Research Article
Orthostatic Intolerance and Postural Orthostatic
Tachycardia Syndrome in Joint Hypermobility
Syndrome/Ehlers-Danlos Syndrome, Hypermobility Type:
Neurovegetative Dysregulation or Autonomic Failure?
Claudia Celletti,
1
Filippo Camerota,
1
Marco Castori,
2
Federica Censi,
3
Laura Gioffrè,
4
Giovanni Calcagnini,
3
and Stefano Strano
4
1
Physical Medicine and Rehabilitation, Umberto I Hospital, Rome, Italy
2
Unit of Clinical Genetics, San Camillo-Forlanini Hospital, Rome, Italy
3
Department of Cardiovascular, Dysmetabolic and Aging-Associated Diseases, Italian Institute of Health, Rome, Italy
4
Department of Heart and Great Vessels “A. Reale”, Sapienza University of Rome, Rome, Italy
Correspondence should be addressed to Claudia Celletti; clacelletti@gmail.com
Received 31 October 2016; Revised 16 January 2017; Accepted 19 January 2017; Published 12 February 2017
Academic Editor: Ramazan Akdemir
Copyright © 2017 Claudia Celletti et al. Tis is an open access article distributed under the Creative Commons Attribution License,
which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.
Background. Joint hypermobility syndrome/Ehlers-Danlos syndrome, hypermobility type (JHS/EDS-HT), is a hereditary connec-
tive tissue disorder mainly characterized by generalized joint hypermobility, skin texture abnormalities, and visceral and vascular
dysfunctions, also comprising symptoms of autonomic dysfunction. Tis study aims to further evaluate cardiovascular autonomic
involvement in JHS/EDS-HT by a battery of functional tests. Methods. Te response to cardiovascular refex tests comprising deep
breathing, Valsalva maneuver, 30/15 ratio, handgrip test, and head-up tilt test was studied in 35 JHS/EDS-HT adults. Heart rate and
blood pressure variability was also investigated by spectral analysis in comparison to age and sex healthy matched group. Results.
Valsalva ratio was normal in all patients, but 37.2% of them were not able to fnish the test. At tilt, 48.6% patients showed postural
orthostatic tachycardia, 31.4% orthostatic intolerance, 20% normal results. Only one patient had orthostatic hypotension. Spectral
analysis showed signifcant higher barorefex sensitivity values at rest compared to controls. Conclusions. Tis study confrms the
abnormal cardiovascular autonomic profle in adults with JHS/EDS-HT and found the higher barorefex sensitivity as a potential
disease marker and clue for future research.
1. Introduction
Ehlers-Danlos syndrome (EDS) is an umbrella term for a
group of heritable sof connective tissue disorders mainly
characterized by generalized joint hypermobility, skin texture
abnormalities, and visceral and vascular fragility or dysfunc-
tions [1]. Current nosology identifes six major EDS variants,
with the classical, hypermobility, and vascular types being the
most common [1]. More recently, diferent studies suggested
and partly demonstrated a clinical overlap between EDS,
hypermobility type (EDS-HT), and the joint hypermobility
syndrome (JHS) [2, 3]. JHS is to date defned as an underdiag-
nosed rheumatologic condition showing generalized joint
hypermobility, chronic musculoskeletal pain, and additional
fndings and presenting a common familial aggregation [4].
Te diagnosis of EDS is confrmed by molecular tools in
most types except for EDS-HT and the cognate JHS, both
remaining clinical diagnoses based on available criteria [4, 5].
At the moment, the prevalent opinion is to provisionally
consider JHS and EDS-HT a single entity (i.e., JHS/EDS-
HT) [2]. In JHS/EDS-HT, generalized joint hypermobility
with recurrent joint dislocations and chronic moderate to
severe pain were the most frequent and severe complaints, but
also muscle cramps, tendinitis, headache, and fatigue are fre-
quently reported [6]. Impaired proprioception, postural con-
trol, and muscular strength are factors that may contribute
to joint instability [7]. Te clinical spectrum associated
with JHS/EDS-HT is not limited to the musculoskeletal
Hindawi Publishing Corporation
BioMed Research International
Volume 2017, Article ID 9161865, 7 pages
http://dx.doi.org/10.1155/2017/9161865