Cognitive Development in Single-Suture Craniosynostosis – A Systematic Review

Figures & data

Figure 1. Flowchart of literature search and inclusion procedure.Reasons for exclusion:Reason 1: No cognitive measureReason 2: Not SSC individuals Reason 3: Not an empirical studyReason 4: Duplicate sample

Table 1. Description of GRADE scores by level of confidence.

Score	Confidence level	Description
+	Very low	The true effect is probably markedly different from the estimated effect.
++	Low	The true effect might be markedly different from the estimated effect.
+++	Moderate	The authors believe that the true effect is probably close to the estimated effect.
++++	High	The authors have a lot of confidence that the true effect is similar to the estimated effect.

Table 2. Summary of overall features of the included studies.

Design (cross-sectional/ longitudinal)	Primary age band (<2 y/2–5 y/ 6–12 y/13–18 y/> 18 y)	Comparison (test norms/ control group)	Sample source (single center /multi-center/ unknown)	Subtype of SSC (mixed/sagittal/ metopic/coronal/ lambdoid/unknown)
27/21	13/9/26/2/0	36/12	27/20/1	26/10/8/2/0/2

Note. y = years; SSC = Single-Suture Craniosynostosis.

Table 3. Summary of included studies with description of participants as well as cognitive measures used.

First author (year)	No. of cases	Status of surgery	Design (cross-sectional/ longitudinal)	Comparison (test norms/ control group)	Gender (male/ female)	Age of partici-pants at evaluation	Subtype of SSC (mixed/ sagittal/ metopic/coronal/ lambdoid)	Cognitive domains assessed	Cognitive measures used	Main findings	Final GRADE score
Alperovich et al. (2021)	75	Post	Cross-sectional	Test norms	48/26	10.3 y average	Sagittal	IQ, Academic achieve-ment Visuo-motor	WASI, WF Beery VMI	Higher IQ and visuomotor t scores in patients having undergone cranial vault remodeling.	++
Arnaud et al. (2002)	99	Pre+ Post	Longitudinal	Test norms	35/64	Unknown (>6 m)	Coronal	IQ	Brunet-Lézine test New Metric test	Higher IQ scores in subjects operated on before one year of age. Overall mean within normal range.	++
Becker et al. (2005)	214	Pre+ Post	Cross-sectional	Test norms	126/88	6 y 4 m average	All subtypes	DQ/IQ	WISC/ BSID/ Cattell scales	47% of subjects with test scores > 1 sd below mean, persistently low post surgery in 27% of cases	++
Bellew et al. (2005)	28 + 28 contr	Pre+ Post	Longitudinal	Controls	23/5	6.9 to 60 m	Sagittal	DQ	Griffiths scales	Subjects lower gross locomotor DQ pre-op but not post-op.	++
Bellew et al. (2011)	32 + 23 contr	Post	Cross-sectional (partial data overlap with Bellew et al., 2005)	Controls	25/8	64.2 m average	Sagittal	DQ	Griffiths scales	DQ within normal range in operated subjects, no sign. diff from controls. Lower DQ scores in unoperated subjects.	++
Bellew and Chumas (2015)	91	Post	Cross-sectional	Test norms	61/30	10.2 y average	Sagittal, unicor-onal and metopic	IQ	WISC-IV	Case test scores within average range. Sagittal subtype significantly higher IQ than other subgroups withhigher VIQ than PIQ.	++
Boltshauser et al. (2003)	30 + 17 contr	Unop-erated	Cross-sectional	Sibling control group	23/7	Unknown (from 2,5 y upwards)	Sagittal	IQ Attention Learning Memory Language Executive function	K-ABC/ WISC-III/WAIS-R/TAP VLMT/ VDLT Mottier, Corsi block Hooper VOT DVTP−2	Generally average test scores. 6/30 cases lower learning/memory scores than controls, 12/30 for attention	+
Bottero et al. (1998)	76	Post	Cross-sectional	Test norms	Un-known	6.5 y average	Metopic	IQ	Brunet-Lezine test, Nouvelle Echelle Metrique test, WISC-R	Significantly higher incidence of low IQ in subjects operated post age 1 year	++
Byun et al. (2018)	266	Post + unop-erated	Longitudinal	Test norms	157/ 109	Unknown (after 2 years of age)	All subtypes	DQ/PDI/MDI	BSID-II	Mean MDI/PDI 1 sd below norm mean. No significant difference across subtypes.	++
Chieffo et al. (2010)	65 + 30 contr	Post	Cross-sectional	Control group	48/17	Mean of 13,1 y	Sagittal+ Uni-coronal	IQ,Visual motor, memory/attention skills Verbal fluency Working memory	WISC-R Purdue Pegboard DTVMPI RCFT Tower of London	Mean scores within average range. Lower scores for verbal IQ and fluency in left unicoronal group. Lower perceptual IQ in sagittal group.	++
Chuang et al. (2021)	47	Post	Cross-sectional	Test norms	Un-known	Range 5–16 y	Sagittal	IQ, visuo-motor	WASI, Beery VMI	Average mean test scores. Higher scores in early surgery subjects, no difference between reoperated and other subjects.	++
Cohen (2004)	22	Pre+ Post	Longitudinal	Test norms	12/10	2 m before surgery + 1 y post	Sagittal + Coronal + Metopic	DQ/MDI/PDI	BSID-II	Cases MDI+PDI 1 sd below norm meanSignificant.positive effect of surgery on PDI but not on MDI.	+
Collett et al. (2017)	179 + 183 contr	Post	Cross-sectional	Controls	113/66	7.5 y average	All subtypes	IQ, attention, Academic achieve-ment, executive function	WISC-IV, WF, TEA-Ch, NEPSY-II	Small effects but significantly lower scores on all measures in subjects, largest for inhibition.	+++
Da Costa et al. (2006)	18	Post	Cross-sectional	Test norms	Un-known	10.9 y average	Sagittal, metopic, uni-coronal	IQ	WISC-IV, Leiter-R	Average group test scores, ca 0.3 sd > mean.	++
Da Costa et al. (2012)	64	Pre+Post	Longitudinal	Test norms	46/18	Mean of 8,5 m pre and 21,2 m post op	All subtypes	DQ/MDI/PDI	BSID-II	Preop MDI slightly below norm mean, MDI significantly below mean. Both measures significantly below postop	++
Cradock et al. (2015)	180 + 183 contr	Post	Cross-sectional	Control group	113/67	7,4 y average	All subtypes	IQ Academic achieve-ment Language	WISC-IV WRAT−4 TOWRE CTOPP	Lower scores for cases on majority of measures (.2 to .6 sd mean diff). Similar sex differences in both cases and controls	++++
Fontana (2017)	5	Pre+Post	Longitudinal	Test norms	4/1	2 y average preop, 3 y postop	Metopic + Coronal	DQ	Bayley-III	4/5 cases in delayed range preop. 3/5 cases improved by 0.67 sd postop	+
Hashim et al. (2014)	70	Post	Cross-sectional	Test norms	Un-known	Mean 10 y	Sagittal	IQ, Acad. achieve-ment Visuo-motor	WASI, WF, Beery VMI	Higher test scores in subjects having undergone whole-vault cranioplasty.	+++
Junn (2021)	26	Post	Cross-sectional	Test norms	21/5	Range 8.7–10 y	Sagittal + Metopic	IQ,Visuo-motor, acad. ach.	WASI-II, Beery VI, Kaufman EA−3	Preoperative ERP not correlated with school age IQ. Higher IQ in early operated cases.	++
Kapp-Simon (1998)	84	Pre+Post+ Unop-erated	Longitudinal	Test norms	56/38	Mean age 8, 21 and 50 months	Metopic + Sagittal + Uni-coronal	DQ/MDI/PDI/GCI	BSID MSCA	Majority of cases had score in normal range. Mean MDI in the metopic group .8 to 1 sd < mean.	++
Kapp-Simon et al. (2005)	100	Pre	Cross-sectional	Test norms	64/36	1,7 to 30,6 months	All subtypes	DQ/MDI/PDI Language	BSID PLS	Cases scoring significantly below norm means (.6 to 1.1 sd). Smallest differences for language, largest for psychomotor.	+++
Kapp-Simon et al. (2016)	174 + 183 cont	Post	Cross-sectional	Control group	113/66	Mean age 7,5 years	All subtypes	IQ LanguageMemory	WISC-IV Token test CMS NEPSY-II CCC−2	All case groups within normal range, but significantly lower scores than controls. Small to moderate effect sizes (d=.0–.4)	++++
Kljajić et al. (2019)	73	Post	Cross-sectional	Test norms	49/24	7–15 years of age	Sagittal + Metopic	IQ/Verbal and perc. Function/ Working memory/ Process-ing speed	WISC-IV	Group means within normal range. Sagittal group sign. lower working memory scores (approx. −.5 sd)	+++
Kljajić et al. (2020)	61	Post	Cross-sectional	Test norms	43/18	8–16 years of age	Sagittal + Metopic	Attention Vigilance	CPT−3	Both groups scoring significantly lower on majority of measures, effect sizes generally in the moderate range (d .4 to .6–7)	++
Magge et al. (2002)	16	Post	Longitudinal	Test norms	Un-known	6–16 years of age	Sagittal	DQ/IQ	WISC-III WRAT CPT DTVMI Vineland-II	Group mean within normal range, but high incidence of learning disorders (50%).	+
Mathijssen et al. (2006)	220	Pre+Post	Longitudinal	Test norms	66/154	Below 16 years of age	All subtypes	IQ	Brunet-Lézine test New Metric test	Group means within normal range. No effect on DQ/IQ of age at surgery, ICP or genetics.	++
van der Meulen et al. (2008)	95	Post	Longitudinal	Test norms	Un-known	8,4 y	All subtypes	DQ Language	SON-R WPPSI-R	Nonsyndromic IQ mean within normal range, significantly higher than syndromic group. No correlation with presence of cerebral BCP.	++
Naumann et al. (2012)	70	Pre+Post	Cross-sectional	Test norms	43/27	3 y	All subtypes	DQ	BSID-II PLS−3	DQ scores approx. 1 sd below mean pre-surgery, no significant positive effect postsurgery. Significant correlations DQ-length of surgery anesthesia	+++
Panchal et al. (2001)	21 + 42 contr	Pre	Cross-sectional	Test norms and deform. Plagioceph. Control group	Unknown	Before 1 y	Sagittal	DQ Language	BSID-II	Mental and psychomotor development score distributions lower in SSC group.	++
Patel et al. (2014)	70	Post	Cross-sectional	Test norms	Unknown	5–25 y	Sagittal	IQ Academic achieve-ment Visuo-motor function	WASI WF Beery VMI BRIEF, BASC	Mean IQs within normal range. Cases operated on earlier had significantly higher test scores and lower rates of learning disorders.	+++
Shimoji (2014)	34	Pre+Post	Cross-sectional	Test norms	32/2	2–4 y	Sagittal	IQ Language	Kyoto form develop-mental test PARS NRC S-S J-CBCL	Mean Cognitive scores > 2 sd below normal range.	+
Shipster (2007)	76	Pre+Post	Longitudinal	Test norms	61/15	Mean 9 m to 15 y 7 m	Metopic	IQ	BSID-II WPPSI-R WISC-III PLS−3 REEL−3 BPVS RAPT/EAT RWFVS CELF	High prevalence of speech and/or language impairment (28%)	++
Speltz et al. (1997)	19 + 19 contr	Pre+Post	Longitudinal	Control group	Unknown	4–24 m	Sagittal	DQ	BSID	All cases obtained scores within normal range, no significant difference to controls. Trend of lower scores in late operated cases.	++
Speltz et al. (2007)	125 + 125 contr	Pre	Cross-sectional	Control group	76/49	2–24 months	All subtypes	IQ Academic achievement	BSID-II PLS−3	Cases IQ and math computation scores significantly lower than controls but small effect sizes (d=.2–.3)	++++
Speltz et al. (2015)	182 + 183 contr	Post	Longitudinal	Control group	114/68	Mean 7,5 y	All subtypes	DQ Language	WISC-IV WRAT−4 TOWRE CTOPP	Case DQ and math scores lower than controls after adjustment; prior to it case DQ, math, reading, and phono-logical awareness lower (d= .1 to .2)	++++
Starr et al. (2007)	168 + 115 contr	Pre+Post	Longitudinal	Test norms	107/61	6–18 m	Metopic	DQ Language	BSID-II PLS−3	Lower case scores after adjustments bth small effect sizes (<.2 sd).	++++
Starr et al. (2010)	49	Pre+Post	Longitudinal	Control group	34/15	18–36 m	All subtypes	DQ Language	BSID-II PLS−3	DQs of cases below norm means (−0,65 to 1,1 sd below mean). No association with severity of skull malformation	+++
Starr et al. (2012)	209 + 222 contr	Pre+Post	Longitudinal	Control group	136/73	Mean 3 y	All subtypes	DQ	BSID-II PLS−3	Small but significant differences with lower test score in cases (− .2 to .4 sd)	++++
Toth et al. (2008)	200 + 149 contr	Pre+Post	Cross-sectional	Control group	131/69	12 to 24 months of age	All subtypes	DQ Memory Response inhibition	BSID-II PLS−3 A-B/ABID Tasks	No differences in memory or response inhibition between cases and controls	++++
Wallace et al. (2016)	179 + 183 contr	Post	, Cross -sectional	Test norms	113/66	Mean age 7,5 y	All subtypes	Visuomotor	NEPSY-II Arrows Beery VMI Purdue Pegboard	Cases lower scores on manual dexterity measures.	++++
Warschausky et al. (2005)	22	Untreated	Retrospective/Cross-sectional	Test norms	19/3	3,6 to 25,3 m	Sagittal	Aspects of verbal IQ, Mem-ory, fine/visuo-motor,	BSID I+II	MDI within normal range, but low language quotient (−1,5 sd)	++
Virtanen et al. (1999)	18 + 18 contr	Post	Cross-sectional	Test norms	13/5	7,8 to 16,3 y	Metopic	IQ	WISC-R Benton Visual Retention Beery VMI Purdue Pegboard	Cases lower scores on general verbal comprehension and auditory memory than controls, higher on Similarities subtest of WISC-R.	++
J. J. B. van der Vlugt et al. (2012)	82	Post	Longitudinal	Test norms	68/14	4 to 18 y	Metopic	IQ	Mullen scales WPPSI-III/WISC-III/WAIS-III SCQ/DISC	Average mean IQ but with high variability (range of IQ 52–147). No association between width of ventricles at age 7 months and IQ.	++
J. J. B. van der Vlugt et al. (2017)	72	Post	Cross-sectional	Test norms	Unknown	3–9 y	Uni-coronal	IQ Academic achieve-ment Visuo-motor function	Mullen scales WPPSI-III/WISC-III	IQ scores above test norm averages. Higher verbal than perceptual functioning, right-sided coronal CS higher verbal scores than left-sided. Visuomotor skills below test norm mean.	++
J. van der Vlugt et al. (2021)	52	Post	Cross-sectional	Test norms	Unknown	Mean of 8.1 y	Metopic	IQ, Visuo-motor	WASI, Brief VMI	Group IQ within normal range, Small negative effect on visuomotor function.
R. T. Wu, Gabrick, et al. (2020)	20	Post	Cross-sectional	Test norms	9/11	Mean of 12,1 y	Metopic + Sagittal	IQ Academic achievement Visuo-motor function	WF WASI Beery VMI BRIEF/BASC	Correlations between test scores and age at testing and at surgery, parental income and education. Cases with SMAD6 mutation significantly lower general and performance IQ and numerical operations skills.	+++
R. T. Wu,Timberlake, et al. (2020)	28	Post	Cross-sectional	Test norms	24/4	Mean of 10 y	All subtypes	IQ Academic achieve-ment	WF WASI Beery VMI BRIEF/BASC	Cases with SMAD6 genetic mutation significantly lower general and performance IQ and numerical operations skills.	++
J. van der Vlugt et al. (2021)	11	Post	Cross-sectional	Test norms	8/3	Mean of 8 y	All subtypes	IQ Academic achieve-ment	WASI WF	Significant correlations between test scores and left frontal EEG recordings.	++

CS= Craniosynostosis, SSC= Single-Suture Craniosynostosis, m=months, y=years, DQ=Developmental Quotient, IQ= Intelligence Quotient, PDI= Performance Developmental Index, MDI= Motor Developmental Index, EEG= Electroencephalogram, BSID= Bayley Scales of Infant Development, WPPSI= Wechsler Preschool and Primary Scale of Intelligence, WISC=Wechsler Intelligence Scale for Children, WASI= Wechsler Abbreviated Scale of Intelligence, WF= Wechsler Fundamentals, K-ABC= Kaufman Assessment Battery for Children, TAP= Test for Attentional Performance, VLMT= Verbal Learning and Memory Test, VDLT= Visual Design and Memory Test, VOT= Visual Orientation Test, DVTP= Developmental Test of Visual Perception, DTVMPI= Developmental Test of Visual, Motor and Perceptual Integration, RCFT= Rey Complex Figure Test, WRAT= Wide Range Assessment Test, TOWRE= Test of Word Reading Efficiency, CTOPP= Comprehensive Test of Phonological Processing, PLS= Preschool Language Scale, MSCA= McCarthy Scale of Children´s Abilities, CMS= Children´s Memory Scale, WPT= Wonderlic Personnel Test, CCC= Children´s Communication Checklist, CPT= Continuous Performance Test, SON= Snijder Oomen Nonverbal intelligence test, BASC= Behavior Assessment System for Children, BRIEF= Behavior Rating Inventory of Executive Function, PARS= Pervasive Developmental Disorders Autism Society Japan Rating Sale, NRC S-S= National Rehabilitation Center Sign-Significance test, CBCL= Child Behavior Check List, REEL= Receptive and Expressive Emergent Language test, PACS= Phonological Assessment of Children´s Speech, EAT=Edinburgh Articulation Test, BPVS= British Picture Vocabulary Scale, RAPT= Renfrew Action picture Test, CELF= Clinical Evaluation of Language Fundamentals, RWFVS= Renfrew Word Finding Vocabulary Scale, NEPSY= A Developmental Neuropsychological Assessment, SCQ= Social Communication Questionnaire, DISC= Diagnostic Interview Schedule for Children.

Table 4. Summary of quality assessments and final GRADE score of the included studies.

First author (year)	Appropriate and clearly stated objective?	Clearly defined and representative sample?	Sufficient sample size and/or power analysis?	Use of appropriate controls?	Ensurances of reliability, validity and if possible blinding of measures used?	Measurement and statistical adjustment for key confounding variables?	GRADE score
Alperovich et al. (2021)	Partly	Yes	No	No	Yes	Yes	++
Arnaud et al. (2002)	Yes	Partly	Partly	No	No	No	++
Becker et al. (2005)	Yes	Yes	Partly	No	No	No	++
Bellew et al. (2005)	Yes	Partly	No	Yes	Partly	No	++
Bellew et al. (2011)	Yes	Partly	No	Partly	No	No	++
Bellew and Chumas (2015)	Partly	Partly	Partly	No	Yes	No	++
Boltshauser et al. (2003)	Yes	Partly	No	Partly	No	No	+
Bottero et al. (1998)	Partly	Yes	Partly	No	No	Partly	++
Byun et al. (2018)	Yes	Partly	Partly	No	Partly	No	++
Chieffo et al. (2010)	Yes	Partly	Partly	Yes	Partly	No	++
Chuang et al. (2021)	Yes	Partly	Partly	No	Partly	No	++
Cohen (2003)	Yes	Partly	No	No	Partly	No	+
Collett et al. (2017)	Yes	Yes	Partly	Yes	Partly	Yes	+++
Da Costa et al. (2006)	Yes	Partly	No	No	Partly	Partly	++
Da Costa et al. (2012)	Yes	Partly	Partly	No	Partly	Partly	++
Cradock et al. (2015)	Yes	Yes	Partly	Yes	Yes	Yes	++++
Fontana (201)	Yes	Partly	No	No	Partly	No	+
Hashim (2014)	Yes	Yes	Yes	No	Partly	Yes	+++
Junn (2021)	Yes	Partly	No	No	Partly	Yes	++
Kapp-Simon (1998)	Yes	Yes	Partly	No	Partly	No	++
Kapp-Simon et al. (2005)	Yes	Yes	Partly	No	Yes	Yes	+++
Kapp-Simon et al. (2016)	Yes	Yes	Party	Yes	Yes	Yes	++++
Kljajić et al. (2019)	Yes	Partly	Partly	No	Yes	Partly	+++
Kljajić et al. (2020)	Yes	Partly	Partly	No	No	No	++
Magge et al. (2002)	Yes	Partly	No	No	No	No	+
Mathijssen et al. (2006)	Partly	Yes	Partly	No	No	No	++
van der Meulen et al. (2008)	Yes	Partly	Partly	No	No	No	++
Naumann et al. (2012)	Yes	Partly	Partly	No	Yes	Partly	+++
Panchal et al. (2001)	Yes	Partly	No	Yes	Partly	Partly	++
Patel et al. (2014)	Yes	Partly	Yes	No	No	Yes	+++
Shimoji (2015)	Partly	Partly	No	No	No	No	+
Shipster (2007)	Yes	Partly	Partly	No	Partly	Partly	++
Speltz et al. (1997)	Yes	Yes	No	Yes	No	No	++
Speltz et al. (2007)	Yes	Yes	Yes	Yes	Yes	Yes	++++
Speltz et al. (2015)	Yes	Yes	Partly	Yes	Yes	Yes	++++
Starr et al. (2007)	Yes	Yes	Partly	Yes	Yes	Yes	++++
Starr et al. (2010)	Yes	Partly	Partly	No	Yes	Partly	+++
Starr et al. (2012)	Yes	Yes	Partly	Yes	Yes	Yes	++++
Toth et al. (2008)	Yes	Yes	Yes	Yes	Yes	Yes	++++
Wallace et al. (2016)	Yes	Partly	Yes	Yes	Yes	Yes	++++
Warschausky et al. (2005)	Yes	Partly	No	No	No	Partly	++
Virtanen et al. (1999)	Partly	Partly	No	Yes	Partly	No	++
J. J. B. van der Vlugt et al. (2012)	Yes	Yes	Partly	No	No	No	++
J. J. B. van der Vlugt et al. (2017)	Yes	Partly	No	No	No	Yes	++
J. van der Vlugt et al. (2021)	Yes	Partly	Partly	No	No	No	++
R. T. Wu, Gabrick, et al. (2020)	Yes	Partly	Partly	No	Yes	Yes	+++
R. T. Wu, Timberlake, et al. (2020)	Yes	Yes	No	No	Yes	Yes	++
R. Wu et al. (2021)	Yes	Partly	No	No	Yes	Partly	++

Table A1. A standardized extraction form based on the National Institute of Mental Health (NIMH) Guidelines for assessment of quality in correlational studies.

Reference number
Methods
Participants
Study population
No of participants
Ages of participants
Conditions of participants (type of craniosynostosis)
Treatments related to craniosynostosis
Age at treatment
Comorbid conditions
Study country
Comment/Follow-up
Measures
Cognitive instruments
Cognitive outcomes measured
Comment/Follow-up
Design
Type of study (prospective, retrospective, register)
Was some kind of comparison to a control group performed?
Time of data collection
Comment/Follow-up
Results
Cognitive outcome
Comparison to controls?
Comment/Follow-up
Quality
Was the study question or objective clearly stated?
Was the research question or objective appropriate?
Was the study population clearly specified and defined?
Were the participants in the study representative?
Were all eligible participants that met the prespecified entry criteria enrolled?
Was the sample size sufficiently large to provide confidence in the findings?
Was a sample size justification, power description, or variance and effect estimates provided?
Were controls selected or recruited from the same or similar population that gave rise to the cases (including the same timeframe)?
Was the test clearly described and delivered consistently across the study population?
Were the outcome measures prespecified, clearly defined, valid, reliable, and assessed consistently across all study participants?
Were the people assessing the outcomes blinded to the participants’ exposures/interventions?
Were key potential confounding variables measured and adjusted statistically for their impact on the relationship between exposure(s) and outcome(s)?

Alperovich, M., Runyan, C. M., Gabrick, K. S., Wu, R. T., Morgan, C., Park, S. E., Chapman, L. A., Couture, D. E., David, L. R., & Persing, J. A. (2021). Long-term neurocognitive outcomes of spring-assisted surgery versus cranial vault remodeling for SagittalSynostosis. Plastic and Reconstructive Surgery, 147(3), 661–671. https://doi.org/10.1097/PRS.0000000000007640

 PubMed Web of Science ®Google Scholar

Arnaud, E., Meneses, P., Lajeunie, E., Thorne, J. A., Marchac, D., & Renier, D. (2002). Postoperative mental and morphological outcome for nonsyndromic brachycephaly. Plastic and Reconstructive Surgery, 110(1), 6–13. https://doi.org/10.1097/00006534-200207000-00002

 PubMed Web of Science ®Google Scholar

Becker, D. B., Petersen, J. D., Kane, A. A., Cradock, M. M., Pilgram, T. K., & Marsh, J. L. (2005). Speech, cognitive, and behavioral outcomes in nonsyndromic craniosynostosis. Plastic and Reconstructive Surgery, 116(2), 400–407. https://doi.org/10.1097/01.prs.0000172763.71043.b8

 PubMed Web of Science ®Google Scholar

Bellew, M., Chumas, P., Mueller, R., Liddington, M., & Russell, J. (2005). Pre- and postoperative developmental attainment in sagittal synostosis. Archives of Disease in Childhood, 90(4), 346–350. https://doi.org/10.1136/adc.2003.035824

 PubMed Web of Science ®Google Scholar

Bellew, M., Liddington, M., Chumas, P., & Russell, J. (2011). Preoperative and postoperative developmental attainment in patients with sagittal synostosis: 5-year follow-up. Journal of Neurosurgery Pediatrics, 7(2), 121–126. https://doi.org/10.3171/2010.11.PEDS10216

 PubMed Web of Science ®Google Scholar

Bellew, M., & Chumas, P. (2015). Long-term developmental follow-up in children with nonsyndromic craniosynostosis. Journal of Neurosurgery Pediatrics, 16(4), 445–451. https://doi.org/10.3171/2015.3.PEDS14567

 PubMed Web of Science ®Google Scholar

Boltshauser, E., Ludwig, S., Dietrich, F., & Landolt, M. A. (2003). Sagittal craniosynostosis: Cognitive development, behaviour, and quality of life in unoperated children. Neuropediatrics, 34(6), 293–300. https://doi.org/10.1055/s-2003-44667

 PubMed Web of Science ®Google Scholar

Bottero, L., Lajeunie, E., Arnaud, E., Marchac, D., & Renier, D. (1998). Functional outcome after surgery for trigonocephaly. Plastic and Reconstructive Surgery, 102(4), 952–960. https://doi.org/10.1097/00006534-199809020-00002

 PubMed Web of Science ®Google Scholar

Byun, I. H., Hong, J. W., Hussein, M. A., & Kim, Y. O. (2018). Demographic characteristics of craniosynostosis patients in Asia. Journal of Cranio-Maxillo-Facial Surgery: Official Publication of the European Association for Cranio-Maxillo-Facial Surgery, 46(4), 674–678. https://doi.org/10.1016/j.jcms.2018.02.008

 PubMed Web of Science ®Google Scholar

Chieffo, D., Giovanni, S. D., Rocco, C. D., DiGiovanni, S., Giansanti, C., Caldarelli, M., & DiRocco, C. (2010). Long-term neuropsychological development in single-suture craniosynostosis treated early. Journal of Neurosurgery, 5(3), 232–237. https://doi.org/10.3171/2009.10.PEDS09231

 Web of Science ®Google Scholar

Chuang, C., Chaunzwa, T. L., Wu, R., Singh, A., Patel, A., Yang, J. F., Hashim, P. W., Travieso, R., Terner, J. S., Mayes, L. C., Duncan, C. C., Jane, J. A., Jr., Lin, K. Y., Bridgett, D. J., & Persing, J. A. (2021). Long-term neurocognitive outcomes in sagittal synostosis: The impact of reoperation. The Journal of Craniofacial Surgery, 32(1), 58–61. https://doi.org/10.1097/SCS.0000000000006909

 PubMed Web of Science ®Google Scholar

Cohen, S. R., Cross, K. P., Nichols, S. L., Simms, C., Cross, K. P., & Burstein, F. D. (2004). American Society of Maxillofacial Surgeons Outcome Study. Preoperative and Postoperative Neurodevelopmental Findings in Single-Suture Craniosynostosis. PLASTIC and RECONSTRUCTIVE SURGERY, 114(4), 841–847. https://doi.org/10.1097/01.PRS.0000132854.14237.A8

 PubMed Web of Science ®Google Scholar

Collett, B. R., Kapp-Simon, K. A., Wallace, E., Cradock, M. M., Buono, L., & Speltz, M. L. (2017). Attention and executive function in children with and without single-suture craniosynostosis. Child Neuropsychology: A Journal on Normal & Abnormal Development in Childhood & Adolescence, 23(1), 83–98. https://doi.org/10.1080/09297049.2015.1085005

 PubMed Web of Science ®Google Scholar

Da Costa, A. C., Walters, I., Savarirayan, R., Anderson, V. A., Wrennall, J. A., & Meara, J. G. (2006). Intellectual outcomes in children and adolescents with syndromic and non-syndromic craniosynostosis. Plastic and Reconstructive Surgery, 118(1), 175–181. https://doi.org/10.1097/01.prs.0000221009.93022.50

 PubMed Web of Science ®Google Scholar

Da Costa, A. C., Anderson, V. A., Savarirayan, R., Wrennall, J. A., Chong, D. K., Holmes, A. D., Greensmith, A. L., & Meara, J. G. (2012). Neurodevelopmental functioning of infants with untreated single-suture craniosynostosis during early infancy. Child’s Nervous System: ChNs: Official Journal of the International Society for Pediatric Neurosurgery, 28(6), 869–877. https://doi.org/10.1007/s00381-011-1660-1

 PubMed Web of Science ®Google Scholar

Cradock, M. M., Gray, K. E., Kapp-Simon, K. A., Collett, B. R., Buono, L. A., & Speltz, M. L. (2015). Sex differences in the neurodevelopment of school-age children with and without single-suture craniosynostosis. Child’s Nervous System: ChNs: Official Journal of the International Society for Pediatric Neurosurgery, 31(7), 1103–1111. https://doi.org/10.1007/s00381-015-2671-0

 PubMed Web of Science ®Google Scholar

Fontana, S. C., Belinger, S., Daniels, D., Tuttle, M., Camarata, P. J., & Andrews, B. T. (2017). Longitudinal assessment of developmental outcomes in infants undergoing late craniosynostosis repair. Journal of Craniofacial Surgery, 29(1), 25–28. https://doi.org/10.1097/SCS.0000000000004024

 Web of Science ®Google Scholar

Hashim, P. W., Patel, A., Yang, J. F., Travieso, R., Terner, J., Losee, J. E., Pollack, I., Jane, J., Sr., Jane, J., Jr., Kanev, P., Mayes, L., Duncan, C., Bridgett, D. J., & Persing, J. A. (2014). The effects of whole-vault cranioplasty versus strip craniectomy on long-term neuropsychological outcomes in sagittal craniosynostosis. Plastic and Reconstructive Surgery, 134(3), 491–501. https://doi.org/10.1097/PRS.0000000000000420

 PubMed Web of Science ®Google Scholar

Junn, A., Dinis, J., Park, K. E., Hauc, S., Yang, J. F., Chuang, C., Han, G., McPartland, J. C., Persing, J. A., & Alperovich, M. (2021). Long-term Follow-up of Preoperative Infant Event-related Potentials in School-age Children with Craniosynostosis. Plastic and Reconstructive Surgery Global Open, 9(10), e3844. https://doi.org/10.1097/GOX.0000000000003844

 PubMedGoogle Scholar

Kapp-Simon, K. A. (1998). Mental Development and Learning Disorders in Children with Single Suture Craniosynostosis. 30 october 2020 från https://journals.sagepub.com/doi/10.1597/1545-1569_1998_035_0197_mdaldi_2.3.co_2#articleCitationDownloadContainer

 Google Scholar

Kapp-Simon, K. A., Leroux, B., Cunningham, M., & Speltz, M. L. (2005). Multisite Study of infants with single-suture craniosynostosis: preliminary report of presurgery development. 8.

 Google Scholar

Kapp-Simon, K. A., Wallace, E., Collett, B. R., Cradock, M. M., Crerand, C. E., & Speltz, M. L. (2016). Language, learning, and memory in children with and without single-suture craniosynostosis. Journal of Neurosurgery: Pediatrics, 17(5), 578–588. https://doi.org/10.3171/2015.9.PEDS15238

 PubMed Web of Science ®Google Scholar

Kljajić, M., Maltese, G., Tarnow, P., Sand, P., & Kölby, L. (2019). The cognitive profile of children with non-syndromic craniosynostosis. Plastic and Reconstructive Surgery, 143(5), 1037e–1052e. https://doi.org/10.1097/PRS.0000000000005515

 PubMed Web of Science ®Google Scholar

Kljajić, M., Maltese, G., Tarnow, P., Sand, P., & Kölby, L. (2020). [Formula: See text] Sustained attention and vigilance of children treated for sagittal and metopic craniosynostosis. Child Neuropsycholog: A Journal on Normal and Abnormal Development in Childhood and Adolescence, 26(4), 475–488. https://doi.org/10.1080/09297049.2019.1682130

 Google Scholar

Magge, S. N., Westerveld, M., Pruzinsky, T., & Persing, J. A. (2002). Long-term neuropsychological effects of sagittal craniosynostosis on child development. Journal of Craniofacial Surgery, 13(1), 99–104. https://doi.org/10.1097/00001665-200201000-00023

 PubMed Web of Science ®Google Scholar

Mathijssen, I., Marchac, D., Lajeunie, E., Marchac, D., & Renier, D. (2006). Postoperative cognitive outcome for synostotic frontal plagiocephaly. Journal of Neurosurgery, 105(1), 16–20. https://doi.org/10.3171/ped.2006.105.1.16

 PubMed Web of Science ®Google Scholar

van der Meulen, J., van der Vlugt, J., Okkerse, J., & Hofman, B. (2008). Early beaten-copper pattern: Its long-term effect on intelligence quotients in 95 children with craniosynostosis. Journal of Neurosurgery: Pediatrics, 1(1), 25–30. https://doi.org/10.3171/PED-08/01/025

 PubMed Web of Science ®Google Scholar

Naumann, H. L., Haberkern, C. M., Pietila, K. E., Birgfeld, C. B., Starr, J. R., Kapp-Simon, K. A., Hopper, R. A., & Speltz, M. L. (2012). Duration of exposure to cranial vault surgery: Associations with neurodevelopment among children with single-suture craniosynostosis: Anesthesia exposure and neurodevelopment. Pediatric Anesthesia, 22(11), 1053–1061. https://doi.org/10.1111/j.1460-9592.2012.03843.x

 PubMed Web of Science ®Google Scholar

Panchal, J., Amirsheybani, H., Gurwitch, R., Cook, V., Francel, P., Neas, B., & Levine, N. (2001). Neurodevelopment in children with single-suture craniosynostosis and plagiocephaly without synostosis. Plastic and Reconstructive Surgery, 108(6), 1492–1500. https://doi.org/10.1097/00006534-200111000-00007

 PubMed Web of Science ®Google Scholar

Patel, A., Yang, J. F., Hashim, P. W., Travieso, R., Terner, J., Mayes, L. C., Kanev, P., Duncan, C., Jane, J., Jane, J., Pollack, I., Losee, J. E., Bridgett, D. J., & Persing, J. A. (2014). The impact of age at surgery on long-term neuropsychological outcomes in sagittal craniosynostosis. The Impact of Age at Surgery on Long-Term Neuropsychological Outcomes in Sagittal Craniosynostosis: Plastic and Reconstructive Surgery, 134(4), 608e–617e. https://doi.org/10.1097/PRS.0000000000000511

 Google Scholar

Shimoji, T., Tominaga, D., Shimoji, K., Miyajima, M., & Tasato, K. (2015). Analysis of pre- and post-operative symptoms of patients with mild trigonocephaly using several developmental and psychological tests. Child’s Nervous System: ChNs: Official Journal of the International Society for Pediatric Neurosurgery, 8(3), 433–440. https://doi.org/10.1007/s00381-014-2595-0

 Google Scholar

Shipster, C., Stackhouse, J., & Wade, A. (2007). Speech, language, and cognitive development in children with isolated sagittal synostosis. Developmental Medicine & Child Neurology, 45(1), 34–43. https://doi.org/10.1111/j.1469-8749.2003.tb00857.x

 Google Scholar

Speltz, M. L., Endriga, M. C., & Mouradian, W. E. (1997). Presurgical and postsurgical mental and psychomotor development of infants with sagittal synostosis—Matthew LC. Endriga, Wendy. Speltz, Marya E. Mouradian, 1997. Hämtad 30 oktober 2020, från. https://journals-sagepub-com.ezproxy.its.uu.se/doi/10.1597/1545-1569_1997_034_0374_papmap_2.3.co_2#articleCitationDownloadContainer.

 Google Scholar

Speltz, M. L., Kapp-Simon, K., Collett, B., Keich, Y., Gaither, R., Cradock, M. M., Buono, L., & Cunningham, M. L. (2007). Neurodevelopment of Infants with single-suture craniosynostosis: Presurgery comparisons with case-matched controls. Plastic and Reconstructive Surgery, 119(6), 1874–1881. https://doi.org/10.1097/01.prs.0000259184.88265.3f

 PubMed Web of Science ®Google Scholar

Speltz, M. L., Collett, B. R., Wallace, E. R., Starr, J. R., Cradock, M. M., Buono, L., Cunningham, M., & Kapp-Simon, K. (2015). Intellectual and academic functioning of school-age children with single-suture craniosynostosis. Pediatrics, 135(3), e615–e623. https://doi.org/10.1542/peds.2014-1634

 PubMed Web of Science ®Google Scholar

Starr, J. R., Kapp-Simon, K. A., Cloonan, Y. K., Collett, B. R., Cradock, M. M., Buono, L., Cunningham, M. L., & Speltz, M. L. (2007). Presurgical and postsurgical assessment of the neurodevelopment of infants with single-suture craniosynostosis: Comparison with controls. Journal of Neurosurgery: Pediatrics, 107(2), 103–110. https://doi.org/10.3171/PED-07/08/103

 Google Scholar

Starr, J. R., Lin, H. J., Ruiz-Correa, S., Cunningham, M. L., Ellenbogen, R. G., Collett, B. R., Kapp-Simon, K. A., & Speltz, M. L. (2010). Little evidence of association between severity of trigonocephaly and cognitive development in infants with single-suture metopic synostosis. Neurosurgery, 67(2), 408–416. https://doi.org/10.1227/01.NEU.0000371992.72539.8B

 PubMed Web of Science ®Google Scholar

Starr, J. R., Collett, B. R., Gaither, R., Kapp-Simon, K. A., Cradock, M. M., Cunningham, M. L., & Speltz, M. L. (2012). Multicenter study of neurodevelopment in 3-Year-old children with and without single-suture craniosynostosis. Archives of Pediatrics & Adolescent Medicine, 166(6). https://doi.org/10.1001/archpediatrics.2011.1800

 Google Scholar

Toth, K., Collett, B., Kapp-Simon, K. A., Cloonan, Y. K., Gaither, R., Cradock, M. M., Buono, L., Cunningham, M. L., Dawson, G., Starr, J., & Speltz, M. L. (2008). Memory and response inhibition in young children with single-suture craniosynostosis. Child Neuropsychology, 14(4), 339–352. https://doi.org/10.1080/09297040701594888

 PubMed Web of Science ®Google Scholar

Wallace, E. R., Collett, B. R., Kapp-Simon, K., Starr, J. R., Birgfeld, C., & Speltz, M. L. (2016). Visuomotor function in school-age children with single-suture craniosynostosis. Journal of Developmental & Behavioral Pediatrics: JDBP, 37(6), 483–490. https://doi.org/10.1097/DBP.0000000000000319

 PubMed Web of Science ®Google Scholar

Warschausky, S., Angobaldo, J., Kewman, D., Buchman, S., Muraszko, K. M., & Azengart, A. (2005). Early development of infants with untreated metopic craniosynostosis. Plastic and Reconstructive Surgery, 115(6), 1518–1523. https://doi.org/10.1097/01.PRS.0000160270.27558.64

 PubMed Web of Science ®Google Scholar

Virtanen, R., Korhonen, T., Fagerholm, J., & Viljanto, J. (1999). Neurocognitive sequelae of scaphocephaly. Pediatrics, 103, 791–795. https://doi.org/10.1542/peds.103.4.791

 PubMed Web of Science ®Google Scholar

van der Vlugt, J. J. B., van der Meulen, J. J. N. M., Creemers, H. E., Verhulst, F. C., Hovius, S. E. R., & Okkerse, J. M. E. (2012). Cognitive and behavioral functioning in 82 patients with trigonocephaly. Plastic and Reconstructive Surgery, 130(4), 885–893. https://doi.org/10.1097/PRS.0b013e318262f21f

 PubMed Web of Science ®Google Scholar

van der Vlugt, J. J. B., van der Meulen, J. J. M. N., van den Braak, R. R. J. C., Vermeij-Keers, C., Horstman, E. G. C., Hovius, S. E. R., Verhulst, F. C., Wierdsma, A. I., Lequin, M. H., & Okkerse, J. M. E. (2017). Insight into the pathophysiologic mechanisms behind cognitive dysfunction in trigonocephaly. Plastic and Reconstructive Surgery, 139(4), 954e–964e. https://doi.org/10.1097/PRS.0000000000003179

 PubMed Web of Science ®Google Scholar

van der Vlugt, J., Coebergh van den Braak, R., van der Meulen, J., Hovius, S., Verhulst, F. C., Okkerse, J., Wierdsma, A. I., Kushner, S. A., & Klimek, M. (2021). Prolonged surgical duration in open craniofacial surgery: Detrimental to cognitive functioning? Journal of Plastic, Reconstructive & Aesthetic Surgery: JPRAS, 74(12), 3443–3476. https://doi.org/10.1016/j.bjps.2021.08.017

 PubMed Web of Science ®Google Scholar

Wu, R. T., Gabrick, K. S., Singh, A., Landi, N., Taylor, J. A., Bartlett, S. P., Persing, J. A., & Alperovich, M. (2020). Comparison of neurocognitive outcomes in postoperative adolescents with unilateral coronal synostosis. Plastic and Reconstructive Surgery, 146(3), 614–619. https://doi.org/10.1097/PRS.0000000000007067

 PubMed Web of Science ®Google Scholar

Wu, R. T., Timberlake, A. T., Abraham, P. F., Gabrick, K. S., Lu, X., Peck, C. J., Sawh-Martinez, R. F., Steinbacher, D. M., Alperovich, M. A., & Persing, J. A. (2020). SMAD6 genotype predicts neurodevelopment in non-syndromic craniosynostosis. Plastic and ReconstructiveSurgery, 145(1), 117e–125e. https://doi.org/10.1097/PRS.0000000000006319

 PubMed Web of Science ®Google Scholar

Wu, R., Nie, J., Abraham, P., Halligan, T., Gabrick, K., Peck, C. J., Sawh-Martinez, R., Steinbacher, D. M., Alperovich, M., McPartland, J., & Persing, J. A. (2021). Neurologic characterization of craniosynostosis: Can direct brain recordings predict language development? The Journal of Craniofacial Surgery, 32(1), 78–82. https://doi.org/10.1097/SCS.0000000000007004

 PubMed Web of Science ®Google Scholar