Prospective study of children with cyanotic and pallid breath-holding spells
Prospective study of children with cyanotic and pallid breath-holding spells
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DOI：
10.1542/peds.107.2.265
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发表时间：
2001-02-01
期刊：
影响因子：
8
通讯作者：
DiMario, FJ
中科院分区：
文献类型：
--
作者：
DiMario, FJ
Objective.在这项研究中，我们试图前瞻性地记录在神经科会诊的紫绀和苍白BHS儿童中严重屏气发作（BHS）的自然史。前瞻性队列研究。在最初咨询时和随后的1年时间间隔进行结构化访谈，内容涉及BHS类型、法术频率、相关现象、后遗症、家族史和法术终止时的年龄。共有95名儿童（48名男孩，47名女孩）与BHS进行了鉴定，并随访了9年。性别间无显著差异。中位发病年龄在6至12个月之间，15%的患者发病年龄小于6个月。法术的平均频率是每周一次，30%的人每天经历1次或更多的法术。高峰频率的中位年龄为12至18个月，范围从6个月至4岁。在BHS缓解时间>12个月的患者中（n = 67），最后一次缓解发生在中位年龄37至42个月时。在那些仍在发生BHS的儿童中，最近一次发作时的最大年龄为7岁。在所有参与者中，低于或等于15%的低血糖惊厥与BHS相关。BHS阳性家族史在34%的家系中检出，父方和母方的阳性率相等。在这项研究中，有关BHS的自然历史的新数据已经确定。这些信息对于家庭咨询和确定干预策略非常重要，并可作为基线数据来评估未来治疗方法的有效性。
Objective. In this investigation, we sought to prospectively document the natural history of severe breath-holding spells (BHS) among children with both cyanotic and pallid BHS who were referred for neurologic consultation.Design. Prospective cohort study.Methods. A structured interview was undertaken at the time of initial consultation and at subsequent 1-year intervals regarding type of BHS, frequency of spells, associated phenomenon, sequelae, family history, and age at termination of spells.Results. A total of 95 children (48 boys, 47 girls) with BHS were identified and followed over a 9-year interval. There were no significant differences between genders. Median onset age was between 6 and 12 months old with 15% presenting younger than 6 months. A median frequency of spells was weekly with 30% experiencing 1 or more spells per day. The median age at peak frequency was between 12 and 18 months old with a range extending from 6 months to 4 years of age. Of the patients whose BHS had remitted for >12 months' time (n = 67), the last spell occurred at a median age of 37 to 42 months. Of those children whose BHS were still occurring, the oldest age at time of latest spell was at 7 years old. Hypoxic convulsions were associated with BHS in less than or equal to 15% of all participants. A positive family history of BHS was identified in 34% of all families with equal frequency distributed between paternal and maternal sides.Conclusions. In this study, new data concerning the natural history of BHS have been determined. The information is important for family counseling and identifying intervention strategies, and serves as baseline data to evaluate the efficacy of future treatment approaches.