Background & Aims

Juvenile Fibromyalgia (JFM) is a chronic pain condition characterized by persistent widespread musculoskeletal pain, fatigue, and sleep disturbances, which particularly affects adolescent girls (Yunus and Masi, 1985, Kashikar-Zuck, et al., 2016). Adolescents with JFM patients also report multisensory hypersensitivities (Ting et al. 2016, Bennett et al. 2014) and previous studies (López-Solà et al., 2014; 2017; Wilbarger and Cook, 2011; Wang and Frey-Law, 2022) have shown that adult patients with fibromyalgia experience greater sensory sensitivities in response to non-painful sensory stimuli of multiple modalities. A better understanding of multisensory sensitivities and their neurobiological basis may help guide treatment selection on an individualized basis (Wang and Frey-Law, 2022). Here, we investigate whether patients with JFM exhibit augmented unpleasantness to non-painful multisensory stimulation.

Methods

Forty-six adolescent girls (16.56 ± 1.01 years) diagnosed with JFM and forty-four healthy girls (16.09 ± 1.06 years) completed validated measures of multisensory and auditory hypersensitivities in daily life (Adolescent/Adult Sensory Profile and Questionnaire on Hypersensitivity to Sound) (Brown et al., 2001; Nelting et al., 2002; Blasing et al., 2010). We used an auditory task with two tests: US Digits in noise (US DIN) to assess auditory impairments and speech recognition abilities in noise and the Test of Attention in Listening (TAiL) to measure suppression of distracting irrelevant auditory information and conflict resolution. Last, we used a multisensory task involving four trials of alternating 30 seconds of simultaneous non-pain multisensory stimulation (visual, auditory, and tactile-motor finger opposition task) and rest periods ranging from 20-30 seconds in a functional magnetic resonance imaging examination. Two-sample t-tests were used to see the differences between groups.

Results

Compared to healthy participants, JFM patients reported higher levels of multisensory and auditory hypersensitivities in daily life (AASP Auditory: t=6.99, P<0.0001, AASP Visual: t=5.50, P<0.0001, AASP Touch: t=7.06, P<0.0001 and QHS: t=7.45, P<0.0001) and augmented unpleasantness during the multisensory task (t=4.38 P<0.0001). Furthermore, the multisensory and auditory hypersensitivities in daily life reported by JFM patients positively correlated with augmented unpleasantness during the multisensory task. Importantly, there were no significant differences between groups in the auditory task (US DIN: t=0.09, P=0.92, TAiL distraction frequency: t=1.44, P=0.15, TAiL distraction location: t=0.92, P=0.35, TAiL conflict frequency: t=1, P=0.32 and TAiL conflict location: t=1.2, P=0.2), suggesting there are no specific sensory auditory impairments nor problems related with attention in listening.

Conclusions

Our findings support the role of non-painful multisensory increased perceived unpleasantness in JFM. Crucially, the auditory task revealed no basic sensory auditory impairments nor attention problems in listening in JFM patients. Thus, the sensory hypersensitivities might not be directly related to issues in the auditory system, nor in peripheral auditory processing, and points towards a potentially more important role of central processing of auditory input, and maybe even with regards to brain regions dedicated to response to auditory signals (instead of primary sensory processing of auditory input per se). This study highlights the potential role of sensory unpleasantness amplification when studying, diagnosing, and treating JFM.

References

Bennett, R. M., Friend, R., Marcus, D., Bernstein, C., Han, B. K., Yachoui, R., … & Jones, K. D. (2014). Criteria for the diagnosis of fibromyalgia: validation of the modified 2010 preliminary American College of Rheumatology criteria and the development of alternative criteria. Arthritis care & research, 66(9), 1364-1373.

Bläsing L, Goebel G, Flötzinger U, Berthold A, Kröner-Herwig B. Hypersensitivity to sound in
tinnitus patients: an analysis of a construct based on questionnaire and audiological data.
International journal of audiology. 2010;49:518–26.

Brown, C., Tollefson, N., Dunn, W., Cromwell, R., & Filion, D. (2001). The adult sensory profile: Measuring patterns of sensory processing. The American Journal of Occupational Therapy, 55(1), 75-82.

Kashikar-Zuck, S., King, C., Ting, T. V., & Arnold, L. M. (2016). Juvenile fibromyalgia: different from the adult chronic pain syndrome?. Current rheumatology reports, 18(4), 19.

López?Solà, M., Pujol, J., Wager, T. D., Garcia?Fontanals, A., Blanco?Hinojo, L., Garcia?Blanco, S., … & Deus, J. (2014). Altered functional magnetic resonance imaging responses to nonpainful sensory stimulation in fibromyalgia patients. Arthritis & rheumatology, 66(11), 3200-3209.

López-Solà, M., Woo, C. W., Pujol, J., Deus, J., Harrison, B. J., Monfort, J., & Wager, T. D. (2017). Towards a neurophysiological signature for fibromyalgia. Pain, 158(1), 34.

Nelting M, Rienhoff NK, Hesse G, Lamparter U. Die Erfassung des subjektiven Leidens unter
Hyperakusis mit einem Selbstbeurteilungsbogen zur Ger.uschu?berempfindlichkeit (GÜF).
Laryngo-Rhino-Otol 2002;81:327-334.

Ting, T. V., Barnett, K., Lynch-Jordan, A., Whitacre, C., Henrickson, M., & Kashikar-Zuck, S. (2016). 2010 American College of Rheumatology adult fibromyalgia criteria for use in an adolescent female population with juvenile fibromyalgia. The Journal of pediatrics, 169, 181-187.

Wang, D., & Frey-Law, L. A. (2022). Multisensory sensitivity differentiates between multiple chronic pain conditions and pain-free individuals. Pain, 10-1097.

Wilbarger, J. L., & Cook, D. B. (2011). Multisensory hypersensitivity in women with fibromyalgia: implications for well being and intervention. Archives of physical medicine and rehabilitation, 92(4), 653-656.

Yunus, M. B. & Masi, A. T. Juvenile primary fibromyalgia syndrome. A clinical study of thirty-three patients and matched normal controls. Arthritis & Rheumatology 28, 138–145 (1985)

Presenting Author

Laura Martín-Herrero

Poster Authors

Laura Martín Herrero

MSc

University of Barcelona

Lead Author

E-mail

Maria Suñol

PhD

Institut of Neurosciences, Department of Medicine, School of Medicine, University of Barcelona

Lead Author

E-mail

Hannah J. Stewart

PhD

Department of Psychology, Lancaster University, Bailrigg, Lancaster, UK

Lead Author

E-mail

Lucia Blanc

BSc

University of Barcelona

Lead Author

E-mail

Tracy V. Ting

Medical Doctor

University of Cincinnati College of Medicine; Cincinnati Children's Hospital Medical Center, Ohio

Lead Author

E-mail

Jonathan Dudley

PhD

Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio

Lead Author

E-mail

Saül Pascual-Diaz

PhD

University of Barcelona

Lead Author

E-mail

Susmita Kashikar-Zuck

PhD

Cincinnati Children's Hospital Medical Center, Cincinnati, OH, US

Lead Author

E-mail

Robert Coghill

Cincinnati Children's Hospital Med. Ctr.

Lead Author

E-mail

Marina López-Sola

Phd

University of Barcelona

Lead Author

E-mail

Topics

  • Pain Imaging