Exploring signs of central sensitization in adolescents with hypermobility Spectrum disorder or hypermobile Ehlers-Danlos syndrome.

Elke Schubert-Hjalmarsson, Ann-Charlott Söderpalm, Mari Lundberg, Anders Fasth, Kelly Ickmans

Journal: European journal of pain (London, England) 2024;29(1):e4754

PMID: 39529262

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Abstract

BACKGROUND

Hypermobility Spectrum Disorder (HSD) and Hypermobile Ehlers-Danlos Syndrome (hEDS) are two overlapping heritable connective tissue disorders characterized by joint hypermobility, chronic pain, impaired body perception, and musculoskeletal symptoms. Central sensitization has been proposed as a plausible explanation for symptoms like widespread pain, fatigue, mood disorders, and sleep disturbances in patients with HSD/hEDS.

OBJECTIVE

The aim of this study was to investigate signs of central sensitization, including exercise-induced hypoalgesia (EIH), and fatigue severity in adolescents with HSD/hEDS.

METHODS

In this prospective, experimental, case-control study, thirty-seven adolescents with HSD/hEDS and 47 healthy adolescents (all aged 13-17 years) were included. Pressure pain thresholds (PPTs) were measured at four muscle groups using a pressure algometer. EIH was evaluated by measuring PPTs on two muscle groups immediately after an exercise test on a bicycle ergometer. Participants also completed questionnaires on fatigue and cognitive/emotional factors.

RESULTS

The study demonstrated significantly lower PPTs in four different muscle groups in adolescents with HSD/hEDS compared to the healthy control group. Both groups achieved a significantly higher PPTs after exercise in the muscle involved in the activity. Adolescents with HSD/hEDS reported higher fatigue levels and more cognitive/emotional difficulties than the control group.

CONCLUSION

Adolescents with HSD/hEDS showed generalized hyperalgesia measured through PPTs at different body sites. EIH was partly affected in adolescents with HSD/hEDS, presenting as unchanged pain sensitivity in the remote muscle. Pain should be considered as a phenomenon that is influenced by different biopsychosocial factors, including possible central sensitization, which increase its complexity.

SIGNIFICANCE STATEMENT

This study breaks new ground by showing signs of central sensitization, including diminished EIH, in adolescents with HSD or hEDS. Given that exercise is a key element in pain management, these findings offer valuable insights when developing treatment plans for adolescents with HSD or hEDS.

Address: Division of Physiotherapy, Department of Health and Rehabilitation, Institute of Neuroscience and Physiology, University of Gothenburg, Gothenburg, Sweden.; Department of Physiotherapy, Queen Silvia Children's Hospital, Sahlgrenska University Hospital, Gothenburg, Region Västra Götaland, Sweden.; Department of Pediatrics, Institute of Clinical Sciences, Sahlgrenska Academy, University of Gothenburg, Gothenburg, Sweden.; Department of Rheumatology and Immunology, Queen Silvia Children's Hospital, Sahlgrenska University Hospital, Gothenburg, Sweden.; Pain in Motion (PAIN) Research Group, Department of Physiotherapy, Human Physiology and Anatomy, Faculty of Physical Education and Physiotherapy, Vrije Universiteit Brussel, Brussels, Belgium.; Department of Physical Medicine and Physiotherapy, University Hospital Brussels, Brussels, Belgium.; Movement and Nutrition for Health and Performance Research Group, Department of Movement and Sport Sciences, Faculty of Physical Education and Physiotherapy, Vrije Universiteit Brussel, Brussels, Belgium.; Department of Orthopaedics, Institute for Clinical Sciences, Sahlgrenska Academy, University of Gothenburg, Mölndal, Sweden.; Pain in Motion (PAIN) Research Group, Department of Physiotherapy, Human Physiology and Anatomy, Faculty of Physical Education and Physiotherapy, Vrije Universiteit Brussel, Brussels, Belgium.; Department of Health Promoting Science, Sophiahemmet University, Stockholm, Sweden.; University of Gothenburg Centre for Person-Centred Care (GPCC), Sahlgrenska Academy, University of Gothenburg, Gothenburg, Sweden.

DOI: 10.1002/ejp.4754

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MeSH Terms: Humans, Adolescent, Ehlers-Danlos Syndrome, Female, Male, Central Nervous System Sensitization, Case-Control Studies, Joint Instability, Prospective Studies, Pain Threshold, Fatigue, Exercise, Pain Measurement