{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,6,16]],"date-time":"2026-06-16T22:06:45Z","timestamp":1781647605292,"version":"3.54.5"},"reference-count":23,"publisher":"Wiley","issue":"4","license":[{"start":{"date-parts":[[2016,6,13]],"date-time":"2016-06-13T00:00:00Z","timestamp":1465776000000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"funder":[{"DOI":"10.13039\/100007046","name":"Greater Milwaukee Foundation","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100007046","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100001582","name":"Dermatology Foundation","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100001582","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Pediatric Dermatology"],"published-print":{"date-parts":[[2016,7]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:sec><jats:title>Background<\/jats:title><jats:p>Practitioners who work with children with posterior fossa, facial hemangiomas, arterial anomalies, cardiovascular anomalies, and abnormalities of the eye (<jats:styled-content style=\"fixed-case\">PHACE<\/jats:styled-content>) syndrome need information about neurodevelopmental outcomes to provide appropriate anticipatory guidance and education for parents. This study aimed to determine the neurodevelopmental outcomes in children with <jats:styled-content style=\"fixed-case\">PHACE<\/jats:styled-content> syndrome and identify which children may be at greatest risk for delays.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>Children with a diagnosis of <jats:styled-content style=\"fixed-case\">PHACE<\/jats:styled-content> syndrome (ages 4\u201318 yrs) were recruited from the <jats:styled-content style=\"fixed-case\">PHACE<\/jats:styled-content> Syndrome International Clinical Registry and Genetic Repository. Participants (<jats:italic>n<\/jats:italic> = 25) underwent a neurodevelopmental evaluation at a children's hospital tertiary care referral center between 2009 and 2013. Children completed standardized neurocognitive tests assessing multiple domains. Parents completed standardized questionnaires assessing behavioral and emotional functioning.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>Results were analyzed according to cohort and individual subject. Mean scores for the cohort did not differ significantly from test norms in most domains. The only subtest that the cohort scored lower on than test norms was Word Structure, a language task. Forty\u2010four percent of the sample scored within the normal range in all domains, 28% had one score in the at\u2010risk range (1\u20132 standard deviations [<jats:styled-content style=\"fixed-case\">SD<\/jats:styled-content>s] below the mean), 12% had two or more scores in the at\u2010risk range, and 16% had at least one score in the impaired range (&gt;2 <jats:styled-content style=\"fixed-case\">SD<\/jats:styled-content>s below the mean).<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusion<\/jats:title><jats:p>Although most children in this cohort of patients with <jats:styled-content style=\"fixed-case\">PHACE<\/jats:styled-content> syndrome did not have significant neurodevelopmental deficits, a subset of patients had delays in multiple areas. Practitioners who work with these children should routinely ask about neurocognitive and developmental skills. Children with more severe phenotypes should be referred for appropriate evaluations and intervention services.<\/jats:p><\/jats:sec>","DOI":"10.1111\/pde.12870","type":"journal-article","created":{"date-parts":[[2016,6,13]],"date-time":"2016-06-13T05:54:08Z","timestamp":1465797248000},"page":"415-423","source":"Crossref","is-referenced-by-count":17,"title":["Neurodevelopmental Outcomes in Children with <scp>PHACE<\/scp> Syndrome"],"prefix":"10.1111","volume":"33","author":[{"given":"Cheryl L.","family":"Brosig","sequence":"first","affiliation":[{"name":"Department of Pediatrics Medical College of Wisconsin Milwaukee Wisconsin"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Dawn H.","family":"Siegel","sequence":"additional","affiliation":[{"name":"Department of Pediatrics Medical College of Wisconsin Milwaukee Wisconsin"},{"name":"Department of Dermatology Medical College of Wisconsin Milwaukee Wisconsin"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Anita N.","family":"Haggstrom","sequence":"additional","affiliation":[{"name":"Department of Dermatology School of Medicine Indiana University Bloomington Indiana"},{"name":"Department of Pediatrics School of Medicine Indiana University Bloomington Indiana"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ilona J.","family":"Frieden","sequence":"additional","affiliation":[{"name":"Department of Dermatology University of California, San Francisco San Francisco California"},{"name":"Department of Pediatrics University of California, San Francisco San Francisco California"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Beth A.","family":"Drolet","sequence":"additional","affiliation":[{"name":"Department of Pediatrics Medical College of Wisconsin Milwaukee Wisconsin"},{"name":"Department of Dermatology Medical College of Wisconsin Milwaukee Wisconsin"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2016,6,13]]},"reference":[{"key":"e_1_2_6_2_1","doi-asserted-by":"publisher","DOI":"10.1001\/archderm.1996.03890270083012"},{"key":"e_1_2_6_3_1","doi-asserted-by":"publisher","DOI":"10.1067\/mpd.2001.114880"},{"key":"e_1_2_6_4_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.31189"},{"key":"e_1_2_6_5_1","unstructured":"HaggstromA BaselgaE ChamlinSet al.Large facial hemangiomas: what is the risk of PHACE?17th International Workshop on Vascular Anomalies June 21\u201324 2008 Boston MA [abstract]."},{"key":"e_1_2_6_6_1","unstructured":"HessCP FullertonHJ AugsteNet al.The spectrum of cerebrovascular anomalies in PHACES. 17th International Workshop on Vascular Anomalies June 21\u201324 2008 Boston MA [abstract]."},{"key":"e_1_2_6_7_1","doi-asserted-by":"publisher","DOI":"10.1542\/peds.2005-1683"},{"key":"e_1_2_6_8_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.pediatrneurol.2006.06.021"},{"key":"e_1_2_6_9_1","doi-asserted-by":"publisher","DOI":"10.1161\/STROKEAHA.107.485185"},{"key":"e_1_2_6_10_1","doi-asserted-by":"publisher","DOI":"10.1177\/0883073812450073"},{"key":"e_1_2_6_11_1","doi-asserted-by":"publisher","DOI":"10.1542\/peds.2009-0082"},{"key":"e_1_2_6_12_1","doi-asserted-by":"publisher","DOI":"10.1002\/mrdd.10043"},{"key":"e_1_2_6_13_1","volume-title":"Wechsler preschool and primary scale of intelligence","author":"Wechsler D","year":"2002"},{"key":"e_1_2_6_14_1","volume-title":"Wechsler intelligence scale for children","author":"Wechsler D","year":"2003"},{"key":"e_1_2_6_15_1","volume-title":"Wechsler abbreviated scale of intelligence","author":"Wechsler D","year":"1999"},{"key":"e_1_2_6_16_1","volume-title":"The Beery\u2010Buktenica developmental test of visual\u2010motor integration","author":"Beery KE","year":"2004"},{"key":"e_1_2_6_17_1","volume-title":"NEPSY: a developmental neuropsychological assessment manual","author":"Korkman M","year":"1998"},{"key":"e_1_2_6_18_1","volume-title":"Conners\u2019 rating scales, revised user's manual","author":"Conners CK","year":"1997"},{"key":"e_1_2_6_19_1","volume-title":"Manual for the ASEBA preschool forms and profiles","author":"Achenbach TM","year":"2000"},{"key":"e_1_2_6_20_1","doi-asserted-by":"publisher","DOI":"10.1177\/073428299000800313"},{"key":"e_1_2_6_21_1","volume-title":"Clinical evaluation of language fundamentals\u2014preschool","author":"Wiig EH","year":"2004"},{"key":"e_1_2_6_22_1","doi-asserted-by":"publisher","DOI":"10.1111\/pde.12447"},{"key":"e_1_2_6_23_1","doi-asserted-by":"publisher","DOI":"10.1597\/1545-1569(2001)038<0068:DCCFAA>2.0.CO;2"},{"key":"e_1_2_6_24_1","unstructured":"BlackorbyJ SchillerE MallikSet al.Patterns in the identification of and outcomes for children and youth with disabilities.http:\/\/ies.ed.gov\/pubsearch\/pubsinfo.asp?pubid=NCEE20104005(accessed on January 13 2016)."}],"container-title":["Pediatric Dermatology"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.wiley.com\/onlinelibrary\/tdm\/v1\/articles\/10.1111%2Fpde.12870","content-type":"unspecified","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/pde.12870","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,9,26]],"date-time":"2023-09-26T15:43:29Z","timestamp":1695743009000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1111\/pde.12870"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2016,6,13]]},"references-count":23,"journal-issue":{"issue":"4","published-print":{"date-parts":[[2016,7]]}},"alternative-id":["10.1111\/pde.12870"],"URL":"https:\/\/doi.org\/10.1111\/pde.12870","archive":["Portico"],"relation":{},"ISSN":["0736-8046","1525-1470"],"issn-type":[{"value":"0736-8046","type":"print"},{"value":"1525-1470","type":"electronic"}],"subject":[],"published":{"date-parts":[[2016,6,13]]}}}