Effects of Combined Exercise on Hypoalgesia and Autonomic Variables in a Patient with Hypermobile Ehlers-Danlos Syndrome: A Case Study
Alberto Melis
Resumen
Ehlers-Danlos syndromes (EDS) are a heterogeneous group of connective tissue disorders characterized by joint hypermobility, skin hyperextensibility, and tissue fragility. In addition to experiencing chronic pain, musculoskeletal injuries, and recurrent dislocations, their condition often involves symptoms related to dysautonomia, which can dominate the pathology and lead to significant disability.
A 26-year-old woman diagnosed with hypermobile EDS with chronic pain and dysautonomia symptoms of 3 years' duration was evaluated. Pain pressure thresholds, heart rate, peripheral skin temperature, and skin conductance were assessed during a Tilt Test and modified Bruce protocols for this study. Two measurements were taken, one before and one after 7 weeks of treatment.
The treatment, based on interval-type cardiovascular exercise and general strength training, produced significant changes in all the variables mentioned above. Decreases in heart rate, temperature, and skin conductance were observed, as well as an increase in pain pressure thresholds in certain cases.
The results of this case may indicate that an exercise-based intervention is capable of producing changes in variables related to the autonomic nervous system and descending pain modulation systems in a patient diagnosed with hypermobile EDS.
Cómo citar
Referencias
Alsouhibani, A., & Hoeger Bement, M. (2022). Impaired conditioned pain modulation was restored after a single exercise session in individuals with and without fibromyalgia. PAIN Reports, 7(3), e996. https://doi.org/10.1097/PR9.0000000000000996
Alves, E. S., Souza, H. S., Rosa, J. P. P., Lira, F. S., Pimentel, G. D., Santos, R. V. T., Oyama, L. M., Damaso, A. R., Oller do Nascimento, C. M., Viana, V. a. R., Boscolo, R. A., Grassmann, V., Santana, M. G., Tufik, S., & de Mello, M. T. (2012). Chronic exercise promotes alterations in the neuroendocrine profile of elderly people. Hormone and Metabolic Research = Hormon- Und Stoffwechselforschung = Hormones Et Metabolisme, 44(13), 975-979. https://doi.org/10.1055/s-0032-1316294
Assumpção, A., Matsutani, L. A., Yuan, S. L., Santo, A. S., Sauer, J., Mango, P., & Marques, A. P. (2018). Muscle stretching exercises and resistance training in fibromyalgia: Which is better? A three-arm randomized controlled trial. European Journal of Physical and Rehabilitation Medicine, 54(5), 663-670. https://doi.org/10.23736/s1973-9087.17.04876-6
Benarroch, E. E. (2012). Postural Tachycardia Syndrome: A Heterogeneous and Multifactorial Disorder. Mayo Clinic Proceedings, 87(12), 1214-1225. https://doi.org/10.1016/j.mayocp.2012.08.013
Besnier, F., Labrunée, M., Pathak, A., Pavy-Le Traon, A., Galès, C., Sénard, J.-M., & Guiraud, T. (2017). Exercise training-induced modification in autonomic nervous system: An update for cardiac patients. Annals of Physical and Rehabilitation Medicine, 60(1), 27-35. https://doi.org/10.1016/j.rehab.2016.07.002
Bobos, P., Ziebart, C., Furtado, R., Lu, Z., & MacDermid, J. C. (2020). Psychometric properties of the global rating of change scales in patients with low back pain, upper and lower extremity disorders. A systematic review with meta-analysis. Journal of Orthopaedics, 21, 40-48. https://doi.org/10.1016/j.jor.2020.01.047
Bonello, C., Girdwood, M., De Souza, K., Trinder, N. K., Lewis, J., Lazarczuk, S. L., Gaida, J. E., Docking, S. I., & Rio, E. K. (2021). Does isometric exercise result in exercise induced hypoalgesia in people with local musculoskeletal pain? A systematic review. Physical Therapy in Sport: Official Journal of the Association of Chartered Physiotherapists in Sports Medicine, 49, 51-61. https://doi.org/10.1016/j.ptsp.2020.09.008
Buryk-Iggers, S., Mittal, N., Santa Mina, D., Adams, S. C., Englesakis, M., Rachinsky, M., Lopez-Hernandez, L., Hussey, L., McGillis, L., McLean, L., Laflamme, C., Rozenberg, D., & Clarke, H. (2022). Exercise and Rehabilitation in People With Ehlers-Danlos Syndrome: A Systematic Review. Archives of Rehabilitation Research and Clinical Translation, 4(2), 100189. https://doi.org/10.1016/j.arrct.2022.100189
Cagnie, B., Coppieters, I., Denecker, S., Six, J., Danneels, L., & Meeus, M. (2014). Central sensitization in fibromyalgia? A
systematic review on structural and functional brain MRI. Seminars in Arthritis and Rheumatism, 44(1), 68-75. https://doi.org/10.1016/j.semarthrit.2014.01.001
Çakır, T., Evcik, F. D., Subaşı, V., Demirdal, Ü. S., & Kavuncu, V. (2015). Investigation of the H reflexes, F waves and sympathetic skin response with electromyography (EMG) in patients with stroke and the determination of the relationship with functional capacity. Acta Neurologica Belgica, 115(3), 295-301. https://doi.org/10.1007/s13760-014-0397-5
Charkoudian, N. (2003). Skin blood flow in adult human thermoregulation: How it works, when it does not, and why. Mayo Clinic Proceedings, 78(5), 603-612. https://doi.org/10.4065/78.5.603
Chesterton, L. S., Sim, J., Wright, C. C., & Foster, N. E. (2007). Interrater reliability of algometry in measuring pressure pain thresholds in healthy humans, using multiple raters. The Clinical Journal of Pain, 23(9), 760-766. https://doi.org/10.1097/AJP.0b013e318154b6ae
Clausen, J. P. (1977). Effect of physical training on cardiovascular adjustments to exercise in man. Physiological Reviews, 57(4), 779-815. https://doi.org/10.1152/physrev.1977.57.4.779
de Abreu, S. B., Lenhard, A., Mehanna, A., de Souza, H. C. D., Correa, F. M. de A., Hasser, E. M., & Martins-Pinge, M. C. (2009). Role of paraventricular nucleus in exercise training-induced autonomic modulation in conscious rats. Autonomic Neuroscience: Basic & Clinical, 148(1-2), 28-35. https://doi.org/10.1016/j.autneu.2009.02.007
De Wandele, I., Rombaut, L., Leybaert, L., Van de Borne, P., De Backer, T., Malfait, F., De Paepe, A., & Calders, P. (2014). Dysautonomia and its underlying mechanisms in the hypermobility type of Ehlers-Danlos syndrome. Seminars in Arthritis and Rheumatism, 44(1), 93-100. https://doi.org/10.1016/j.semarthrit.2013.12.006
De Wandele, I., Rombaut, L., Malfait, F., De Backer, T., De Paepe, A., & Calders, P. (2013). Clinical heterogeneity in patients with the hypermobility type of Ehlers-Danlos Syndrome. Research in Developmental Disabilities, 34(3), 873-881. https://doi.org/10.1016/j.ridd.2012.11.018
Demmler, J. C., Atkinson, M. D., Reinhold, E. J., Choy, E., Lyons, R. A., & Brophy, S. T. (2019). Diagnosed prevalence of Ehlers-Danlos syndrome and hypermobility spectrum disorder in Wales, UK: A national electronic cohort study and case–control comparison. BMJ Open, 9(11), e031365. https://doi.org/10.1136/bmjopen-2019-031365
Dutkiewicz, J., Szlufik, S., & Friedman, A. (2019). “New methods of assessing autonomic disorders in Parkinson disease patients: Skin-galvanic reaction”. Journal of Neural Transmission, 126(11), 1421-1424. https://doi.org/10.1007/s00702-019-02060-6
Fedorowski, A., & Melander, O. (2013). Syndromes of orthostatic intolerance: A hidden danger. Journal of Internal Medicine, 273(4), 322-335. https://doi.org/10.1111/joim.12021
Figueroa, A., Kingsley, J. D., McMillan, V., & Panton, L. B. (2008). Resistance exercise training improves heart rate variability in women with fibromyalgia. Clinical Physiology and Functional Imaging, 28(1), 49-54. https://doi.org/10.1111/j.1475-097X.2007.00776.x
Fogarty, J. S. (1997). Reactance theory and patient noncompliance. Social Science & Medicine (1982), 45(8), 1277-1288. https://doi.org/10.1016/s0277-9536(97)00055-5
Freeman, R. (2002). The chronic fatigue syndrome is a disease of the autonomic nervous system. Sometimes. Clinical Autonomic Research, 12(4), 231-233. https://doi.org/10.1007/s10286-002-0058-2
Fu, Q., & Levine, B. D. (2018). Exercise and Non-Pharmacological Treatment of POTS. Autonomic neuroscience : basic & clinical, 215, 20-27. https://doi.org/10.1016/j.autneu.2018.07.001
Hinrichs, R., Michopoulos, V., Winters, S., Rothbaum, A. O., Rothbaum, B. O., Ressler, K. J., & Jovanovic, T. (2017). Mobile assessment of heightened skin conductance in posttraumatic stress disorder: H INRICHS ET AL . Depression and Anxiety, 34(6), 502-507. https://doi.org/10.1002/da.22610
Jackson, K., Silva, H. M. V., Zhang, W., Michelini, L. C., & Stern, J. E. (2005). Exercise Training Differentially Affects Intrinsic Excitability of Autonomic and Neuroendocrine Neurons in the Hypothalamic Paraventricular Nucleus. Journal of Neurophysiology, 94(5), 3211-3220. https://doi.org/10.1152/jn.00277.2005
Jakobsen, M. D., Sundstrup, E., Persson, R., Andersen, C. H., & Andersen, L. L. (2014). Is Borg’s perceived exertion scale a useful indicator of muscular and cardiovascular load in blue-collar workers with lifting tasks? A cross-sectional workplace study. European Journal of Applied Physiology, 114(2), 425-434. https://doi.org/10.1007/s00421-013-2782-9
Ke, J.-Q., Shao, S.-M., Zheng, Y.-Y., Fu, F.-W., Zheng, G.-Q., & Liu, C.-F. (2017). Sympathetic skin response and heart rate variability in predicting autonomic disorders in patients with Parkinson disease. Medicine, 96(18), e6523. https://doi.org/10.1097/MD.0000000000006523
Kosek, E., & Hansson, P. (1997). Modulatory influence on somatosensory perception from vibration and heterotopic noxious conditioning stimulation (HNCS) in fibromyalgia patients and healthy subjects. Pain, 70(1), 41-51. https://doi.org/10.1016/s0304-3959(96)03295-2
Loew, L., Brosseau, L., Kenny, G. P., Durand-Bush, N., Poitras, S., De Angelis, G., & Wells, G. A. (2017). An evidence-based walking program among older people with knee osteoarthritis: The PEP (participant exercise preference) pilot randomized controlled trial. Clinical Rheumatology, 36(7), 1607-1616. https://doi.org/10.1007/s10067-017-3606-9
Malfait, F., Francomano, C., Byers, P., Belmont, J., Berglund, B., Black, J., Bloom, L., Bowen, J. M., Brady, A. F., Burrows, N. P., Castori, M., Cohen, H., Colombi, M., Demirdas, S., De Backer, J., De Paepe, A., Fournel-Gigleux, S., Frank, M., Ghali, N., … Tinkle, B. (2017). The 2017 international classification of the Ehlers–Danlos syndromes. American Journal of Medical Genetics Part C: Seminars in Medical Genetics, 175(1), 8-26. https://doi.org/10.1002/ajmg.c.31552
Martinez-Lavin, M. (2007). Biology and therapy of fibromyalgia. Stress, the stress response system, and fibromyalgia. Arthritis Research & Therapy, 9(4), 216. https://doi.org/10.1186/ar2146
Martínez-Lavín, M., & Hermosillo, A. G. (2000). Autonomic nervous system dysfunction may explain the multisystem features of fibromyalgia. Seminars in Arthritis and Rheumatism, 29(4), 197-199. https://doi.org/10.1016/S0049-0172(00)80008-6
Martins-Pinge, M. C. (2011). Cardiovascular and autonomic modulation by the central nervous system after aerobic exercise training. Brazilian Journal of Medical and Biological Research = Revista Brasileira De Pesquisas Medicas E Biologicas, 44(9), 848-854. https://doi.org/10.1590/s0100-879x2011007500102
Martins-Pinge, M. C., Becker, L. K., Garcia, M. R. L., Zoccal, D. B., Neto, R. V., Basso, L. S., de Souza, H. C. D., & Lopes, O. U. (2005). Attenuated pressor responses to amino acids in the rostral ventrolateral medulla after swimming training in conscious rats. Autonomic Neuroscience: Basic & Clinical, 122(1-2), 21-28. https://doi.org/10.1016/j.autneu.2005.07.007
McCrae, C. S., Craggs, J. G., Curtis, A. F., Nair, N., Kay, D., Staud, R., Berry, R. B., & Robinson, M. E. (2022). Neural activation changes in response to pain following cognitive behavioral therapy for patients with comorbid fibromyalgia and insomnia: A pilot study. Journal of Clinical Sleep Medicine: JCSM: Official Publication of the American Academy of Sleep Medicine, 18(1), 203-215. https://doi.org/10.5664/jcsm.9540
Moss, P., Sluka, K., & Wright, A. (2007). The initial effects of knee joint mobilization on osteoarthritic hyperalgesia. Manual Therapy, 12(2), 109-118. https://doi.org/10.1016/j.math.2006.02.009
Nelson, R., & Petersen, C. (2017). The reliability and minimal detectable change of the cardiovascular response and self-selected exercise intensity during forward and backward treadmill exercise in individuals with Parkinson disease. SAGE Open Medicine, 5, 2050312117736229. https://doi.org/10.1177/2050312117736229
Parsaik, A., Allison, T. G., Singer, W., Sletten, D. M., Joyner, M. J., Benarroch, E. E., Low, P. A., & Sandroni, P. (2012). Deconditioning in patients with orthostatic intolerance. Neurology, 79(14), 1435-1439. https://doi.org/10.1212/WNL.0b013e31826d5f95
Ra, W., T, C., Cm, S., & Pa, V. (2022, agosto 7). Selected Heritable Disorders of Connective Tissue and Disability. PubMed. https://doi.org/10.17226/26431
Ramos, J. S., Dalleck, L. C., Borrani, F., Beetham, K. S., Mielke, G. I., Dias, K. A., Wallen, M. P., Keating, S. E., Fassett, R. G., & Coombes, J. S. (2017). High-intensity interval training and cardiac autonomic control in individuals with metabolic syndrome: A randomised trial. International Journal of Cardiology, 245, 245-252. https://doi.org/10.1016/j.ijcard.2017.07.063
Reychler, G., De Backer, M.-M., Piraux, E., Poncin, W., & Caty, G. (2021). Physical therapy treatment of hypermobile Ehlers-Danlos syndrome: A systematic review. American Journal of Medical Genetics. Part A, 185(10), 2986-2994. https://doi.org/10.1002/ajmg.a.62393
Rombaut, L., Malfait, F., Cools, A., De Paepe, A., & Calders, P. (2010). Musculoskeletal complaints, physical activity and health-related quality of life among patients with the Ehlers–Danlos syndrome hypermobility type. Disability and Rehabilitation, 32(16), 1339-1345. https://doi.org/10.3109/09638280903514739
Roque Marçal, I., Teixeira Do Amaral, V., Fernandes, B., Martins de Abreu, R., Alvarez, C., Veiga Guimarães, G., Cornelissen, V. A., & Gomes Ciolac, E. (2022). Acute high-intensity interval exercise versus moderate-intensity continuous exercise in heated water-based on hemodynamic, cardiac autonomic, and vascular responses in older individuals with hypertension. Clinical and Experimental Hypertension (New York, N.Y.: 1993), 44(5), 427-435. https://doi.org/10.1080/10641963.2022.2065288
Sarma, S., & Levine, B. D. (2016). Beyond the Bruce Protocol: Advanced Exercise Testing for the Sports Cardiologist. Cardiology Clinics, 34(4), 603-608. https://doi.org/10.1016/j.ccl.2016.06.009
Schachter, C. L., Busch, A. J., Peloso, P. M., & Sheppard, M. S. (2003). Effects of short versus long bouts of aerobic exercise in sedentary women with fibromyalgia: A randomized controlled trial. Physical Therapy, 83(4), 340-358.
Skyba, D. A., Radhakrishnan, R., Rohlwing, J. J., Wright, A., & Sluka, K. A. (2003). Joint manipulation reduces hyperalgesia by activation of monoamine receptors but not opioid or GABA receptors in the spinal cord. Pain, 106(1-2), 159-168. https://doi.org/10.1016/s0304-3959(03)00320-8
Sladek, C. D., Michelini, L. C., Stachenfeld, N. S., Stern, J. E., & Urban, J. H. (2015). Endocrine-Autonomic Linkages. Comprehensive Physiology, 5(3), 1281-1323. https://doi.org/10.1002/cphy.c140028
Song, J. S., Yamada, Y., Kataoka, R., Wong, V., Spitz, R. W., Bell, Z. W., & Loenneke, J. P. (2022). Training-induced hypoalgesia and its potential underlying mechanisms. Neuroscience and Biobehavioral Reviews, 141, 104858. https://doi.org/10.1016/j.neubiorev.2022.104858
Staud, R., & Domingo, M. (2001). Evidence for abnormal pain processing in fibromyalgia syndrome. Pain Medicine (Malden, Mass.), 2(3), 208-215. https://doi.org/10.1046/j.1526-4637.2001.01030.x
Sutton, R., Fedorowski, A., Olshansky, B., Gert van Dijk, J., Abe, H., Brignole, M., de Lange, F., Kenny, R. A., Lim, P. B., Moya, A., Rosen, S. D., Russo, V., Stewart, J. M., Thijs, R. D., & Benditt, D. G. (2021). Tilt testing remains a valuable asset. European Heart Journal, 42(17), 1654-1660. https://doi.org/10.1093/eurheartj/ehab084
Tanaka, H., Monahan, K. D., & Seals, D. R. (2001). Age-predicted maximal heart rate revisited. Journal of the American College of Cardiology, 37(1), 153-156. https://doi.org/10.1016/s0735-1097(00)01054-8
van Campen, C. (Linda) M. C., Rowe, P. C., & Visser, F. C. (2021). Deconditioning does not explain orthostatic intolerance in ME/CFS (myalgic encephalomyelitis/chronic fatigue syndrome). Journal of Translational Medicine, 19(1), 193. https://doi.org/10.1186/s12967-021-02819-0
van de Borne, P., Montano, N., Pagani, M., Oren, R., & Somers, V. K. (1997). Absence of low-frequency variability of sympathetic nerve activity in severe heart failure. Circulation, 95(6), 1449-1454. https://doi.org/10.1161/01.cir.95.6.1449
Vicenzino, B., Collins, D., & Wright, A. (1996). The initial effects of a cervical spine manipulative physiotherapy treatment on the pain and dysfunction of lateral epicondylalgia. Pain, 68(1), 69-74. https://doi.org/10.1016/S0304-3959(96)03221-6
Voogt, L., de Vries, J., Meeus, M., Struyf, F., Meuffels, D., & Nijs, J. (2015). Analgesic effects of manual therapy in patients with musculoskeletal pain: A systematic review. Manual Therapy, 20(2), 250-256. https://doi.org/10.1016/j.math.2014.09.001
Wewege, M. A., & Jones, M. D. (2021). Exercise-Induced Hypoalgesia in Healthy Individuals and People With Chronic Musculoskeletal Pain: A Systematic Review and Meta-Analysis. The Journal of Pain, 22(1), 21-31. https://doi.org/10.1016/j.jpain.2020.04.003
Yang, L., Li, W., Xie, Y., Ma, S., Zhou, X., Huang, X., & Tan, S. (2023). Impact of dysautonomic symptom burden on the quality of life in Neuromyelitis optica spectrum disorder patients. BMC Neurology, 23(1), 112. https://doi.org/10.1186/s12883-023-03162-1