Hirabaru Keiko, Matsuo Muneaki
Department of Pediatrics, Faculty of Medicine, Saga University, Nabeshima, Saga, Japan.
Pediatr Int. 2018 Jan;60(1):70-75. doi: 10.1111/ped.13388. Epub 2017 Oct 3.
The aim of this study was to determine the frequency of central nervous system comorbidities in children with neurofibromatosis type 1 (NF1).
We performed a nationwide survey to investigate neurological comorbidities in 3-15-year-old children with NF1 in Japan by sending questionnaires to pediatricians and pediatric neurologists. A secondary questionnaire was sent to the parents of identified NF1 patients to assess neurological comorbidities including headache, attention deficit-hyperactivity disorder (ADHD) Rating Scale (RS), and the Social Responsiveness Scale 2.
The primary survey identified 760 NF1 patients, and the parents of 565 patients were sent the secondary questionnaire. The parental response rate was 25.7% (145; 63 girls, 81 boys, one unspecified). Among the patients, 42.9% (55/128; 35 girls, 20 boys) were reported to exhibit intellectual problems. On the ADHD-RS, 40.2% (47/117) of NF1 patients aged 6-15 had ADHD (RS score >93rd percentile), with a rate of 47.7% in boys and 30.8% in girls. Furthermore, 20.2% of patients had suspected autism spectrum disorder (29/143; 10 girls, 19 boys), with Social Responsiveness Scale score ≥76. Headache was reported by 49.6% (61/123) of children over 5 years old, and 25.2% (31/123; 10 girls, 21 boys) reported migraine. Other neurological comorbidities included 20 cases of epilepsy (13.8%), 11 cases of optic nerve glioma (7.6%), five cases of brain tumor (3.4%), six cases of cerebrovascular disease (4.1%), and two cases of hydrocephalus (1.4%).
Intellectual problems, ADHD, autism spectrum disorder, and migraine are major neurological comorbidities in NF1.
本研究的目的是确定1型神经纤维瘤病(NF1)患儿中枢神经系统合并症的发生率。
我们通过向儿科医生和儿科神经科医生发送问卷,对日本3至15岁的NF1患儿的神经合并症进行了全国性调查。向确诊的NF1患者的父母发送了一份二级问卷,以评估神经合并症,包括头痛、注意力缺陷多动障碍(ADHD)评定量表(RS)和社会反应量表2。
初步调查确定了760例NF1患者,并向565例患者的父母发送了二级问卷。父母的回复率为25.7%(145例;63名女孩,81名男孩,1例未注明性别)。在这些患者中,42.9%(55/128;35名女孩,20名男孩)被报告有智力问题。在ADHD-RS量表上,6至15岁的NF1患者中有40.2%(47/117)患有ADHD(RS评分>第93百分位),男孩的发生率为47.7%,女孩为30.8%。此外,20.2%的患者疑似患有自闭症谱系障碍(29/143;10名女孩,19名男孩),社会反应量表评分≥76。5岁以上儿童中有49.6%(61/123)报告有头痛,25.2%(31/123;10名女孩,21名男孩)报告有偏头痛。其他神经合并症包括2例癫痫(13.8%)、11例视神经胶质瘤(7.6%)、5例脑肿瘤(3.4%)、6例脑血管疾病(4.1%)和2例脑积水(1.4%)。
智力问题、ADHD、自闭症谱系障碍和偏头痛是NF1的主要神经合并症。