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In the tooth, <jats:italic>Notum<\/jats:italic> is known to be expressed in odontoblasts, and severe dentin defects and irregular tooth roots have been reported in <jats:italic>Notum<\/jats:italic>-deficient mice. However, the precise expression pattern of <jats:italic>Notum<\/jats:italic> in early tooth development, and the role of <jats:italic>Notum<\/jats:italic> in crown and root patterns remain elusive. In the present study, we identified a novel <jats:italic>Notum<\/jats:italic> expression in primary enamel knot (EK), secondary EKs, and dental papilla during tooth development. <jats:italic>Notum<\/jats:italic>-deficient mice exhibited enlarged secondary EKs, resulting in broader cusp tips, altered cusp patterns, and reduced concavity in crown outline. These alterations in crown outline led to a reduction in cervical tongue length, thereby inducing root fusion in <jats:italic>Notum<\/jats:italic>-deficient mice. 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