Patient-perceived progression in multiple system atrophy: natural history of quality of life
| dc.rights.license | open | en_US |
| hal.structure.identifier | Bordeaux population health [BPH] | |
| dc.contributor.author | SAULNIER, Tiphaine
ORCID: 0000-0001-8551-4200 | |
| dc.contributor.author | FABBRI, Margherita | |
| hal.structure.identifier | Bordeaux population health [BPH] | |
| dc.contributor.author | LE GOFF, Melanie
ORCID: 0000-0003-2848-6287 | |
| hal.structure.identifier | Bordeaux population health [BPH] | |
| dc.contributor.author | HELMER, Catherine | |
| dc.contributor.author | PAVY-LE TRAON, Anne | |
| hal.structure.identifier | Institut des Maladies Neurodégénératives [Bordeaux] [IMN] | |
| dc.contributor.author | MEISSNER, Wassilios
IDREF: 113664761 | |
| dc.contributor.author | RASCOL, Olivier | |
| hal.structure.identifier | Bordeaux population health [BPH] | |
| dc.contributor.author | PROUST LIMA, Cecile | |
| hal.structure.identifier | Institut des Maladies Neurodégénératives [Bordeaux] [IMN] | |
| hal.structure.identifier | Bordeaux population health [BPH] | |
| dc.contributor.author | SAMIER FOUBERT, Alexandra | |
| dc.date.accessioned | 2024-05-07T07:31:57Z | |
| dc.date.available | 2024-05-07T07:31:57Z | |
| dc.date.issued | 2024-03-07 | |
| dc.identifier.issn | 0022-3050 | en_US |
| dc.identifier.uri | https://oskar-bordeaux.fr/handle/20.500.12278/199686 | |
| dc.description.abstractEn | BACKGROUND: Health-related quality of life (Hr-QoL) scales provide crucial information on neurodegenerative disease progression, help improve patient care and constitute a meaningful endpoint for therapeutic research. However, Hr-QoL progression is usually poorly documented, as for multiple system atrophy (MSA), a rare and rapidly progressing alpha-synucleinopathy. This work aimed to describe Hr-QoL progression during the natural course of MSA, explore disparities between patients and identify informative items using a four-step statistical strategy. METHODS: We leveraged the data of the French MSA cohort comprising annual assessments with the MSA-QoL questionnaire for more than 500 patients over up to 11 years. A four-step strategy (1) determined the subdimensions of Hr-QoL, (2) modelled the subdimension trajectories over time, (3) mapped item impairments with disease stages and (4) identified most informative items. RESULTS: Four dimensions were identified. In addition to the original motor, non-motor and emotional domains, an oropharyngeal component was highlighted. While the motor and oropharyngeal domains deteriorated rapidly, the non-motor and emotional aspects were already impaired at cohort entry and deteriorated slowly over the disease course. Impairments were associated with sex, diagnosis subtype and delay since symptom onset. Except for the emotional domain, each dimension was driven by key identified items. CONCLUSION: The multidimensional Hr-QoL deteriorates progressively over the course of MSA and brings essential knowledge for improving patient care. As exemplified with MSA, the thorough description of Hr-QoL over time using the four-step strategy can provide perspectives on neurodegenerative diseases' management to ultimately deliver better support focused on the patient's perspective. | |
| dc.description.sponsorship | Modèles Dynamiques pour les Etudes Epidémiologiques Longitudinales sur les Maladies Chroniques - ANR-18-CE36-0004 | en_US |
| dc.language.iso | EN | en_US |
| dc.rights | Attribution 3.0 United States | * |
| dc.rights.uri | http://creativecommons.org/licenses/by/3.0/us/ | * |
| dc.subject.en | Multisystem Atrophy | |
| dc.subject.en | Neuropathy | |
| dc.subject.en | Quality Of Life | |
| dc.subject.en | Scales | |
| dc.title.en | Patient-perceived progression in multiple system atrophy: natural history of quality of life | |
| dc.title.alternative | J Neurol Neurosurg Psychiatry | en_US |
| dc.type | Article de revue | en_US |
| dc.identifier.doi | 10.1136/jnnp-2023-332733 | en_US |
| dc.subject.hal | Sciences du Vivant [q-bio]/Santé publique et épidémiologie | en_US |
| dc.identifier.pubmed | 38453477 | en_US |
| bordeaux.journal | Journal of Neurology, Neurosurgery and Psychiatry | en_US |
| bordeaux.hal.laboratories | Bordeaux Population Health Research Center (BPH) - UMR 1219 | en_US |
| bordeaux.institution | Université de Bordeaux | en_US |
| bordeaux.institution | INSERM | en_US |
| bordeaux.institution | CNRS | |
| bordeaux.team | ACTIVE_BPH | en_US |
| bordeaux.team | LEHA_BPH | en_US |
| bordeaux.team | BIOSTAT_BPH | en_US |
| bordeaux.peerReviewed | oui | en_US |
| bordeaux.inpress | non | en_US |
| bordeaux.identifier.funderID | Conseil Régional Aquitaine | en_US |
| hal.popular | non | en_US |
| hal.audience | Internationale | en_US |
| dc.rights.cc | Pas de Licence CC | en_US |
| bordeaux.COinS | ctx_ver=Z39.88-2004&rft_val_fmt=info:ofi/fmt:kev:mtx:journal&rft.jtitle=Journal%20of%20Neurology,%20Neurosurgery%20and%20Psychiatry&rft.date=2024-03-07&rft.eissn=0022-3050&rft.issn=0022-3050&rft.au=SAULNIER,%20Tiphaine&FABBRI,%20Margherita&LE%20GOFF,%20Melanie&HELMER,%20Catherine&PAVY-LE%20TRAON,%20Anne&rft.genre=article |
