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Study on animal model for oGular Goloboma

眼眼球戈洛瘤动物模型的研究

基本信息

批准号：
22500394
负责人：
MATSUURA Tetsuro
金额：
$ 2.83万
依托单位：
Setsunan University
依托单位国家：
日本
项目类别：
Grant-in-Aid for Scientific Research (C)
财政年份：
2010
资助国家：
日本
起止时间：
2010 至 2012
项目状态：
已结题

来源：
https://kaken.nii.ac.jp/grant/KAKENHI-PROJECT-22500394/
关键词：
先天異常 Coloboma 動物モデル病理

项目摘要

The RCS strain ofrats has a low incidence ofmicrophthalmia. Ocular coloboma is sometimes accompanied by corectopia in humans and therefore ectopic pupil may indicateocular coloboma in experimental animals. We found that inferior ectopic pupil is associated exclusively with small-sized eyes in this strain. The objective of the current study was to evaluate whether inferiorectopic pupil is associated with iridal coloboma and other types of ocular coloboma in RCS rats. Ectopicpupils in RCS rats were found exclusively in an inferior position, where the iris was shortened. Fundicexamination revealed severe chorioretinal coloboma in all cases of inferior ectopic pupil. The morphologic characteristics closely resembled those of chorioretinal coloboma in humans. The RCS strain is a suitable model for human ocular coloboma, and inferior ectopic pupil appears to be a strong indicator of ocular coloboma.FLS mice have been shown to develop a hereditary disorder characterized by localized retinocho … More roidal defects of the ventral fundus very similar to human typical ocular colobomawithout microphthalmia. The objective of this study was to determine when and how the failure ofthe optic fissure closure occurs, andto clarify the disturbed mechanism of basement membrane disintegration during embryonal stage in FLS mice. The inner and outer layers of the optic cup did not normally fuse at midlenticular levels ofthe opticfissure in almost 70% of FLS fetuses by GD l5.5,whereas both margins were completely fused in all normal BALB/c fetuses of the same gestational day.The basement membrane persisted after the close contact of both sides of the fissure margins during GD 11.5 and 15.5. UltrastructUrally, the basal lamina was not disintegrated and mesenchymal cells intervened between the two neuroepithelial layers, resulting in complete separation of both fissure margins at GD l 3.0. It is highly probable that the disturbed basement membrane disintegration right before optic fissure closure causes mild ocular coloboma without microphthalmia in FLS mice. Less

RCS品系大鼠的小眼症发生率较低。人类的眼缺损有时伴有瞳孔异位，因此，在实验动物中，异位瞳孔可能提示眼缺损。我们发现，下异位瞳孔是专门与小尺寸的眼睛在这个应变。本研究的目的是评估RCS大鼠的直肠下瞳孔是否与虹膜缺损和其他类型的眼部缺损相关。RCS大鼠的异位瞳孔仅位于下方，虹膜缩短。眼底检查发现所有下异位瞳孔的病例都有严重的脉络膜视网膜缺损。其形态特征与人类脉络膜视网膜缺损非常相似。RCS品系是一种适合于人类眼缺损的模型，下异位瞳孔似乎是眼缺损的一个强有力的指标。 ...更多信息腹侧眼底的球状缺陷与人类典型的无小眼畸形的眼缺损非常相似。本研究的目的是确定FLS小鼠视裂闭合失败的发生时间和方式，并阐明胚胎期基底膜崩解紊乱的机制。在GD 15.5时，近70%的FLS胎儿视杯内外层在视裂中晶状体水平不正常融合，而在同一胎龄的正常BALB/c胎儿视杯内外层均完全融合，在GD 11.5和15.5时，视裂两侧边缘紧密接触后，基底膜仍存在。超微结构上，基底层未被破坏，间充质细胞介入两个神经上皮层之间，导致GD 13.0时两个裂缘完全分离。在FLS小鼠中，视裂闭合前基底膜崩解紊乱极有可能导致轻度眼缺损而无小眼球。少