Petters R M, Alexander C A, Wells K D, Collins E B, Sommer J R, Blanton M R, Rojas G, Hao Y, Flowers W L, Banin E, Cideciyan A V, Jacobson S G, Wong F
Department of Animal Science, North Carolina State University, Raleigh, USA.
Nat Biotechnol. 1997 Oct;15(10):965-70. doi: 10.1038/nbt1097-965.
Patients with retinitis pigmentosa (RP) typically develop night blindness early in life due to loss of rod photoreceptors. The remaining cone photoreceptors are the mainstay of their vision; however, over years or decades, these cones slowly degenerate, leading to blindness. We created transgenic pigs that express a mutated rhodopsin gene (Pro347Leu). Like RP patients with the same mutation, these pigs have early and severe rod loss; initially their cones are relatively spared, but these surviving cones slowly degenerate. By age 20 months, there is only a single layer of morphologically abnormal cones and the cone electroretinogram is markedly reduced. Given the strong similarities in phenotype to that of RP patients, these transgenic pigs will provide a large animal model for study of the protracted phase of cone degeneration found in RP and for preclinical treatment trials.
患有色素性视网膜炎(RP)的患者通常在生命早期因视杆光感受器丧失而出现夜盲症。剩余的视锥光感受器是他们视力的主要支撑;然而,经过数年或数十年,这些视锥细胞会慢慢退化,导致失明。我们培育出了表达突变视紫红质基因(Pro347Leu)的转基因猪。与具有相同突变的RP患者一样,这些猪早期就出现严重的视杆细胞丧失;最初它们的视锥细胞相对未受影响,但这些存活的视锥细胞会慢慢退化。到20个月大时,仅剩下一层形态异常的视锥细胞,并且视锥细胞视网膜电图明显降低。鉴于其表型与RP患者的表型有很强的相似性,这些转基因猪将为研究RP中视锥细胞退化的长期阶段以及临床前治疗试验提供一个大型动物模型。