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Recurrent stroke in a child with incontinentia pigmenti

Michael S Cartwright1, David L White, Lewis M Miller

  • 1Department of Neurology, Wake Forest University School of Medicine, Winston-Salem, North Carolina 27157, USA. mcartwri@wfubmc.edu

Journal of Child Neurology
|January 20, 2009
PubMed

Insights

Incontinentia pigmenti (IP) can cause recurrent infant strokes, even with antiplatelet therapy. Further research into IP pathophysiology is crucial for effective stroke treatment in affected children.

Area of Science:

  • Pediatric Neurology
  • Vascular Neurology
  • Genetics

Background:

  • Incontinentia pigmenti (IP) is a rare genetic disorder affecting the skin, eyes, nervous system, and teeth.
  • Vascular complications, including stroke, are known but less commonly reported in neonates and infants.

Observation:

  • A neonate diagnosed with genetically confirmed incontinentia pigmenti experienced bilateral strokes within the first week of life.
  • Recurrent acute strokes were observed on serial magnetic resonance imaging (MRI) at 10 days and 3 months of age.

Findings:

  • Initial MRI at 5 days revealed multiple restricted diffusion areas in both cerebral hemispheres.
  • Despite initiating antiplatelet therapy after the second stroke, a third infarct occurred by 3 months.
  • Follow-up MRI at 10 months showed infarct maturation without new stroke events.

Implications:

  • This case highlights a unique presentation of recurrent stroke in an infant with incontinentia pigmenti.
  • It underscores the need for enhanced understanding of IP's underlying mechanisms to guide targeted stroke prevention and treatment strategies.
  • Further investigation into the pathophysiology of vascular events in IP is warranted.

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