Radiographic severity is associated with worse executive function in metopic craniosynostosis.

Behavior Executive function Metopic nonsyndromic craniosynostosis Radiographic severity

Journal

Child's nervous system : ChNS : official journal of the International Society for Pediatric Neurosurgery
ISSN: 1433-0350
Titre abrégé: Childs Nerv Syst
Pays: Germany
ID NLM: 8503227

Informations de publication

Date de publication:
21 Jun 2024
Historique:
received: 05 03 2024
accepted: 05 06 2024
medline: 21 6 2024
pubmed: 21 6 2024
entrez: 21 6 2024
Statut: aheadofprint

Résumé

Children with metopic synostosis have been found to have more neurocognitive and behavioral difficulties. The variables that may affect future neurodevelopmental outcomes, including presenting morphologic severity, have not been fully studied. In the largest study to date, we aimed to assess what portends worse neurocognitive and behavioral outcomes at school age. Children 6-18 years old with surgically corrected metopic nonsyndromic craniosynostosis underwent neurocognitive testing. Parents completed behavior rating surveys about their child: Conners-3 (ADHD), Social Responsiveness Scale-2 (autism spectrum disorder), Behavior Rating Inventory of Executive Function-2 (BRIEF-2: executive function), and Child's Behavior Checklist (overall behavior). The endocranial bifrontal angle (EBA), adjusted EBA (aEBA), frontal angle (FA), and AI-derived metopic severity score (MSS) were determined on pre-operative CT images. Multivariate linear regressions were used to evaluate the association of age at surgery and severity. There were 87 children who underwent neurocognitive testing (average age 10.9 ± 3.3 years) of whom 67 also completed behavioral assessments. Greater phenotypical severity of metopic synostosis (lower FA, aEBA, and EBA) was associated with worse scores on the subscales of the BRIEF-2 (executive function) and executive subscale of the Conners-3. Increasing age at surgery was associated with worse executive function subscale scores of the Conners-3 when controlling for each severity measurement and sociodemographic risk. Children with greater phenotypic severity of metopic synostosis have worse executive function at school age. The majority of children with metopic synostosis have signs of ADHD. Later surgeries (greater than 12 months) may impact executive functioning, regardless of the degree of severity. Future research should aim at identifying the direct structural changes to the brain.

Sections du résumé

BACKGROUND BACKGROUND
Children with metopic synostosis have been found to have more neurocognitive and behavioral difficulties. The variables that may affect future neurodevelopmental outcomes, including presenting morphologic severity, have not been fully studied. In the largest study to date, we aimed to assess what portends worse neurocognitive and behavioral outcomes at school age.
METHODS METHODS
Children 6-18 years old with surgically corrected metopic nonsyndromic craniosynostosis underwent neurocognitive testing. Parents completed behavior rating surveys about their child: Conners-3 (ADHD), Social Responsiveness Scale-2 (autism spectrum disorder), Behavior Rating Inventory of Executive Function-2 (BRIEF-2: executive function), and Child's Behavior Checklist (overall behavior). The endocranial bifrontal angle (EBA), adjusted EBA (aEBA), frontal angle (FA), and AI-derived metopic severity score (MSS) were determined on pre-operative CT images. Multivariate linear regressions were used to evaluate the association of age at surgery and severity.
RESULTS RESULTS
There were 87 children who underwent neurocognitive testing (average age 10.9 ± 3.3 years) of whom 67 also completed behavioral assessments. Greater phenotypical severity of metopic synostosis (lower FA, aEBA, and EBA) was associated with worse scores on the subscales of the BRIEF-2 (executive function) and executive subscale of the Conners-3. Increasing age at surgery was associated with worse executive function subscale scores of the Conners-3 when controlling for each severity measurement and sociodemographic risk.
CONCLUSION CONCLUSIONS
Children with greater phenotypic severity of metopic synostosis have worse executive function at school age. The majority of children with metopic synostosis have signs of ADHD. Later surgeries (greater than 12 months) may impact executive functioning, regardless of the degree of severity. Future research should aim at identifying the direct structural changes to the brain.

Identifiants

pubmed: 38904768
doi: 10.1007/s00381-024-06493-7
pii: 10.1007/s00381-024-06493-7
doi:

Types de publication

Journal Article

Langues

eng

Sous-ensembles de citation

IM

Informations de copyright

© 2024. The Author(s), under exclusive licence to Springer-Verlag GmbH Germany, part of Springer Nature.

Références

Achenbach TM (2009) The Achenbach System of Empirically Based Assessment (ASEBA): development, findings, theory, and applications. Burlington, VT: University of Vermont Research Center for Children, YOuth, & Families
Beckett JS, Chadha P, Persing JA, Steinbacher DM (2012) Classification of trigonocephaly in metopic synostosis. Plast Reconstr Surg 130(3):442e–447e
doi: 10.1097/PRS.0b013e31825dc244 pubmed: 22929268
Beery K, Buktenica N, Beery N (2010) The Beery-Buktenica developmental test of visual motor inegration: adminisation, scoring and teaching manual (6th edition). Minneapolis, MN: NCS Pearson
Bellew M, Mandela RJ, Chumas PD (2019) Impact of age at surgery on neurodevelopmental outcomes in sagittal synostosis. J Neurosurg Pediatr 23(4):434–441
doi: 10.3171/2018.8.PEDS18186 pubmed: 30660108
Best JR, Miller PH (2010) A developmental perspective on executive function. Child Dev 81(6):1641–1660
doi: 10.1111/j.1467-8624.2010.01499.x pubmed: 21077853 pmcid: 3058827
Bhalodia R, Dvoracek LA, Ayyash AM, Kavan L, Whitaker R, Goldstein JA (2020) Quantifying the severity of metopic craniosynostosis: a pilot study application of machine learning in craniofacial surgery. J Craniofac Surg 31(3):697–701
doi: 10.1097/SCS.0000000000006215 pubmed: 32011542 pmcid: 7202995
Blair C (2017) Educating executive function. Wiley Interdiscip Rev Cog Sci 8(1–2). https://doi.org/10.1002/wcs.1403
Blum JD, Beiriger J, Kalmar C, Avery RA, Lang SS et al (2022) Relating metopic craniosynostosis severity to intracranial pressure. J Craniofac Surg 33(8):2372–2378
doi: 10.1097/SCS.0000000000008748 pubmed: 35864584
Bottero L, Lajeunie E, Arnaud E, Marchac D, Renier D (1998) Functional outcome after surgery for trigonocephaly. Plast Reconstr Surg 102(4):952–8 discussion 959–60
doi: 10.1097/00006534-199809020-00002 pubmed: 9734408
Bruni TP (2014) Test review: Social Responsiveness Scale-Second Edition (SRS-2). J Psychoeduc Assess 32(4):365–369
doi: 10.1177/0734282913517525
Chandler L, Park KE, Allam O, Mozaffari MA, Khetpal S et al (2021) Distinguishing craniomorphometric characteristics and severity in metopic synostosis patients. Int J Oral Maxillofac Surg 50(8):1040–1046
doi: 10.1016/j.ijom.2020.11.022 pubmed: 33483210
Collett BR, Kapp-Simon KA, Wallace E, Cradock MM, Buono L, Speltz ML (2017) Attention and executive function in children with and without single-suture craniosynostosis. Child Neuropsychol 23(1):83–98
doi: 10.1080/09297049.2015.1085005 pubmed: 26381123
Conners CK, Pitkanen J, Rzepa SR, Conners 3rd Edition (Conners 3; Conners 2008), in Encyclopedia of Clinical Neuropsychology, J.S. Kreutzer, J. DeLuca, and B. Caplan, Editors (2011) Springer New York: New York, NY. p. 675–678
Cornelissen M, Ottelander B, Rizopoulos D, van der Hulst R, Mink van der Molen A et al (2016) Increase of prevalence of craniosynostosis. J Craniomaxillofac Surg 44(9):1273–1279
doi: 10.1016/j.jcms.2016.07.007 pubmed: 27499511
Cronin BJ, Brandel MG, McKee RM, Hashmi A, Oviedo P et al (2020) A comparison of intracranial volume growth in normal children and patients with metopic craniosynostosis. J Craniofac Surg 31(1):142–146
doi: 10.1097/SCS.0000000000005946 pubmed: 31652215
Edwards-Bailey L, Piggott K, Dalton L, Horton J, Parks C, Wright G, Kearney A (2023) The Craniofacial Collaboration UK: developmental outcomes in 5-year-old children with metopic synostosis. J Craniofac Surg 34(3):855–859
doi: 10.1097/SCS.0000000000009095 pubmed: 36329005
Gabrick KS, Wu RT, Singh A, Persing JA, Alperovich M (2020) Radiographic severity of metopic craniosynostosis correlates with long-term neurocognitive outcomes. Plast Reconstr Surg 145(5):1241–1248
doi: 10.1097/PRS.0000000000006746 pubmed: 32332546
Hendrickson NK, McCrimmon AW (2019) Test review: Behavior Rating Inventory of Executive Function®, Second Edition (BRIEF®2) by Gioia, G. A., Isquith, P. K., Guy, S. C., & Kenworthy, L. Canadian J School Psychol 34(1):73–78
doi: 10.1177/0829573518797762
Junn A, Dinis J, Hauc SC, Bruce MK, Park KE et al (2023) Validation of artificial intelligence severity assessment in metopic craniosynostosis. Cleft Palate Craniofac J 60(3):274–279
doi: 10.1177/10556656211061021 pubmed: 34787505
Junn AH, Long AS, Hauc SC, Almeida MN, Alper DP, Rivera JC, Mayes L, Persing JA, Alperovich M (2023) Long-term neurocognitive outcomes in 204 single-suture craniosynostosis patients. Childs Nerv Syst 39(7):1921–1928
doi: 10.1007/s00381-023-05908-1 pubmed: 36877207
Kalmar CL, Lang S-S, Heuer GG, Schreiber JE, Tucker AM, Swanson JW, Beslow LA (2022) Neurocognitive outcomes of children with non-syndromic single-suture craniosynostosis. Childs Nerv Syst 38(5):893–901
doi: 10.1007/s00381-022-05448-0 pubmed: 35192026
Kapp-Simon KA, Collett BR, Barr-Schinzel MA, Cradock MM, Buono LA, Pietila KE, Speltz ML (2012) Behavioral adjustment of toddler and preschool-aged children with single-suture craniosynostosis. Plast Reconstr Surg 130(3):635–647
doi: 10.1097/PRS.0b013e31825dc18b pubmed: 22929249 pmcid: 3431507
Kaufman A, Kaufman N, K-TEA: Kaufman Test of Educational Achievement-3 (1998) Circle Pines, MN: American Guidance Service
Kljajic M, Maltese G, Tarnow P, Sand P, Kölby L (2019) The cognitive profile of children with nonsyndromic craniosynostosis. Plast Reconstr Surg 143(5):1037e–1052e
doi: 10.1097/PRS.0000000000005515 pubmed: 30789480
Kronig ODM, Kronig SAJ, Van Adrichem LNA (2022) Intracranial volume not correlated with severity in trigonocephaly. Cleft Palate Craniofac J 59(6):794–799
doi: 10.1177/10556656211025185 pubmed: 34137298
Kunz M, Lehner M, Heger A, Armbruster L, Weigand H, Mast G, Peraud A (2014) Neurodevelopmental and esthetic results in children after surgical correction of metopic suture synostosis: a single institutional experience. Childs Nerv Syst 30(6):1075–1082
doi: 10.1007/s00381-013-2340-0 pubmed: 24337567
McKee RM, Kamel GN, Cronin BJ, Ewing E, Lance SH, Gosman AA (2021) A comparison of intracranial volumes and metopic index in patients with isolated metopic ridge, metopic craniosynostosis, and normal healthy children. J Craniofac Surg 32(1):108–112
doi: 10.1097/SCS.0000000000007044 pubmed: 33186289
Osborn AJ, Roberts RM, Mathias JL, Anderson PJ, Flapper WJ (2019) Cognitive, behavioral and psychological functioning in children with metopic synostosis: a meta-analysis examining the impact of surgical status. Child Neuropsychol 25(2):263–277
doi: 10.1080/09297049.2018.1441821 pubmed: 29482450
Peck CJ, Junn A, Park KE, Khetpal S, Cabrejo R, Singh A, Mayes L, Alperovich M, Persing JA (2022) Longitudinal outpatient and school-based service use among children with nonsyndromic craniosynostosis. Plast Reconstr Surg 150(6):1309–1317
doi: 10.1097/PRS.0000000000009678 pubmed: 36126215
Sajadi S, Raheb G, Maarefvand M, Alhosseini KA (2020) Family problems associated with conduct disorder perceived by patients, families and professionals. J Educ Health Promot 9:184
doi: 10.4103/jehp.jehp_110_20 pubmed: 32953911 pmcid: 7482631
Selber J, Reid RR, Chike-Obi CJ, Sutton LN, Zackai EH, McDonald-McGinn D, Sonnad SS, Whitaker LA, Bartlett SP (2008) The changing epidemiologic spectrum of single-suture synostoses. Plast Reconstr Surg 122(2):527–533
doi: 10.1097/PRS.0b013e31817d548c pubmed: 18626371
Shimamura AP (2000) The role of the prefrontal cortex in dynamic filtering. Psychobiology 28(2):207–218
doi: 10.3758/BF03331979
Sidoti EJ Jr, Marsh JL, Marty-Grames L, Noetzel MJ (1996) Long-term studies of metopic synostosis: frequency of cognitive impairment and behavioral disturbances. Plast Reconstr Surg 97(2):276–281
doi: 10.1097/00006534-199602000-00002 pubmed: 8559809
Speltz ML, Collett BR, Wallace ER, Kapp-Simon K (2016) Behavioral Adjustment of school-age children with and without single-suture craniosynostosis. Plast Reconstr Surg 138(2):435–445
doi: 10.1097/PRS.0000000000002383 pubmed: 27465166
Tandon D, Skolnick GB, Naidoo SD, Grames LM, Cradock MM, Smyth MD, Patel KB (2021) Morphologic severity of craniosynostosis: implications for speech and neurodevelopment. Cleft Palate Craniofac J 58(11):1361–1369
doi: 10.1177/1055665620984643 pubmed: 33467909
van der Meulen J (2012) Metopic synostosis. Childs Nerv Syst 28(9):1359–1367
doi: 10.1007/s00381-012-1803-z pubmed: 22872249 pmcid: 3413823
van der Vlugt JJB, van der Meulen JJNM, Creemers HE, Verhulst FC, Hovius SER, Okkerse JME (2012) Cognitive and behavioral functioning in 82 patients with trigonocephaly. Plast Reconstr Surg 130(4):885–893
doi: 10.1097/PRS.0b013e318262f21f pubmed: 23018698
Wechsler D (2011) Wechsler Abbreviated Scale of Intelligence, Second Ediiton (WASI-II). San Antonio, TX: NSC Pearson
Wu RT, Park KE, Chandler L, Ahmad M, Allam O, Timberlake AT, Persing JA, Alperovich M (2022) Virtual neurocognitive testing in craniofacial surgery. Plast Reconstr Surg 149(6):1413–1416
doi: 10.1097/PRS.0000000000009145 pubmed: 35413035
Yule AM, DiSalvo M, Wilens TE, Wozniak J, Faraone SV, Lyons RM, Woodworth KY, Biederman J (2020) High correspondence between child behavior checklist rule breaking behavior scale with conduct disorder in males and females. Child Psychiatry Hum Dev 51(6):978–985
doi: 10.1007/s10578-020-00978-7 pubmed: 32172405 pmcid: 7492452

Auteurs

Mariana N Almeida (MN)

Division of Plastic Surgery, Department of Surgery, Yale School of Medicine, 330 Cedar Street, Boardman Building, New Haven, CT, 06510, USA.

David P Alper (DP)

Division of Plastic Surgery, Department of Surgery, Yale School of Medicine, 330 Cedar Street, Boardman Building, New Haven, CT, 06510, USA.

Carlos Barrero (C)

Division of Plastic Surgery, Children's Hospital of Philadelphia and University of Pennsylvania, Philadelphia, PA, USA.

Neil Parikh (N)

Division of Plastic Surgery, Department of Surgery, Yale School of Medicine, 330 Cedar Street, Boardman Building, New Haven, CT, 06510, USA.

Sacha C Hauc (SC)

Division of Plastic Surgery, Department of Surgery, Yale School of Medicine, 330 Cedar Street, Boardman Building, New Haven, CT, 06510, USA.

Jake Moscarelli (J)

Division of Plastic Surgery, Department of Surgery, Yale School of Medicine, 330 Cedar Street, Boardman Building, New Haven, CT, 06510, USA.

Michael Golinko (M)

Division of Plastic Surgery, Department of Surgery, Vanderbilt University School of Medicine, Nashville, TN, USA.

John Persing (J)

Division of Plastic Surgery, Department of Surgery, Yale School of Medicine, 330 Cedar Street, Boardman Building, New Haven, CT, 06510, USA.

Jordan Swanson (J)

Division of Plastic Surgery, Children's Hospital of Philadelphia and University of Pennsylvania, Philadelphia, PA, USA.

Michael Alperovich (M)

Division of Plastic Surgery, Department of Surgery, Yale School of Medicine, 330 Cedar Street, Boardman Building, New Haven, CT, 06510, USA. Michael.alperovich@yale.edu.

Classifications MeSH