Evidence map›Paper›PMID 39512431›Full record

ArticleJIMD reports2024

Disease burden among patients with Arginase 1 deficiency and their caregivers: A multinational, cross-sectional survey.

Sara Olofsson, Sofia Löfvendahl, Julia Widén, Lena Jacobson, Peter Lindgren, Karolina M Stepien, Jean-Baptiste Arnoux, Maria Luz Couce Pico, Elisa Leão Teles, Mattias Rudebeck

Abstract read
In one paragraph

Article in JIMD reports, 2024. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 1 paper.

0numbers the graph read from it
0cells of the map it votes in
1citing papers in PubMed
–field-weighted citation impact
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

1 citing paper in PubMed.

  1. 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

10 authors.

Sara OlofssonThe Swedish Institute for Health Economics, IHE Lund Sweden.ORCID https://orcid.org/0000-0003-2609-2065
Sofia LöfvendahlThe Swedish Institute for Health Economics, IHE Lund Sweden.
Julia WidénImmedica Pharma AB Stockholm Sweden.
Lena JacobsonImmedica Pharma AB Stockholm Sweden.
Peter LindgrenThe Swedish Institute for Health Economics, IHE Lund Sweden.
Karolina M StepienSalford Royal Organization, Northern Care Alliance NHS Foundation Trust Salford UK.
Jean-Baptiste ArnouxNecker Hospital Paris France.
Maria Luz Couce PicoHospital Clinico Universitario de Santiago de Compostela, IDIS, MetabERN Santiago de Compostela Spain.
Elisa Leão TelesCentro Hospitalar Universitário de São João, MetabERN Porto Portugal.
Mattias RudebeckImmedica Pharma AB Stockholm Sweden.

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Arginase 1 deficiency (ARG1-D) is an ultrarare, metabolic disease which may cause spastic paraplegia, cognitive deficiency, seizures, and ultimately severe disability. The aim of this study was to assess disease burden in ARG1-D by performing a cross-sectional survey of patients with ARG1-D and their caregivers in four European countries (France, Portugal, Spain, and the United Kingdom). Patients were enrolled at participating clinics and data were collected using a web-based questionnaire. The findings indicate that there is a significant share of patients who experience severe cognitive and mobility impairment but also that there is a considerable variance in symptom severity among patients. Disease management was mostly in line with treatment guidelines and self-reported adherence to treatment was reported to be high among a majority although following diet restrictions was perceived as difficult. However, despite this, since a large share of patients experienced severe cognitive and mobility impairment an unmet need among this patient population is suggested. The introduction of disease-modifying therapies and early identification and diagnosis may help alleviate the disease burden associated with ARG1-D in the future.

Indexed as

ARG1‐Darginase 1 deficiencycaregiverdisease burdenpatient perspectiveurea cycle disorder

Identifiers

PMID39512431
PMCPMC11540579

What OpenQuestion holds

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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.