{
  "abstract": "Regeneration programs enable animals to restore damaged or lost tissues, and the range of stimuli for these programs is incompletely understood. Here, we used zebrafish, a vertebrate species with exceptional regenerative capacity, to identify chemically induced mutations that alter regeneration-associated gene activation. Transgenic zebrafish with a permissive promoter and EGFP cassette inserted in the vicinity of the pro-regenerative factor gene fgf20a were mutagenized, and larvae homozygous for ENU-induced mutations were assessed for disruptions in fgf20a-directed reporter gene expression following fin fold amputation. One line was identified with heritable, elevated fgf20a:EGFP presence in the absence of experimental injury, localized to regions of fin fold tissue undergoing degeneration. Whole-genome sequencing identified a mutation within exon 72 of the fraser syndrome 1 (fras1) gene, mutated in patients with inherited skin disease. fras1 mutant larvae spontaneously displayed broader signatures of regeneration, and zebrafish crispants for homologs of other genes mutated in human developmental diseases also displayed regeneration-associated gene expression in regions of dysmorphology. Tempering Fgf signaling by transgenic expression of a dominant-negative Fgf receptor in fras1 mutants exacerbated the disease phenotype. Our findings provide evidence that regeneration programs are harnessed in response to developmental defects caused by genetic mutations, potentially buffering deleterious phenotypes.\n\nRegeneration programs enable animals to restore damaged or lost tissues, but the stimuli that regulate these programs remain unclear. This study shows that mutations causing developmental defects activate regeneration responses that buffer the deleterious effects of congenital disease in zebrafish embryos.",
  "abstract_shown": true,
  "affiliations": [
    "Morgridge Institute for Research, Madison, Wisconsin, United States of America",
    "Department of Cell and Regenerative Biology, University of Wisconsin--Madison, Madison, Wisconsin, United States of America",
    "Department of Cell Biology, Duke University School of Medicine, Durham, North Carolina, United States of America"
  ],
  "article_type": "Short Reports",
  "arxiv": null,
  "author_total": 5,
  "authors": [
    {
      "affs": [
        0
      ],
      "name": "Kazunori Ando"
    },
    {
      "affs": [
        1
      ],
      "name": "Sushant Bangru"
    },
    {
      "affs": [
        2
      ],
      "name": "John Welsby"
    },
    {
      "name": "John D. Thompson"
    },
    {
      "name": "Kenneth D. Poss"
    }
  ],
  "bibtex": "https://research.draconitic.com/paper/71twkj2zbpy1p.bib",
  "cited_by": 0,
  "doi": "10.1371/journal.pbio.3004011",
  "fulltext_url": "https://journals.plos.org/plosbiology/article?id=10.1371/journal.pbio.3004011",
  "id": "71twkj2zbpy1p",
  "issns": [
    "1545-7885"
  ],
  "landing_url": "https://journals.plos.org/plosbiology/article?id=10.1371/journal.pbio.3004011",
  "language": null,
  "licence": "CC-BY-4.0",
  "licence_label": "CC BY 4.0",
  "licence_source": "journal",
  "licence_url": "https://creativecommons.org/licenses/by/4.0/",
  "metadata_licence": null,
  "native_subjects": [
    [
      "openalex",
      "1307|Zebrafish Biomedical Research Applications"
    ],
    [
      "plos",
      "/Biology and life sciences/Developmental biology/Life cycles/Larvae"
    ],
    [
      "plos",
      "/Biology and life sciences/Developmental biology/Morphogenesis/Regeneration"
    ],
    [
      "plos",
      "/Biology and life sciences/Developmental biology/Organism development/Regeneration"
    ],
    [
      "plos",
      "/Biology and life sciences/Genetics/Gene expression"
    ],
    [
      "plos",
      "/Biology and life sciences/Genetics/Heredity/Homozygosity"
    ],
    [
      "plos",
      "/Biology and life sciences/Genetics/Phenotypes"
    ],
    [
      "plos",
      "/Biology and life sciences/Organisms/Eukaryota/Animals/Vertebrates/Fish"
    ],
    [
      "plos",
      "/Biology and life sciences/Organisms/Eukaryota/Animals/Vertebrates/Fish/Osteichthyes/Zebrafish"
    ],
    [
      "plos",
      "/Biology and life sciences/Zoology/Animals/Vertebrates/Fish"
    ],
    [
      "plos",
      "/Biology and life sciences/Zoology/Animals/Vertebrates/Fish/Osteichthyes/Zebrafish"
    ],
    [
      "plos",
      "/Medicine and health sciences/Surgical and invasive medical procedures/Surgical amputation"
    ],
    [
      "plos",
      "/Research and analysis methods/Animal studies/Experimental organism systems/Animal models/Zebrafish"
    ],
    [
      "plos",
      "/Research and analysis methods/Animal studies/Experimental organism systems/Model organisms/Zebrafish"
    ],
    [
      "plos",
      "/Research and analysis methods/Model organisms/Zebrafish"
    ]
  ],
  "openalex": "W7213878919",
  "pdf_url": "https://journals.plos.org/plosbiology/article/file?id=10.1371/journal.pbio.3004011&type=printable",
  "pmcid": null,
  "pmid": null,
  "published": "2026-09-21",
  "publisher": "Public Library of Science",
  "ris": "https://research.draconitic.com/paper/71twkj2zbpy1p.ris",
  "sources": [
    {
      "first_seen": "2026-10-30T12:17:43Z",
      "native_id": "10.1371/journal.pbio.3004011",
      "source": "plos",
      "url": "https://journals.plos.org/plosbiology/article?id=10.1371/journal.pbio.3004011"
    }
  ],
  "subjects": [
    {
      "field": "biology",
      "origin": "plos:developmental biology",
      "subfield": "developmental-biology"
    },
    {
      "field": "biology",
      "origin": "plos:genetics",
      "subfield": "genetics-genomics"
    },
    {
      "field": "biology",
      "origin": "plos:zoology",
      "subfield": "zoology-behaviour"
    }
  ],
  "title": "Regeneration programs buffer genetic defects in animal development",
  "title_text": "Regeneration programs buffer genetic defects in animal development",
  "type": "peer-reviewed",
  "updated_at": "2026-10-30T12:17:43Z",
  "url": "https://research.draconitic.com/paper/71twkj2zbpy1p",
  "venue": "PLOS Biology",
  "versions": []
}