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Prophylactic recombinant factor VIIa administration to an infant with congenital systemic juvenile xanthogranuloma
Jesus De Santiago1, Ernesto Martinez-Garcia, Jorge Giron
1Department of Anesthesiology, Children University Hospital Niño Jesus, Madrid, Spain. jdesantiago@telefonica.net
Insights
Recombinant factor VIIa (rFVIIa) effectively prevented bleeding during invasive procedures in an infant with congenital systemic juvenile xanthogranuloma. This therapy proved successful when other treatments for coagulopathy failed.
Area of Science:
- Hematology
- Pediatrics
- Oncology
Background:
- Congenital systemic juvenile xanthogranuloma (s-XG) can present with significant coagulopathy.
- Infants with s-XG may require invasive procedures like central venous access system implantation and biopsies.
- Managing coagulopathy refractory to standard treatments poses a clinical challenge.
Observation:
- A pediatric patient with s-XG exhibited impaired liver function, thrombocytopenia, and refractory coagulopathy.
- The patient was scheduled for central venous access system implantation and liver/bone marrow biopsies.
- Standard treatments including fresh-frozen plasma and platelet infusions were ineffective.
Findings:
- Recombinant factor VIIa (rFVIIa) was administered prophylactically at 80 microg/kg every 2 hours.
- rFVIIa was given pre-procedure, intra-procedure, and post-procedure (30 min before to 6 hours after).
- Only minor bleeding was observed during the invasive procedures.
Implications:
- rFVIIa can be an effective prophylactic agent for invasive procedures in pediatric patients with refractory coagulopathy.
- This case highlights a potential therapeutic option for managing bleeding risks in complex pediatric cases.
- Successful rFVIIa use may improve outcomes for children with s-XG undergoing necessary interventions.
Abstract:
We report the case of an infant affected with congenital systemic juvenile xanthogranuloma scheduled for central venous access system implantation (Port-a-Cath) and a liver and bone marrow biopsy. The patient had impaired liver function, thrombocytopenia, and coagulopathy which was refractory to daily fresh-frozen plasma and platelet infusions: 80 microg x kg(-1) dose(-1) of recombinant factor VIIa (rFVIIa) was administered i.v. every 2 h starting 30 min before the procedure and ending 6 h afterwards. Very minor bleeding was observed during the procedure. In conclusion, rFVIIa therapy was effective as prophylaxis for both invasive procedures in this patient with a coagulopathy which was refractory to other different therapies.
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