Jacks T, Shih T S, Schmitt E M, Bronson R T, Bernards A, Weinberg R A
Center for Cancer Research, Massachusetts Institute of Technology, Cambridge 02139.
Nat Genet. 1994 Jul;7(3):353-61. doi: 10.1038/ng0794-353.
Human neurofibromatosis type 1 is a dominant disease caused by the inheritance of a mutant allele of the NF1 gene. In order to study NF1 function, we have constructed a mouse strain carrying a germline mutation in the murine homologue. Heterozygous animals do not exhibit the classical symptoms of the human disease, but are highly predisposed to the formation of various tumour types, notably phaeochomocytoma, a tumour of the neural crest-derived adrenal medulla, and myeloid leukaemia, both of which occur with increased frequency in human NF1 patients. The wild-type Nf1 allele is lost in approximately half of the tumours from heterozygous animals. In addition, homozygosity for the Nf1 mutation leads to abnormal cardiac development and mid-gestational embryonic lethality.
人类1型神经纤维瘤病是一种显性疾病,由NF1基因突变等位基因的遗传引起。为了研究NF1的功能,我们构建了一种在小鼠同源物中携带种系突变的小鼠品系。杂合动物不表现出人类疾病的典型症状,但极易形成各种肿瘤类型,尤其是嗜铬细胞瘤,一种源自神经嵴的肾上腺髓质肿瘤,以及髓系白血病,这两种肿瘤在人类NF1患者中发生频率均增加。野生型Nf1等位基因在杂合动物的大约一半肿瘤中丢失。此外,Nf1突变的纯合性会导致心脏发育异常和妊娠中期胚胎致死。