Evidence map›Paper›PMID 27722092›Full record

ArticleMolecular genetics and metabolism reports2016

Stability is maintained in adults with Gaucher disease type 1 switched from velaglucerase alfa to eliglustat or imiglucerase: A sub-analysis of the eliglustat ENCORE trial.

Rebecca Pleat, Timothy M Cox, T Andrew Burrow, Pilar Giraldo, Ozlem Goker-Alpan, Barry E Rosenbloom, Laura R Croal, Lisa H Underhill, Sebastiaan J M Gaemers, M Judith Peterschmitt

Registry-linked trialOpen access · goldAbstract read
In one paragraph

Article in Molecular genetics and metabolism reports, 2016. The graph could read no effect estimate from its abstract, so it casts no vote on the map. It is linked to trial NCT00943111 (A Phase 3, Randomized, Multi-Center, Multi-National, Open-Label, Active Comparator Study to Evaluate the Efficacy and Safety of Genz-112638 in Patients With Gaucher Disease Type 1 Who Have Reached Therapeutic Goals With Enzyme Replacement Therapy), which is not on this map. Cited by 7 papers, 1 of them a synthesis that pooled it.

0numbers the graph read from it
0cells of the map it votes in
7citing papers in PubMed, 1 pooled it
1.2field-weighted citation impact, top 20% of its field
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

NCT00943111 phase3completednot on this map

A Phase 3, Randomized, Multi-Center, Multi-National, Open-Label, Active Comparator Study to Evaluate the Efficacy and Safety of Genz-112638 in Patients With Gaucher Disease Type 1 Who Have Reached Therapeutic Goals With Enzyme Replacement Therapy (ENCORE)

TypeinterventionalSponsorGenzyme, a Sanofi CompanyRan2009 to 2015Enrolled160ConditionsGaucher Disease, Type 1ArmsEliglustat tartrate, Imiglucerase
3 · Its place in the literature

Who cites it

7 citing papers in PubMed, 1 synthesis or guideline pooled it, 18 citations in OpenAlex.

  1. Hematopoietic stem cell transplantation for Gaucher disease.The Cochrane database of systematic reviews · 2017
    Pooled it
  2. Article
  3. Glucosylsphingosine (Lyso-GbInternational journal of molecular sciences · 2022
    Article
  4. Article
  5. Review
  6. Article
  7. Review
4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

10 authors at 5 institutions in 3 countries.

Rebecca PleatSanofi Genzyme, Cambridge, MA, USA.
Timothy M CoxDepartment of Medicine, University of Cambridge, Addenbrooke's Hospital, Cambridge, UK.
T Andrew BurrowCincinnati Children's Hospital Medical Center, Cincinnati, OH, USA.
Pilar GiraldoCentro de Investigación Biomédica en Red de Enfermedades Raras (CIBERER), IISAragón, Zaragoza, Spain.
Ozlem Goker-AlpanLysosomal Disorders Research and Treatment Unit, Center for Clinical Trials, O&O Alpan, LLC, Fairfax, VA, USA.
Barry E RosenbloomCedars-Sinai/Tower Hematology Oncology, Beverly Hills, CA, USA.
Laura R CroalSanofi Genzyme, Cambridge, MA, USA.
Lisa H UnderhillSanofi Genzyme, Cambridge, MA, USA.
Sebastiaan J M GaemersSanofi Genzyme, Cambridge, MA, USA.
M Judith PeterschmittSanofi Genzyme, Cambridge, MA, USA.
Sanofi (United States) · USAddenbrooke's Hospital · GBCincinnati Children's Hospital Medical Center · USInstituto de Investigación Sanitaria Aragón · ESLysosomal and Rare Disorders Research and Treatment Center · US

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Gaucher disease type 1 is an autosomal recessive disorder caused by deficient activity of the lysosomal enzyme acid β-glucosidase resulting in accumulation of glucosylceramide and clinical manifestations of anemia, thrombocytopenia, hepatosplenomegaly, and skeletal disease. The historic standard of care is intravenous recombinant enzyme therapy with imiglucerase. Eliglustat, an oral substrate reduction therapy, is a first-line treatment for adults with Gaucher disease type 1 who have a compatible CYP2D6-metabolizer phenotype (≈ 95% of patients). The 12-month ENCORE trial (NCT00943111) found eliglustat non-inferior to imiglucerase in maintaining stability in adult Gaucher patients previously stabilized after ≥ 3 years of enzyme therapy (imiglucerase or velaglucerase alfa). This post-hoc analysis examined safety and efficacy in the 30 ENCORE patients who were receiving velaglucerase alfa at study entry and were randomized to eliglustat (n = 22) or imiglucerase (n = 8). Efficacy and safety in velaglucerase alfa-transitioned patients were consistent with the full ENCORE trial population; 90% of patients switched to eliglustat and 88% of patients switched to imiglucerase met the composite endpoint (stable hemoglobin concentration, platelet count, spleen volume, and liver volume). Clinical stability was maintained for 12 months in Gaucher disease type 1 patients in the ENCORE trial who switched from velaglucerase alfa to either eliglustat or imiglucerase.

Indexed as

Composite endpointEliglustatGaucher disease type 1ImigluceraseNon-inferiority trialVelaglucerase alfa

Identifiers

PMID27722092
PMCPMC5050260
OpenAlexW2526502055

What OpenQuestion holds

Textmetadata
LicenceCC BY-NC-ND
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Registered trials

Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.