Psychological interventions for individuals with Ehlers-Danlos syndrome and hypermobility spectrum disorder: a scoping review

Jessica Z Song; Dorothy Luong; Estée C H Feldman; Susan Tran; Laure Perrier; Kathleen Eubanks; Mark Bayley; Monika Kastner; Maxwell Slepian; Sarah E P Munce

doi:10.1186/s13023-023-02799-y

Psychological interventions for individuals with Ehlers-Danlos syndrome and hypermobility spectrum disorder: a scoping review

Orphanet J Rare Dis. 2023 Aug 31;18(1):254. doi: 10.1186/s13023-023-02799-y.

Authors

Jessica Z Song¹, Dorothy Luong², Estée C H Feldman³, Susan Tran⁴, Laure Perrier⁵, Kathleen Eubanks⁶, Mark Bayley², Monika Kastner^{7

8}, Maxwell Slepian^{9

10

11

12}, Sarah E P Munce^{2

8

13}

Affiliations

¹ KITE Research Institute, Toronto Rehabilitation Institute - University Health Network, Toronto, ON, Canada. jzsong348@gmail.com.
² KITE Research Institute, Toronto Rehabilitation Institute - University Health Network, Toronto, ON, Canada.
³ Behavioral Medicine and Clinical Psychology, Cincinnati Children's Hospital Medical Center, Cincinnati, OH, USA.
⁴ Department of Psychology, DePaul University, Chicago, IL, USA.
⁵ Dalla Lana School of Public Health, Institute of Health Policy, Management and Evaluation, University of Toronto, Toronto, ON, Canada.
⁶ Ehlers-Danlos Syndrome Canada, Toronto, ON, Canada.
⁷ North York General Hospital, Toronto, ON, Canada.
⁸ Institute of Health Policy, Management and Evaluation, University of Toronto, Toronto, ON, Canada.
⁹ GoodHope Ehlers-Danlos Syndrome Clinic, Toronto General Hospital, University Health Network, Toronto, ON, Canada.
¹⁰ Department of Anesthesia and Pain Management, Toronto General Hospital, University Health Network, Toronto, ON, Canada.
¹¹ Krembil Research Institute, University Health Network, Toronto, ON, Canada.
¹² Department of Anesthesiology and Pain Medicine, University of Toronto, Toronto, ON, Canada.
¹³ Department of Occupational Science and Occupational Therapy, University of Toronto, Toronto, ON, Canada.

Abstract

Purpose: To identify the nature and extent of the evidence on psychological interventions among individuals with Ehlers-Danlos Syndrome (EDS) and Hypermobility Spectrum Disorder (HSD).

Materials and methods: Eligible studies reported on psychological interventions for individuals of all ages with EDS and/or HSD. All studies published in English were included, with no restrictions to publication year or status. MEDLINE, CINAHL, EMBASE, and PsycINFO were searched. Two reviewers independently screened studies and abstracted data.

Results: This scoping review included 10 studies reporting on EDS, HSD, or both. Only cohort studies and case studies were identified. Four studies investigated Cognitive Behavioural Therapy (CBT), one investigated Dialectical Behavioural Therapy (DBT), two investigated psychoeducation, two investigated Intensive Interdisciplinary Pain Treatment (IIPT), and one investigated Acceptance Commitment Therapy (ACT). Interventions targeted pain management, self-destructive behaviours, and related psychological issues (e.g., depression/anxiety). Sample sizes were small (n < 50) for most studies and interventions were generally poorly described.

Conclusions: There is a critical need for high-quality research surrounding psychological interventions for individuals with EDS/HSD. Psychological interventions for these individuals are understudied and existing studies lack validity. Researchers should investigate psychological interventions for individuals with all types of EDS/HSD with high-quality studies to validate findings from the existing studies.

Keywords: Chronic pain; Cognitive behavioural therapy; Ehlers-Danlos syndrome; Hypermobility spectrum disorder; Joint hypermobility; Joint hypermobility syndrome; Psychological interventions.

Publication types

Review
Research Support, Non-U.S. Gov't

MeSH terms

Ehlers-Danlos Syndrome* / therapy
Humans
Pain
Psychosocial Intervention*
Sample Size