van Asperen C J, Overweg-Plandsoen W C, Cnossen M H, van Tijn D A, Hennekam R C
Institute for Human Genetics, Academic Medical Centre, Amsterdam, The Netherlands.
J Med Genet. 1998 Apr;35(4):323-7. doi: 10.1136/jmg.35.4.323.
The simultaneous occurrence of familial neurofibromatosis type 1 (NF1) and an overgrowth syndrome resembling Weaver syndrome was observed in two related cases (a mother and her son). NF1 was confirmed by molecular genetic analysis showing a large deletion at 17q11.2, encompassing the entire NF1 gene. The other symptoms in the two cases were similar to the features reported in Weaver syndrome. Although the combination of NF1 and an overgrowth syndrome resembling Weaver syndrome in this family may be fortuitous, we favour the hypothesis that the deletion of the entire gene has caused this combined phenotype. Possible pathogenetic mechanisms are discussed. The observation suggests a relation between NF1 with an extraordinarily large gene deletion and a Weaver(-like) syndrome. This warrants investigation for deletions in the 17q11.2 region in Weaver(-like) syndrome patients.
在两个相关病例(一位母亲和她的儿子)中观察到1型神经纤维瘤病(NF1)与类似韦弗综合征的过度生长综合征同时出现。通过分子遗传学分析证实为NF1,显示17q11.2存在大片段缺失,涵盖整个NF1基因。这两个病例中的其他症状与韦弗综合征报道的特征相似。尽管该家族中NF1与类似韦弗综合征的过度生长综合征的组合可能是偶然的,但我们倾向于这样的假说,即整个基因的缺失导致了这种联合表型。讨论了可能的发病机制。该观察结果提示NF1伴异常大片段基因缺失与韦弗(样)综合征之间存在关联。这值得对韦弗(样)综合征患者的17q11.2区域缺失情况进行研究。