Bone marrow involvement in Gaucher disease at MRI : what long-term evolution can we expect under enzyme replacement therapy?

Eur Radiol. 2015 Oct;25(10):2969-75. doi: 10.1007/s00330-015-3715-2. Epub 2015 Apr 16.

Abstract

Purpose: To study the long-term evolution of the bone marrow burden (BMB) score at MRI in patients with Gaucher disease (GD) under enzyme replacement therapy (ERT).

Material and methods: Forty patients treated for GD were retrospectively studied in a referral centre. BMB scores were assessed on spine and femur MR examinations performed between January 2003 and June 2014. The long-term evolution of the BMB scores was analyzed using a linear mixed model.

Results: A total of 121 MRI examinations were performed during the study period with a mean follow-up of 7.1 years ± 5.6, an average rate of 3.1 MR examinations ± 1.7 per patient and an interval of 2.3 years ± 1.1 between examinations. Patients had received ERT during 12 years on average ± 6.7. The trend of BMB scores with time decreased significantly by 15% (P = 0.008) during the total study period and 39% (P = 0.01) during the first 5 years of treatment. No changes in BMB scores were observed after five years of treatment.

Conclusion: In Gaucher patients, the trend of MRI BMB scores with time decreased significantly under ERT the first 5 years of treatment before a long-term stabilization.

Key points: • Bone marrow infiltration of Gaucher patients responds to enzyme replacement therapy • MRI BMB score decreases mainly during the first five years of treatment • MRI BMB score tends to stabilize after five years of treatment • MR examinations could be limited after five years of treatment.

MeSH terms

  • Adolescent
  • Adult
  • Aged
  • Bone Marrow Diseases / etiology*
  • Bone Marrow Diseases / pathology
  • Child
  • Child, Preschool
  • Enzyme Replacement Therapy / methods*
  • Female
  • Femur
  • Gaucher Disease / complications*
  • Gaucher Disease / drug therapy
  • Gaucher Disease / pathology
  • Glucosylceramidase / therapeutic use
  • Humans
  • Infant
  • Magnetic Resonance Imaging
  • Male
  • Middle Aged
  • Physical Examination / methods
  • Referral and Consultation
  • Retrospective Studies
  • Spine
  • Young Adult

Substances

  • Glucosylceramidase