Chiari I malformation risk across anatomical subtypes of sagittal synostosis

Abstract

BackgroundSagittal synostosis is increasingly recognized as a heterogeneous condition. Whether distinct anatomical phenotypes differ in Chiari type I malformation (CM-I) burden, and how posterior fossa morphology contributes to this heterogeneity, remains unclear.MethodsWe retrospectively studied 241 children with sagittal synostosis who underwent preoperative three-dimensional CT volumetry and MRI between January 2023 and December 2025. Patients were classified as anterior fossa–involved (Group A, n = 63), posterior fossa–involved (Group B, n = 53), or isolated sagittal synostosis (Group C, n = 125). Residualized posterior fossa volume (PFV_adj) was derived from PFV regressed on preoperative intracranial volume and age at surgery. ANCOVA and sequential logistic regression models were used, with Firth penalized regression as a sensitivity analysis.ResultsCM-I prevalence differed across subtypes (P = 0.012) and was highest in Group B (28.3%) versus Group A (11.1%) and Group C (12.0%). In the subtype-only model, Group B had higher CM-I odds than Group C (OR = 2.89, 95% CI 1.29–6.48; P = 0.010). Group B also showed significantly lower PFV_adj (P < 0.001). In morphology-adjusted analyses, greater tentorial angle (OR = 3.20 per 5°, 95% CI 2.10–4.88; P < 0.001) and lower PFV_adj (OR = 0.74 per 10 mL, 95% CI 0.62–0.89; P = 0.002) were associated with CM-I. Model AUC increased numerically from 0.869 to 0.885 after adding PFV_adj. Firth regression yielded consistent results.ConclusionsThe posterior fossa–involved phenotype carries the greatest overall CM-I burden and is characterized by relative posterior fossa deficiency and a steeper tentorial configuration. PFV_adj provides a developmentally adjusted measure that complements anatomical subtype classification.

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BibTeX RIS

BibTeX

@article{kong2026chiari,
  title = {{Chiari I malformation risk across anatomical subtypes of sagittal synostosis}},
  author = {Chenxu Kong and Linzhao Min and Jiajia Wang and Jia Wei and Zhenjiang Li and Xiaoqiang Wang},
  journal = {Frontiers in Surgery},
  year = {2026},
  month = oct,
  doi = {10.3389/fsurg.2026.1949189},
  url = {https://doi.org/10.3389/fsurg.2026.1949189},
  note = {CC BY},
}

RIS

TY  - JOUR
TI  - Chiari I malformation risk across anatomical subtypes of sagittal synostosis
AU  - Chenxu Kong
AU  - Linzhao Min
AU  - Jiajia Wang
AU  - Jia Wei
AU  - Zhenjiang Li
AU  - Xiaoqiang Wang
PY  - 2026
DA  - 2026/10/23
JO  - Frontiers in Surgery
DO  - 10.3389/fsurg.2026.1949189
UR  - https://doi.org/10.3389/fsurg.2026.1949189
AB  - BackgroundSagittal synostosis is increasingly recognized as a heterogeneous condition. Whether distinct anatomical phenotypes differ in Chiari type I malformation (CM-I) burden, and how posterior fossa morphology contributes to this heterogeneity, remains unclear.MethodsWe retrospectively studied 241 children with sagittal synostosis who underwent preoperative three-dimensional CT volumetry and MRI between January 2023 and December 2025. Patients were classified as anterior fossa–involved (Group A, n = 63), posterior fossa–involved (Group B, n = 53), or isolated sagittal synostosis (Group C, n = 125). Residualized posterior fossa volume (PFV_adj) was derived from PFV regressed on preoperative intracranial volume and age at surgery. ANCOVA and sequential logistic regression models were used, with Firth penalized regression as a sensitivity analysis.ResultsCM-I prevalence differed across subtypes (P = 0.012) and was highest in Group B (28.3%) versus Group A (11.1%) and Group C (12.0%). In the subtype-only model, Group B had higher CM-I odds than Group C (OR = 2.89, 95% CI 1.29–6.48; P = 0.010). Group B also showed significantly lower PFV_adj (P < 0.001). In morphology-adjusted analyses, greater tentorial angle (OR = 3.20 per 5°, 95% CI 2.10–4.88; P < 0.001) and lower PFV_adj (OR = 0.74 per 10 mL, 95% CI 0.62–0.89; P = 0.002) were associated with CM-I. Model AUC increased numerically from 0.869 to 0.885 after adding PFV_adj. Firth regression yielded consistent results.ConclusionsThe posterior fossa–involved phenotype carries the greatest overall CM-I burden and is characterized by relative posterior fossa deficiency and a steeper tentorial configuration. PFV_adj provides a developmentally adjusted measure that complements anatomical subtype classification.
SN  - 2296-875X
ER  -

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