Italian validation of the functional difficulties questionnaire (FDQ-9) and its correlation with major determinants of quality of life in adults with hypermobile Ehlers-Danlos syndrome/hypermobility spectrum disorder
Open accessAbstract
The 2017 nosology defines the new criteria for hypermobile Ehlers–Danlos syndrome (hEDS), which is now considered one end of a continuous spectrum encompassing isolated, nonsyndromic joint hypermobility (JH) and hypermobility spectrum disorders (HSDs). Preliminary data indicate a link between JH and neurodevelopmental disorders and, in particular, developmental coordination disorder (DCD) in children. Assessing DCD in adults is difficult and the recently described functional difficulties questionnaire 9 (FDQ‐9) is one of the few available tools. The aims of this study are to (a) validate FDQ‐9 written in Italian and present normal values in 230 Italian controls; (b) explore the relationship of FDQ‐9 with the brief pain inventory, composite autonomic symptom score 31, multidimensional fatigue inventory, attention deficit/hyperactivity disorder self‐report version 1.1, and the SF‐36 for quality of life in 105 Italian adults with hEDS/HSD. Validation of the FDQ‐9 in Italian was carried out by translation, cross‐cultural adaptation and test/retest reliability analysis. A case–control study was performed comparing the FDQ‐9 outcome between 105 patients and 105 sex‐ and age‐matched controls. Fifty‐nine percent of the patients resulted positive compared to the 3.8% of controls (p value < .00001). In patients, FDQ‐9 positive result associated with positive attention deficit/hyperactivity disorder self‐report version 1.1 (OR = 4.04). Multivariate regression analysis comparing FDQ‐9 with the other questionnaires demonstrated a strong association between positive FDQ‐9 and the number of painful joints. Our preliminary data open wider management and therapeutic perspectives for coordination difficulties in hypermobile individuals.
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Fatigue occurs in up to 84% of patients with HSDs.
“Fatigue (up to 84%)”
From Hypermobility spectrum disorders: A review (2023) · cites this paper as [44]
A positive FDQ-9 score is associated with different features affecting QoL in adults with hEDS/HSD.
“We already demonstrated that a positive FDQ-9 score is associated with different features affecting QoL in adults with hEDS/HSD [6].”
From Severity classes in adults with hypermobile Ehlers–Danlos syndrome/hypermobility spectrum disorders: a pilot study of 105 Italian patients (2019) · cites this paper as [6]
Dyspraxia, learning disorder, and attention deficit hyperactivity disorder contribute relevantly to quality of life in children and adults with symptomatic joint hypermobility and hEDS/HSD.
“features, including dyspraxia, learning disorder and attention deficit hyperactivity disorder, were demonstrated to relevantly contribute to QoL in both children and adults with symptomatic JH and hEDS/HSD [6,8].”
From Severity classes in adults with hypermobile Ehlers–Danlos syndrome/hypermobility spectrum disorders: a pilot study of 105 Italian patients (2019) · cites this paper as [6]
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- Authors
- Morlino, S., Dordoni, C., Sperduti, I., Clark, C. J., Piedimonte, C., Fontana, A., Colombi, M., Grammatico, P., Copetti, M., & Castori, M.
- Affiliations
- Sapienza University of Rome (IT)
- University of Brescia (IT)
- San Gallicano Hospital (IT)
- Bournemouth University (GB)
- Casa Sollievo della Sofferenza (IT)
- Corresponding author
- Castori, Marco
- ORCID
- Venue
- American Journal of Medical Genetics Part B: Neuropsychiatric Genetics
- Publisher
- Wiley
- Published
- 2018-11-23
- Volume
- 180
- Issue
- 1
- Pages
- 25-34
- ISSN
- 1552-4841 (ISSN-L)
- 1552-485X
- OpenAlex
- W2901656810
- Cited by
- 12
- References
- 47
- Citations by year
- 2023: 5 · 2022: 1 · 2021: 2 · 2020: 2 · 2019: 1
- FWCI
- 0.76
- Type
- journal article
- Language
- en
- Open access
- bronze
- doi.org
- Locations
- American Journal of Medical Genetics Part B Neuropsychiatric Genetics (publishedVersion)
- PubMed (publishedVersion)
- Bournemouth University Research Online (Bournemouth University) (acceptedVersion)
- Institutional Research Information System (Università degli Studi di Brescia) (submittedVersion)
- IRIS Research product catalog (Sapienza University of Rome) (submittedVersion)
- Topics
- Life Sciences › Biochemistry, Genetics and Molecular Biology › Genetics › Connective tissue disorders research
- Health Sciences › Medicine › Pharmacology › Musculoskeletal pain and rehabilitation
- Health Sciences › Medicine › Psychiatry and Mental health › Attention Deficit Hyperactivity Disorder
- Keywords
- hypermobile Ehlers-Danlos syndrome, hypermobility spectrum disorders, joint hypermobility, developmental coordination disorder, attention deficit hyperactivity disorder, quality of life, cross-cultural adaptation, Italian validation, pain, autonomic symptoms, fatigue, neurodevelopmental disorders
- MeSH terms
- Adolescent, Adult, Ehlers-Danlos Syndrome, Female, Humans, Italy, Male, Middle Aged, Quality of Life, Surveys and Questionnaires, Reproducibility of Results, Case-Control Studies, Self Report
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