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Congenital perisylvian dysfunction - is it a spectrum?

Maria Clark1, Wui K Chong, Tim Cox

  • 1Neurosciences Unit, University College London, Institute of Child Health, London, UK.

Developmental Medicine and Child Neurology
|June 25, 2009
PubMed
Summary

This study explores the connection between bulbar cerebral palsy (Worster-Drought syndrome) and perisylvian polymicrogyria in children. Findings suggest a spectrum of congenital perisylvian dysfunction, with clinical signs being key for prognosis.

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Area of Science:

  • Pediatric Neurology
  • Neuroimaging
  • Developmental Neuroscience

Background:

  • Bulbar cerebral palsy, also known as Worster-Drought syndrome (WDS), is a condition affecting children.
  • Perisylvian polymicrogyria is a brain malformation characterized by excessive folding of the cerebral cortex.

Purpose of the Study:

  • To investigate the overlap and relationship between WDS and perisylvian polymicrogyria in pediatric patients.
  • To understand the clinical spectrum and prognostic indicators associated with these conditions.

Main Methods:

  • Retrospective analysis of clinical data and magnetic resonance imaging (MRI) from 121 children.
  • Categorization of participants into groups: WDS with normal imaging, congenital bilateral perisylvian polymicrogyria (CBPP), and congenital unilateral perisylvian polymicrogyria (CUPP).

Main Results:

  • Shared etiological markers were observed across groups, including male sex and antenatal events.
  • Pseudobulbar palsy with mild limb pyramidal signs was a common phenotype, present in WDS, CBPP, and CUPP groups, often with learning disability, epilepsy, and behavioral issues.
  • Pseudobulbar palsy was a stronger predictor of additional impairments than polymicrogyria findings.

Conclusions:

  • Congenital perisylvian dysfunction is proposed as a spectrum that includes WDS and perisylvian polymicrogyria.
  • Clinical phenotype is a more reliable indicator of prognosis than imaging findings alone.
  • Epileptic encephalopathy can lead to acquired perisylvian dysfunction.