Nonsyndromic cleft lip and palate (NSCLP) encompasses a group of orofacial abnormalities. Emerging evidence has revealed the presence of structural brain abnormalities in affected individuals. Previous studies have performed structure-based volumetric analysis of the brain assessing gross lobular subdivisions of the cerebral cortex and white matter which may have only vague relationships to the functional subregions implicated in behavioral and cognitive deficits observed in NSCLP patients. High-resolution magnetic resonance imaging structural data were acquired to provide a detailed characterization of the brain with respect to both regional cortical volume and thickness in 26 children with NSCLP and 26 age- and demographically matched typically developing children. Children with NSCLP exhibited abnormally large cerebral cortex grey matter volumes with decreased volumes of subcortical grey matter and cerebral white matter structures. Hemisphere-specific patterns of cortical volume and thickness abnormalities were identified. This study is the first to examine cortical thickness abnormalities in NSCLP. Overall, these findings suggest that the brains of children with NSCLP are less mature than those of their age-matched peers. Gender-specific comparisons reveal that NSCLP females were more immature compared to their typically developing peers compared to NSCLP males.

Keywords:
Nonsyndromic cleft lip and palate, Magnetic resonance imaging, Volume, Cortex, Thickness
1.
Broder H, Richman L, Matheson P: Learning disability, school achievement, and grade retention among children with cleft: a two-center study. Cleft Palate Craniofac J 1998;35:127-131.
2.
Conrad A, Richman L, Nopoulos P, Dailey S: Neuropsychological functioning in children with non-syndromic cleft of the lip and/or palate. Child Neuropsychol 2009;2:1-14.
3.
Endriga M, Kapp-Simon K: Psychological issues in craniofacial care: state of the art. Cleft Palate Craniofac J 1999;36:3-11.
4.
Millard T, Richman L: Different cleft conditions, facial appearance, and speech: relationship to psychological variables. Cleft Palate Craniofac J 2001;38:68-75.
5.
Richman L: Cognitive patterns and learning disabilities of cleft palate children with verbal deficits. J Speech Hear Res 1980;23:447-456.
6.
Richman L, Eliason M: Disorders of communications: developmental language disorders and cleft palate; in Walker C, Roberts M (eds): Handbook of Child Clinical Psychology. New York, John Wiley & Sons, 2001, pp 603-617.
7.
Nopoulos P, Boes AD, Jabines A, Conrad AL, Canady J, Richman L, Dawson JD: Hyperactivity, impulsivity, and inattention in boys with cleft lip and palate: relationship to ventromedial prefrontal cortex morphology. J Neurodev Disord 2010;2:235-242.
8.
Richman LC, McCoy TE, Conrad AL, Nopoulos P: Neuropsychological, behavioral, and academic sequelae of cleft: early developmental, school age, and adolescent/young adult outcomes. Cleft Palate Craniofac J 2012;49:387-396.
9.
Mueller A, Sader R, Honigmann K, Zeilhofer H, Schwenzer-Zimmerer K: Central nervous malformations in presence of clefts reflect developmental interplay. Int J Oral Maxillofac Surg 2007;36:289-295.
10.
Nopoulos P, Berg S, Canady J, Richman L, Van Demark D, Andreasen N: Abnormal brain morphology in patients with isolated cleft lip, cleft palate, or both: a preliminary analysis. Cleft Palate Craniofac J 2000;37:441-446.
11.
Nopoulos P, Berg S, Canady J, Richman L, Van Demark D, Andreasen N: Structural brain abnormalities in adult males with clefts of the lip and/or palate. Genet Med 2002;4:1-9.
12.
Nopoulos P, Berg S, Van Demark D, Richman L, Canady J: Increased incidence of a midline brain anomaly in patients with nonsyndromic clefts of the lip and/or palate. J Neuroimaging 2001;11:418-424.
13.
Nopoulos P, Choe I, Berg S, Van Demark D, Canady J, Richman L: Ventral frontal cortex morphology in adult males with isolated orofacial clefts: relationship to abnormalities in social function. Cleft Palate Craniofac J 2005;42:138-144.
14.
Nopoulos P, Langbehn D, Canady J, Magnotta V, Richman L: Abnormal brain structure in children with isolated clefts of the lip or palate. Arch Pediatr Adolesc Med 2007;161:753-758.
15.
Desikan RS, Ségonne F, Fischl B, Quinn BT, Dickerson BC, Blacker D, Buckner RL, Dale AM, Maguire RP, Hyman BT, Albert MS, Killiany RJ: An automated labeling system for subdividing the human cerebral cortex on MRI scans into gyral based regions of interest. Neuroimage 2006;31:968-980.
16.
Destrieux C, Fischl B, Dale A, Halgren E: Automatic parcellation of human cortical gyri and sulci using standard anatomical nomenclature. Neuroimage 2010;53:1-15.
17.
Anderson V, Beauchamp MH, Yeates KO, Crossley L, Hearps SJC, Catroppa C: Social competence at 6 months following childhood traumatic brain injury. J Int Neuropsychol Soc 2013;19:539-550.
18.
Fischl B, Salat DH, Busa E, Albert M, Dieterich M, Haselgrove C, van der Kouwe A, Killiany R, Kennedy D, Klaveness S, Montillo A, Makris N, Rosen B, Dale AM: Whole brain segmentation: automated labeling of neuroanatomical structures in the human brain. Neuron 2002;33:341-355.
19.
Rencher AC, Scott DT: Assessing the contribution of individual variables following rejection of a multivariate hypothesis. Commun Stat Simul Comput 1990;19:535-553.
20.
Kirk RE: Practical significance: a concept whose time has come. Educ Psychol Meas 1996;56:746-759.
21.
Gogtay N, Giedd JN, Lusk L, Hayashi KM, Greenstein D, Vaituzis AC, Nugent TF, Herman DH, Clasen LS, Toga AW, Rapoport JL, Thompson PM: Dynamic mapping of human cortical development during childhood through early adulthood. Proc Natl Acad Sci USA 2004;101:8174-8179.
22.
Tamnes CK, Østby Y, Fjell AM, Westlye LT, Due-Tønnessen P, Walhovd KB: Brain maturation in adolescence and young adulthood: regional age-related changes in cortical thickness and white matter volume and microstructure. Cerebral Cortex 2010;20:534-548.
23.
Westlye LT, Walhovd KB, Dale AM, Bjørnerud A, Due-Tønnessen P, Engvig A, Grydeland H, Tamnes CK, Østby Y, Fjell AM: Life-span changes of the human brain white matter: diffusion tensor imaging (DTI) and volumetry. Cereb Cortex 2010;20:2055-2068.
24.
Richman L, Ryan S, Wilgenbusch T, Millard T: Overdiagnosis and medication for attention-deficit hyperactivity disorder in children with cleft: diagnostic examination and follow-up. Cleft Palate Craniofac J 2004;41:351-354.
25.
Fox MD, Corbetta M, Snyder AZ, Vincent JL, Raichle ME: Spontaneous neuronal activity distinguishes human dorsal and ventral attention systems. Proc Natl Acad Sci USA 2006;103:10046-10051.
26.
Ptak R, Schnider A: The dorsal attention network mediates orienting toward behaviorally relevant stimuli in spatial neglect. J Neurosci 2010;30:12557-12565.
27.
Corbetta M, Kincade JM, Ollinger JM, McAvoy MP, Shulman GL: Voluntary orienting is dissociated from target detection in human posterior parietal cortex. Nat Neurosci 2000;3:292-297.
© 2014 S. Karger AG, Basel
2014
Copyright / Drug Dosage / Disclaimer
Copyright: All rights reserved. No part of this publication may be translated into other languages, reproduced or utilized in any form or by any means, electronic or mechanical, including photocopying, recording, microcopying, or by any information storage and retrieval system, without permission in writing from the publisher.
Drug Dosage: The authors and the publisher have exerted every effort to ensure that drug selection and dosage set forth in this text are in accord with current recommendations and practice at the time of publication. However, in view of ongoing research, changes in government regulations, and the constant flow of information relating to drug therapy and drug reactions, the reader is urged to check the package insert for each drug for any changes in indications and dosage and for added warnings and precautions. This is particularly important when the recommended agent is a new and/or infrequently employed drug.
Disclaimer: The statements, opinions and data contained in this publication are solely those of the individual authors and contributors and not of the publishers and the editor(s). The appearance of advertisements or/and product references in the publication is not a warranty, endorsement, or approval of the products or services advertised or of their effectiveness, quality or safety. The publisher and the editor(s) disclaim responsibility for any injury to persons or property resulting from any ideas, methods, instructions or products referred to in the content or advertisements.
You do not currently have access to this content.