Evidence map›Paper›PMID 28510034›Full record

ArticleJIMD reports2018

Improvement of Fabry Disease-Related Gastrointestinal Symptoms in a Significant Proportion of Female Patients Treated with Agalsidase Beta: Data from the Fabry Registry.

William R Wilcox, Ulla Feldt-Rasmussen, Ana Maria Martins, Alberto Ortiz, Roberta M Lemay, Ana Jovanovic, Dominique P Germain, Carmen Varas, Katherine Nicholls, Frank Weidemann and 1 more

Registry-linked trialAbstract read
In one paragraph

Article in JIMD reports, 2018. The graph could read no effect estimate from its abstract, so it casts no vote on the map. It is linked to trial NCT00196742 (Fabry Disease Registry Protocol), which is not on this map. Cited by 14 papers, 1 of them a synthesis that pooled it.

0numbers the graph read from it
0cells of the map it votes in
14citing papers in PubMed, 1 pooled it
9.3field-weighted citation impact, top 2% 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.

NCT00196742 recruitingnot on this map

Fabry Disease Registry Protocol

Typeobservational_patient_registrySponsorGenzyme, a Sanofi CompanyRan2001 to 2034Enrolled9,000ConditionsFabry Disease
3 · Its place in the literature

Who cites it

14 citing papers in PubMed, 1 synthesis or guideline pooled it, 28 citations in OpenAlex.

  1. Pooled it
  2. Article
  3. Review
  4. Review
  5. Review
  6. Article
  7. Review
  8. Gastrointestinal Involvement in Anderson-Fabry Disease: A Narrative Review.International journal of environmental research and public health · 2021
    Review
  9. Article
  10. Article
  11. Article
  12. Article
  13. Review
  14. 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

11 authors at 9 institutions in 8 countries.

William R WilcoxDepartment of Human Genetics, Emory University School of Medicine, 615 Michael Street, Room 305H, Atlanta, GA, 30322, USA. william.wilcox@emory.edu.
Ulla Feldt-RasmussenDepartment of Medical Endocrinology, Rigshospitalet, Copenhagen University Hospital, Copenhagen, Denmark.
Ana Maria MartinsReference Center for Inborn Errors of Metabolism, Federal University of São Paulo, São Paulo, Brazil.
Alberto OrtizUnidad de Dialisis, IIS-Fundacion Jimenez Diaz, Universidad Autonoma de Madrid, Madrid, Spain.
Roberta M LemayStrategic Epidemiology and Biostatistics, Rare Diseases, Sanofi Genzyme, Cambridge, MA, USA.
Ana JovanovicMark Holland Metabolic Unit, Salford Royal NHS Foundation Trust, Salford, UK.
Dominique P GermainDivision of Medical Genetics, University of Versailles - St Quentin en Yvelines, Paris-Saclay University, Montigny, France.
Carmen VarasFabry Disease Multidisciplinary Team, Departamento Dermatología y ETS, Hospital San Pablo de Coquimbo, Coquimbo, Chile.
Katherine NichollsDepartment of Nephrology, Royal Melbourne Hospital, Parkville, VIC, Australia.
Frank WeidemannDepartment of Internal Medicine II, Katharinen-Hospital Unna, Unna, Germany.
Robert J HopkinDivision of Human Genetics, Cincinnati Children's Hospital Medical Center, Cincinnati, OH, USA.
Cincinnati Children's Hospital Medical Center · USEmory University · USKatharinenhospital · DERigshospitalet · DKSalford Royal NHS Foundation Trust · GBThe Royal Melbourne Hospital · AUUniversidad Autónoma de Madrid · ESUniversidade Federal de São Paulo · BRUniversité Paris-Saclay · FR

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Fabry disease, an X-linked inherited lysosomal storage disorder, is caused by mutations in the gene encoding α-galactosidase, GLA. In patients with Fabry disease, glycosphingolipids accumulate in various cell types, triggering a range of cellular and tissue responses that result in a wide spectrum of organ involvement. Although variable, gastrointestinal symptoms are among the most common and significant early clinical manifestations; they tend to persist into adulthood if left untreated. To further understand the effects of sustained enzyme replacement therapy (ERT) with agalsidase beta on gastrointestinal symptoms in heterozygotes, a data analysis of female patients enrolled in the Fabry Registry was conducted. To be included, females of any age must have received agalsidase beta (average dose 1.0 mg/kg every 2 weeks) for at least 2.5 years. Measured outcomes were self-reported gastrointestinal symptoms (abdominal pain, diarrhea). Outcomes at baseline and last follow-up, and their change from baseline to last follow-up, were assessed. Relevant data were available for 168 female patients. Mean age at the start of ERT was 43 years and mean treatment duration 5.7 years. Baseline pre-treatment abdominal pain was reported by 45% of females and diarrhea by 39%. At last follow-up, 31% reported abdominal pain (p < 0.01) and 27% diarrhea (p < 0.01). The results of this Fabry Registry analysis suggest that while on sustained treatment with agalsidase beta (1.0 mg/kg every 2 weeks), both abdominal pain and diarrhea improved in many female patients with Fabry disease.

Indexed as

Abdominal painAgalsidase betaDiarrheaEnzyme replacement therapyFabry diseaseFabry registryGastrointestinal symptoms

Identifiers

PMID28510034
PMCPMC5874211
OpenAlexW2614337540

What OpenQuestion holds

Textmetadata
Read underepoch 390

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.