Evidence map›Paper›PMID 32722587›Full record

ReviewBiomolecules2020

Precision Medicine for Lysosomal Disorders.

Filippo Pinto E Vairo, Diana Rojas Málaga, Francyne Kubaski, Carolina Fischinger Moura de Souza, Fabiano de Oliveira Poswar, Guilherme Baldo, Roberto Giugliani

Open access · goldAbstract readReview
In one paragraph

Review in Biomolecules, 2020. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 13 papers.

0numbers the graph read from it
0cells of the map it votes in
13citing papers in PubMed
1.9field-weighted citation impact, top 14% 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

13 citing papers in PubMed, 24 citations in OpenAlex.

  1. Article
  2. Review
  3. Article
  4. Review
  5. Article
  6. Adult-Onset Neuronal Ceroid Lipofuscinosis:Tremor and other hyperkinetic movements (New York, N.Y.) · 2024
    Article
  7. Review
  8. Article
  9. Article
  10. Article
  11. Article
  12. Article
  13. 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

7 authors at 4 institutions in 2 countries.

Filippo Pinto E VairoCenter for Individualized Medicine, Mayo Clinic, Rochester, 55905 MN, USA.ORCID 0000-0001-6030-1903
Diana Rojas MálagaGrupo Fleury, São Paulo 04344-070, Brazil.
Francyne KubaskiMedical Genetics Service, HCPA, Porto Alegre 90035-903, Brazil.ORCID 0000-0002-9330-0154
Carolina Fischinger Moura de SouzaMedical Genetics Service, HCPA, Porto Alegre 90035-903, Brazil.
Fabiano de Oliveira PoswarMedical Genetics Service, HCPA, Porto Alegre 90035-903, Brazil.ORCID 0000-0002-0804-2876
Guilherme BaldoPostgraduate Program in Genetics and Molecular Biology, UFRGS, Porto Alegre 91501-970, Brazil.
Roberto GiuglianiMedical Genetics Service, HCPA, Porto Alegre 90035-903, Brazil.
Universidade Federal do Rio Grande do Sul · BRNational Institute on Population Medical Genetics · BRFleury S.A. (Brazil) · BRMayo Clinic · US

Funding

Conselho Nacional de Desenvolvimento Científico e Tecnológico 303219/19-0Conselho Nacional de Desenvolvimento Científico e Tecnológico 465549/2014-4Coordenação de Aperfeiçoamento de Pessoal de Nível Superior 88887.136366/2017-00FIPE/HCPA 2017-0664FIPE/HCPA 2017-0685Fundação de Amparo à Pesquisa do Estado do Rio Grande do Sul 17/2551-0000521-0
6 · The paper itself

Abstract

Precision medicine (PM) is an emerging approach for disease treatment and prevention that accounts for the individual variability in the genes, environment, and lifestyle of each person. Lysosomal diseases (LDs) are a group of genetic metabolic disorders that include approximately 70 monogenic conditions caused by a defect in lysosomal function. LDs may result from primary lysosomal enzyme deficiencies or impairments in membrane-associated proteins, lysosomal enzyme activators, or modifiers that affect lysosomal function. LDs are heterogeneous disorders, and the phenotype of the affected individual depends on the type of substrate and where it accumulates, which may be impacted by the type of genetic change and residual enzymatic activity. LDs are individually rare, with a combined incidence of approximately 1:4000 individuals. Specific therapies are already available for several LDs, and many more are in development. Early identification may enable disease course prediction and a specific intervention, which is very important for clinical outcome. Driven by advances in omics technology, PM aims to provide the most appropriate management for each patient based on the disease susceptibility or treatment response predictions for specific subgroups. In this review, we focused on the emerging diagnostic technologies that may help to optimize the management of each LD patient and the therapeutic options available, as well as in clinical developments that enable customized approaches to be selected for each subject, according to the principles of PM.

Indexed as

AnimalsEnzyme Replacement TherapyGenetic TherapyHumansLysosomal Storage DiseasesLysosomesPrecision Medicineenzyme replacement therapygene therapy.lysosomal diseasespharmacological chaperonesprecision medicine

Identifiers

PMID32722587
PMCPMC7463721
OpenAlexW3045172616

What OpenQuestion holds

Textmetadata
LicenceCC BY
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.