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Cerebral vein thrombosis in a four year old with Behçet's disease

Duygu Ovunc Hacihamdioglu1, Murat Demiriz2, Gungor Sobaci3

  • 1Gulhane Military Medical Academy, Medicine School, Department of Pediatrics, Division of Pediatric Nephrology and Rheumatology, Turkey.

Reumatologia Clinica
|January 7, 2014
PubMed

Insights

This study presents the youngest case of Behçet's disease (BD) with sagittal sinus thrombosis in a four-year-old child. Infliximab therapy showed promise for treating this rare pediatric neurological complication.

Area of Science:

  • Vascular Medicine
  • Pediatric Neurology
  • Rheumatology

Background:

  • Behçet's disease (BD) is a multisystemic inflammatory disorder characterized by vasculitis affecting various blood vessels.
  • Central nervous system (CNS) involvement occurs in 5-10% of BD patients, with neurological symptoms typically appearing after 3-6 years of disease onset.
  • Pediatric BD, though rare, can lead to increased morbidity and mortality, with a mean age of onset around 7 years.

Observation:

  • A four-year-old Turkish boy, the youngest reported case, presented with Behçet's disease and sagittal sinus thrombosis.
  • The patient exhibited papilledema but no overt neurological signs at presentation.
  • This case highlights an unusual early presentation of neurological involvement in pediatric BD.

Findings:

  • Treatment with infliximab was initiated for the sagittal sinus thrombosis in this pediatric BD patient.
  • Initial response to infliximab suggests potential efficacy in managing this severe complication.
  • The patient's presentation represents the earliest documented instance of sagittal sinus thrombosis in childhood Behçet's disease.

Implications:

  • Infliximab may represent a viable therapeutic option for refractory sagittal sinus thrombosis in pediatric Behçet's disease.
  • This case expands the understanding of the clinical spectrum and early manifestations of CNS involvement in childhood BD.
  • Further long-term studies are warranted to confirm the sustained efficacy and safety of infliximab in this population.

Keywords:
Behçet's diseaseChildrenEnfermedad de Behçet'sInfliximabNiñosThrombosisTrombosis

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