Cognitive, emotional, and behavioral considerations for chronic pain management in the Ehlers–Danlos syndrome hypermobility-type: a narrative review
Abstract
BACKGROUND: Ehlers-Danlos syndrome (EDS) hypermobility-type is the most common hereditary disorder of the connective tissue. The tissue fragility characteristic of this condition leads to multi-systemic symptoms in which pain, often severe, chronic, and disabling, is the most experienced. Clinical observations suggest that the complex patient with EDS hypermobility-type is refractory toward several biomedical and physical approaches. In this context and in accordance with the contemporary conceptualization of pain (biopsychosocial perspective), the identification of psychological aspects involved in the pain experience can be useful to improve interventions for this under-recognized pathology. PURPOSE: Review of the literature on joint hypermobility and EDS hypermobility-type concerning psychological factors linked to pain chronicity and disability. METHODS: A comprehensive search was performed using scientific online databases and references lists, encompassing publications reporting quantitative and qualitative research as well as unpublished literature. RESULTS: Despite scarce research, psychological factors associated with EDS hypermobility-type that potentially affect pain chronicity and disability were identified. These are cognitive problems and attention to body sensations, negative emotions, and unhealthy patterns of activity (hypo/hyperactivity). CONCLUSIONS: As in other chronic pain conditions, these aspects should be more explored in EDS hypermobility-type, and integrated into chronic pain prevention and management programs. Implications for Rehabilitation Clinicians should be aware that joint hypermobility may be associated with other health problems, and in its presence suspect a heritable disorder of connective tissue such as the Ehlers-Danlos syndrome (EDS) hypermobility-type, in which chronic pain is one of the most frequent and invalidating symptoms. It is necessary to explore the psychosocial functioning of patients as part of the overall chronic pain management in the EDS hypermobility-type, especially when they do not respond to biomedical approaches as psychological factors may be operating against rehabilitation. Further research on the psychological factors linked to pain chronicity and disability in the EDS hypermobility-type is needed.
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More research is needed on relevant psychosocial strategies for individuals with EDS.
“it has been suggested that more research is needed on relevant psychosocial strategies for individuals with EDS [31].”
From Psychological interventions for individuals with Ehlers-Danlos syndrome and hypermobility spectrum disorder: a scoping review (2023) · cites this paper as [31]
Cites
The Need for a New Medical Model: A Challenge for Biomedicine
Engel, G. L. (1977) · Science · doi:10.1126/science.847460 · Cited by 12,887 · see citing works
Fear-avoidance and its consequences in chronic musculoskeletal pain: a state of the art.
Vlaeyen, J. W. S., & Linton, S. J. (2000) · Pain · doi:10.1016/s0304-3959(99)00242-0 · Cited by 4,931 · see citing works
The 2017 international classification of the Ehlers–Danlos syndromes
Malfait, F., Francomano, C., Byers, P., Belmont, J., Berglund, B., Black, J., Bloom, L., Bowen, J. M., Brady, A. F., Burrows, N. P., et al. (2017) · American Journal of Medical Genetics Part C: Seminars in Medical Genetics · doi:10.1002/ajmg.c.31552 · Cited by 1,936 · cited 2× · see citing works
“Unlike other EDS subtypes, the genetic etiology of EDS-H remains unclear [5,6].”
BackgroundPincus, T., Burton, A. K., Vogel, S., & Field, A. P. (2002) · Spine · doi:10.1097/00007632-200203010-00017 · Cited by 1,633 · cited 2× · see citing works
“These aspects of body awareness and catastrophizing thoughts about body sensations might influence the perception and tolerance of pain [60,61], and negatively affect its trajectory [28].”
BackgroundBehavioral methods for chronic pain and illness
Fordyce, W. E. (1977) · Pain · doi:10.1016/0304-3959(77)90029-x · Cited by 1,630 · see citing works
Prevalence and correlates of adult attention-deficit hyperactivity disorder: meta-analysis
Simon, V., Czobor, P., Bálint, S., Mészáros, Á., & Bitter, I. (2009) · British Journal of Psychiatry · doi:10.1192/bjp.bp.107.048827 · Cited by 1,601 · see citing works
Mindfulness Meditation for Chronic Pain: Systematic Review and Meta-analysis
Hilton, L., Hempel, S., Ewing, B. A., Apaydin, E., Xenakis, L., Newberry, S., Colaiaco, B., Maher, A. R., Shanman, R. M., Sorbero, M. E., et al. (2017) · Annals of Behavioral Medicine · doi:10.1007/s12160-016-9844-2 · Cited by 977 · see citing works
Mechanisms of mindfulness training: Monitor and Acceptance Theory (MAT
Lindsay, E. K., & Creswell, J. D. (2017) · Clinical Psychology Review · doi:10.1016/j.cpr.2016.10.011 · Cited by 919 · cited 1× · see citing works
“These interventions promote passing from rational or irrational thinking concerning physical symptoms and hypervigilance to a non-judgmental way of observing experiences through the development of monitoring attention skills and a mental attitude of acceptance [58,63].”
BackgroundEhde, D. M., Dillworth, T. M., & Turner, J. A. (2014) · American Psychologist · doi:10.1037/a0035747 · Cited by 879 · see citing works
Behavioral Methods for Chronic Pain and Illness
Quimby, C. W. (1976) · Anesthesiology · doi:10.1097/00000542-197610000-00030 · Cited by 866 · see citing works
Impact of psychological factors in the experience of pain
Linton, S. J., & Shaw, W. S. (2011) · Physical Therapy · doi:10.2522/ptj.20100330 · Cited by 819 · see citing works
Veehof, M. M., Trompetter, H. R., Bohlmeijer, E. T., & Schreurs, K. M. G. (2016) · Cognitive Behaviour Therapy · doi:10.1080/16506073.2015.1098724 · Cited by 737 · cited 1× · see citing works
“Recent systematic reviews and a meta-analysis highlight the contribution of the “third wave” of CBT to the treatment of chronic pain patients [64,65].”
BackgroundBody awareness: construct and self-report measures.
Mehling, W. E., Gopisetty, V., Daubenmier, J., Price, C. J., Hecht, F. M., & Stewart, A. (2009) · PLoS ONE · doi:10.1371/journal.pone.0005614 · Cited by 653 · see citing works
The influence of negative emotions on pain: Behavioral effects and neural mechanisms
Wiech, K., & Tracey, I. (2009) · NeuroImage · doi:10.1016/j.neuroimage.2009.05.059 · Cited by 614 · see citing works
A framework for the classification of joint hypermobility and related conditions
Castori, M., Tinkle, B., Levy, H., Grahame, R., Malfait, F., & Hakim, A. (2017) · American Journal of Medical Genetics Part C: Seminars in Medical Genetics · doi:10.1002/ajmg.c.31539 · Cited by 602 · cited 1× · see citing works
“Generalized JH is probably due to an altered collagen condition, and is more often a congenital and inherited trait [4].”
BackgroundBiopsychosocial perspective on chronic pain.
Turk, D. C., & Monarch, E. S. (1996) · Cited by 439 · see citing works
Hakim, A., & Grahame, R. (2003) · International Journal of Clinical Practice · doi:10.1111/j.1742-1241.2003.tb10455.x · Cited by 312 · cited 2× · see citing works
“Joint hypermobility (JH), which is defined as an increased distensibility of the joints, is a somatic characteristic very common in the general population, with a prevalence ranging between 10% and 40% [1], being more frequent in women and decreasing with age.”
BackgroundSensory modulation dysfunction in children with attention-deficit–hyperactivity disorder
Mangeot, S., Miller, L. J., McIntosh, D. N., McGrath‐Clarke, J., Simon, J., Hagerman, R. J., & Goldson, E. (2001) · Developmental Medicine & Child Neurology · doi:10.1017/s0012162201000743 · Cited by 302 · see citing works
Neural substrates linking balance control and anxiety
BALABAN, C. (2002) · Physiology & Behavior · doi:10.1016/s0031-9384(02)00935-6 · Cited by 269 · see citing works
Pain in Ehlers-Danlos Syndrome Is Common, Severe, and Associated with Functional Impairment
Voermans, N. C., Knoop, H., Bleijenberg, G., & van Engelen, B. G. (2010) · Journal of Pain and Symptom Management · doi:10.1016/j.jpainsymman.2009.12.026 · Cited by 237 · see citing works
The role of avoidance, pacing, and other activity patterns in chronic pain
McCracken, L. M., & Samuel, V. M. (2007) · Pain · doi:10.1016/j.pain.2006.11.016 · Cited by 234 · see citing works
Castori, M. (2012) · ISRN Dermatology · doi:10.5402/2012/751768 · Cited by 222 · see citing works
Quality of Life in Patients With Postural Tachycardia Syndrome
Benrud-Larson, L. M., Dewar, M. S., Sandroni, P., Rummans, T. A., Haythornthwaite, J. A., & Low, P. A. (2002) · Mayo Clinic Proceedings · doi:10.4065/77.6.531 · Cited by 217 · see citing works
The role of psychological interventions in the management of patients with chronic pain
Roditi, D., & Roditi (2011) · Psychology Research and Behavior Management · doi:10.2147/prbm.s15375 · Cited by 217 · see citing works
Gratacòs, M., Nadal, M., Martín‐Santos, R., Pujana, M. Á., Gago, J., Peral, B., Armengol, L., Ponsa, I., Miró, R., Bulbena, A., et al. (2001) · Cell · doi:10.1016/s0092-8674(01)00447-0 · Cited by 215 · see citing works
Self-citationPain management in the Ehlers–Danlos syndromes
Chopra, P., Tinkle, B., Hamonet, C., Brock, I., Gompel, A., Bulbena, A., & Francomano, C. (2017) · American Journal of Medical Genetics Part C: Seminars in Medical Genetics · doi:10.1002/ajmg.c.31554 · Cited by 209 · see citing works
Self-citationAndrews, N. E., Strong, J., & Meredith, P. J. (2012) · Archives of Physical Medicine and Rehabilitation · doi:10.1016/j.apmr.2012.05.029 · Cited by 204 · see citing works
A diathesis-stress model of chronic pain and disability following traumatic injury.
Turk, D. (2002) · Pain Research and Management · doi:10.1155/2002/252904 · Cited by 200 · see citing works
Psychological mechanisms in the transition from acute to chronic pain: over- or underrated?
Hasenbring, M. I., Hallner, D., & Klasen, B. (2001) · Der Schmerz · doi:10.1007/s004820100030 · Cited by 198 · see citing works
Pain in the body. Altered interoception in chronic pain conditions: a systematic review
Di Lernia, D., Serino, S., & Riva, G. (2016) · Neuroscience & Biobehavioral Reviews · doi:10.1016/j.neubiorev.2016.09.015 · Cited by 190 · see citing works
Psychological Approaches to Pain Management: A Practitioner's Handbook
Merskey, H. (1997) · Journal of Neuropsychiatry · doi:10.1176/jnp.9.4.623 · Cited by 185 · see citing works
Premorbid “overactive” lifestyle in chronic fatigue syndrome and fibromyalgia
Houdenhove, B. V., Neerinckx, E., Onghena, P., Lysens, R., & Vertommen, H. (2001) · Journal of Psychosomatic Research · doi:10.1016/s0022-3999(01)00247-1 · Cited by 175 · see citing works
Joint hypermobility–asset or liability? A study of joint mobility in ballet dancers.
Grahame, R., & Jenkins, J. (1972) · Annals of the Rheumatic Diseases · doi:10.1136/ard.31.2.109 · Cited by 169 · see citing works
Gastrointestinal involvement in the Ehlers–Danlos syndromes
Fikree, A., Chelimsky, G., Collins, H., Kovacic, K., & Aziz, Q. (2017) · American Journal of Medical Genetics Part C: Seminars in Medical Genetics · doi:10.1002/ajmg.c.31546 · Cited by 159 · see citing works
Castori, M., Morlino, S., Celletti, C., Celli, M., Morrone, A., Colombi, M., Camerota, F., & Grammatico, P. (2012) · American Journal of Medical Genetics Part A · doi:10.1002/ajmg.a.35483 · Cited by 151 · see citing works
Psychiatric and psychological aspects in the Ehlers–Danlos syndromes
Bulbena, A., Baeza‐Velasco, C., Bulbena‐Cabré, A., Pailhez, G., Critchley, H., Chopra, P., Mallorquí‐Bagué, N., Frank, C., & Porges, S. (2017) · American Journal of Medical Genetics Part C: Seminars in Medical Genetics · doi:10.1002/ajmg.c.31544 · Cited by 149 · see citing works
Self-citationMallik, A., Ferrell, W. R., McDonald, A. G., & Sturrock, R. D. (1994) · Lara D. Veeken · doi:10.1093/rheumatology/33.7.631 · Cited by 146 · see citing works
Cederlöf, M., Larsson, H., Lichtenstein, P., Almqvist, C., Serlachius, E., & Ludvigsson, J. F. (2016) · BMC Psychiatry · doi:10.1186/s12888-016-0922-6 · Cited by 142 · cited 1× · see citing works
“[73] reported an increased risk of having ADHD (risk ratio =5.”
BackgroundBiobehavioral factors affecting pain and disability in low back pain: mechanisms and assessment.
Feuerstein, M., & Beattie, P. F. (1995) · Physical Therapy · doi:10.1093/ptj/75.4.267 · Cited by 142 · see citing works
Proprioception and muscle torque deficits in children with hypermobility syndrome.
Fatoye, F., Palmer, S., MacMillan, F., Rowe, P., & van der Linden, M. L. (2008) · Rheumatology · doi:10.1093/rheumatology/ken435 · Cited by 139 · see citing works
Association Between Joint Hypermobility Syndrome and Panic Disorder
Martín‐Santos, R., Bulbena, A., Porta, M. S., Gago, J., Balaguer, L. M., & Duró, J. C. (1998) · American Journal of Psychiatry · doi:10.1176/ajp.155.11.1578 · Cited by 137 · see citing works
Self-citationSmith, T. O., Easton, V., Bacon, H., Jerman, E., Armon, K., Poland, F., & Macgregor, A. J. (2014) · Rheumatology · doi:10.1093/rheumatology/ket317 · Cited by 130 · see citing works
Colombi, M., Dordoni, C., Chiarelli, N., & Ritelli, M. (2015) · American Journal of Medical Genetics Part C: Seminars in Medical Genetics · doi:10.1002/ajmg.c.31429 · Cited by 127 · see citing works
Pollatos, O., Füstös, J., & Critchley, H. D. (2012) · Pain · doi:10.1016/j.pain.2012.04.030 · Cited by 127 · see citing works
Rombaut, L., Malfait, F., De Wandele, I., Cools, A., Thijs, Y., De Paepe, A., & Calders, P. (2011) · Archives of Physical Medicine and Rehabilitation · doi:10.1016/j.apmr.2011.01.016 · Cited by 124 · see citing works
Kindermans, H. P. J., Roelofs, J., Goossens, M. E. J. B., Huijnen, I. P. J., Verbunt, J. A., & Vlaeyen, J. W. S. (2011) · The Journal of Pain · doi:10.1016/j.jpain.2011.04.009 · Cited by 124 · see citing works
Active despite pain: the putative role of stop-rules and current mood
Vlaeyen, J. W. S., & Morley, S. (2004) · Pain · doi:10.1016/j.pain.2004.04.037 · Cited by 121 · see citing works
Rombaut, L., Malfait, F., Wandele, I. D., Thijs, Y., Palmans, T., Paepe, A. D., & Calders, P. (2011) · Arthritis Care & Research · doi:10.1002/acr.20557 · Cited by 110 · see citing works
The relationship between activity and chronic back pain
LINTON, S. J. (1985) · Pain · doi:10.1016/0304-3959(85)90092-2 · Cited by 106 · see citing works
Scheper, M., de Vries, J. E., Verbunt, J., & Engelbert, R. H. H. (2015) · Journal of Pain Research · doi:10.2147/jpr.s64251 · Cited by 105 · see citing works
High symptom reporters are less interoceptively accurate in a symptom-related context
Bogaerts, K., Millen, A., Wan, L., Peuter, S. D., Diest, I. V., Vlemincx, E., Fannes, S., & den Bergh, O. V. (2008) · Journal of Psychosomatic Research · doi:10.1016/j.jpsychores.2008.03.019 · Cited by 103 · see citing works
Di Stefano, G., Celletti, C., Baron, R., Castori, M., Di Franco, M., La Cesa, S., Leone, C., Pepe, A., Cruccu, G., Truini, A., et al. (2016) · European Journal of Pain · doi:10.1002/ejp.856 · Cited by 100 · see citing works
Brain structure and joint hypermobility: Relevance to the expression of psychiatric symptoms
Eccles, J. A., Beacher, F. D. C., Gray, M. A., Jones, C. L., Minati, L., Harrison, N. A., & Critchley, H. D. (2012) · British Journal of Psychiatry · doi:10.1192/bjp.bp.111.092460 · Cited by 96 · see citing works
Ehlers–Danlos syndrome, hypermobility type: A characterization of the patients' lived experience
Murray, B., Yashar, B. M., Uhlmann, W. R., Clauw, D. J., & Petty, E. M. (2013) · American Journal of Medical Genetics Part A · doi:10.1002/ajmg.a.36293 · Cited by 92 · see citing works
Rombaut, L., Malfait, F., De Paepe, A., Rimbaut, S., Verbruggen, G., De Wandele, I., & Calders, P. (2011) · Arthritis & Rheumatism · doi:10.1002/art.30337 · Cited by 91 · see citing works
Rombaut, L., Scheper, M., De Wandele, I., De Vries, J., Meeus, M., Malfait, F., Engelbert, R., & Calders, P. (2015) · Clinical Rheumatology · doi:10.1007/s10067-014-2499-0 · Cited by 91 · see citing works
Psychosocial functioning in the Ehlers–Danlos syndrome
Lumley, M. A., Jordan, M., Rubenstein, R., Tsipouras, P., & Evans, M. I. (1994) · American Journal of Medical Genetics · doi:10.1002/ajmg.1320530206 · Cited by 90 · see citing works
Berglund, B., Pettersson, C., Pigg, M., & Kristiansson, P. (2015) · BMC Musculoskeletal Disorders · doi:10.1186/s12891-015-0549-7 · Cited by 89 · see citing works
Sinibaldi, L., Ursini, G., & Castori, M. (2015) · American Journal of Medical Genetics Part C: Seminars in Medical Genetics · doi:10.1002/ajmg.c.31430 · Cited by 87 · see citing works
Bathen, T., Hångmann, A. B., Hoff, M., Andersen, L. Ø., & Rand‐Hendriksen, S. (2013) · American Journal of Medical Genetics Part A · doi:10.1002/ajmg.a.36060 · Cited by 86 · see citing works
Pain, distress and joint hyperlaxity.
Grahame, R. (2000) · PubMed · Cited by 84 · see citing works
Heritable disorders of connective tissue
Grahame, R. (2000) · Best Practice & Research Clinical Rheumatology · doi:10.1053/berh.1999.0069 · Cited by 82 · see citing works
Hershenfeld, S. A., Wasim, S., McNiven, V., Parikh, M., Majewski, P., Faghfoury, H., & So, J. (2016) · Rheumatology International · doi:10.1007/s00296-015-3375-1 · Cited by 81 · see citing works
Celletti, C., Castori, M., La Torre, G., & Camerota, F. (2013) · BioMed Research International · doi:10.1155/2013/580460 · Cited by 81 · see citing works
Voermans, N. C., & Knoop, H. (2011) · Disability and Rehabilitation · doi:10.3109/09638288.2010.531373 · Cited by 81 · see citing works
Joint hypermobility syndrome: problems that require psychological intervention
Baeza‐Velasco, C., Gély-Nargeot, M.-C., Bulbena, A., & Bravo, J. F. (2011) · Rheumatology International · doi:10.1007/s00296-011-1839-5 · Cited by 79 · see citing works
Self-citationJOINT HYPERMOBILITY SYNDROME AND ANXIETY DISORDERS
Bulbena, A., Duró, J. C., Mateo, A., Porta, M. S., & Vallejo, J. (1988) · The Lancet · doi:10.1016/s0140-6736(88)90514-4 · Cited by 77 · see citing works
Self-citationSomatosensory Amplification and Its Relationship to Heartbeat Detection Ability
Mailloux, J., & Brener, J. M. (2002) · Psychosomatic Medicine · doi:10.1097/00006842-200203000-00020 · Cited by 74 · see citing works
High frequency of childhood ADHD history in women with fibromyalgia
Reyero, F., Ponce, G., Rodriguez-Jimenez, R., Fernandez-Dapica, P., Taboada, D., Martin, V., Navio, M., Jimenez-Arriero, M. A., Hoenicka, J., & Palomo, T. (2011) · European Psychiatry · doi:10.1016/j.eurpsy.2010.03.012 · Cited by 73 · see citing works
Peur du mouvement chez des accidentés du travail: L'Échelle de Kinésiophobie de Tampa (EKT).
French, D. J., Roach, P. J., & Mayes, S. (2002) · Canadian Journal of Behavioural Science/Revue canadienne des sciences du comportement · doi:10.1037/h0087152 · Cited by 69 · see citing works
Baeza-Velasco, C., Pailhez, G., Bulbena, A., & Baghdadli, A. (2015) · General Hospital Psychiatry · doi:10.1016/j.genhosppsych.2014.10.002 · Cited by 67 · see citing works
Self-citation“It's very hard to change yourself”
Andrews, N. E., Strong, J., Meredith, P. J., Gordon, K., & Bagraith, K. S. (2015) · Pain · doi:10.1097/j.pain.0000000000000161 · Cited by 66 · see citing works
Oral and mandibular manifestations in the Ehlers–Danlos syndromes
Mitakides, J., & Tinkle, B. T. (2017) · American Journal of Medical Genetics Part C: Seminars in Medical Genetics · doi:10.1002/ajmg.c.31541 · Cited by 66 · see citing works
Attention management as a treatment for chronic pain
Elomaa, M. M., Williams, A. C. D. C., & Kalso, E. A. (2009) · European Journal of Pain · doi:10.1016/j.ejpain.2008.12.002 · Cited by 62 · see citing works
Neuropathic pain is a common feature in Ehlers-Danlos syndrome
Camerota, F., Celletti, C., Castori, M., Grammatico, P., & Padua, L. (2011) · Journal of Pain and Symptom Management · doi:10.1016/j.jpainsymman.2010.09.012 · Cited by 61 · see citing works
Bulbena, A., Pailhez, G., Bulbena-Cabré, A., Mallorquí-Bagué, N., & Baeza-Velasco, C. (2015) · Advances in Psychosomatic Medicine · doi:10.1159/000369113 · Cited by 60 · see citing works
Self-citationScheper, M. C., Pacey, V., Rombaut, L., Adams, R. D., Tofts, L., Calders, P., Nicholson, L. L., & Engelbert, R. H. H. (2017) · Arthritis Care & Research · doi:10.1002/acr.22998 · Cited by 58 · see citing works
Understanding the Psychosocial Profile of Women with Fibromyalgia Syndrome
Shuster, J., McCormack, J., Riddell, R. P., & Toplak, M. E. (2009) · Pain Research and Management · doi:10.1155/2009/134808 · Cited by 57 · see citing works
The perception of bodily sensations, with special reference to hypochondriasis.
Steptoe, A., & Noll, A. (1997) · Behaviour Research and Therapy · doi:10.1016/s0005-7967(97)00055-7 · Cited by 57 · see citing works
Bart, O., Bar-Haim, Y., Weizman, E., Levin, M., Sadeh, A., & Mintz, M. (2009) · Research in Developmental Disabilities · doi:10.1016/j.ridd.2008.07.008 · Cited by 56 · see citing works
The disabling role of fluctuations in physical activity in patients with chronic low back pain
Huijnen, I. P. J., Verbunt, J., Roelofs, J., Goossens, M. E. J. B., & Peters, M. L. (2009) · European Journal of Pain · doi:10.1016/j.ejpain.2008.12.008 · Cited by 54 · see citing works
Baeza-Velasco, C., Gely-Nargeot, M.-C., Vilarrasa, A. B., Fenetrier, C., & Bravo, J. F. (2011) · The International Journal of Psychiatry in Medicine · doi:10.2190/pm.41.2.g · Cited by 52 · see citing works
Self-citationCOGNITIVE THERAPY FOR CHRONIC PAIN
Wootton, J., & Warfield, C. (2009) · Elsevier eBooks · doi:10.1016/b978-1-4160-4836-7.00072-9 · Cited by 51 · see citing works
The Cognitive Functions in Adults with Chronic Pain: A Comparative Study
Nadar, M. S., Jasem, Z. A., & Manee, F. (2016) · Pain Research and Management · doi:10.1155/2016/5719380 · Cited by 50 · see citing works
Association between joint hypermobility syndrome and panic disorder: a case-control study.
Campayo, J. G., Asso, E., Alda, M., Andrés, E., & Sobradiel, N. (2010) · Psychosomatics · doi:10.1176/appi.psy.51.1.55 · Cited by 47 · see citing works
Pain in Ehlers-Danlos syndromes: manifestations, therapeutic strategies and future perspectives
Castori, M. (2016) · Expert Opinion on Orphan Drugs · doi:10.1080/21678707.2016.1238302 · Cited by 46 · cited 4× · see citing works
“More research is needed to identify an optimal non-pharmacological approach to pain management in EDS-H [14].”
BackgroundInfluentialBenign joint hypermobility syndrome in patients with attention deficit / hyperactivity disorders
Sebnem (2011) · DOAJ (DOAJ: Directory of Open Access Journals) · doi:10.5152/tjr.2011.029 · Cited by 42 · see citing works
Biopsychosocial Profiles of Different Pain Diagnostic Groups
Porter-Moffitt, S., Gatchel, R. J., Robinson, R. C., Deschner, M., Posamentier, M. T., Polatin, P. B., & Lou, L. (2006) · Journal of Pain · doi:10.1016/j.jpain.2005.12.003 · Cited by 42 · see citing works
Benign Joint Hypermobility Syndrome in Patients with Attention Deficit/Hyperactivity Disorders
DOĞAN, Ş. K., TANER, Y., & EVCİK1, D. (2011) · Archives of Rheumatology · doi:10.5606/tjr.2011.029 · Cited by 42 · see citing works
Baeza-Velasco, C., Van den Bossche, T., Grossin, D., & Hamonet, C. (2016) · Eating and Weight Disorders - Studies on Anorexia, Bulimia and Obesity · doi:10.1007/s40519-015-0232-x · Cited by 39 · see citing works
Self-citationArthur, K. B., Caldwell, K. L., Forehand, S., & Davis, K. M. (2015) · Disability and Rehabilitation · doi:10.3109/09638288.2015.1092175 · Cited by 37 · see citing works
How do people with chronically painful joint hypermobility syndrome make decisions about activity?
Schmidt, A., Corcoran, K., Grahame, R., & Williams, A. C. D. C. (2014) · British Journal of Pain · doi:10.1177/2049463714554112 · Cited by 32 · see citing works
Current issues in behavioral and pharmacological approaches to smoking cessation
Hájek, P. (1996) · Addictive Behaviors · doi:10.1016/0306-4603(96)00029-9 · Cited by 30 · see citing works
Baeza‐Velasco, C., Gély-Nargeot, M.-C., Pailhez, G., & Bulbena, A. (2013) · Current Sports Medicine Reports · doi:10.1249/jsr.0b013e3182a4b933 · Cited by 29 · see citing works
Self-citationEfficacy of an out-patient pain management programme for people with joint hypermobility syndrome
Rahman, A., Daniel, C., & Grahame, R. (2014) · Clinical Rheumatology · doi:10.1007/s10067-014-2539-9 · Cited by 26 · see citing works
Shiari, R., Saeidifard, F., & Zahed, G. (2013) · Annals of Paediatric Rheumatology · doi:10.5455/apr.032420131219 · Cited by 26 · see citing works
Proprioceptive acuity into knee hypermobile range in children with Joint Hypermobility Syndrome
Pacey, V., Adams, R. D., Tofts, L., Munns, C. F., & Nicholson, L. L. (2014) · Pediatric Rheumatology · doi:10.1186/1546-0096-12-40 · Cited by 24 · see citing works
Derksen, M. T., Vreeling, M. J. W., & Tchetverikov, I. (2015) · PubMed · Cited by 24 · see citing works
Mallorquí‐Bagué, N., Bulbena, A., Roé-Vellvé, N., Hoekzema, E. A., Carmona, S., Barba‐Müller, E., Fauquet, J., Pailhez, G., & Vilarroya, Ó. (2015) · European Psychiatry · doi:10.1016/j.eurpsy.2015.01.004 · Cited by 23 · see citing works
Self-citationHealth care professionals' understanding of chronic pain: a grounded theory study
Thunberg, K., Carlsson, S. G., & Hallberg, L. R. ‐. (2001) · Scandinavian Journal of Caring Sciences · doi:10.1046/j.1471-6712.2001.1510099.x · Cited by 21 · see citing works
Generalised joint hypermobility and neurodevelopmental traits in a non-clinical adult population
Glans, M., Bejerot, S., & Humble, M. B. (2017) · BJPsych Open · doi:10.1192/bjpo.bp.116.004325 · Cited by 21 · see citing works
Hamonet, C., Gompel, A., Raffray, Y., Zeitoun, J. D., Delarue, M., Vlamynck, E., Haidar, R., & Mazaltarine, G. (2014) · Douleurs : Evaluation - Diagnostic - Traitement · doi:10.1016/j.douler.2014.09.003 · Cited by 11 · see citing works
Baeza‐Velasco, C., Bourdon, C., Polanco-Carrasco, R., de Jouvencel, M., Gély-Nargeot, M.-C., Gompel, A., & Hamonet, C. (2017) · Rheumatology International · doi:10.1007/s00296-017-3659-8 · Cited by 11 · see citing works
Self-citationBehavioral and Representational Components of “Hyperactivity” in Fibromyalgia Syndrome Patients
Grisart, J. M., Masquelier, É., Desmedt, A., Scaillet, N., & Luminet, O. (2010) · Journal of Musculoskeletal Pain · doi:10.3109/10582450903568563 · Cited by 8 · see citing works
Pain management and cognitive behavioural therapy
Daniel, H. C. (2010) · Elsevier eBooks · doi:10.1016/b978-0-7020-3005-5.00012-4 · Cited by 8 · see citing works
Body perception in a sample of nonclinical youngsters with joint hypermobility
Bulbena‐Cabré, A., Pailhez, G., Cabrera, A., Baeza‐Velasco, C., Porges, S. W., & Bulbena, A. (2017) · Ansiedad y Estrés · doi:10.1016/j.anyes.2017.07.002 · Cited by 8 · see citing works
Self-citationCelletti, C., Castori, M., Morico, G., & Camerota, F. (2014) · International Journal of Clinical Rheumatology · doi:10.2217/ijr.13.74 · Cited by 3 · see citing works
Impaired quality of life and functional status in patients with benign joint hypermobility syndrome
Şahin, E., Gülbahar, S., Baydar, M., Söylev, G. Ö., Bircan, Ç., El, Ö., Kızıl, R., & Peker, Ö. (2015) · MYOPAIN · doi:10.3109/10582452.2016.1143074 · Cited by 3 · see citing works
Historia y Actualidad del Síndrome de Ehlers-Danlos-Tschernogobow
Hamonet, C., Ducret, L., Layadi, K., & Baeza‐Velasco, C. (2016) · Dialnet (Universidad de la Rioja) · Cited by 3 · see citing works
Self-citationRecognizing and treating Ehlers-Danlos syndrome(s): the need for a multidisciplinary approach
Morlino, S., Piedimonte, C., & Castori, M. (2016) · Cuadernos de neuropsicología · Cited by 2 · see citing works
Barfiwala, K. N. (2016) · OhioLink ETD Center (Ohio Library and Information Network) · Cited by 1 · see citing works
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Wasim, S., Suddaby, J. S., Parikh, M., Leylachian, S., Ho, B., Guerin, A., & So, J. (2019) · Rheumatology International · doi:10.1007/s00296-019-04293-w · Cited by 44 · see citing works
Bulbena‐Cabré, A., Baeza‐Velasco, C., Rosado‐Figuerola, S., & Bulbena, A. (2021) · American Journal of Medical Genetics Part C: Seminars in Medical Genetics · doi:10.1002/ajmg.c.31955 · Cited by 41 · see citing works
Self-citationPain Phenotypes in Rare Musculoskeletal and Neuromuscular Diseases
Tucker-Bartley, A., Lemme, J. D., Gomez-Morad, A. D., Shah, N. A., Veliu, M., Birklein, F., Storz, C., Rutkove, S. B., Kronn, D., Boyce, A. M., et al. (2021) · Neuroscience & Biobehavioral Reviews · doi:10.1016/j.neubiorev.2021.02.009 · Cited by 42 · see citing works
Psychosocial Influence of Ehlers–Danlos Syndrome in Daily Life of Patients: A Qualitative Study
Palomo-Toucedo, I., Leon-Larios, F., Reina-Bueno, M., Vázquez-Bautista, M., Munuera-Martínez, P., & Domínguez-Maldonado, G. (2020) · International Journal of Environmental Research and Public Health · doi:10.3390/ijerph17176425 · Cited by 36 · see citing works
Hypermobile Ehlers-Danlos Syndrome and Hypermobility Spectrum Disorders.
Yew, K. S., Kamps-Schmitt, K. A., & Borge, R. (2021) · PubMed · Cited by 33 · see citing works
Molander, P., Novo, M., Hållstam, A., Löfgren, M., Stålnacke, B., & Gerdle, B. U. C. (2020) · Journal of Clinical Medicine · doi:10.3390/jcm9072143 · Cited by 33 · see citing works
Ehlers-Danlos Syndrome in the Field of Psychiatry: A Review
Ishiguro, H., Yagasaki, H., & Horiuchi, Y. (2022) · Frontiers in Psychiatry · doi:10.3389/fpsyt.2021.803898 · Cited by 31 · see citing works
Demes, J. S., McNair, B., & Taylor, M. R. G. (2020) · American Journal of Medical Genetics Part A · doi:10.1002/ajmg.a.61837 · Cited by 30 · see citing works
Pain Symptomatology and Management in Pediatric Ehlers–Danlos Syndrome: A Review
Feldman, E. C. H., Hivick, D. P., Slepian, P. M., Tran, S. T., Chopra, P., & Greenley, R. N. (2020) · Children · doi:10.3390/children7090146 · Cited by 24 · see citing works
Baeza‐Velasco, C. (2021) · American Journal of Medical Genetics Part C: Seminars in Medical Genetics · doi:10.1002/ajmg.c.31946 · Cited by 17 · see citing works
Self-citationThe psychological burden associated with Ehlers-Danlos syndromes: a systematic review
Kennedy, M., Loomba, K., Ghani, H., & Riley, B. A. (2022) · Journal of Osteopathic Medicine · doi:10.1515/jom-2021-0267 · Cited by 16 · see citing works
Psychosocial and Motor Characteristics of Patients With Hypermobility
Lamari, M. M., Lamari, N. M., Filho, G. M. D. A., de Medeiros, M. P., Marques, V. R. P., & Pavarino, É. C. (2022) · Frontiers in Psychiatry · doi:10.3389/fpsyt.2021.787822 · Cited by 16 · see citing works
Rocchetti, M., Bassotti, A., Corradi, J., Damiani, S., Pasta, G., Annunziata, S., Guerrieri, V., Mosconi, M., Gentilini, D., & Brondino, N. (2021) · Healthcare · doi:10.3390/healthcare9111472 · Cited by 14 · see citing works
Clark, N., Kainth, G. S., Johnson, M., Rangan, A., Kottam, L., & Swainston, K. (2023) · Rheumatology International · doi:10.1007/s00296-023-05503-2 · Cited by 15 · see citing works
Exploring pain mechanisms in hypermobile Ehlers‐Danlos syndrome: A case–control study
Wandele, I. D., Colman, M., Hermans, L., Oosterwijck, J. V., Meeus, M., Rombaut, L., Brusselmans, G., Syx, D., Calders, P., & Malfait, F. (2022) · European Journal of Pain · doi:10.1002/ejp.1956 · Cited by 12 · see citing works
Guedry, S. E., Langley, B. O., Schaefer, K., & Hanes, D. A. (2022) · Disability and Rehabilitation · doi:10.1080/09638288.2022.2131003 · Cited by 12 · see citing works
Song, J. Z., Luong, D., Feldman, E. C. H., Tran, S., Perrier, L., Eubanks, K., Bayley, M., Kastner, M., Slepian, M., & Munce, S. E. P. (2023) · Orphanet Journal of Rare Diseases · doi:10.1186/s13023-023-02799-y · Cited by 9 · see citing works
“Despite these promising outcomes, it has been suggested that more research is needed on relevant psychosocial strategies for individuals with EDS [31].”
BackgroundA qualitative study of pain and related symptoms experienced by people with Ehlers-Danlos syndromes
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Black, W. R., Jones, J. T., Rush, E. T., Malloy-Walton, L. E., & Harding, A. (2023) · Journal of Multidisciplinary Healthcare · doi:10.2147/jmdh.s396221 · Cited by 8 · see citing works
Schubert-Hjalmarsson, E., Fasth, A. L., Ickmans, K., Söderpalm, A., & Lundberg, M. (2024) · European Journal of Pain · doi:10.1002/ejp.4754 · Cited by 8 · see citing works
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Lattimore, P. J., & Harrison, F. (2022) · Disability and Rehabilitation · doi:10.1080/09638288.2022.2140843 · Cited by 5 · see citing works
Molander, P., Novo, M., Ringqvist, Å., Hållstam, A., Hesser, H., Löfgren, M., Stålnacke, B., & Gerdle, B. U. C. (2024) · Journal of Rehabilitation Medicine · doi:10.2340/jrm.v56.12431 · Cited by 8 · see citing works
Ahimaz, P. R., Kramer, T., Swaroop, P., Mitchell, M., Hernan, R., Anyane‐Yeboa, K., & Pereira, E. M. (2022) · American Journal of Medical Genetics Part A · doi:10.1002/ajmg.a.62945 · Cited by 5 · see citing works
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Sousa, A., Jung, M., Allera, A., & Riley, B. A. (2025) · Brain and Behavior · doi:10.1002/brb3.70603 · Cited by 4 · see citing works
Chuchin, J., & Ornstein, T. J. (2023) · Disability and Rehabilitation · doi:10.1080/09638288.2023.2268520 · Cited by 4 · see citing works
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Dockrell, D. M., Berg, K. M., & Ralston, S. H. (2021) · Rheumatology Advances in Practice · doi:10.1093/rap/rkab046 · Cited by 3 · see citing works
Chew, M. T., İlhan, E., Nicholson, L. L., Kobayashi, S., & Chan, C. L. (2024) · Disability and Rehabilitation · doi:10.1080/09638288.2024.2351180 · Cited by 3 · see citing works
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Ishiguro, H., Honobe, N., Suzuki, T., Tamai, M., & Nakane, T. (2019) · Case Reports in Psychiatry · doi:10.1155/2019/7472301 · Cited by 3 · see citing works
Tariq, A., Khan, M. N., Jamali, A. G., Basra, M. A., Mahato, A., Imran, S. M., Mohsin, Z., Singla, S., Mirani, I. K., Asmat, U., et al. (2025) · Cureus · doi:10.7759/cureus.86123 · Cited by 2 · see citing works
Guerrerio, A. L., Mateja, A., MacCarrick, G. L., Fintzi, J., Brittain, E., Frischmeyer‐Guerrerio, P. A., & Dietz, H. C. (2024) · PLoS ONE · doi:10.1371/journal.pone.0298272 · Cited by 2 · see citing works
Chew, M. T., İlhan, E., Nicholson, L. L., Kobayashi, S., Pacey, V., Hakim, A. J., & Chan, C. L. (2025) · European Journal of Pain · doi:10.1002/ejp.70030 · Cited by 2 · see citing works
Kaabi, N. A., Camacho, E., Orchanian‐Cheff, A., Silano, V., McGillis, L., Truong, W. T., Reid, W. D., Chow, C., Ryan, C. M., Slepian, P. M., et al. (2025) · Journal of Clinical Medicine · doi:10.3390/jcm14124126 · Cited by 2 · see citing works
Foster, J., Walklet, E., & Stephens-Lewis, D. (2025) · Rare · doi:10.1016/j.rare.2025.100107 · Cited by 1 · see citing works
Emerging Clinical Concepts in Pain Management of Ehlers-Danlos Syndrome
Banerjee, R. C., Ramsey, M. E., Buras, B. E., Potharaju, P., Shekoohi, S., Abd-Elsayed, A. A., & Kaye, A. D. (2026) · Current Pain and Headache Reports · doi:10.1007/s11916-026-01532-1 · Cited by 1 · see citing works
Camerota, F., Mariani, R., Petronelli, G., Rabissi, B., Vizzini, M. A. S., Trani, M. D., Roselli, V., Pasquini, M., Renzi, A., & Celletti, C. (2025) · Brain Sciences · doi:10.3390/brainsci15020202 · Cited by 1 · see citing works
Timmons, W., Sproule, J., & Mulholland, R. (2021) · Research in Dance Education · doi:10.1080/14647893.2021.1940916 · Cited by 1 · see citing works
Physiotherapy for Joint Hypermobility Disorders
Lamari, N. M., & Beighton, P. (2023) · In clinical practice · doi:10.1007/978-3-031-34914-0_13
Ehlers-Danlos Syndrome - living with chronic pain. Current knowledge of the disease
Olszak, J., Kapłan, W., Rachwał, D., PIĄTEK, E., Zalewa, K., Bartoszek, L., Czelej, M., Stachowicz, H., & Mazurek, J. (2023) · Journal of Education Health and Sport · doi:10.12775/jehs.2023.19.01.015
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Oman, E. A., Jessica, S., Timothy, B., & Ali, K. (2023) · IEEE MIT Undergraduate Research Technology Conference (URTC) · doi:10.1109/urtc60662.2023.10534886
Palomo-Toucedo, I. C., Reina‐Bueno, M., Munuera-Martínez, P. V., Vázquez-Bautista, M. D. C., Domínguez-Maldonado, G., & León-Larios, F. (2024) · Healthcare · doi:10.3390/healthcare12232392
Renzi, A., Celletti, C., Trani, M. D., Vizzini, M. A. S., Colaboni, L., Petronelli, G., Pasquini, M., Camerota, F., & Mariani, R. (2025) · Healthcare · doi:10.3390/healthcare13060636
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DePace, N. L., Soloway, S., Yayac, M. F., & Colombo, J. (2025) · Springer eBooks · doi:10.1007/978-3-031-88769-7_13
Frye, W. S., Ward, S. R., Cucchiaro, G., & Crenshaw, M. (2025) · PM&R · doi:10.1002/pmrj.70050
Henning, M., Hock, M., Shukri, A., Löw, V., Kernich, N., Rauterberg, J., Klapproth, H., & Tantcheva-Poór, I. (2026) · Orphanet Journal of Rare Diseases · doi:10.1186/s13023-026-04242-4
Schenke, K. C., Foster, J., & Stephens-Lewis, D. (2026) · Journal of Health Psychology · doi:10.1177/13591053251410690
- Authors
- Baeza-Velasco, C., Bulbena, A., Polanco-Carrasco, R., & Jaussaud, R.
- Affiliations
- Laboratory of Psychopathology and Health Processes, University Paris Descartes - Sorbonne Paris Cité, Boulogne Billancourt, France;
- Universitat Autònoma de Barcelona (ES)
- Chilean Association of Scientific Journals of Psychology, Santiago, Chile;
- Centre Hospitalier Régional et Universitaire de Nancy (FR)
- Venue
- Disability and Rehabilitation
- Publisher
- Informa UK Limited
- Published
- 2018-01-22
- Volume
- 41
- Issue
- 9
- Pages
- 1110-1118
- ISSN
- 0963-8288 (ISSN-L)
- 1464-5165
- OpenAlex
- W2793482931
- Cited by
- 51
- References
- 111
- Citations by year
- 2026: 3 · 2025: 10 · 2024: 8 · 2023: 7 · 2022: 9 · 2021: 7 · 2020: 5 · 2019: 2
- FWCI
- 0.41
- Type
- journal article
- Language
- en
- Open access
- closed
- Locations
- Disability and Rehabilitation (publishedVersion)
- PubMed (publishedVersion)
- Topics
- Health Sciences › Medicine › Pharmacology › Musculoskeletal pain and rehabilitation
- Life Sciences › Biochemistry, Genetics and Molecular Biology › Genetics › Connective tissue disorders research
- Health Sciences › Medicine › Pediatrics, Perinatology and Child Health › Pediatric Pain Management Techniques
- Keywords
- Ehlers-Danlos syndrome hypermobility type, chronic pain, joint hypermobility, connective tissue diseases, pain chronicity, disability, cognitive factors, negative emotions, psychosocial factors, biopsychosocial model, pain management, rehabilitation
- MeSH terms
- Adaptation, Psychological, Anxiety, Depression, Ehlers-Danlos Syndrome, Fear, Frustration, Humans, Pain, Chronic Pain, Pain Management, Cognitive Dysfunction
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