Assessment and Characterization of Hyaloid Vessels in Mice

Wang, Zhongxiao; Liu, Chi-Hsiu; Huang, Shuo; Chen, Jing

doi:10.3791/59222

“… 40 For mice, regression of the hyaloid vasculature is complete by P21. 41 In the vitreous of WT and Adamts10 G661R/G661R mice, cell nuclei were detected at P10 ( Figs. 7 A, 7 D, 7 G, 7 J) but not at 3 or 24 months of age ( Figs.…”

Section: Resultsmentioning

confidence: 99%

Altered Ocular Fibrillin Microfibril Composition in Mice With a Glaucoma-Causing Mutation of Adamts10

Wu

¹

,

Mortlock

²

,

Kuchtey

³

et al. 2021

Invest. Ophthalmol. Vis. Sci.

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Purpose Previously, we identified a G661R mutation of ADAMTS10 (a disintegrin-like and metalloprotease with thrombospondin type 1 motif 10) as being disease causative in a colony of Beagles with inherited primary open-angle glaucoma (POAG). Mutations in ADAMTS10 are known to cause Weill–Marchesani syndrome (WMS), which is also caused by mutations in the fibrillin-1 gene ( FBN1 ), suggesting functional linkage between ADAMTS10 and fibrillin-1, the principal component of microfibrils. Here, we established a mouse line with the G661R mutation of Adamts10 ( Adamts10 G661R/G661R ) to determine if they develop features of WMS and alterations of ocular fibrillin microfibrils. Methods Intraocular pressure (IOP) was measured using a TonoLab rebound tonometer. Central cornea thickness (CCT), anterior chamber depth (ACD) and axial length (AL) of the eye were examined by spectral-domain optical coherence tomography. Sagittal eye sections from mice at postnatal day 10 (P10) and at 3 and 24 months of age were stained with antibodies against fibrillin-1, fibrillin-2, and ADAMTS10. Results IOP was not elevated in Adamts10 G661R/G661R mice. Adamts10 G661R/G661R mice had smaller bodies, thicker CCT, and shallower ACD compared to wild-type mice but normal AL. Adamts10 G661R/G661R mice displayed persistent fibrillin-2 and enhanced fibrillin-1 immunofluorescence in the lens zonules and in the hyaloid vasculature and its remnants in the vitreous. Conclusions Adamts10 G661R/G661R mice recapitulate the short stature and ocular phenotypes of WMS. The altered fibrillin-1 and fibrillin-2 immunoactivity in Adamts10 G661R/G661R mice suggests that the G661R mutation of Adamts10 perturbs regulation of the fibrillin isotype composition of microfibrils in the mouse eye.

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“…WNT7B potentially interacts with other key members in the WNT signaling pathway, including FZD4, LRP5, and Norrin, in congenital eye disorders such as persistent hyperplastic primary vitreous, neural tube defects, and Norrie disease. 35 , 37 , 38 Further studies are warranted.…”

Section: Discussionmentioning

confidence: 99%

Association ofWNT7BandRSPO1with Axial Length in School Children

Lu

¹

,

Tang

²

,

Li

³

et al. 2020

Invest. Ophthalmol. Vis. Sci.

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Purpose To evaluate the association between single-nucleotide polymorphisms (SNPs) in the ZC3H11B , RSPO1 , C3orf26 , GJD2 , ZNRF3 , and WNT7B genes and myopia endophenotypes in children. Methods Seven SNPs identified in previous genome-wide association studies of axial length (AL) were genotyped in 2883 Southern Han Chinese children. Multiple linear regression analyses were conducted to evaluate the genotype association with AL, spherical equivalent (SE), corneal curvature (CC), and central corneal thickness (CCT). Results Two SNPs—namely, rs12144790 in RSPO1 (allele T, P = 0.0066, β = 0.062) and rs10453441 in WNT7B (allele A, P = 8.03 × 10 –6 , β = 0.103)—were significantly associated with AL. The association of rs4373767 in ZC3H11B (allele C, P = 0.030, β = –0.053) could not withstand the correction for multiple testing. WNT7B rs10453441 showed a strong association with CC ( P = 1.17 × 10 –14 , β = 0.053) and with CCT ( P = 0.0026, β = 2.65). None of the tested SNPs was significantly associated with SE. The C allele of SNP rs12321 in ZNRF3 was associated with CC ( P = 0.0060, β = –0.018). Conclusions This study revealed that the RSPO1 SNP rs12144790 was associated with AL, whereas WNT7B rs10453441 was associated with AL, CC, and CCT in children. A novel association between ZNRF3 rs12321 and CC was discovered. Our data suggest that the RSPO1 and WNT7B genes might exert their effects on multiple aspects of eye growth during childhood. Potential differences in the genetic profiles of AL between children and adults should be explored in larger cohorts.

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“… 76 , 77 The vascular regression of hyaloid vessels during early development may also contribute to the difference in regions adjacent and distal to the ONH. 78 In addition, specific maturational processes occurring at the cellular and vascular levels of the retinal layers may also lead to thickness changes in regions distal and adjacent to the ONH. Through the entire retina, angiogenesis and vascular remodeling produce changes in the vascular network density and distribution of vessels.…”

Section: Discussionmentioning

confidence: 99%

Morphometric and Microstructural Changes During Murine Retinal Development Characterized Using In Vivo Optical Coherence Tomography

Brais-Brunet

¹

,

Heckel

²

,

Kanniyappan

³

et al. 2021

Invest. Ophthalmol. Vis. Sci.

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Purpose The purpose of this study was to develop an in vivo optical coherence tomography (OCT) system capable of imaging the developing mouse retina and its associated morphometric and microstructural changes. Methods Thirty-four wild-type mice (129S1/SvlmJ) were anesthetized and imaged between postnatal (P) day 7 and P21. OCT instrumentation was developed to optimize signal intensity and image quality. Semi-automatic segmentation tools were developed to quantify the retinal thickness of the nerve fiber layer (NFL), inner plexiform layer (IPL), inner nuclear layer (INL), and the outer retinal layers (ORL), in addition to the total retina. The retinal maturation was characterized by comparing layer thicknesses between consecutive time points. Results From P7 to P10, the IPL increased significantly, consistent with retinal synaptogenesis. From P10 to P12, the IPL and ORL also increased, which is coherent with synaptic connectivity and photoreceptor maturation. In contrast, during these periods, the INL decreased significantly, consistent with cellular densification and selective apoptotic “pruning” of the tissue during nuclear migration. Thereafter from P12 to P21, the INL continued to thin (significantly from P17 to P21) whereas the other layers remained unchanged. No time-dependent changes were observed in the NFL. Overall, changes in the total retina were attributed to those in the IPL, INL, and ORL. Regions of the retina adjacent to the optic nerve head were thinner than distal regions during maturation. Conclusions Changes in retinal layer thickness are consistent with retinal developmental mechanisms. Accordingly, this report opens new horizons in using our system in the mouse to characterize longitudinally developmental digressions in models of human diseases.

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Assessment and Characterization of Hyaloid Vessels in Mice

Cited by 11 publications

References 29 publications

Altered Ocular Fibrillin Microfibril Composition in Mice With a Glaucoma-Causing Mutation of Adamts10

Altered Ocular Fibrillin Microfibril Composition in Mice With a Glaucoma-Causing Mutation of Adamts10

Association ofWNT7BandRSPO1with Axial Length in School Children

Morphometric and Microstructural Changes During Murine Retinal Development Characterized Using In Vivo Optical Coherence Tomography

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