run_metadata: 60827
This data as json
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| 60827 | SRR12536650 | SRX9026395 | SRS7277845 | SRP279118 | PRJNA659812 | Defects in the zonule in loxl1 knock out zebrafish | PRJNA659812 | Other | Purposes: To investigate the roles of lysyl oxidase like 1 loxl1 in zebrafish eye development and the potency of loxl1 knock out in mimicking the ocular manifestations of Exfoliation Syndrome.Methods: CRISPR/Cas9 technology was used to generate a frameshift deletion shifting code deletion in zebrafish loxl1. Expression profiles and ocular manifestations of wildtype WT heterozygous mutant loxl1+/ and homozygous mutant loxl1 / zebrafish were analyzed in a range of developmental stages from whole zebrafish larvae to dissected adult zebrafish eyes using western blot immunofluorescence real time RT PCR and transcriptome resequencing analysis RNA seq. Scanning electron microscopy SEM was used to study the zonular structure and corneal fiber organization of adult zebrafish eyes.Results: The loxl1 mutation caused zonular bundling disorders in juvenile zebrafish and accumulation of pearl like particles adhering to the adult zebrafish zonule. SEM revealed more numerous particles but of smaller size in loxl1 / zebrafish compared with those in loxl1+/ although light microscopy showed that the overall staining of the zonule in loxl1+/ was darker than that in loxl1 / . Expression of transforming growth factor b tgfb pathway related proteins changed at the protein level but not the mRNA level. The phosphorylated Smad3 pSmad3 level was locally activated in the iris side trabecular meshwork and ciliary body of loxl1 / adults.Conclusions: Defects in the zonular bundling found in loxl1 knock out zebrafish indicate dysplasia. Compared with adult loxl1+/ zebrafish more numerous but smaller pearl like particles adhered to the zonule in adult loxl1 / zebrafish. loxl1 / zebrafish is a promising animal model of XFS zonular pathology. | 3 larvae loxl1 / | strain:AB|dev stage:7 dpf|sex:male|tissue:larvae|source material id:3|BioSampleModel:Model organism or animal | 7dpf ko | loxl1 zebrafish3 | loxl1 zebrafish3 | materials and methods | RNA-Seq | TRANSCRIPTOMIC | RANDOM | SINGLE | ILLUMINA | Illumina HiSeq 4000 | SRP279118 | larvae_ko_20190920NA_ATAGCGAC_S101_L001_R1_001.fastq.gz larvae_ko_20190920NA_ATAGCGAC_S101_L001_R2_001.fastq.gz | fastq fastq | 8570236936.0 | 28378268.0 | larvae ko 20190920NA ATAGCGAC S101 L001 R1 001.fastq.gz | 0:151 1:151 | A:2291363400;C:1985315062;G:2023225375;T:2269717794;N:615305 | 151 | 151 | 2291363400 | 1985315062 | 2023225375 | 2269717794 | 615305 | SRX9026395 | SRS7277845 | SRA1118248 | Eye and ENT Hospital of Fudan University|Ophthalmology and Vision Science | Eye and ENT Hospital of Fudan University | 2 | 0.95641 | 0.95018 | 0.09997 | 0.0968 | 0.68436 | 0.68627 | 0.45419 | 0.45754 | 151 | 151 | B | B | biological fallback assumption | illumina | hiseq_era | unknown | random_priming | unknown | bulk | unknown | unknown | China | 2020-08-27 | Larval | Larval | Undetermined | Undetermined |