Evidence map›Paper›PMID 34943802›Full record

ArticleCells2021

Benefits and Toxicity of Disulfiram in Preclinical Models of Nephropathic Cystinosis.

Anna Taranta, Mohamed A Elmonem, Francesco Bellomo, Ester De Leo, Sara Boenzi, Manoe J Janssen, Amer Jamalpoor, Sara Cairoli, Anna Pastore, Cristiano De Stefanis and 10 more

Open access · goldAbstract read
In one paragraph

Article in Cells, 2021. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 8 papers.

0numbers the graph read from it
0cells of the map it votes in
8citing papers in PubMed
1.2field-weighted citation impact, top 21% 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

8 citing papers in PubMed, 11 citations in OpenAlex.

  1. Long-term outcomes in nephropathic cystinosis: a review.Pediatric nephrology (Berlin, Germany) · 2026
    Review
  2. Cystinosis and Cellular Energy Failure: Mitochondria at the Crossroads.International journal of molecular sciences · 2026
    Review
  3. Can disulfiram modulate liver injury in CCl4-induced fibrosis in rats?Gastroenterology and hepatology from bed to bench · 2025
    Article
  4. Article
  5. Review
  6. Article
  7. Article
  8. 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

20 authors at 5 institutions in 5 countries.

Anna TarantaRenal Diseases Research Unit, Genetics and Rare Diseases Research Division, Bambino Gesù Children's Hospital, IRCCS, 00165 Rome, Italy.
Mohamed A ElmonemDepartment of Clinical and Chemical Pathology, Faculty of Medicine, Cairo University, Cairo 11956, Egypt.ORCID 0000-0002-3154-1948
Francesco BellomoRenal Diseases Research Unit, Genetics and Rare Diseases Research Division, Bambino Gesù Children's Hospital, IRCCS, 00165 Rome, Italy.ORCID 0000-0003-2912-8764
Ester De LeoRenal Diseases Research Unit, Genetics and Rare Diseases Research Division, Bambino Gesù Children's Hospital, IRCCS, 00165 Rome, Italy.
Sara BoenziLaboratory of Metabolic Biochemistry Unit, Department of Pediatric Medicine, Bambino Gesù Children's Hospital, IRCCS, 00165 Rome, Italy.
Manoe J JanssenDivision of Pharmacology, Utrecht Institute for Pharmaceutical Sciences, Utrecht University, 3584 CG Utrecht, The Netherlands.
Amer JamalpoorDivision of Pharmacology, Utrecht Institute for Pharmaceutical Sciences, Utrecht University, 3584 CG Utrecht, The Netherlands.
Sara CairoliLaboratory of Metabolic Biochemistry Unit, Department of Pediatric Medicine, Bambino Gesù Children's Hospital, IRCCS, 00165 Rome, Italy.
Anna PastoreGenetics and Rare Diseases Research Division, Bambino Gesù Children's Hospital, IRCCS, 00165 Rome, Italy.ORCID 0000-0002-0990-9419
Cristiano De StefanisHistology-Core Facility, Bambino Gesù Children's Hospital, IRCCS, 00165 Rome, Italy.
Manuela ColucciRenal Diseases Research Unit, Genetics and Rare Diseases Research Division, Bambino Gesù Children's Hospital, IRCCS, 00165 Rome, Italy.
Laura R RegaRenal Diseases Research Unit, Genetics and Rare Diseases Research Division, Bambino Gesù Children's Hospital, IRCCS, 00165 Rome, Italy.
Isabella GiovannoniDepartment of Pathology, Bambino Gesù Children's Hospital, IRCCS, 00165 Rome, Italy.ORCID 0000-0002-9980-6594
Paola FrancalanciDepartment of Pathology, Bambino Gesù Children's Hospital, IRCCS, 00165 Rome, Italy.ORCID 0000-0001-9047-2383
Lambertus P van den HeuvelLaboratory of Pediatric Nephrology, Department of Development and Regeneration, KU Leuven, 3000 Leuven, Belgium.
Carlo Dionisi-ViciLaboratory of Metabolic Biochemistry Unit, Department of Pediatric Medicine, Bambino Gesù Children's Hospital, IRCCS, 00165 Rome, Italy.
Bianca M GoffredoLaboratory of Metabolic Biochemistry Unit, Department of Pediatric Medicine, Bambino Gesù Children's Hospital, IRCCS, 00165 Rome, Italy.
Rosalinde MasereeuwDivision of Pharmacology, Utrecht Institute for Pharmaceutical Sciences, Utrecht University, 3584 CG Utrecht, The Netherlands.ORCID 0000-0002-1560-1074
Elena LevtchenkoLaboratory of Pediatric Nephrology, Department of Development and Regeneration, KU Leuven, 3000 Leuven, Belgium.ORCID 0000-0002-8352-7312
Francesco EmmaRenal Diseases Research Unit, Genetics and Rare Diseases Research Division, Bambino Gesù Children's Hospital, IRCCS, 00165 Rome, Italy.
Bambino Gesù Children's Hospital · ITUtrecht University · NLCairo University · EGKU Leuven · BERadboud University Nijmegen · NL

Funding

Cystinosis Research Foundation #CRFS-2014E-rare European Union #E-Rare-2 JCT 2014Foundation of Scientific Research Flanders #11Y5216NItalian Ministry of Health #RC2015
6 · The paper itself

Abstract

Nephropathic cystinosis is a rare disease caused by mutations of the CTNS gene that encodes for cystinosin, a lysosomal cystine/H+ symporter. The disease is characterized by early-onset chronic kidney failure and progressive development of extra-renal complications related to cystine accumulation in all tissues. At the cellular level, several alterations have been demonstrated, including enhanced apoptosis, altered autophagy, defective intracellular trafficking, and cell oxidation, among others. Current therapy with cysteamine only partially reverts some of these changes, highlighting the need to develop additional treatments. Among compounds that were identified in a previous drug-repositioning study, disulfiram (DSF) was selected for in vivo studies. The cystine depleting and anti-apoptotic properties of DSF were confirmed by secondary in vitro assays and after treating

Indexed as

Toxicity TestsAcetylcysteineAnimalsApoptosisCystineCystinosisDisease Models, AnimalDisulfidesDisulfiramEmbryo, NonmammalianHumansKidney DiseasesLarvaMiceMice, KnockoutZebrafishAcetylcysteineCystineDisulfidesDisulfiramcystinosisdisulfirammicezebrafish

Identifiers

PMID34943802
PMCPMC8699074
OpenAlexW3217028541

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.