Evidence map›Paper›PMID 39762099›Full record

Observational studyBMJ open2025

Exploring fatigue in Marfan and hypermobile Ehlers-Danlos syndromes: an analytical cross-sectional study in two Italian healthcare centres.

Nathasha Udugampolage, Jacopo Taurino, Alessandra Bassotti, Alessandro Pini, Rosario Caruso, Edward Callus, Arianna Magon, Gianluca Conte, Giada De Angeli, Giulia Paglione and 2 more

Registry-linked trialAbstract readMulticenter StudyObservational Study
In one paragraph

Observational study in BMJ open, 2025. The graph could read no effect estimate from its abstract, so it casts no vote on the map. It is linked to trial NCT05712564 (Observational Survey on Fatigue Levels in Relation to Depressive Disorders and Insomnia in Adult Patients with Marfan Syndrome and Hypermobile Ehlers-Danlos Syndrome), which is not on this map. Cited by 2 papers.

0numbers the graph read from it
0cells of the map it votes in
2citing 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.

NCT05712564 completednot on this map

Observational Survey on Fatigue Levels in Relation to Depressive Disorders and Insomnia in Adult Patients with Marfan Syndrome and Hypermobile Ehlers-Danlos Syndrome

TypeobservationalSponsorIRCCS Policlinico S. DonatoRan2023 to 2023Enrolled282ConditionsRare Diseases, Fatigue, Insomnia, DepressionArmsQuestionnaire
3 · Its place in the literature

Who cites it

2 citing papers in PubMed.

  1. Article
  2. 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

12 authors.

Nathasha UdugampolageCardiovascular Genetic Centre, IRCCS Policlinico San Donato, San Donato Milanese, Italy.
Jacopo TaurinoCardiovascular Genetic Centre, IRCCS Policlinico San Donato, San Donato Milanese, Italy.
Alessandra BassottiRegional Center of Ehlers-Danlos Syndrome, Fondazione IRCCS Ca' Granda Ospedale Maggiore Policlinico, Milan, Italy.
Alessandro PiniCardiovascular Genetic Centre, IRCCS Policlinico San Donato, San Donato Milanese, Italy.ORCID http://orcid.org/0000-0002-8325-6057
Rosario CarusoDepartment of Biomedical Sciences for Health, University of Milan, Milano, Italy rosario.caruso@unimi.it.ORCID http://orcid.org/0000-0002-7736-6209
Edward CallusDepartment of Biomedical Sciences for Health, University of Milan, Milano, Italy.
Arianna MagonHealth Professions Research and Development Unit, IRCCS Policlinico San Donato, San Donato Milanese, Italy.
Gianluca ConteHealth Professions Research and Development Unit, IRCCS Policlinico San Donato, San Donato Milanese, Italy.ORCID http://orcid.org/0000-0002-8171-8203
Giada De AngeliHealth Professions Research and Development Unit, IRCCS Policlinico San Donato, San Donato Milanese, Italy.ORCID http://orcid.org/0000-0003-2826-0181
Giulia PaglioneHealth Professions Research and Development Unit, IRCCS Policlinico San Donato, San Donato Milanese, Italy.
Irene BaroniHealth Professions Research and Development Unit, IRCCS Policlinico San Donato, San Donato Milanese, Italy.ORCID http://orcid.org/0000-0002-1326-8642
Giuliana TrifiròRegional Center of Ehlers-Danlos Syndrome, Fondazione IRCCS Ca' Granda Ospedale Maggiore Policlinico, Milan, Italy.

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

objectiveThis study investigates the prevalence and underlying factors of fatigue in individuals with Marfan syndrome (MFS) and hypermobile Ehlers-Danlos syndromes (hEDS), highlighting the necessity for focused research on this symptom within these patient populations.

designCross-sectional, multicentre study.

settingData were collected from participants diagnosed with MFS or hEDS across multiple healthcare centres.

participantsThe study enrolled 282 participants (127 with MFS and 155 with hEDS). PRIMARY AND SECONDARY OUTCOME MEASURES: Fatigue was measured using the Fatigue Severity Scale (FSS). Additional assessments included the Patient Health Questionnaire-9 (PHQ-9) for depression and the Insomnia Severity Index (ISI) for sleep disturbances.

resultsParticipants with hEDS exhibited significantly higher median fatigue scores (FSS median=5.9, IQR=5.00-6.44) compared with the MFS group (FSS median=4.0, IQR=2.88-5.00). Significant predictors of fatigue included being female, having hEDS, participating in psychotherapy, and elevated scores on depression and insomnia scales. In the overall sample, hEDS significantly predicted fatigue (B=0.430, p=0.022), with depression and insomnia as strong influencers (PHQ-9: B=0.12, p<0.001; ISI: B=0.092, p<0.001). Notably, 80% of the hEDS group reported clinically relevant fatigue levels, compared with 31.5% in the MFS group. Daily persistence of fatigue was especially pronounced in hEDS, with 72.2% reporting everyday fatigue versus 25.2% in MFS. Temporal fatigue patterns also differed, with a more evenly distributed pattern throughout the day in hEDS, correlating with higher insomnia scores.

conclusionsThe results underscore the severe impact of fatigue on individuals with hEDS compared with those with MFS, suggesting the need for targeted, multidisciplinary management strategies to enhance quality of life. TRIAL REGISTRATION NUMBER: NCT05712564.

Indexed as

Ehlers-Danlos SyndromeFatigueMarfan SyndromeAdultCross-Sectional StudiesDepressionFemaleHumansItalyMaleMiddle AgedPrevalenceSeverity of Illness IndexSleep Initiation and Maintenance DisordersSurveys and QuestionnairesYoung AdultCARDIOLOGYFatigueFrailtyNursing Care

Identifiers

PMID39762099
PMCPMC11749496

What OpenQuestion holds

Textmetadata
LicenceCC BY-NC
Read underepoch 390

Registered trials

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.