Evaluation of treatment response to enzyme replacement therapy with Velaglucerase alfa in patients with Gaucher disease using whole-body magnetic resonance imaging

Blood Cells Mol Dis. 2016 Mar:57:35-41. doi: 10.1016/j.bcmd.2015.11.003. Epub 2015 Nov 27.

Abstract

Objective: This was a retrospective data analysis to evaluate the treatment response to enzyme replacement therapy (ERT) with Velaglucerase alfa using whole-body magnetic resonance imaging (MRI).

Materials and methods: A baseline and follow-up MRI were performed on 18 Gaucher Type 1 patients at an interval of 11.6 months. The MRI score systems determined the Bone-Marrow-Burden (BMB) score, the Düsseldorf-Gaucher score (DGS), and the Vertebra-Disc-Ratio (VDR). The Severity Score Index Type 1 (GD-DS3) was also assessed.

Results: The baseline MRI medians were: BMB, 7.00; DGS, 3.00; and VDR: 1.70; while, the follow-up MRI medians were: BMB, 7.00; DGS, 3.00; and VDR: 1.73. The baseline GD-DS3 median was 2.40 (BMB excl.: 0.50) and the follow-up median was 2.00 (BMB excl.: 0.50). There was weak statistical significance with the Wilcoxon signed-rank test for the DGS (p=0.034) and GD-DS3 (p=0.047) between both MRIs.

Conclusion: Velaglucerase alfa therapy is a effective long-term treatment for Gaucher Type 1 patients who are newly diagnosed or switching therapies. Measurements with whole-body MRI and an objective scoring system were reliable tools for detecting early stage bone marrow activity. Further research is needed to evaluate the "Booster-Effect" of Velaglucerase alfa therapy in Gaucher skeletal disease.

Keywords: Gaucher disease; Score systems; Velaglucerase alfa; Whole-body MRI.

MeSH terms

  • Adult
  • Aged
  • Bone Marrow / drug effects
  • Bone Marrow / pathology
  • Enzyme Replacement Therapy / methods*
  • Female
  • Follow-Up Studies
  • Gaucher Disease / diagnosis*
  • Gaucher Disease / drug therapy*
  • Gaucher Disease / pathology
  • Glucosylceramidase / therapeutic use*
  • Humans
  • Magnetic Resonance Imaging / methods*
  • Male
  • Middle Aged
  • Platelet Count
  • Recombinant Proteins / therapeutic use
  • Retrospective Studies
  • Severity of Illness Index
  • Treatment Outcome
  • Whole Body Imaging / methods

Substances

  • Recombinant Proteins
  • Glucosylceramidase
  • Velaglucerase alfa, human