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Orthostatic intolerance and fatigue in the hypermobility type of Ehlers-Danlos syndrome

Inge De Wandele UGent, Lies Rombaut UGent, Tine De Backer UGent, Wim Peersman UGent, Hellen Da Silva Van den Bosch UGent, Sophie De Mits UGent, Anne De Paepe UGent, Patrick Calders UGent and Fransiska Malfait UGent (2016) RHEUMATOLOGY. 55(8). p.1412-1420
abstract
OBJECTIVE: To investigate whether orthostatic intolerance (OI) is a significant predictor for fatigue in Ehlers-Danlos Syndrome, hypermobility type (EDS-HT). METHODS: Eighty patients with EDS-HT and 52 controls participated in the first part of the study, which consisted of questionnaires. Fatigue was evaluated using the Checklist Individual Strength (CIS). As possible fatigue determinants OI [Autonomic Symptom Profile (ASP)], habitual physical activity (Baecke), affective distress [Hospital Anxiety and Depression Scale (HADS)], pain (SF36), medication use and generalized hypermobility (5-point score of Grahame and Hakim regarding generalized joint hypermobility) were studied. Next, a 20 min head-up tilt (70°) was performed in a subsample of 39 patients and 35 controls, while beat-to-beat heart rate and blood pressure were monitored (Holter, Finometer Pro). Before and after tilt, fatigue severity was assessed using a numeric rating scale. RESULTS: Patients scored significantly higher on the CIS [total score: EDS: 98.2 (18.63)vscontrols: 45.8 (16.62), P < 0.001] and on the OI domain of the ASP [EDS: 22.78 (7.16)vscontrols: 6.5 (7.78)]. OI was prevalent in EDS-HT (EDS: 74.4%, controls: 34.3%, P = 0.001), and frequently expressed as postural orthostatic tachycardia (41.0% of the EDS group). Patients responded to tilt with a higher heart rate and lower total peripheral resistance (p < 0.001; p = 0.032). This altered response correlated with fatigue in daily life (CIS). In the EDS-HT group, tilt provoked significantly more fatigue [numeric rating scale increase: EDS: +3.1 (1.90), controls: +0.5 (1.24), P < 0.001]. Furthermore, the factors OI, pain, affective distress, decreased physical activity and sedative use explained 47.7% of the variance in fatigue severity. CONCLUSION: OI is an important determinant of fatigue in EDS-HT.
Please use this url to cite or link to this publication:
author
organization
year
type
journalArticle (original)
publication status
published
subject
keyword
head-up tilt, hypermobility, fatigue, Ehlers-Danlos syndrome, orthostatic intolerance, cardiovascular dysautonomia, NEURALLY-MEDIATED HYPOTENSION, POSTURAL TACHYCARDIA SYNDROME, CONSENSUS STATEMENT, PHYSICAL-ACTIVITY, BRAIN FOG, QUESTIONNAIRE, DEFINITION, SYMPTOMS, MIDODRINE, MARFAN
journal title
RHEUMATOLOGY
Rheumatology
volume
55
issue
8
pages
1412 - 1420
Web of Science type
Article
Web of Science id
000383847200010
JCR category
RHEUMATOLOGY
JCR impact factor
4.818 (2016)
JCR rank
4/30 (2016)
JCR quartile
1 (2016)
ISSN
1462-0324
DOI
10.1093/rheumatology/kew032
language
English
UGent publication?
yes
classification
A1
copyright statement
I have transferred the copyright for this publication to the publisher
id
7205617
handle
http://hdl.handle.net/1854/LU-7205617
date created
2016-05-09 16:05:31
date last changed
2017-06-16 14:29:39
@article{7205617,
  abstract     = {OBJECTIVE: To investigate whether orthostatic intolerance (OI) is a significant predictor for fatigue in Ehlers-Danlos Syndrome, hypermobility type (EDS-HT).
METHODS: Eighty patients with EDS-HT and 52 controls participated in the first part of the study, which consisted of questionnaires. Fatigue was evaluated using the Checklist Individual Strength (CIS). As possible fatigue determinants OI [Autonomic Symptom Profile (ASP)], habitual physical activity (Baecke), affective distress [Hospital Anxiety and Depression Scale (HADS)], pain (SF36), medication use and generalized hypermobility (5-point score of Grahame and Hakim regarding generalized joint hypermobility) were studied. Next, a 20 min head-up tilt (70{\textdegree}) was performed in a subsample of 39 patients and 35 controls, while beat-to-beat heart rate and blood pressure were monitored (Holter, Finometer Pro). Before and after tilt, fatigue severity was assessed using a numeric rating scale.
RESULTS: Patients scored significantly higher on the CIS [total score: EDS: 98.2 (18.63)vscontrols: 45.8 (16.62), P {\textlangle} 0.001] and on the OI domain of the ASP [EDS: 22.78 (7.16)vscontrols: 6.5 (7.78)]. OI was prevalent in EDS-HT (EDS: 74.4\%, controls: 34.3\%, P = 0.001), and frequently expressed as postural orthostatic tachycardia (41.0\% of the EDS group). Patients responded to tilt with a higher heart rate and lower total peripheral resistance (p {\textlangle} 0.001; p = 0.032). This altered response correlated with fatigue in daily life (CIS). In the EDS-HT group, tilt provoked significantly more fatigue [numeric rating scale increase: EDS: +3.1 (1.90), controls: +0.5 (1.24), P {\textlangle} 0.001]. Furthermore, the factors OI, pain, affective distress, decreased physical activity and sedative use explained 47.7\% of the variance in fatigue severity.
CONCLUSION: OI is an important determinant of fatigue in EDS-HT.},
  author       = {De Wandele, Inge and Rombaut, Lies and De Backer, Tine and Peersman, Wim and Da Silva Van den Bosch, Hellen and De Mits, Sophie and De Paepe, Anne and Calders, Patrick and Malfait, Fransiska},
  issn         = {1462-0324},
  journal      = {RHEUMATOLOGY},
  keyword      = {head-up tilt,hypermobility,fatigue,Ehlers-Danlos syndrome,orthostatic intolerance,cardiovascular dysautonomia,NEURALLY-MEDIATED HYPOTENSION,POSTURAL TACHYCARDIA SYNDROME,CONSENSUS STATEMENT,PHYSICAL-ACTIVITY,BRAIN FOG,QUESTIONNAIRE,DEFINITION,SYMPTOMS,MIDODRINE,MARFAN},
  language     = {eng},
  number       = {8},
  pages        = {1412--1420},
  title        = {Orthostatic intolerance and fatigue in the hypermobility type of Ehlers-Danlos syndrome},
  url          = {http://dx.doi.org/10.1093/rheumatology/kew032},
  volume       = {55},
  year         = {2016},
}

Chicago
De Wandele, Inge, Lies Rombaut, Tine De Backer, Wim Peersman, Hellen Da Silva Van den Bosch, Sophie De Mits, Anne De Paepe, Patrick Calders, and Fransiska Malfait. 2016. “Orthostatic Intolerance and Fatigue in the Hypermobility Type of Ehlers-Danlos Syndrome.” Rheumatology 55 (8): 1412–1420.
APA
De Wandele, I., Rombaut, L., De Backer, T., Peersman, W., Da Silva Van den Bosch, H., De Mits, S., De Paepe, A., et al. (2016). Orthostatic intolerance and fatigue in the hypermobility type of Ehlers-Danlos syndrome. RHEUMATOLOGY, 55(8), 1412–1420.
Vancouver
1.
De Wandele I, Rombaut L, De Backer T, Peersman W, Da Silva Van den Bosch H, De Mits S, et al. Orthostatic intolerance and fatigue in the hypermobility type of Ehlers-Danlos syndrome. RHEUMATOLOGY. 2016;55(8):1412–20.
MLA
De Wandele, Inge, Lies Rombaut, Tine De Backer, et al. “Orthostatic Intolerance and Fatigue in the Hypermobility Type of Ehlers-Danlos Syndrome.” RHEUMATOLOGY 55.8 (2016): 1412–1420. Print.