Evidence map›Paper›PMID 40646614›Full record

ArticleOrphanet journal of rare diseases2025

Determinants of fatigue in patients with Marfan syndrome: a study using PROMS.

Ines Cavalier, Leslie Toko Kamga, Laurence Morin, Laurence Bal, Laurence Faivre, Guillaume Jondeau, Viet-Thi Tran, Olivier Milleron

Abstract readLetter
In one paragraph

Article in Orphanet journal of rare diseases, 2025. 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

8 authors.

Ines CavalierDepartment of Epidemiology, Biostatistics and Clinical Research, Hôpital Bichat- Claude Bernard, INSERM CIC-EC 1425, AP-HP.Nord-Université Paris-Cité, Paris, France.
Leslie Toko KamgaCentre d'Épidémiologie Clinique, Hôpital Hôtel-Dieu, Assistance Publique-Hôpitaux de Paris (AP-HP), Paris, France.
Laurence MorinAssociation Marfans, 121 rue de la Convention, Paris, 75018, France.
Laurence BalCRMR Marfan et Apparentés, Centre Aorte Timone, CHU Timone, Aix-Marseille Université , 264 Rue Saint-Pierre , Marseille Cedex 5, 13385, France.
Laurence FaivreCentre de Génétique et Centre de Référence Anomalies du Développement et Syndromes Malformatifs FHU TRANSLAD, INSERM UMR1231 Hôpital d'Enfants CHU Dijon, 14 Rue Paul Gaffarel, Dijon, 21000, France.
Guillaume JondeauCentre national de référence pour le syndrome de Marfan et pathologies apparentés Assistance Publique Hôpitaux de Paris, Service de Cardiologie, Hôpital Bichat Paris, 264 Rue Saint-Pierre, Paris, France.
Viet-Thi TranCentre d'Épidémiologie Clinique, Hôpital Hôtel-Dieu, Assistance Publique-Hôpitaux de Paris (AP-HP), Paris, France.
Olivier MilleronCentre national de référence pour le syndrome de Marfan et pathologies apparentés Assistance Publique Hôpitaux de Paris, Service de Cardiologie, Hôpital Bichat Paris, 264 Rue Saint-Pierre, Paris, France. olivier.milleron@aphp.fr.ORCID http://orcid.org/0000-0002-6517-0559

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

backgroundFatigue is often reported by individuals with Marfan syndrome (MFS). However, the determinants of fatigue and its impact on the daily lives of patients with MFS remain poorly understood. We sought to assess the level of fatigue and its determinants in individuals with MFS.

methodsWe conducted a cross-sectional study in ComPaRe Marfan, an e-cohort of MFS patients. Fatigue was assessed using the FACIT questionnaire already used in a wide range of chronic diseases. Pain and its interference with daily life were assessed using the Brief Pain Inventory. We performed univariate and multivariable linear regressions to identify determinants of fatigue.

resultsA total of 162 people with MFS completed the FACIT-Fatigue questionnaire. The median age was 46 and 59% were women. The median FACIT-Fatigue score was 31(IQR 22-39) and over ¾ of the cohort had a FACIT-Fatigue score below 40 corresponding to at least some level of fatigue. In the multivariate model, only pain interference was associated with fatigue (Coeff= -0.34, CI95%: [-0.46; -0.22], p < 0.001). Beta-blocker treatment, history of aortic, lens or scoliosis surgery were not associated with FACIT fatigue score. In addition, the FACIT fatigue scores was repeated every month during 3 months and were stable over time.

conclusionFatigue is common in patients with MFS and is associated with pain that interferes with daily life. Therefore, pain management in MFS patients could improve quality of life and fatigue.

Indexed as

FatigueMarfan SyndromeAdultCross-Sectional StudiesFemaleHumansMaleMiddle AgedPainQuality of LifeSurveys and Questionnaires

Identifiers

PMID40646614
PMCPMC12255052

What OpenQuestion holds

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LicenceCC BY
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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.