良性发作性斜颈：表型、自然病史和生活质量。

Benign paroxysmal torticollis: phenotype, natural history, and quality of life.

机构信息

Child & Adolescent Headache Program, Department of Neurology, University of California, San Francisco, San Francisco, CA, USA.

Division of Pediatric Neurology, Oregon Health & Science University, Portland, OR, USA.

出版信息

Pediatr Res. 2021 Nov;90(5):1044-1051. doi: 10.1038/s41390-020-01309-1. Epub 2021 Feb 9.

DOI:10.1038/s41390-020-01309-1

PMID:33564129

原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC8349923/

Abstract

BACKGROUND

Benign paroxysmal torticollis (BPT) is characterized by attacks of head tilt associated with vomiting, irritability, and/or ataxia in early childhood. BPT is associated with migraine but risk factors are unknown. Impact on quality of life is also unknown.

METHODS

Parents/caregivers of children with ongoing or resolved BPT participated in telephone interviews (n = 73). Those with ongoing BPT completed the Infant Toddler Quality of Life questionnaire (ITQoL).

RESULTS

Median age of children at the time of interview was 2.9 years (range 0.25-23). BPT was ongoing in 52% (n = 38). Nineteen percent (n = 14) developed migraine (median age 9.25 years, range 2.5-23) and 63% (n = 46) developed another episodic syndrome associated with migraine. Proportion of patients who developed migraine was higher among those with certain migrainous symptoms during BPT attacks vs. those without: phonophobia (58 vs. 21%, p = 0.02), photophobia and phonophobia (55 vs. 23%, p = 0.05), and photophobia, phonophobia, and motion sensitivity (60 vs. 22%, p = 0.02). ITQoL results showed significant impact of BPT on quality of life.

CONCLUSIONS

Children with BPT may develop migraine or other episodic syndromes associated with migraine. Presence of migrainous features during BPT episodes may increase likelihood of developing migraine. Though characterized as "benign," BPT can significantly impact children and families.

IMPACT

Benign paroxysmal torticollis (BPT) is a rare condition of early childhood characterized by episodes of head tilt associated with vomiting, irritability, ataxia, pallor, and/or malaise. This cohort study describes the phenotypic spectrum of BPT, variable treatment, natural history and association with migraine, and impact on development and quality of life. Children with BPT may go on to develop migraine or episodic syndromes that may be associated with migraine; presence of migrainous features during attacks may increase odds of developing migraine. BPT can have significant impact on quality of life, demonstrated by findings from the Infant Toddler Quality of Life questionnaire.

摘要

背景

良性阵发性斜颈（BPT）的特征是在幼儿期出现与呕吐、烦躁和/或共济失调相关的头部倾斜发作。BPT 与偏头痛有关，但风险因素尚不清楚。对生活质量的影响也不清楚。

方法

正在接受或已缓解 BPT 的儿童的父母/照顾者参加了电话访谈（n=73）。那些正在接受 BPT 的儿童完成了婴儿和幼儿生活质量问卷（ITQoL）。

结果

访谈时儿童的中位年龄为 2.9 岁（范围 0.25-23）。52%（n=38）的 BPT 持续存在。19%（n=14）发展为偏头痛（中位年龄 9.25 岁，范围 2.5-23），63%（n=46）发展为另一种与偏头痛相关的发作性综合征。在 BPT 发作期间出现某些偏头痛症状的患者中，偏头痛的发病率较高：畏声（58%比 21%，p=0.02）、畏光和畏声（55%比 23%，p=0.05），以及畏光、畏声和运动敏感（60%比 22%，p=0.02）。ITQoL 结果表明 BPT 对生活质量有显著影响。

结论

患有 BPT 的儿童可能会发展为偏头痛或其他与偏头痛相关的发作性综合征。BPT 发作期间出现偏头痛特征可能会增加偏头痛的发病几率。尽管 BPT 被描述为“良性”，但它会对儿童及其家庭产生重大影响。

影响

良性阵发性斜颈（BPT）是一种罕见的幼儿期疾病，其特征是与呕吐、烦躁、共济失调、苍白和/或不适相关的头部倾斜发作。本队列研究描述了 BPT 的表型谱、可变治疗、自然史以及与偏头痛的关系，以及对发育和生活质量的影响。患有 BPT 的儿童可能会发展为偏头痛或可能与偏头痛相关的发作性综合征；发作期间出现偏头痛特征可能会增加偏头痛的发病几率。BPT 会对生活质量产生重大影响，婴儿和幼儿生活质量问卷的调查结果证实了这一点。