Tam, O. H., Pennisi, D., Wilkinson, L., Little, M. H., Wazin, F., Wan, V. L., Lovicu, F. J. (May 2018) Crim1 is required for maintenance of the ocular lens epithelium. Exp Eye Res, 170. pp. 58-66. ISSN 0014-4835
Abstract
The development and growth of the vertebrate ocular lens is dependent on the regulated proliferation of an anterior monolayer of epithelial cells, and their subsequent differentiation into elongate fiber cells. The growth factor rich ocular media that bathes the lens mediates these cellular processes, and their respective intracellular signaling pathways are in turn regulated to ensure that the proper lens architecture is maintained. Recent studies have proposed that Cysteine Rich Motor Neuron 1 (Crim1), a transmembrane protein involved in organogenesis of many tissues, might influence cell adhesion, polarity and proliferation in the lens by regulating integrin-signaling. Here, we characterise the lens and eyes of the Crim1(KST264) mutant mice, and show that the loss of Crim1 function in the ocular tissues results in inappropriate differentiation of the lens epithelium into fiber cells. Furthermore, restoration of Crim1 levels in just the lens tissue of Crim1(KST264) mice is sufficient to ameliorate most of the dysgenesis observed in the mutant animals. Based on our findings, we propose that tight regulation of Crim1 activity is required for maintenance of the lens epithelium, and its depletion leads to ectopic differentiation into fiber cells, dramatically altering lens structure and ultimately leading to microphthalmia and aphakia.
| Item Type: | Paper |
|---|---|
| Uncontrolled Keywords: | Crim1 Fiber differentiation Lens epithelium Lens fibers TGFbeta |
| Subjects: | bioinformatics > genomics and proteomics > genetics & nucleic acid processing > protein structure, function, modification > protein expression organs, tissues, organelles, cell types and functions > tissues types and functions > transport > trans-membrane transport |
| CSHL Authors: | |
| Communities: | CSHL labs > Hammell M. lab |
| Depositing User: | Matt Covey |
| Date: | 16 May 2018 |
| Date Deposited: | 07 Mar 2018 14:27 |
| Last Modified: | 14 Jun 2018 19:44 |
| Related URLs: | |
| URI: | https://repository.cshl.edu/id/eprint/36211 |
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