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We studied 24 participants with likely pathogenic or pathogenic variants in <jats:italic>DYRK1A<\/jats:italic> through the Simons Searchlight study and systematically assessed their medical history and development using standardized instruments: Vineland Adaptive Behavior Scale II (VABS\u2010II) and Child Behavior Checklists\/1.5\u20105 and 6\u201018 (CBCL\/1.5\u20105, CBCL\/6\u201018). All of the individuals in the cohort had neurological manifestations including intellectual disability or developmental delay, microcephaly, autism spectrum disorder, and\/or seizures. The severity of the neurodevelopmental disorder was variable with a few children scoring in the moderately low range on the adaptive behavior composite score on the VABS\u2010II. This study confirms the association of <jats:italic>DYRK1A<\/jats:italic> haploinsufficiency with neurodevelopmental disabilities, microcephaly, autism spectrum disorder, and epilepsy and quantifies the range of adaptive behaviors.<\/jats:p>","DOI":"10.1002\/ajmg.a.62721","type":"journal-article","created":{"date-parts":[[2022,3,14]],"date-time":"2022-03-14T07:34:01Z","timestamp":1647243241000},"page":"1954-1963","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":10,"title":["Characterization of phenotypic range in <scp><i>DYRK1A<\/i><\/scp> haploinsufficiency syndrome using standardized behavioral measures"],"prefix":"10.1002","volume":"188","author":[{"given":"Rebecca","family":"Fenster","sequence":"first","affiliation":[{"name":"Department of Pediatrics Columbia University Irving Medical Center  New York New York 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