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A diffusion tensor imaging comparison of white matter development in nonsyndromic craniosynostosis to neurotypical infants

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Abstract

Purpose

Nonsyndromic craniosynostosis (NSC) is associated with neurocognitive deficits, and intervention at infancy is standard of care to limit the negative effects of NSC on brain development. In this study, diffusion tensor imaging (DTI) was implemented to investigate white matter microstructure in infants with NSC undergoing cranial vault remodeling, and a comparison was made with white matter development in neurotypical controls.

Methods

Infants presenting with NSC (n = 12) underwent DTI scans before and after cranial vault remodeling. Neurotypical infants (n = 5), age matched to NSC patients at preoperative scans, were compared to preoperative DTI scans. Pre- and postoperative NSC scans were compared in aggregate, and the sagittal synostosis (n = 8) patients were evaluated separately. Finally, neurotypical infants from the University of North Carolina/University of New Mexico Baby Connectome Project (BCP), who underwent DTI scans at timepoints matching the NSC pre- and postoperative DTI scans, were analyzed (n = 9). Trends over the same time period were compared between NSC and BCP scans.

Results

No significant differences were found between preoperative NSC scans and controls. White matter development was more limited in NSC patients than in BCP patients, with microstructural parameters of the corpus body and genu and inferior and superior longitudinal fasciculi consistently lagging behind developmental changes observed in healthy patients.

Conclusion

Infant white matter development appears more limited in NSC patients undergoing cranial vault remodeling relative to that in neurotypical controls. Further investigation is needed to explore these differences and the specific effects of early surgical intervention.

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Data availability

The datasets analyzed during the current study are available from the corresponding author on reasonable request.

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Funding

Dr. Alperovich receives funding from CTSA Grant Number KL2 TR001862 from the National Center for Advancing Translational Science (NCATS), a component of the National Institutes of Health (NIH) and consults for Johnson & Johnson and LifeNet Health.

Author information

Author notes

  1. Jake Moscarelli and Mariana N. Almeida contributed equally as co-first authors.

Authors and Affiliations

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

    Jake Moscarelli, Mariana N. Almeida, Kevin G. Hu, Jacqueline M. H. Ihnat, Neil Parikh, John A. Persing & Michael Alperovich

  2. Department of Diagnostic Radiology, Yale School of Medicine, New Haven, CT, USA

    Cheryl Lacadie

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  1. Jake Moscarelli
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Contributions

M.N.A. and M.A. conceived the experiment. M.N.A., K.G.H., J.M.H.I., and N.P. performed patient recruitment and data collection. J.M. planned the analysis with C.L. and with guidance from M.A., M.N.A., and J.A.P. C.L. performed the analysis. J.M. drafted the manuscript with critical input and revision from all authors (M.N.A., C.L., K.G.H., J.M.H.I., N.P., J.A.P., M.A.). M.A. supervised and directed the study.

Corresponding author

Correspondence to Michael Alperovich.

Ethics declarations

Ethics approval

All procedures performed in studies involving human participants were in accordance with the ethical standards of the institutional and/or national research committee and with the 1964 Helsinki declaration and its later amendments or comparable ethical standards. The study was approved by the Yale Institutional Review Board (HIC # 2000030969).

Consent to participate

Informed consent was obtained from legal guardians for all individual participants included in this study.

Competing interests

The authors declare no competing interests.

Disclaimer

The manuscript contents are solely the responsibility of the authors and do not necessarily represent the official view of NIH. Research reported in this publication was also supported by the Richard K. Gershon Endowed Medical Student Research Fellowship and Yale School of Medicine Fellowship for Medical Student Research. The content is solely the responsibility of the authors and does not necessarily represent the official views of Richard K. Gershon Endowed Medical Student Research Fellowship and Yale School of Medicine Fellowship for Medical Student Research.

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Moscarelli, J., Almeida, M.N., Lacadie, C. et al. A diffusion tensor imaging comparison of white matter development in nonsyndromic craniosynostosis to neurotypical infants. Childs Nerv Syst 40, 1477–1487 (2024). https://doi.org/10.1007/s00381-023-06262-y

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