Generalized hyperalgesia in children and adults diagnosed with hypermobility syndrome and Ehlers-Danlos syndrome hypermobility yype: a discriminative analysis

M. C. Scheper, V. Pacey, L. Rombaut, R. D. Adams, L. Tofts, P. Calders, L. L. Nicholson, R. H. H. Engelbert

Research output: Contribution to journalArticleResearchpeer-review

Abstract

Objective: Lowered pressure-pain thresholds have been demonstrated in adults with Ehlers-Danlos syndrome hypermobility type (EDS-HT), but whether these findings are also present in children is unclear. Therefore, the objectives of the study were to determine whether generalized hyperalgesia is present in children with hypermobility syndrome (HMS)/EDS-HT, explore potential differences in pressure-pain thresholds between children and adults with HMS/EDS-HT, and determine the discriminative value of generalized hyperalgesia. Methods: Patients were classified in 1 of 3 groups: HMS/EDS-HT, hypermobile (Beighton score ≥4 of 9), and healthy controls. Descriptive data of age, sex, body mass index, Beighton score, skin laxity, and medication usage were collected. Generalized hyperalgesia was quantified by the average pressure-pain thresholds collected from 12 locations. Confounders collected were pain locations/intensity, fatigue, and psychological distress. Comparisons between children with HMS/EDS-HT and normative values, between children and adults with HMS/EDS-HT, and corrected confounders were analyzed with multivariate analysis of covariance. The discriminative value of generalized hyperalgesia employed to differentiate between HMS/EDS-HT, hypermobility, and controls was quantified with logistic regression. Results: Significantly lower pressure-pain thresholds were found in children with HMS/EDS-HT compared to normative values (range −22.0% to −59.0%; P ≤ 0.05). When applying a threshold of 30.8 N/cm2 for males and 29.0 N/cm2 for females, the presence of generalized hyperalgesia discriminated between individuals with HMS/EDS-HT, hypermobility, and healthy controls (odds ratio 6.0). Conclusion: Children and adults with HMS/EDS-HT are characterized by hypermobility, chronic pain, and generalized hyperalgesia. The presence of generalized hyperalgesia may indicate involvement of the central nervous system in the development of chronic pain.

LanguageEnglish
Pages421-429
Number of pages9
JournalArthritis Care and Research
Volume69
Issue number3
DOIs
Publication statusPublished - Mar 2017

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Ehlers-Danlos Syndrome
Hyperalgesia
Pain Threshold
Pressure
Chronic Pain
Ehlers-Danlos syndrome type 3
Fatigue
Body Mass Index
Multivariate Analysis
Central Nervous System
Logistic Models
Odds Ratio
Psychology

Cite this

Scheper, M. C. ; Pacey, V. ; Rombaut, L. ; Adams, R. D. ; Tofts, L. ; Calders, P. ; Nicholson, L. L. ; Engelbert, R. H. H. / Generalized hyperalgesia in children and adults diagnosed with hypermobility syndrome and Ehlers-Danlos syndrome hypermobility yype : a discriminative analysis. In: Arthritis Care and Research. 2017 ; Vol. 69, No. 3. pp. 421-429.
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title = "Generalized hyperalgesia in children and adults diagnosed with hypermobility syndrome and Ehlers-Danlos syndrome hypermobility yype: a discriminative analysis",
abstract = "Objective: Lowered pressure-pain thresholds have been demonstrated in adults with Ehlers-Danlos syndrome hypermobility type (EDS-HT), but whether these findings are also present in children is unclear. Therefore, the objectives of the study were to determine whether generalized hyperalgesia is present in children with hypermobility syndrome (HMS)/EDS-HT, explore potential differences in pressure-pain thresholds between children and adults with HMS/EDS-HT, and determine the discriminative value of generalized hyperalgesia. Methods: Patients were classified in 1 of 3 groups: HMS/EDS-HT, hypermobile (Beighton score ≥4 of 9), and healthy controls. Descriptive data of age, sex, body mass index, Beighton score, skin laxity, and medication usage were collected. Generalized hyperalgesia was quantified by the average pressure-pain thresholds collected from 12 locations. Confounders collected were pain locations/intensity, fatigue, and psychological distress. Comparisons between children with HMS/EDS-HT and normative values, between children and adults with HMS/EDS-HT, and corrected confounders were analyzed with multivariate analysis of covariance. The discriminative value of generalized hyperalgesia employed to differentiate between HMS/EDS-HT, hypermobility, and controls was quantified with logistic regression. Results: Significantly lower pressure-pain thresholds were found in children with HMS/EDS-HT compared to normative values (range −22.0{\%} to −59.0{\%}; P ≤ 0.05). When applying a threshold of 30.8 N/cm2 for males and 29.0 N/cm2 for females, the presence of generalized hyperalgesia discriminated between individuals with HMS/EDS-HT, hypermobility, and healthy controls (odds ratio 6.0). Conclusion: Children and adults with HMS/EDS-HT are characterized by hypermobility, chronic pain, and generalized hyperalgesia. The presence of generalized hyperalgesia may indicate involvement of the central nervous system in the development of chronic pain.",
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Generalized hyperalgesia in children and adults diagnosed with hypermobility syndrome and Ehlers-Danlos syndrome hypermobility yype : a discriminative analysis. / Scheper, M. C.; Pacey, V.; Rombaut, L.; Adams, R. D.; Tofts, L.; Calders, P.; Nicholson, L. L.; Engelbert, R. H. H.

In: Arthritis Care and Research, Vol. 69, No. 3, 03.2017, p. 421-429.

Research output: Contribution to journalArticleResearchpeer-review

TY - JOUR

T1 - Generalized hyperalgesia in children and adults diagnosed with hypermobility syndrome and Ehlers-Danlos syndrome hypermobility yype

T2 - Arthritis Care and Research

AU - Scheper, M. C.

AU - Pacey, V.

AU - Rombaut, L.

AU - Adams, R. D.

AU - Tofts, L.

AU - Calders, P.

AU - Nicholson, L. L.

AU - Engelbert, R. H. H.

PY - 2017/3

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N2 - Objective: Lowered pressure-pain thresholds have been demonstrated in adults with Ehlers-Danlos syndrome hypermobility type (EDS-HT), but whether these findings are also present in children is unclear. Therefore, the objectives of the study were to determine whether generalized hyperalgesia is present in children with hypermobility syndrome (HMS)/EDS-HT, explore potential differences in pressure-pain thresholds between children and adults with HMS/EDS-HT, and determine the discriminative value of generalized hyperalgesia. Methods: Patients were classified in 1 of 3 groups: HMS/EDS-HT, hypermobile (Beighton score ≥4 of 9), and healthy controls. Descriptive data of age, sex, body mass index, Beighton score, skin laxity, and medication usage were collected. Generalized hyperalgesia was quantified by the average pressure-pain thresholds collected from 12 locations. Confounders collected were pain locations/intensity, fatigue, and psychological distress. Comparisons between children with HMS/EDS-HT and normative values, between children and adults with HMS/EDS-HT, and corrected confounders were analyzed with multivariate analysis of covariance. The discriminative value of generalized hyperalgesia employed to differentiate between HMS/EDS-HT, hypermobility, and controls was quantified with logistic regression. Results: Significantly lower pressure-pain thresholds were found in children with HMS/EDS-HT compared to normative values (range −22.0% to −59.0%; P ≤ 0.05). When applying a threshold of 30.8 N/cm2 for males and 29.0 N/cm2 for females, the presence of generalized hyperalgesia discriminated between individuals with HMS/EDS-HT, hypermobility, and healthy controls (odds ratio 6.0). Conclusion: Children and adults with HMS/EDS-HT are characterized by hypermobility, chronic pain, and generalized hyperalgesia. The presence of generalized hyperalgesia may indicate involvement of the central nervous system in the development of chronic pain.

AB - Objective: Lowered pressure-pain thresholds have been demonstrated in adults with Ehlers-Danlos syndrome hypermobility type (EDS-HT), but whether these findings are also present in children is unclear. Therefore, the objectives of the study were to determine whether generalized hyperalgesia is present in children with hypermobility syndrome (HMS)/EDS-HT, explore potential differences in pressure-pain thresholds between children and adults with HMS/EDS-HT, and determine the discriminative value of generalized hyperalgesia. Methods: Patients were classified in 1 of 3 groups: HMS/EDS-HT, hypermobile (Beighton score ≥4 of 9), and healthy controls. Descriptive data of age, sex, body mass index, Beighton score, skin laxity, and medication usage were collected. Generalized hyperalgesia was quantified by the average pressure-pain thresholds collected from 12 locations. Confounders collected were pain locations/intensity, fatigue, and psychological distress. Comparisons between children with HMS/EDS-HT and normative values, between children and adults with HMS/EDS-HT, and corrected confounders were analyzed with multivariate analysis of covariance. The discriminative value of generalized hyperalgesia employed to differentiate between HMS/EDS-HT, hypermobility, and controls was quantified with logistic regression. Results: Significantly lower pressure-pain thresholds were found in children with HMS/EDS-HT compared to normative values (range −22.0% to −59.0%; P ≤ 0.05). When applying a threshold of 30.8 N/cm2 for males and 29.0 N/cm2 for females, the presence of generalized hyperalgesia discriminated between individuals with HMS/EDS-HT, hypermobility, and healthy controls (odds ratio 6.0). Conclusion: Children and adults with HMS/EDS-HT are characterized by hypermobility, chronic pain, and generalized hyperalgesia. The presence of generalized hyperalgesia may indicate involvement of the central nervous system in the development of chronic pain.

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