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Pseudotumor cerebri in kaposiform lymphangiomatosis: a case report and pathogenetic hypothesis

Cristina Barceló-López1, Antonio L López-Guerrero1, Antonio García-López1

  • 1Regional Service of Neurosurgery, Virgen de la Arrixaca University Hospital, 30120, Murcia, Spain.

Child'S Nervous System : Chns : Official Journal of the International Society for Pediatric Neurosurgery
|April 15, 2018
PubMed

Insights

A child with kaposiform lymphangiomatosis (KLA) developed pseudotumor cerebri, a rare condition. A ventriculoperitoneal shunt improved the patient, suggesting a link between KLA and increased intracranial pressure.

Area of Science:

  • Neurology
  • Pediatrics
  • Vascular Malformations

Background:

  • Kaposiform lymphangiomatosis (KLA) is a rare vascular tumor.
  • Pseudotumor cerebri (PTC) involves increased intracranial pressure.
  • The association between KLA and PTC is not well-established.

Observation:

  • A 4-year-old boy with KLA presented with headaches and papilledema.
  • Initial treatment with acetazolamide was ineffective.
  • Clinical deterioration necessitated a ventriculoperitoneal shunt.

Findings:

  • Neuroimaging ruled out cranial or meningeal KLA infiltration.
  • The patient's PTC was likely caused by thoracic venous compression due to KLA.
  • This case represents the first reported instance of PTC in a patient with KLA.

Implications:

  • This case highlights a potential, previously unrecognized complication of KLA.
  • It suggests a possible mechanism linking KLA to elevated intracranial venous pressure.
  • Further research is warranted to explore this association and its management.
Abstract

Keywords:
Benign intracranial hypertensionIntracranial lymphatic systemKaposiform lymphangiomatosisPseudotumor cerebriVentriculoperitoneal shunting

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