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Genetic murine models of craniosynostosis exist, but often imperfectly model human patients. Case, cohort, and surveillance studies have identified excess thyroid hormone as an agent that can either cause or exacerbate human cases of craniosynostosis.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>Here we investigate the influence of <jats:italic>in utero<\/jats:italic> and <jats:italic>in vitro<\/jats:italic> exogenous thyroid hormone exposure on a murine model of craniosynostosis, <jats:italic>Twist 1 +\/\u2212<\/jats:italic>.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>By 15 days post\u2010natal, there was evidence of coronal suture fusion in the <jats:italic>Twist 1<\/jats:italic> +\/\u2212 model, regardless of exposure. With the exception of craniofacial width, there were no significant effects of exposure; however, the <jats:italic>Twist 1 +\/\u2212<\/jats:italic> phenotype was significantly different from the wild\u2010type control. <jats:italic>Twist 1 +\/\u2212<\/jats:italic> cranial suture cells did not respond to thyroxine treatment as measured by proliferation, osteogenic differentiation, and gene expression of osteogenic markers. However, treatment of these cells did result in modulation of thyroid associated gene expression.<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusion<\/jats:title><jats:p>Our findings suggest the phenotypic effects of the genetic mutation largely outweighed the effects of thyroxine exposure in the <jats:italic>Twist 1<\/jats:italic> +\/\u2212 model. These results highlight difficultly in experimentally modeling gene\u2013environment interactions for craniosynostotic phenotypes. 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