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Surgical Timing and Neurocognitive Development among Patients with Craniosynostosis: Analysis of Confounders
Jeremy V Lynn1, Lauren K Buchman1, Christopher J Breuler1
1From the Section of Plastic Surgery, University of Michigan.
Insights
Children with craniosynostosis undergoing cranial vault remodeling (CVR) later in life may have pre-existing delays. Socioeconomic factors and comorbidities, not surgical timing alone, may influence neurocognitive outcomes in CVR patients.
Area of Science:
- Pediatric Neurosurgery
- Developmental Pediatrics
- Medical Ethics
Background:
- Craniosynostosis is a condition where skull sutures fuse prematurely.
- Previous research suggested delayed neurocognitive development in children with craniosynostosis treated after 12 months of age.
- Potential confounding factors in this relationship were not fully explored.
Purpose of the Study:
- To identify factors that may confound the relationship between the timing of cranial vault remodeling (CVR) and neurocognitive development in children.
- To investigate the hypothesis that socioeconomic disadvantages and comorbid conditions influence the timing of CVR and subsequent neurocognitive outcomes.
Main Methods:
- A retrospective study of 227 patients with nonsyndromic single-suture craniosynostosis who underwent CVR between 2009 and 2020.
- Patients were divided into two groups: early CVR (before 12 months) and late CVR (after 12 months).
- Sociodemographic and clinical variables, including preoperative cognitive and language assessments, were analyzed.
Main Results:
- The late CVR group (70 patients) had a higher proportion of non-White patients, those qualifying for financial assistance, preterm births, and comorbid conditions compared to the early CVR group (157 patients).
- Preoperative testing revealed a greater prevalence of baseline cognitive and language delays in the late CVR group.
- Statistical significance was observed for these differences (P < 0.05).
Conclusions:
- Socioeconomic disadvantages and comorbid conditions are associated with delayed CVR.
- These factors may introduce a selection bias, contributing to observed differences in neurocognitive development.
- Future research on CVR timing and neurocognitive outcomes must account for these confounding variables.
Background:
In 1988, Renier and Marchac asserted that children with craniosynostosis who undergo cranial vault remodeling (CVR) after 12 months of age experience delayed neurocognitive development compared to children who undergo CVR before 12 months of age. The purpose of this study was to identify factors potentially confounding this cause-and-effect relationship. The authors hypothesize that children with socioeconomic disadvantages or comorbid conditions are more likely to undergo CVR after 12 months and may represent a selection bias toward delayed neurocognitive development.
Methods:
Patients with nonsyndromic single-suture craniosynostosis who underwent CVR between 2009 and 2020 at Michigan Medicine were included ( n = 227). Sociodemographic and clinical variables were documented. The sample was dichotomized to compare patients who underwent CVR before (early) and after (late) 12 months of age. Statistical analysis was performed at P < 0.05 significance.
Results:
The early and late groups contained 157 patients and 70 patients, respectively. Compared to the early group, the late group contained a larger proportion of patients who identified as non-White ( P = 0.03), qualified for need-based financial assistance ( P = 0.03), were born preterm ( P < 0.01), or had a comorbid condition ( P < 0.01). Based on preoperative testing, the late group contained a larger proportion of patients with baseline cognitive ( P < 0.001) and language ( P = 0.008) delays relative to the early group.
Conclusions:
This study demonstrates that socioeconomic disadvantages and comorbid conditions are prevalent among patients who undergo delayed CVR and may represent a selection bias toward delayed neurocognitive development. Future studies evaluating the relationship between surgical timing and neurocognitive development must control for these factors.
Clinical Question/Level Of Evidence:
Risk, II.
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