Splenic nodules in paediatric Gaucher disease treated by enzyme replacement therapy

Pediatr Radiol. 2008 Jun;38(6):657-60. doi: 10.1007/s00247-008-0811-3. Epub 2008 Apr 1.

Abstract

Background: The natural history of focal splenic lesions in paediatric Gaucher disease (GD) is unknown and these lesions are thought to persist despite enzyme replacement therapy (ERT).

Objective: To assess the prevalence, evolution and resolution of splenic nodules in a cohort of paediatric Gaucher patients treated with ERT.

Materials and methods: The US findings in 37 children with GD were retrospectively reviewed. A total of 28 children underwent serial abdominal US examinations as part of their initial assessment and during routine follow-up. All patients received ERT.

Results: Six children (21%) had splenic nodules on US examination, either at presentation or on follow-up examination. In all six patients, the nodules had resolved on follow-up imaging, with resolution taking 17 months to 4 years 8 months. Two sets of siblings developed nodules that resolved over a similar time period.

Conclusion: Disappearance of focal splenic lesions in children with GD during follow-up has not been previously reported. The development of new focal splenic lesions in children with GD whilst on ERT has not been previously documented.

MeSH terms

  • 1-Deoxynojirimycin / administration & dosage
  • 1-Deoxynojirimycin / analogs & derivatives*
  • Child
  • Child, Preschool
  • Cohort Studies
  • Enzyme Inhibitors / administration & dosage
  • Female
  • Follow-Up Studies
  • Gaucher Disease / complications
  • Gaucher Disease / drug therapy*
  • Glucosylceramidase / administration & dosage*
  • Humans
  • Infant
  • Male
  • Retrospective Studies
  • Spleen / diagnostic imaging*
  • Spleen / pathology
  • Splenic Diseases / diagnosis*
  • Splenic Diseases / etiology
  • Splenomegaly / etiology
  • Splenomegaly / pathology
  • Ultrasonography

Substances

  • Enzyme Inhibitors
  • 1-Deoxynojirimycin
  • miglustat
  • Glucosylceramidase
  • imiglucerase