2022
Zebrafish models of Alx-linked frontonasal dysplasia reveal a role for Alx1 and Alx3 in the anterior segment and vasculature of the developing eye
Yoon B, Yeung P, Santistevan N, Bluhm L, Kawasaki K, Kueper J, Dubielzig R, Vanoudenhove J, Cotney J, Liao E, Grinblat Y. Zebrafish models of Alx-linked frontonasal dysplasia reveal a role for Alx1 and Alx3 in the anterior segment and vasculature of the developing eye. Biology Open 2022, 11: bio059189. PMID: 35142342, PMCID: PMC9167625, DOI: 10.1242/bio.059189.Peer-Reviewed Original ResearchConceptsALX geneAnterior neurocraniumZebrafish modelGenetic mechanismsNovel roleAnterior segment formationHomeobox transcription factorCranial neural crestOxidative stress responseParalogous genesConserved roleAnterior segment defectsAbsence of eyesEthanol toxicityTranscription factorsTranscriptomic analysisLineage labelingAlx1Midfacial morphogenesisKey regulatorNeural crestStress responseSegment formationMutantsVascular development
2018
High-Resolution Epigenomic Atlas of Human Embryonic Craniofacial Development
Wilderman A, VanOudenhove J, Kron J, Noonan JP, Cotney J. High-Resolution Epigenomic Atlas of Human Embryonic Craniofacial Development. Cell Reports 2018, 23: 1581-1597. PMID: 29719267, PMCID: PMC5965702, DOI: 10.1016/j.celrep.2018.03.129.Peer-Reviewed Original ResearchConceptsRegulatory sequencesEmbryonic developmentEmbryonic craniofacial developmentEmbryonic craniofacial tissueGene regulatory programsNormal facial variationHuman embryonic developmentCraniofacial abnormalitiesEpigenomic annotationsEpigenomic atlasCraniofacial developmentIntronic sequencesCraniofacial tissuesRegulatory programsCraniofacial researchersMultiple tissuesCell typesSignificant enrichmentSystematic identificationCommon variantsCausal regionOrofacial cleftingEmbryonic periodSequenceCraniofacial complex