Long-term neurocognitive outcomes in 204 single-suture craniosynostosis patients

Junn, Adam H.; Long, Aaron S.; Hauc, Sacha C.; Almeida, Mariana N.; Alper, David P.; Rivera, Jean Carlo; Mayes, Linda C.; Persing, John A.; Alperovich, Michael

doi:10.1007/s00381-023-05908-1

Cited by 8 publications

(2 citation statements)

References 53 publications

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“…Another study reported deficits in intelligence quotient and math computation in patients who had undergone open repair, compared with unaffected controls. Notably, deficits were more severe in patients with unicoronal or metopic craniosynostosis than in those with sagittal craniosynostosis, a finding that was also reported more recently . Bellew et al found that surgery done at younger ages was associated with better neurocognitive outcomes when measured at 10 and 15 years of age.…”

Section: Introductionmentioning

confidence: 67%

“…Notably, deficits were more severe in patients with unicoronal or metopic craniosynostosis than in those with sagittal craniosynostosis, 18 a finding that was also reported more recently. 19 Bellew et al 20 found that surgery done at younger ages was associated with better neurocognitive outcomes when measured at 10 and 15 years of age.…”

Section: Introductionmentioning

confidence: 99%

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Cognitive Outcomes of Children With Sagittal Craniosynostosis Treated With Either Endoscopic or Open Calvarial Vault Surgery

Magge,

Fotouhi,

Allhusen

et al. 2024

JAMA Netw Open

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ImportanceSeveral studies have reported a higher incidence of neurodevelopmental delays and cognitive deficits in patients with single-suture craniosynostosis; however, there are few studies examining the associations of repair type with cognitive outcomes.ObjectiveTo measure differences in neuropsychological outcomes between school-age children who were treated for sagittal craniosynostosis and unaffected controls and explore differences in cognitive function among children with sagittal craniosynostosis who were previously treated with either endoscopic strip craniectomy or open calvarial vault surgery.Design, Setting, and ParticipantsThis cohort study was performed between 2018 and 2022. Eligible participants included patients aged 5 to 17 years who had previously been seen as infants or toddlers (&lt;3 years) at 1 of 3 surgical centers for craniosynostosis repair with either endoscopic surgery or open calvarial vault surgery. A separate cohort of unaffected controls were included for comparison. Data analysis was conducted from November 2023 to February 2024.ExposuresOpen calvarial vault surgery or endoscopic repair for single-suture craniosynostosis.Main Outcomes and MeasuresThe primary outcome was the Differential Ability Scales-II (DAS-II) General Conceptual Ability (GCA) score, an index for overall intellectual ability. Secondary outcomes included DAS-II subscale scores (Verbal Ability, Nonverbal Reasoning, Spatial Ability, Working Memory, and Processing Speed), and Patient-Reported Outcomes Measurement Information System (PROMIS) cognitive function scores.ResultsA total of 81 patients with sagittal craniosynostosis (59 male [73%]; 22 female [27%]) and 141 controls (81 male [57%]; 60 female [43%]) were included. Of the 81 participants with sagittal craniosynostosis, 46 underwent endoscopic repair and 35 underwent open repair. Median (range) age at time of follow-up assessment was 7.7 (5.0-14.8) years for children with sagittal craniosynostosis and median age at assessment was 8.5 (7.7-10.5) years for controls. After controlling for age at assessment, sex, and socioeconomic status, there was no statistically significant or clinically meaningful difference in GCA scores between children who underwent endoscopic repair (adjusted mean score, 100; 95% CI, 96-104) and open repair (adjusted mean score, 103; 95% CI, 98-108) (P &gt; .99). We found no significant difference in PROMIS scores between repair types (median [range] for endoscopic repair 54 [31-68] vs median [range] for open repair 50 [32-63]; P = .14). When comparing the treatment groups with the unaffected controls, differences in subscale scores for GCA and working memory were observed but were within normal range.Conclusions and RelevanceIn this cohort study, there were no statistically or clinically significant differences in cognitive outcomes among school-age children by and type of surgical procedure used to repair nonsyndromic sagittal craniosynostosis. These findings suggest primary care clinicians should be educated about different options for craniosynostosis surgery to ensure early referral of these patients so that all treatment options remain viable.

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Section: Introductionmentioning

confidence: 67%

Section: Introductionmentioning

confidence: 99%

Cognitive Outcomes of Children With Sagittal Craniosynostosis Treated With Either Endoscopic or Open Calvarial Vault Surgery

Magge,

Fotouhi,

Allhusen

et al. 2024

JAMA Netw Open

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Complications in Intracranial and Extracranial Surgery of Craniosynostosis

Rasse

2024

Surgery of Craniosynostosis and Related Midface Deformities

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Radiomorphologic profiles of nonsyndromic sagittal craniosynostosis

Skadorwa,

Wierzbieniec,

Sośnicka

et al. 2023

Childs Nerv Syst

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Purpose Numerous classification systems of nonsyndromic sagittal craniosynostosis (NSC) are applied but none has gained a wide acceptance, since each classification is focused on distinct aspects of cranial dysmorphology. The goal of this study was to depict the most common combinations of radiomorphologic characteristics of NSC and to separate groups where the patients were morphologically similar to one another and at the same time significantly different from others. Methods The study was conducted on anonymized thin-cut CT scans of 131 children with NSC aged 1–12 months (mean age 5.42 months). The type of cranial dysmorphology was assessed using four criteria: skull shape, pattern of sagittal suture fusion, morphologic features and cerebrospinal fluid (CSF) spaces alterations. After assigning the categories, an unsupervised k-modes clustering algorithm was applied to identify distinct patients clusters representing radiomorphologic profiles determined by investigated characteristics. Results Cluster analysis revealed three distinct radiomorphologic profiles including the most common combinations of features. The profiles were not influenced by sex nor age but were significantly determined by skull shape (V = 0.58, P < 0.0001), morphologic features (V = 0.50, P < 0.0001) and pattern of sagittal suture fusion (V = 0.47, P < 0.0001). CSF alterations did not significantly correlate with the profiles (P = 0.3585). Conclusion NSC is a mosaic of radiologic and morphologic features. The internal diversity of NSC results in dissimilar groups of patients defined by unique combinations of radiomorphologic characteristics, from which the skull shape is the most differentiating factor. Radiomorphologic profiles support the idea of clinical trials targeted at more selective outcomes assessment.

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Long-term neurocognitive outcomes in 204 single-suture craniosynostosis patients

Cited by 8 publications

References 53 publications

Cognitive Outcomes of Children With Sagittal Craniosynostosis Treated With Either Endoscopic or Open Calvarial Vault Surgery

Cognitive Outcomes of Children With Sagittal Craniosynostosis Treated With Either Endoscopic or Open Calvarial Vault Surgery

Complications in Intracranial and Extracranial Surgery of Craniosynostosis

Radiomorphologic profiles of nonsyndromic sagittal craniosynostosis

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