Evidence map›Paper›PMID 28653488›Full record

ArticleBritish journal of clinical pharmacology2017

Drugs for rare disorders.

Serge Cremers, Jeffrey K Aronson

Open access · bronzeAbstract readEditorial
In one paragraph

Article in British journal of clinical pharmacology, 2017. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 14 papers.

0numbers the graph read from it
0cells of the map it votes in
14citing papers in PubMed
2.4field-weighted citation impact, top 11% 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.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

14 citing papers in PubMed, 22 citations in OpenAlex.

  1. Review
  2. Article
  3. Review
  4. Article
  5. Article
  6. Profile of Drug Utilization in Patients with Rare Diseases in Tuscany, Italy: A Population-Based Study.International journal of environmental research and public health · 2023
    Article
  7. Article
  8. Article
  9. Article
  10. Review
  11. Article
  12. Pharmacometrics and systems pharmacology for metabolic bone diseases.British journal of clinical pharmacology · 2019
    Review
  13. Article
  14. Drugs for rare disorders.British journal of clinical pharmacology · 2017
    Article
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

2 authors at 2 institutions in 2 countries.

Serge CremersDepartments of Pathology & Cell Biology and Medicine, and Irving Institute for Clinical and Translational Research, Columbia University Medical Center, New York, NY, 10027, USA.
Jeffrey K AronsonCentre for Evidence Based Medicine, Nuffield Department of Primary Care Health Sciences, Radcliffe Infirmary, Woodstock Road, Oxford,, OX2 6GG, UK.ORCID 0000-0002-6800-5532
Columbia University Irving Medical Center · USUniversity of Oxford · GB

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Estimates of the frequencies of rare disorders vary from country to country; the global average defined prevalence is 40 per 100 000 (0.04%). Some occur in only one or a few patients. However, collectively rare disorders are fairly common, affecting 6-8% of the US population, or about 30 million people, and a similar number in the European Union. Most of them affect children and most are genetically determined. Diagnosis can be difficult, partly because of variable presentations and partly because few clinicians have experience of individual rare disorders, although they may be assisted by searching databases. Relatively few rare disorders have specific pharmacological treatments (so-called orphan drugs), partly because of difficulties in designing trials large enough to determine benefits and harms alike. Incentives have been introduced to encourage the development of orphan drugs, including tax credits and research aids, simplification of marketing authorization procedures and exemption from fees, and extended market exclusivity. Consequently, the number of applications for orphan drugs has grown, as have the costs of using them, so much so that treatments may not be cost-effective. It has therefore been suggested that not-for-profit organizations that are socially motivated to reduce those costs should be tasked with producing them. A growing role for patient organizations, improved clinical and translational infrastructures, and developments in genetics have also contributed to successful drug development. The translational discipline of clinical pharmacology is an essential component in drug development, including orphan drugs. Clinical pharmacologists, skilled in basic pharmacology and its links to clinical medicine, can be involved at all stages. They can contribute to the delineation of genetic factors that determine clinical outcomes of pharmacological interventions, develop biomarkers, design and perform clinical trials, assist regulatory decision making, and conduct postmarketing surveillance and pharmacoepidemiological and pharmacoeconomic assessments.

Indexed as

Drug ApprovalChildCost-Benefit AnalysisDrug DiscoveryEuropean UnionHumansJapanLegislation, DrugMotivationOrphan Drug ProductionPrevalenceRare DiseasesTranslational Research, BiomedicalUnited Statesclinical pharmacologydrug developmentorphan drugsorphan productsrare diseasesrare disorders

Identifiers

PMID28653488
PMCPMC5510061
OpenAlexW2724249652

What OpenQuestion holds

Textmetadata
Read underepoch 390

Registered trials

None linked

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.