Zonular defects in loxl1-deficient zebrafish.
In: Clinical & Experimental Ophthalmology, Jg. 50 (2022), Heft 1, S. 62-73
Online
academicJournal
Zugriff:
Background: To investigate the roles of the lysyl oxidase-like 1 (loxl1) gene in zebrafish eye development and the potency of loxl1 deficiency in mimicking the ocular manifestations of exfoliation syndrome (XFS). Methods: CRISPR/Cas9 technology was used to generate a frameshift coding deletion in zebrafish loxl1. Expression profiles and ocular manifestations of the wildtype, heterozygous mutant (loxl1+/-) and homozygous mutant (loxl1-/-) zebrafish were analysed in a range of developmental stages from zebrafish larvae to dissected adult zebrafish eyes. Results: The loxl1 deficiency caused zonular bundling disorders in juvenile zebrafish and accumulation of pearl-like particles adhering to the adult zebrafish zonule. The bundles appeared to lack form and were thinner in both loxl1+/- and loxl1-/- zebrafish compared with the wildtype (p < 0.01 for all Bonferroni post-hoc analyses). The zonule of loxl1-/- zebrafish appeared stretched, ragged and torn, with isolated fibres also detected. The particles in loxl1-/- zebrafish were more numerous (counts: 92.33 ± 10.02/100 µm2 vs. 58.33 ± 5.03/100 µm2, p = 0.006), but smaller in size (diameter: 0.21 ± 0.03 µm vs. 0.43 ± 0.04 µm, p = 0.002) compared with those in loxl1+/-. Transmission electron microscopy revealed thinning or even loss of elastic lamina in loxl1+/- Bruch's membrane (BM) (thickness of elastic lamina: 92.94 ± 18.19 nm in the wildtype vs 35.65 ± 14.76 nm in loxl1+/-, p = 0.003). The breakage of BM was observed in loxl1-/-. Conclusions: The loxl1-/- zebrafish is a promising animal model of XFS zonular pathology. [ABSTRACT FROM AUTHOR]
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Zonular defects in loxl1-deficient zebrafish.
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Autor/in / Beteiligte Person: | Zhang, Min ; Sun, Shaoyang ; Wang, Lei ; Wang, Xu ; Chen, Tianhui ; Chen, Zexu ; Jiang, Yongxiang |
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Zeitschrift: | Clinical & Experimental Ophthalmology, Jg. 50 (2022), Heft 1, S. 62-73 |
Veröffentlichung: | 2022 |
Medientyp: | academicJournal |
ISSN: | 1442-6404 (print) |
DOI: | 10.1111/ceo.14017 |
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