Long-term developmental follow-up in children with nonsyndromic craniosynostosis
Open accessAbstract
OBJECT: The aim of this study was to determine the distribution of Full Scale IQ (FSIQ) by type of craniosynostosis and to verify the finding that at long-term follow-up, verbal IQ (VIQ) is significantly higher than performance IQ (PIQ) in patients with single-suture sagittal synostosis (SS) despite falling within the "average" range for intelligence. Whether this also occurs in other types of craniosynostosis and whether surgery and sex are relevant were also determined. The relationship between age at time of surgery and later IQ was ascertained. METHODS: The data for 91 children with craniosynostosis (47 sagittal, 15 unicoronal, 13 metopic, 9 multisuture, and 7 bicoronal) were collected at their routine, 10 years of age IQ assessment (mean age 123.8 months). The patients included 61 males and 30 females; 62 patients had undergone surgery and 29 had not. RESULTS: The mean FSIQ for all types of craniosynostosis combined (96.2) fell within the average range for the general population. Some variation was evident across the different types of craniosynostosis: the SS group showed the highest FSIQs and a "normal" distribution of bandings; the other types had a higher proportion of FSIQs in the lower bandings. The data confirmed the finding that VIQ is greater than PIQ despite falling within the average range for intelligence, with a difference of 5.0 for all types of craniosynostosis combined (p = 0.001), 7.6 for the SS group (p = 0.001), and 6.9 for the unicoronal group (p = 0.029). This VIQ > PIQ effect was not found with multisuture craniosynostosis. The VIQ > PIQ discrepancy occurred regardless of whether the patient had undergone surgery and occurred more often in males than females. In the SS group and the bicoronal group, FSIQ (p = 0.036 and p = 0.046, respectively) and PIQ (p = 0.012 and p = 0.017, respectively), though not VIQ, were higher when surgery had been performed early. CONCLUSIONS: The study confirms that at long-term follow-up, although children with nonsyndromic craniosynostosis fall within the normal range for intelligence, there is a VIQ > PIQ discrepancy above what would be expected in the normal population, which may be indicative of more subtle difficulties in achievement. This discrepancy is affected by type of craniosynostosis, sex, and age at time of surgery.
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IQ scores in children with ISS are usually in the average range, and some children with ISS achieve high or very high IQ scores.
“The scores of the intelligence quotient (IQ) in these children are usually in the average range [27], and there are even studies in which some children with ISS achieved high or very high IQ scores [29,30].”
From Isolated Sagittal Craniosynostosis: A Comprehensive Review (2024) · cites this paper as [29]
Other authors have described poorer gross motor skills and significantly lower non-verbal IQ than verbal IQ in children with ISS compared to controls.
“Other authors have also described poorer gross motor skills and significantly lower non-verbal IQ than verbal IQ in children with ISS compared to controls [29,32].”
From Isolated Sagittal Craniosynostosis: A Comprehensive Review (2024) · cites this paper as [29]
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“The scores of the intelligence quotient (IQ) in these children are usually in the average range [27], and there are even studies in which some children with ISS achieved high or very high IQ scores [29,30].”
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BackgroundResultInfluentialResults Following Adoption of a Modified Melbourne Technique of Total Scaphocephaly Correction
Sharma, J. D., O’Hara, J., Borghi, A., Rodríguez-Flórez, N., Breakey, R. W. F., Ong, J., Jeelani, O., Dunaway, D. J., & James, G. (2018) · Journal of Craniofacial Surgery · doi:10.1097/scs.0000000000004593 · Cited by 18 · see citing works
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Unilateral Coronal Craniosynostosis
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“However, surgery is often offered in craniosynostosis for cosmetic reasons, as there is increasing evidence suggesting an impact on neurodevelopmental outcome.(21) The importance of our study lied on the fact that we examined the development of patients with craniosynostosis who underwent surgery with FOA and PCVE, wh…”
BackgroundIncreased utilization of healthcare services in children with craniosynostosis
Colón, G. D. R., Jin, M. C., Grant, G. A., & Prolo, L. M. (2022) · Journal of Neurosurgery Pediatrics · doi:10.3171/2022.2.peds2253 · Cited by 4 · see citing works
Cognitive performance of preschool children with different types of non-syndromic craniosynostosis
Moreno-Villagómez, J., Yáñez-Téllez, G., Prieto‐Corona, B., Seubert-Ravelo, A. N., & Garcia, A. R. (2020) · Brain Impairment · doi:10.1017/brimp.2020.7 · Cited by 4 · see citing works
The role of genetics on behavioral outcomes in nonsyndromic sagittal synostosis
Alper, D. P., Almeida, M. N., Hu, K. G., Yang, J. F., Timberlake, A. T., Shah, J. D., Persing, J. A., & Alperovich, M. A. (2024) · Journal of Neurosurgery Pediatrics · doi:10.3171/2024.4.peds23510 · Cited by 3 · see citing works
Rożek, A., Larysz, D., & Larysz, P. (2025) · Journal of Craniofacial Surgery · doi:10.1097/scs.0000000000011275 · Cited by 3 · see citing works
Le retentissement neuropsychologique de la craniosténose : cas de la scaphocéphalie non syndromique
Verlut, I., Mottolèse, C., Szathmári, A., Banvillet, E., Poncet, A., Blache, A., Rousselle, C., Gleizal, A. M., Paulus, C., & Rocco, F. D. (2019) · Neurochirurgie · doi:10.1016/j.neuchi.2019.09.018 · Cited by 3 · see citing works
Edwards-Bailey, L., Piggott, K., Dalton, L. J., Horton, J., Parks, C., Lloyd-White, S., Wright, G., & Kearney, A. (2023) · Journal of Craniofacial Surgery · doi:10.1097/scs.0000000000009803 · Cited by 3 · see citing works
Moderne Behandlung von Kraniosynostosen
Schmidt, L., Fassl, V., Erhardt, L., Winter, J., Lollert, A., Heider, J., & Ottenhausen, M. (2023) · Monatsschrift Kinderheilkunde · doi:10.1007/s00112-022-01683-6 · Cited by 2 · see citing works
Baraya, N., Thibodaux, L. K., Martin, A., Stone, C., Strahle, J. M., Patel, K. B., & Chapman, L. A. (2025) · The Cleft Palate-Craniofacial Journal · doi:10.1177/10556656251339879 · Cited by 2 · see citing works
Wijdan, S. A., Yezdan, S. A., Asim, R., Sakrani, R. A., Batool, R., & Abdalla, M. I. (2026) · Annals of Medicine and Surgery · doi:10.1097/ms9.0000000000004377 · Cited by 2 · see citing works
“A cross-sectional study by Bellew and Chumas indicated relatively higher intelligence quotients of postoperative sagittal synostosis patients in contrast to their metopic and unicoronal counterparts [27] .”
Pavicevic, D., Milosevic, J., Petrovic-Markovic, I., Milenković, Z., & Parezanović-Ilić, K. (2020) · Vojnosanitetski pregled · doi:10.2298/vsp171222021p · Cited by 2 · see citing works
Early diagnosis and surgical treatment of craniosynostoses
Ivanov, V. P., Sakhno, L. P., Shchetinina, A. M., Kim, A. V., & Трущелева, С. (2022) · Rossiyskiy Vestnik Perinatologii i Pediatrii (Russian Bulletin of Perinatology and Pediatrics) · doi:10.21508/1027-4065-2022-67-5-62-67 · Cited by 2 · see citing works
Sumkovski, R., Kocevski, I., & Mikjunovikj, M. (2021) · Springer eBooks · doi:10.1007/978-3-030-72338-5_18 · Cited by 1 · see citing works
A New Method for Measuring Cognition in Children With Sagittal Craniosynostosis
Chapman, L. A., Runyan, C. M., Couture, D. E., Thibodaux, L. K., Wright, A., & David, L. R. (2024) · FACE · doi:10.1177/27325016241299005 · Cited by 1 · see citing works
“14 Children treated for sagittal synostosis typically have normal cognitive development, 15-17 with better outcomes in children treated before 7 months of age.”
InfluentialFisch, S., Tudor, A., Benchekroun, E. M., Landsberg, W., Feldstein, N. A., Lamb, M. E., Carpenter, T. O., Rundle, A., Jacobson, J. B., Neugut, A. I., et al. (2025) · Bone · doi:10.1016/j.bone.2025.117488 · Cited by 1 · see citing works
Moreno-Villagómez, J., Guillermina Yáñez, T., Belén Prieto, C., Yaneth Rodríguez, A., & Antonio García, M. (2017) · Revista chilena de neuro-psiquiatría · doi:10.4067/s0717-92272017000100007 · Cited by 1 · see citing works
Price, G. A., Rentzeperis, F., Price, H., Taub, P. J., & Morgenstern, P. F. (2025) · Child s Nervous System · doi:10.1007/s00381-025-06834-0 · Cited by 1 · see citing works
“In terms of the impact of craniosynostosis surgery on neurodevelopment in adolescence, Bellew et al. suggest that surgery performed at a younger age may lead to better neurocognitive outcomes in later childhood [39].”
Rocco, F. D. (2020) · Springer eBooks · doi:10.1007/978-3-319-72168-2_58 · Cited by 1 · see citing works
Hydrocephalus in craniosynostosis
Lo, A. L., Massand, S., & Rizk, E. (2022) · Elsevier eBooks · doi:10.1016/b978-0-12-819507-9.00023-5 · Cited by 1 · see citing works
Ching, J. A., Troy, J. S., Ruas, E. J., Beckman, J. M., & Tuite, G. F. (2018) · The Cleft Palate-Craniofacial Journal · doi:10.1177/1055665618777906
Craniosynostosis: Repercussions on Schooling, Inclusive Education, and Siblings
Colinet, S., Bonnet, P., & Rocco, F. D. (2018) · Springer eBooks · doi:10.1007/978-3-319-31512-6_67-1
Management of Patient with Craniosynostosis
Krishnaney-Davison, R., Chhabada, S., & Castro, P. (2020) · Springer eBooks · doi:10.1007/978-981-15-0458-7_7
Current Controversies in Craniosynostosis Research: A Tribute to Ian Jackson
Smetona, J. T., Junn, A., Dinis, J., Lopez, J., Lu, X., Alperovich, M. A., & Persing, J. A. (2020) · Journal of Craniofacial Surgery · doi:10.1097/scs.0000000000007331
“This finding is consistent with a subsequent study conducted by Bellew and Chumas, which found that early surgical intervention was associated with improved performance IQ.(36) These findings have contributed to a push towards earlier surgery, despite a potential increase in the need for subsequent revision associated…”
ResultUnilateral Coronal Craniosynostosis
Massimi, L., & Rocco, C. D. (2020) · Springer eBooks · doi:10.1007/978-3-319-72168-2_59
Kraniosinostoz Cerrahisinde Anestezi ve Transfüzyonun Değerlendirilmesi: Retrospektif Çalışma
Kılıçarslan, N., & Balkaya, A. N. (2022) · Sağlık Akademisi Kastamonu · doi:10.25279/sak.1021427
Craniosynostosis: Repercussions on Schooling, Inclusive Education, and Siblings
Colinet, S., Bonnet, P., & Rocco, F. D. (2020) · Springer eBooks · doi:10.1007/978-3-319-72168-2_67
Rocco, F. D. (2019) · Springer eBooks · doi:10.1007/978-3-319-31512-6_58-1
A rare case of the scaphocephalic skull of an adult male
Ahmadpour, S., & Foghi, K. (2022) · Egyptian Journal of Forensic Sciences · doi:10.1186/s41935-022-00303-4
“development, neurological disorders, and psychological disturbances (Aldridge et al. 2017; Bellew and Chumas 2015; Adamo and Pollack 2010).”
BackgroundPearson, G. D. N., & Khansa, I. (2024) · FACE · doi:10.1177/27325016241230105
Jung, J., Lam, J., deSouza, R., Anderson, I., & Chumas, P. (2019) · Advances in Clinical Neuroscience & Rehabilitation · doi:10.47795/vdbt8588
Suplemento da IV Jornada da SAMMG & XII Congresso SAMMG
(2024) · Revista Médica de Minas Gerais · doi:10.5935/2238-3182.2024v34s2
Nonsyndromic craniosynostosis associated with bipolar affective disorder and mild cognitive disorder
Deepak, P., Agrawal, M., Yadav, P., & Chauhan, V. S. (2024) · Industrial Psychiatry Journal · doi:10.4103/ipj.ipj_48_24
“[6] Other psychiatric disorders such as psychotic disorders (behavioral abnormalities), bipolar affective disorder, depressive disorder, eating disorder, anxiety disorder, personality disorder, and suicide attempts may also be present in nonsyndromic craniosynostosis.”
Long Follow-Up Results in Craniosynostoses
Salokorpi, N., & Serlo, W. S. (2024) · Springer eBooks · doi:10.1007/978-3-031-69386-1_54
Nadel, J., & Kestle, J. R. W. (2024) · Springer eBooks · doi:10.1007/978-3-031-69386-1_16
Complications and Long-Term Surgical Outcome in Craniosynostoses
Vinchon, M., Beuriat, P.-A., Szathmári, A., & Rocco, F. D. (2024) · Springer eBooks · doi:10.1007/978-3-031-69386-1_52
Quality of Life Metrics in Craniosynostosis: A Guide for Surgeons and Healthcare Practitioners
Warsi, N. M., Riesel, J. N., & Kulkarni, A. V. (2024) · Springer eBooks · doi:10.1007/978-3-031-69386-1_53
Complications and Long-Term Surgical Outcome in Craniosynostoses
Vinchon, M., Beuriat, P.-A., Szathmári, A., & Rocco, F. D. (2024) · Springer eBooks · doi:10.1007/978-3-031-69386-1_23
Natakusuma, T. I. S. D., de Liyis, B. G., Wiyanjana, K. D. F., Maliawan, S., & Mahadewa, T. G. B. (2025) · World Neurosurgery X · doi:10.1016/j.wnsx.2025.100526 · Cited by 1 · see citing works
Moreno-Villagómez, J., Yáñez-Téllez, G., Prieto‐Corona, B., Castillo-Mimila, M., Hernández, E., & García‐Méndez, A. (2026) · Child s Nervous System · doi:10.1007/s00381-026-07155-6
“However, a number of possible factors have been proposed, including the location of the suture [5, 14–17], socioeconomic factors [18, 19], and age at surgical intervention [16, 20, 21].”
Kong, V., Salib, A., Allam, O., Parker, E., Kauke‐Navarro, M., Moscarelli, J. R., Glaeser-Khan, S., & Alperovich, M. A. (2026) · Journal of Craniofacial Surgery · doi:10.1097/scs.0000000000013054
Neurodevelopmental Outcomes Among Nonsyndromic Craniosynostosis Subtypes
Ha, J., Semerjian, C., Tomei, K. L., & Wang, H. D. (2026) · Journal of Craniofacial Surgery · doi:10.1097/scs.0000000000013153
Fever Following Open Surgical Repair of Craniosynostosis
Groff, D., Gilles, A., Oxford, M. A., Hiller, A. R., Ryu, G. S., Kunselman, A. R., Rizk, E. B., & Samson, T. D. (2026) · Journal of Craniofacial Surgery · doi:10.1097/scs.0000000000013423
- Authors
- Bellew, M., & Chumas, P.
- Affiliations
- Departments of Plastic, Reconstructive, and Hand Surgery and
- Leeds General Infirmary (GB)
- Venue
- Journal of Neurosurgery Pediatrics
- Published
- 2015-10
- Volume
- 16
- Issue
- 4
- Pages
- 445-451
- ISSN
- 1933-0707 (ISSN-L)
- 1933-0715
- OpenAlex
- W1520210099
- Cited by
- 88
- References
- 29
- Citations by year
- 2026: 5 · 2025: 7 · 2024: 12 · 2023: 7 · 2022: 17 · 2021: 10 · 2020: 10 · 2019: 10 · 2018: 6 · 2017: 4
- FWCI
- 2.28
- Type
- journal article
- Language
- en
- Open access
- bronze
- doi.org
- Locations
- Journal of Neurosurgery Pediatrics (publishedVersion)
- PubMed (publishedVersion)
- Topics
- Life Sciences › Biochemistry, Genetics and Molecular Biology › Genetics › Craniofacial Disorders and Treatments
- Health Sciences › Medicine › Surgery › Pectus Deformity Diagnosis and Treatment
- Social Sciences › Arts and Humanities › Archeology › Forensic Anthropology and Bioarchaeology Studies
- Keywords
- craniosynostosis, nonsyndromic craniosynostosis, intelligence quotient, verbal IQ, performance IQ, IQ discrepancy, sagittal synostosis, unicoronal synostosis, metopic synostosis, bicoronal synostosis, craniosynostosis surgery, age at surgery, sex differences, long-term developmental outcomes
- MeSH terms
- Adolescent, Age Factors, Brain Damage, Chronic, Child, Developmental Disabilities, Craniosynostoses, Female, Follow-Up Studies, Humans, Intelligence Tests, Language Development Disorders, Male, Movement Disorders, Postoperative Complications, Skull, Treatment Outcome, Plastic Surgery Procedures, Early Medical Intervention
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