Evidence map›Paper›PMID 26067812›Full record

ArticleJournal of inherited metabolic disease2016

Concomitant immune-related events in Wilson disease: implications for monitoring chelator therapy.

Jessica Seessle, Daniel Nils Gotthardt, Mark Schäfer, Annina Gohdes, Jan Pfeiffenberger, Peter Ferenci, Wolfgang Stremmel, Karl Heinz Weiss

Abstract read
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In one paragraph

Article in Journal of inherited metabolic disease, 2016. 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
1.5field-weighted citation impact, top 20% 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, 20 citations in OpenAlex.

  1. Review
  2. Article
  3. Article
  4. Autoantibodies: are they a clue for liver diseases?Clinical and experimental hepatology · 2022
    Article
  5. Article
  6. The Role of Zinc in the Treatment of Wilson's Disease.International journal of molecular sciences · 2022
    Review
  7. Clinical management of Wilson disease.Annals of translational medicine · 2019
    Review
  8. Advances in Treatment of Wilson Disease.Tremor and other hyperkinetic movements (New York, N.Y.) · 2018
    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

8 authors at 3 institutions in 2 countries.

Jessica SeessleDepartment of Gastroenterology, University Hospital Heidelberg, Im Neuenheimer Feld 410, D-69120, Heidelberg, Germany.
Daniel Nils GotthardtDepartment of Gastroenterology, University Hospital Heidelberg, Im Neuenheimer Feld 410, D-69120, Heidelberg, Germany.
Mark SchäferDepartment of Gastroenterology, University Hospital Heidelberg, Im Neuenheimer Feld 410, D-69120, Heidelberg, Germany.
Annina GohdesDepartment of Gastroenterology, University Hospital Heidelberg, Im Neuenheimer Feld 410, D-69120, Heidelberg, Germany.
Jan PfeiffenbergerDepartment of Gastroenterology, University Hospital Heidelberg, Im Neuenheimer Feld 410, D-69120, Heidelberg, Germany.
Peter FerenciDepartment of Gastroenterology, University Hospital Vienna, Vienna, Austria.
Wolfgang StremmelDepartment of Gastroenterology, University Hospital Heidelberg, Im Neuenheimer Feld 410, D-69120, Heidelberg, Germany.
Karl Heinz WeissDepartment of Gastroenterology, University Hospital Heidelberg, Im Neuenheimer Feld 410, D-69120, Heidelberg, Germany. karl-heinz_weiss@med.uni-heidelberg.de.
Heidelberg University · DEUniversity Hospital Heidelberg · DEVienna General Hospital · AT

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

BACKGROUND AND

aimsCurrent guidelines favor the use of chelating agents (d-penicillamine, trientine) in first line therapy of symptomatic Wilson disease patients. Development of chelator induced immunological adverse events are a concern especially under d-penicillamine therapy. This study assessed the prevalence of co-existing or therapy-related immune-mediated diseases in Wilson disease patients, and evaluated the role of antinuclear antibodies in therapy monitoring.

methodsWe retrospectively analyzed 235 Wilson disease patients. Medical regimens were classified and analyzed in relation to adverse events and antinuclear antibody courses.

resultsCoexisting immune-mediated diseases were evident in 19/235 (8.1%) patients, of which 13/235 (5.5%) had pre-existing autoimmune diseases. Six patients (2.6%) developed an autoimmune disease under therapy, all of them under long-term d-penicillamine treatment. Data relating to antinuclear antibody courses during treatment and adverse events were available for patients treated with d-penicillamine (n = 91), trientine (n = 58), and zinc salts (n = 58). No significant increase in antinuclear antibody titers in patients treated with d-penicillamine (16/91; 17.6%), trientine (12/58; 20.7%), and zinc (7/58; 12.1%) were found.

conclusionUnder long-term d-penicillamine therapy a minority of patients developed immune-mediated disease. Elevations in antinuclear antibodies were found frequently, but no correlations were evident between increases in antinuclear antibodies and the development of immune-mediated diseases or medical regimes. Thus, the value of antinuclear antibodies for monitoring adverse events under chelator therapy seems to be limited.

Indexed as

AdolescentAdultAntibodies, AntinuclearAutoimmune DiseasesChelating AgentsChildCross-Sectional StudiesDrug MonitoringDrug-Related Side Effects and Adverse ReactionsFemaleHepatolenticular DegenerationHumansMalePenicillamineRetrospective StudiesTrientineAntibodies, AntinuclearChelating AgentsPenicillamineTrientineZinc

Identifiers

PMID26067812
OpenAlexW573755264

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Registered trials

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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.