run_metadata: 34218
This data as json
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| 34218 | SRR31539743 | SRX26906743 | SRS23386329 | SRP548139 | PRJNA1191617 | Sox10 is required for systemic initiation of bone mineralization | GSE283071 | Transcriptome Analysis | Heterozygous variants in SOX10 cause syndromes affecting pigmentation digestion hearing and neural development primarily attributable to failed differentiation or loss of non skeletal neural crest derivatives. We report here an additional novel requirement for Sox10 in bone mineralization. Neither crest nor mesoderm derived bones initiate mineralization on time in zebrafish sox10 mutants despite normal osteoblast differentiation and matrix production. Mutants are deficient in the Trpv6+ ionocytes that take up calcium from the environment resulting in severe calcium deficiency. As these ionocytes derive from ectoderm not crest we hypothesized that the primary defect resides in a separate organ that systemically regulates ionocyte numbers. RNAseq revealed significantly elevated stanniocalcin Stc1a an anti hypercalcemic hormone in sox10 mutants. Stc1a inhibits calcium uptake in fish by repressing trpv6 expression and Trpv6+ ionocyte proliferation. Epistasis assays confirm excess Stc1a as the proximate cause of the calcium deficit. The pronephros derived glands that synthesize Stc1a interact with sox10+ cells but these cells are missing in mutants. We conclude that sox10+ crest derived cells non autonomously limit Stc1a production to allow the inaugural wave of calcium uptake necessary to initiate bone mineralization. Overall design: Bulk RNA seq was performed on pooled whole body zebrafish wild type and sox10 mutant larvae at 45 hpf 4 dpf and 7 dpf. Total of 18 samples. 45 hpf 3 replicates: controls n = 10 20 & 25; mutants n = 10 15 & 15 embryos 4 dpf 3 replicates: controls n = 10 15 & 12; mutants n = 10 11 & 12 embryos and 7 dpf 3 replicates: controls n = 12 30 & 18; mutants = 18 12 & 7 embryos | pubmed:39791977 | Whole body 45 hpf sox10 mutant embryos biol rep 3 | GSM8655448 | source name:whole body|tissue:whole body|genotype:sox10 / |time:45 hpf|geo loc name:missing|collection date:missing | Whole body 45 hpf sox10 mutant embryos biol rep 3 | The data was processed through the Galaxy project using Trimmomatic version 0.39 Sequences were pseudoaligned and quantified at TPM values using Kallisto. Assembly: GRCz11 Supplementary files format and content: Tab delimited text file includes TPM values for each transcript at 45 hpf 4 dpf and 7 dpf | whole body | RNA was extracted using the RNAqueous 4PCR Total RNA Isolation Kit Invitrogen 150 to 300 ng of total RNA determined by InvitrogenTM QubitTM high sensitivity spectrofluorometric measurement was poly A selected and reverse transcribed using Illumina’s TruSeq® stranded mRNA library preparation kit. | tissue:whole body|genotype:sox10 / |time:45 hpf | GSM8655448 | GSM8655448: Whole body 45 hpf sox10 mutant embryos biol rep 3; Danio rerio; RNA Seq | GSM8655448 r1 | GSM8655448 | 1 | RNA was extracted using the RNAqueous 4PCR Total RNA Isolation Kit Invitrogen 150 to 300 ng of total RNA determined by InvitrogenTM QubitTM high sensitivity spectrofluorometric measurement was poly A selected and reverse transcribed using Illumina's TruSeq® stranded mRNA library preparation kit. | RNA-Seq | TRANSCRIPTOMIC | cDNA | PAIRED | ILLUMINA | Illumina NovaSeq 6000 | SRP548139 | 45 hpf_mut 3_R1.fastq.gz 45 hpf_mut 3_R2.fastq.gz | fastq fastq | 14338321572.0 | 47477886.0 | GSM8655448 r1 | 0:151 1:151 | A:3808719246;C:3382555065;G:3212075099;T:3933758367;N:1213795 | 151 | 151 | 3808719246 | 3382555065 | 3212075099 | 3933758367 | 1213795 | SRX26906743 | SRS23386329 | Barske Lab, Human Genetics, Cincinnati Children's Hospital | B | B | biological fallback assumption | illumina | novaseq_era | unknown | poly_a | trueseq | bulk | bulk | bulk | United States | 2024-11-27 | Multi-stage | Multi-stage | Trunk | Surface Structure |