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Development of a potent embryonic chick lens model for studying congenital cataracts in vivo.

ABSTRACT: Congenital cataracts are associated with gene mutations, yet the underlying mechanism remains largely unknown. Here we reported an embryonic chick lens model that closely recapitulates the process of cataract formation. We adopted dominant-negative site mutations that cause congenital cataracts, connexin, Cx50E48K, aquaporin 0, AQP0R33C, αA-crystallin, CRYAA R12C and R54C. The recombinant retroviruses containing these mutants were microinjected into the occlusive lumen of chick lenses at early embryonic development. Cx50E48K expression developed cataracts associated with disorganized nuclei and enlarged extracellular spaces. Expression of AQP0R33C resulted in cortical cataracts, enlarged extracellular spaces and distorted fiber cell organization. αA crystallin mutations distorted lens light transmission and increased crystalline protein aggregation. Together, retroviral expression of congenital mutant genes in embryonic chick lenses closely mimics characteristics of human congenital cataracts. This model will provide an effective, reliable in vivo system to investigate the development and underlying mechanism of cataracts and other genetic diseases.

SUBMITTER: Li Z

PROVIDER: S-EPMC7952907 | biostudies-literature | 2021 Mar

REPOSITORIES: biostudies-literature

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Development of a potent embryonic chick lens model for studying congenital cataracts in vivo.

Li Zhen Z Gu Sumin S Quan Yumeng Y Varadaraj Kulandaiappan K Jiang Jean X JX

Communications biology 20210311 1

Congenital cataracts are associated with gene mutations, yet the underlying mechanism remains largely unknown. Here we reported an embryonic chick lens model that closely recapitulates the process of cataract formation. We adopted dominant-negative site mutations that cause congenital cataracts, connexin, Cx50E48K, aquaporin 0, AQP0R33C, αA-crystallin, CRYAA R12C and R54C. The recombinant retroviruses containing these mutants were microinjected into the occlusive lumen of chick lenses at early e ...[more]

PMID: 33707565

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Project description:The aim of this study was to evaluate the long-term visual outcomes and factors affecting visual results in children undergoing secondary intraocular lens (IOL) implantation following primary congenital cataract extraction.Children with congenital cataracts who underwent secondary IOL implantation for aphakia at the Eye and ENT Hospital of Fudan University between January 1, 2001, and December 31, 2007, were retrospectively reviewed. One eye was randomly selected in children with bilateral cataracts. Laterality, type of cataract (total or partial opacity), sex, age at primary and secondary surgeries, visual axis opacity (VAO), compliance with amblyopia therapy, postoperative ocular complications, refractive error, ocular alignment, and best corrected visual acuity (BCVA) at last follow-up were recorded; potential factors that might have affected the visual results were evaluated.Seventy-six bilateral and 34 unilateral congenital cataract cases were analyzed; the mean ages of the children were 12.70±5.06 and 12.50±2.71 years at final follow-up, the mean follow-up periods from IOL implantation were 94.93±24.22 and 109.09±18.89 months, and the mean BCVA (Log MAR) values were 0.51±0.37 and 1.05±0.46, respectively. Final BCVA after secondary IOL implantation was significantly associated with laterality, type of cataract, age at primary cataract extraction, compliance with amblyopia therapy, and refractive correction after surgery. No significant associations were found between BCVA and sex, age at secondary IOL implantation, VAO, or other ocular complications. The most common ocular complications were VAO and elevated intraocular pressure after surgery. There were no other complications, with the exception of one eye with IOL dislocation.The results indicate that the important determinants of long-term visual outcomes in children with congenital cataracts undergoing secondary IOL implantation are laterality, cataract type, age at initial cataract extraction, compliance with amblyopia therapy, and refractive error.

Project description:Early colonization of intestinal microbiota plays an important role in intestinal development. However, the microbial succession at an embryonic stage and its assembly patterns induced by prenatal nutrition are unknown. In the present study, we used a chick embryo model to investigate the effects of in ovo feeding (IOF) of L-arginine (Arg) on the intestinal development and microbial succession of embryos. A total of 216 fertile eggs were randomly distributed into 2 groups including the non-injected control group and IOF of Arg group with 7 mg/egg. The results showed that IOF Arg increased the intestinal index, absolute weight of jejunum, and improved jejunal morphology in terms of villus width and surface area (p < 0.05). The relative mRNA expressions of mTOR and 4E-BP1 were up-regulated and accompanied by higher contents of Mucin-2 in the Arg group (p < 0.05). There was a significant elevation in contents of serum glucose and high-density lipoprotein cholesterol, whereas there was a decreased low-density lipoprotein cholesterol in the Arg group (p < 0.05). Additionally, Proteobacteria and Firmicutes were major intestinal bacteria species at the embryonic stage. However, Arg supplementation targeted to shape assembly patterns of microbial succession and then changed microbial composition (p = 0.05). Meanwhile, several short-chain fatty acids (SCFAs)-producing bacteria, such as Roseburia, Blautia, and Ruminococcus were identified as biomarkers in the Arg group (LDA > 3, p < 0.05). Accordingly, significant elevated concentrations of SCFAs, including lactic acid and formic acid, were observed in the Arg group (p < 0.05), accompanied by the higher concentration of butyric acid (0.05 < p < 0.10). In conclusion, prenatal Arg supplementation improved embryonic intestine development by regulating glucose and lipid homeostasis to supply more energy for chick embryos. The possible mechanism could be the roles of Arg in shaping the microbial assembly pattern and succession of the embryonic intestine, particularly the enrichment of potential probiotics. These findings may contribute to exploring nutritional strategies to establish health-promoting microbiota by manipulating prenatal host-microbe interactions for the healthy development of neonates.

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Development of a potent embryonic chick lens model for studying congenital cataracts in vivo.

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Development of a potent embryonic chick lens model for studying congenital cataracts in vivo.

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