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Related Experiment Videos

Bilateral neuropathologic changes in a child with hemimegalencephaly

R Jahan1, P S Mischel, J G Curran

  • 1Department of Radiological Sciences, UCLA School of Medicine, Los Angeles, California 90095-1721, USA.

Pediatric Neurology
|January 22, 1998
PubMed
Summary

Hemimegalencephaly, a rare brain malformation, can present with bilateral cortical dysplasia, not just unilateral. This finding may explain varied outcomes in patients undergoing hemispherectomy for intractable seizures.

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Area of Science:

  • Neurology
  • Developmental Neuroscience
  • Pediatric Pathology

Background:

  • Hemimegalencephaly is a congenital brain malformation characterized by unilateral enlargement of one cerebral hemisphere.
  • It often presents with intractable seizures and developmental delay in infants.
  • Previous understanding suggested primarily unilateral neuropathologic changes.

Observation:

  • A 7-month-old girl with developmental delay and intractable seizures exhibited hemispheric asymmetry and an enlarged right cerebral hemisphere.
  • Despite undergoing a right hemispherectomy due to medically refractory seizures, the patient unfortunately died from surgical complications.
  • Postmortem examination revealed asymmetric right cerebral hemisphere enlargement and, microscopically, bilateral cortical dysplasia.

Findings:

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  • The case demonstrated severe cortical dysplasia in the enlarged right hemisphere and mild dysplasia in the left hemisphere.
  • This represents the first reported instance of bilateral cortical abnormalities in a patient diagnosed with hemimegalencephaly.
  • While white matter abnormalities in the contralateral hemisphere are known, bilateral cortical dysplasia is a novel observation.

Implications:

  • The presence of bilateral cortical dysplasia may contribute to the variable clinical outcomes observed in hemimegalencephaly patients.
  • This finding could necessitate a re-evaluation of treatment strategies and prognostic indicators for this condition.
  • Further research is warranted to understand the pathogenesis and clinical significance of bilateral cortical dysplasia in hemimegalencephaly.