Kaas Bonnie, Huisman Thierry A G M, Tekes Aylin, Bergner Amanda, Blakeley Jaishri O, Jordan Lori C
Johns Hopkins University School of Medicine, Baltimore, MD, USA.
J Child Neurol. 2013 May;28(5):561-9. doi: 10.1177/0883073812448531. Epub 2012 Jul 25.
To describe the spectrum and associated clinical features of peripheral and cerebral vasculopathy in pediatric patients with neurofibromatosis type 1, children seen at a single center from 2000 to 2010 with appropriate imaging studies were identified. Scans were assessed for vascular disease by 2 pediatric neuroradiologists. Of 181 children, 80 had pertinent imaging studies: 77 had brain imaging, 6 had peripheral imaging, and 3 had both. Vasculopathy was identified in 14/80 children (18%, minimum prevalence of 14/181; 8%). Of those with vascular abnormalities, 2/14 had peripheral vasculopathy (1% minimum prevalence) and 12/14 had cerebrovascular abnormalities (7% minimum prevalence). No associations were found between vasculopathy and common clinical features of neurofibromatosis type 1, including optic pathway glioma, plexiform neurofibroma, skeletal abnormalities, attention-deficit hyperactivity disorder (ADHD), or suspected learning disability. Both peripheral and cerebral vasculopathy are important complications of pediatric neurofibromatosis type 1 and should be considered in the management of this complex disease.
为描述1型神经纤维瘤病患儿外周血管病变和脑血管病变的范围及相关临床特征,我们确定了2000年至2010年在单一中心就诊且有适当影像学检查的儿童。由2名儿科神经放射科医生对扫描结果进行血管疾病评估。在181名儿童中,80名有相关影像学检查:77名有脑部影像学检查,6名有外周影像学检查,3名两者都有。在80名儿童中有14名(18%,最低患病率为14/181;8%)被发现有血管病变。在有血管异常的儿童中,2/14有外周血管病变(最低患病率为1%),12/14有脑血管异常(最低患病率为7%)。未发现血管病变与1型神经纤维瘤病的常见临床特征之间存在关联,这些特征包括视神经通路胶质瘤、丛状神经纤维瘤、骨骼异常、注意力缺陷多动障碍(ADHD)或疑似学习障碍。外周血管病变和脑血管病变都是1型儿科神经纤维瘤病的重要并发症,在这种复杂疾病的管理中应予以考虑。