Evidence map›Paper›PMID 37510170›Full record

ReviewDiagnostics (Basel, Switzerland)2023

Copper Deficiency as Wilson's Disease Overtreatment: A Systematic Review.

Tomasz Litwin, Agnieszka Antos, Jan Bembenek, Adam Przybyłkowski, Iwona Kurkowska-Jastrzębska, Marta Skowrońska, Anna Członkowska

Open access · goldAbstract readReview
In one paragraph

Review in Diagnostics (Basel, Switzerland), 2023. 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
3.2field-weighted citation impact, top 8% 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, 20 citations in OpenAlex.

  1. Article
  2. Article
  3. Review
  4. Neuropsychiatric manifestations of Wilson's disease.Neurological sciences : official journal of the Italian Neurological Society and of the Italian Society of Clinical Neurophysiology · 2026
    Review
  5. Article
  6. Feeding-Triggered Seizures in a Newborn withJournal of clinical medicine · 2025
    Article
  7. Article
  8. Article
  9. Cuproptosis and Cu: a new paradigm in cellular death and their role in non-cancerous diseases.Apoptosis : an international journal on programmed cell death · 2024
    Review
  10. Review
  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 2 institutions in 1 country.

Tomasz LitwinSecond Department of Neurology, Institute of Psychiatry and Neurology, 02-957 Warsaw, Poland.ORCID 0000-0003-2670-9651
Agnieszka AntosSecond Department of Neurology, Institute of Psychiatry and Neurology, 02-957 Warsaw, Poland.
Jan BembenekDepartment of Clinical Neurophysiology, Institute of Psychiatry and Neurology, 02-957 Warsaw, Poland.
Adam PrzybyłkowskiDepartment of Gastroenterology and Internal Medicine, Medical University of Warsaw, 02-091 Warsaw, Poland.ORCID 0000-0002-5851-2856
Iwona Kurkowska-JastrzębskaSecond Department of Neurology, Institute of Psychiatry and Neurology, 02-957 Warsaw, Poland.
Marta SkowrońskaSecond Department of Neurology, Institute of Psychiatry and Neurology, 02-957 Warsaw, Poland.
Anna CzłonkowskaSecond Department of Neurology, Institute of Psychiatry and Neurology, 02-957 Warsaw, Poland.ORCID 0000-0002-1956-1866
Institute of Psychiatry and Neurology · PLMedical University of Warsaw · PL

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

backgroundTreatment of Wilson's disease (WD), an inherited disease characterized by copper overload, is lifelong and there is the possibility that copper deficiency (CD) may occur. We systematically reviewed the literature to describe treatment patterns, symptoms and outcomes associated with CD.

methodsUsing preferred reporting items for systematic reviews and meta-analyses (PRISMA) guidelines, the PubMed database was searched up to 6 April 2023.

resultsAcross 17 articles, 20 cases of CD were described, most commonly (15 cases) in WD patients treated with zinc salts (ZS), less often on combined chelator and ZS therapy (3 cases), molybdate salts plus ZS (1), or molybdate alone (1). CD symptoms occurred insidiously, including sideroblastic anemia, neutropenia, axonal sensory neuropathy, posterior cord myelopathy and increased ratio of epileptic seizures (or epilepsy). CD diagnosis was based on symptoms and severely reduced urinary copper excretion (<20 µg/24 h [<0.3 µmol/24 h] on ZS, or <100 µg/24 h [<1.6 µmol/24 h] on chelators) with low total serum copper and ceruloplasmin.

conclusionsAwareness of CD and regular monitoring of copper metabolism is needed during WD treatment. Temporary cessation of anti-copper treatment usually reverses serum copper reductions as well as pancytopenia; however, some symptoms, especially neuropathy and myelopathy, may persist.

Indexed as

coppermyelopathyneuropathypancytopeniaWilson’s disease

Identifiers

PMID37510170
PMCPMC10377829
OpenAlexW4385068871

What OpenQuestion holds

Textmetadata
LicenceCC BY
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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.