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Updated: May 21, 2025

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Isolated Squamosal Synostosis: Defining the Phenotype.

Matthew E Pontell1, Connor S Wagner1, Neil Reddy1

  • 1From the Division of Plastic, Reconstructive, & Oral Surgery, Children's Hospital of Philadelphia, Philadelphia, PA.

Annals of Plastic Surgery
|May 19, 2025
PubMed
Summary
This summary is machine-generated.

Isolated squamosal synostosis (ISS) presentation varies by laterality. Unilateral ISS rarely causes intracranial hypertension, while bilateral ISS, especially complete fusion, frequently requires monitoring for elevated intracranial pressure.

Keywords:
craniosynostosisintracranial pressurepapilledemasquamosalsquamous

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Area of Science:

  • Craniofacial Surgery
  • Pediatric Neurosurgery
  • Medical Imaging

Background:

  • Isolated squamosal synostosis (ISS) is a rare craniosynostosis affecting the squamosal suture.
  • Clinical presentation and management guidelines for ISS require further characterization.

Purpose of the Study:

  • To characterize the clinical manifestations of isolated squamosal synostosis (ISS).
  • To review published literature on ISS.
  • To compare presentation and outcomes based on laterality and fusion type.

Main Methods:

  • Retrospective review of computed tomography (CT) imaging (2008-2022) for patients with ISS.
  • Analysis of patient age, signs of intracranial pressure, and surgical management.
  • Systematic literature review of ISS cases (1990-2023).

Main Results:

  • Eighteen institutional patients and 13 literature cases were analyzed.
  • Unilateral ISS (7 cases) presented early without intracranial hypertension.
  • Bilateral ISS (24 cases) showed higher rates of intracranial hypertension, particularly with complete fusion (66% vs 22%).
  • Surgical vault remodeling was performed in 9/15 bilateral ISS patients.

Conclusions:

  • ISS clinical presentation is influenced by laterality and degree of fusion.
  • Unilateral ISS typically has a benign course regarding intracranial pressure.
  • Bilateral ISS necessitates vigilant monitoring for elevated intracranial pressure, especially in complete fusion cases.