{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,2,7]],"date-time":"2026-02-07T09:09:39Z","timestamp":1770455379573,"version":"3.49.0"},"reference-count":26,"publisher":"Wiley","issue":"5","license":[{"start":{"date-parts":[[2018,10,26]],"date-time":"2018-10-26T00:00:00Z","timestamp":1540512000000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"funder":[{"name":"Cure AHC"},{"name":"Duke University Funds"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Develop Med Child Neuro"],"published-print":{"date-parts":[[2019,5]]},"abstract":"<jats:sec><jats:title>Aim<\/jats:title><jats:p>To determine the neuropsychological abnormalities that occur in alternating hemiplegia of childhood (<jats:styled-content style=\"fixed-case\">AHC<\/jats:styled-content>) and report on our experience in managing them.<\/jats:p><\/jats:sec><jats:sec><jats:title>Method<\/jats:title><jats:p>Patients underwent evaluations according to our standardized <jats:styled-content style=\"fixed-case\">AHC<\/jats:styled-content> pathway. Data were entered into our prospective <jats:styled-content style=\"fixed-case\">AHC<\/jats:styled-content> database and then analyzed.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>Of the cohort of 25 consecutive patients (ages 15mo\u201342y), eight had initial chief complaints about cognition, 14 language, five attention, and 11 behavior. As compared to population norms means, neuropsychological and behavioral assessment tools (including Child Behavior Checklist, Vineland Adaptive Behavior Scales, Peabody Picture Vocabulary, and Wechsler Intelligence Quotient tests) showed significant impairments in multiple domains: cognition, expressive and receptive language, executive function\/attention, and behavior (<jats:italic>p<\/jats:italic>&lt;0.05 in all comparisons). Evaluations generated management recommendations in all patients. Twenty had neuropsychiatric diagnoses: 10 attention\u2010deficit\/hyperactivity disorder (<jats:styled-content style=\"fixed-case\">ADHD<\/jats:styled-content>), seven disruptive behavior, and three anxiety disorder. Eight out of nine patients with <jats:styled-content style=\"fixed-case\">ADHD<\/jats:styled-content> who were prescribed medications responded to pharmacotherapy.<\/jats:p><\/jats:sec><jats:sec><jats:title>Interpretation<\/jats:title><jats:p>Patients with <jats:styled-content style=\"fixed-case\">AHC<\/jats:styled-content> have developmental difficulties related to impairments in multiple neuropsychological domains. This supports the hypothesis that the underlying <jats:styled-content style=\"fixed-case\">AHC<\/jats:styled-content> pathophysiology involves diffuse neuronal dysfunction. Testing generated recommendations to help manage these difficulties. Patients with <jats:styled-content style=\"fixed-case\">AHC<\/jats:styled-content> also have a range of neuropsychiatric diagnoses, the most common being <jats:styled-content style=\"fixed-case\">ADHD<\/jats:styled-content> which responds to pharmacotherapy.<\/jats:p><\/jats:sec><jats:sec><jats:title>What this paper adds<\/jats:title><jats:p>\n<jats:list list-type=\"bullet\">\n\n<jats:list-item><jats:p>Patients with alternating hemiplegia of childhood (AHC) have developmental difficulties with underlying neuropsychological impairments.<\/jats:p><\/jats:list-item>\n\n<jats:list-item><jats:p>The findings in this study are consistent with an underlying AHC pathophysiology which involves diffuse neuronal, probably largely GABAergic, dysfunction.<\/jats:p><\/jats:list-item>\n\n<jats:list-item><jats:p>Patients with AHC have a range of neuropsychiatric diagnoses, the most common being attention\u2010deficit\/hyperactivity disorder.<\/jats:p><\/jats:list-item>\n<\/jats:list>\n<\/jats:p><\/jats:sec>","DOI":"10.1111\/dmcn.14077","type":"journal-article","created":{"date-parts":[[2018,10,26]],"date-time":"2018-10-26T08:23:40Z","timestamp":1540542220000},"page":"547-554","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":33,"title":["Cognitive, adaptive, and behavioral profiles and management of alternating hemiplegia of childhood"],"prefix":"10.1111","volume":"61","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-5469-1092","authenticated-orcid":false,"given":"Joan M","family":"Jasien","sequence":"first","affiliation":[{"name":"Division of Pediatric Neurology Duke University Health System Durham NC USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Melanie","family":"Bonner","sequence":"additional","affiliation":[{"name":"Department of Psychiatry and Behavioral Sciences Duke University Durham NC 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