| LDR | | 00000nam u2200205 4500 |
| 001 | | 000000433136 |
| 005 | | 20200225112632 |
| 008 | | 200131s2019 ||||||||||||||||| ||eng d |
| 020 | |
▼a 9781085619981 |
| 035 | |
▼a (MiAaPQ)AAI13815213 |
| 040 | |
▼a MiAaPQ
▼c MiAaPQ
▼d 247004 |
| 082 | 0 |
▼a 152 |
| 100 | 1 |
▼a Wu, Robin T. |
| 245 | 10 |
▼a Neurodevelopmental Risks of Non-syndromic Craniosynostosis. |
| 260 | |
▼a [S.l.]:
▼b Yale University.,
▼c 2019. |
| 260 | 1 |
▼a Ann Arbor:
▼b ProQuest Dissertations & Theses,
▼c 2019. |
| 300 | |
▼a 86 p. |
| 500 | |
▼a Source: Dissertations Abstracts International, Volume: 81-03, Section: B. |
| 500 | |
▼a Advisor: Persing, John A. |
| 502 | 1 |
▼a Thesis (M.D.)--Yale University, 2019. |
| 506 | |
▼a This item must not be sold to any third party vendors. |
| 506 | |
▼a This item must not be added to any third party search indexes. |
| 520 | |
▼a Purpose: Nonsyndromic craniosynostosis may manifest with complex cognitive, language, behavioral, and emotional sequelae, depending on the suture fusion involved. De-novo or rare transmitted mutations in the SMAD6 gene affect midline synostosis in 7% of patients. Current standards of assessment, such as the Bayley Scales of Infant Development (BSID), may not be predictive of long-term development, paving the way for newer assessments such as functional magnetic resonance imaging (fMRI) and the event related potentials (ERP), which measures passive neurological responses to speech sounds. Methods: Cranially-mature, post-operative unilateral coronal, metopic, midline SMAD6 mutated and age/race/gender/synostosis/operation matched non-SMAD6 controls from the Yale Craniofacial Clinic and the Children's Hospital of Philadelphia (CHOP) completed a double-blinded neurodevelopmental assessment, which included the Wechsler Fundamentals, Wechsler Abbreviated Scale of Intelligence, and Beery- Buktenica Developmental Test. Unilateral coronal (ULC) or metopic synostosis were age/gender/handedness matched to controls and participated in a GoNoGo task under fMRI. Craniosynostosis infants were given the BSID and ERP testing at two points (pre and post operatively), and after they reached >6 years of age, patients completed the Wechsler Abbreviated Scale of Intelligence and Wechsler Fundamentals to measure 5 language functional domains. Results: ULC patients had a mean verbal IQ of 117.3 and performance IQ of 106.4, performed above average on academic achievements except for numerical, but below average on all visual-motor tests. Right ULC had improved spelling compared to left ULC, controlled for exogenous influences (p=0.033). Metopic patients with mild phenotype (endocranial bifrontal angle <124) performed better in word reading (p=0.035) and reading composite (p=0.014) than patients with severe stenosis (>124). After controlling for exogenous factors, midline synostosis patients with SMAD6 mutations performed worse on numerical operations(p=0.046), performance IQ(p=0.018), full IQ(p=0.010), and motor coordination(p=0.043) than those without the mutation. Among seven ULC and six metopic patients that participated in fMRI, metopic patients had decreased bloodoxygenation- level-dependent signal in the posterior cingulate(p=0.017) and middle temporal gyrus(MTG |
| 590 | |
▼a School code: 0265. |
| 650 | 4 |
▼a Medicine. |
| 650 | 4 |
▼a Physiology. |
| 650 | 4 |
▼a Neurosciences. |
| 650 | 4 |
▼a Health sciences. |
| 650 | 4 |
▼a Cognitive psychology. |
| 650 | 4 |
▼a Psychobiology. |
| 650 | 4 |
▼a Physiological psychology. |
| 690 | |
▼a 0564 |
| 690 | |
▼a 0566 |
| 690 | |
▼a 0317 |
| 690 | |
▼a 0633 |
| 690 | |
▼a 0719 |
| 690 | |
▼a 0349 |
| 690 | |
▼a 0989 |
| 710 | 20 |
▼a Yale University.
▼b Yale School of Medicine. |
| 773 | 0 |
▼t Dissertations Abstracts International
▼g 81-03B. |
| 773 | |
▼t Dissertation Abstract International |
| 790 | |
▼a 0265 |
| 791 | |
▼a M.D. |
| 792 | |
▼a 2019 |
| 793 | |
▼a English |
| 856 | 40 |
▼u http://www.riss.kr/pdu/ddodLink.do?id=T15490811
▼n KERIS
▼z 이 자료의 원문은 한국교육학술정보원에서 제공합니다. |
| 980 | |
▼a 202002
▼f 2020 |
| 990 | |
▼a ***1816162 |
| 991 | |
▼a E-BOOK |