{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,20]],"date-time":"2026-09-20T09:42:15Z","timestamp":1789897335526,"version":"4.0.1"},"reference-count":67,"publisher":"Wiley","issue":"3","license":[{"start":{"date-parts":[[2010,1,13]],"date-time":"2010-01-13T00:00:00Z","timestamp":1263340800000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Developmental Dynamics"],"published-print":{"date-parts":[[2010,3]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:p>Apert syndrome (AS) is one of at least nine disorders considered members of the fibroblast growth factor receptor (FGFR) \u20101, \u20102, and \u20103\u2013related craniosynostosis syndromes. Nearly 100% of individuals diagnosed with AS carry one of two neighboring mutations on Fgfr2. The cranial phenotype associated with these two mutations includes coronal suture synostosis, either unilateral (unicoronal synostosis) or bilateral (bicoronal synostosis). Brain dysmorphology associated with AS is thought to be secondary to cranial vault or base alterations, but the variation in brain phenotypes within Apert syndrome is unexplained. Here, we present novel three\u2010dimensional data on brain phenotypes of inbred mice at postnatal day 0 each carrying one of the two Fgfr2 mutations associated with AS. Our data suggest that the brain is primarily affected, rather than secondarily responding to skull dysmorphogenesis. Our hypothesis is that the skull and brain are both primarily affected in craniosynostosis and that shared phenogenetic developmental processes affect both tissues in craniosynostosis of Apert syndrome. Developmental Dynamics 239:987\u2013997, 2010. \u00a9 2010 Wiley\u2010Liss, Inc.<\/jats:p>","DOI":"10.1002\/dvdy.22218","type":"journal-article","created":{"date-parts":[[2010,1,13]],"date-time":"2010-01-13T13:02:41Z","timestamp":1263387761000},"page":"987-997","source":"Crossref","is-referenced-by-count":42,"title":["Brain phenotypes in two FGFR2 mouse models for Apert syndrome"],"prefix":"10.1002","volume":"239","author":[{"given":"Kristina","family":"Aldridge","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Cheryl A.","family":"Hill","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jordan R.","family":"Austin","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Christopher","family":"Percival","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Neus","family":"Martinez\u2010Abadias","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Thomas","family":"Neuberger","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yingli","family":"Wang","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ethylin Wang","family":"Jabs","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Joan T.","family":"Richtsmeier","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2010,1,13]]},"reference":[{"key":"e_1_2_7_2_1","doi-asserted-by":"crossref","first-page":"31","DOI":"10.1046\/j.1469-7580.2002.00074.x","article-title":"Central nervous system phenotypes in craniosynostosis","volume":"201","author":"Aldridge K","year":"2002","journal-title":"J Anat"},{"key":"e_1_2_7_3_1","doi-asserted-by":"crossref","first-page":"690","DOI":"10.1002\/ar.a.20201","article-title":"Brain morphology in nonsyndromic unicoronal craniosynostosis","volume":"285","author":"Aldridge K","year":"2005","journal-title":"Anat Rec"},{"key":"e_1_2_7_4_1","doi-asserted-by":"crossref","first-page":"373","DOI":"10.1111\/j.1469-7580.2005.00397.x","article-title":"Relationship of brain and skull in pre\u2010 and postoperative sagittal synostosis","volume":"206","author":"Aldridge K","year":"2006","journal-title":"J Anat"},{"key":"e_1_2_7_5_1","doi-asserted-by":"crossref","first-page":"1060","DOI":"10.1002\/ajmg.a.31721","article-title":"Differential effects of trisomy on brain shape and volume in related aneuploid mouse models","volume":"143","author":"Aldridge K","year":"2007","journal-title":"Am J Med Genet Part A"},{"key":"e_1_2_7_6_1","doi-asserted-by":"crossref","first-page":"161","DOI":"10.1159\/000081933","article-title":"Analysis of intracranial volume in Apert syndrome genotypes","volume":"40","author":"Anderson P","year":"2004","journal-title":"Pediatr Neurosurg"},{"key":"e_1_2_7_7_1","doi-asserted-by":"crossref","first-page":"83","DOI":"10.1159\/000072258","article-title":"Expression of FGF receptors 1, 2, 3 in the embryonic and postnatal mouse brain compared with Pdgfra, Olig2 and Plp\/dm20: implications for oligodendrocyte development","volume":"25","author":"Bansal R","year":"2003","journal-title":"Dev Neurosci"},{"key":"e_1_2_7_8_1","doi-asserted-by":"crossref","first-page":"151","DOI":"10.1111\/j.1469-1809.1959.tb01728.x","article-title":"Apert's syndrome (a type of acrocephalosyndactyly)\u2010\u2010observations on a British series of thirty\u2010nine cases","volume":"24","author":"Blank C","year":"1960","journal-title":"Ann Hum Genet Lond"},{"key":"e_1_2_7_9_1","doi-asserted-by":"crossref","first-page":"169","DOI":"10.1016\/S8756-3282(03)00222-9","article-title":"A Ser250Trp substitution in mouse fibroblast growth factor receptor 2 (Fgfr2) results in craniosynostosis","volume":"33","author":"Chen L","year":"2003","journal-title":"Bone"},{"key":"e_1_2_7_10_1","first-page":"316","volume-title":"Craniosynostosis: diagnosis, evaluation, and management","author":"Cohen M","year":"2000"},{"key":"e_1_2_7_11_1","doi-asserted-by":"crossref","first-page":"36","DOI":"10.1002\/ajmg.1320350108","article-title":"The central nervous system in the Apert syndrome","volume":"35","author":"Cohen M","year":"1990","journal-title":"Am J Med Genet"},{"key":"e_1_2_7_12_1","first-page":"565","article-title":"Agenesis of the corpus callosum. 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