REGISTRATION

www.clinicaltrials.gov identifier is NCT04482764.

摘要

背景

发绀性屏气发作（CBHS）是幼儿中一种自限性疾病。频繁发作会引起父母的恐惧和焦虑。BHS 罕见报道有癫痫发作、脑损伤和猝死。一些报道称铁或吡拉西坦可减少发作次数。我们评估了丙戊酸（VPA）治疗 CBHS 的疗效和改善的预测因素。

方法

参与者为 90 名（≥4/周）CBHS 儿童（年龄：1.6±0.4 岁），给予 VPA（5mg/kg/d）治疗。在 3-≥6 个月后进行随访。评估自主神经系统功能。

结果

大多数（74.4%）儿童每天发作，19%的儿童≥2 次/天。哭泣或愤怒会引发发作。46.7%的儿童出现体位性低血压。他们的脑电图、QT、QTc 间期或 QTd 或 QTcd 和心率正常。与对照组相比，46.7%、74.4%和 72.2%的儿童在体位性下降时收缩压（>20mmHg）和舒张压（>10mmHg）和平均动脉压（>10mmHg）下降，28.9%的儿童在 0.125%毛果芸香碱下出现瞳孔缩小（P=0.001）。VPA 治疗 3 个月内发作频率降低（P=0.001）。发作频率降低的独立预测因素为愤怒和哭泣减少[比值比（OR）=4.52（95%可信区间[CI]：2.35-6.04），P=0.01]。