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Clinical Phenotypes and the Effects of Interdisciplinary Pain Treatment in Pediatric Complex Regional Pain Syndrome
Violeta Dimova1, Edin T Randall2,3, Amanda Cao3
1Department of Neurology, University Medical Center of the Johannes Gutenberg University Mainz, Mainz, Germany.
Insights
Pediatric complex regional pain syndrome (CRPS) shows phenotypes similar to adults, including peripheral and central types. An intensive interdisciplinary treatment effectively reduced pain and improved function in children with CRPS.
Area of Science:
- Pediatric Pain Medicine
- Neurology
- Rehabilitation Medicine
Background:
- Investigated the replication of adult complex regional pain syndrome (CRPS) phenotypes in pediatric patients.
- Assessed the efficacy of an intensive pain therapeutic approach for pediatric CRPS.
Purpose of the Study:
- To identify and characterize CRPS phenotypes in children.
- To evaluate the impact of a multidisciplinary treatment program on pediatric CRPS symptoms and function.
Main Methods:
- Utilized hierarchical and k-means clustering on neurological examination data from two pediatric CRPS cohorts (N=24 and N=14).
- Quantified phenotype scores for clustering analysis.
- Cohort #2 underwent a 4-6 week intensive program of psychological, physical, and occupational therapy, with pre- and post-treatment assessments.
Main Results:
- Replicated a two-cluster structure of CRPS signs in pediatric patients: 'peripheral' (skin changes, edema) and 'central' (motor, sensory deficits, allodynia).
- K-means clustering identified three phenotypes: 'peripheral' (N=12), 'central' (N=10), and 'mixed' (N=17), mirroring adult findings.
- Treatment demonstrated significant positive effects on pain affect, behavior, psychological distress, depression, pain catastrophizing, physical activity, and strength (Cohen's d = 0.8-1.3).
Conclusions:
- Pediatric CRPS exhibits distinct phenotypes ('peripheral' and 'central') similar to adults, reflecting underlying pathophysiological mechanisms.
- An intensive, interdisciplinary treatment approach is effective in ameliorating pediatric CRPS severity and restoring patient function.
Background:
This investigation aimed to replicate phenotypes in pediatric complex regional pain syndrome (CRPS) previously identified in adult CRPS patients. The impact of an intensive pain therapeutic approach in pediatric CRPS was also evaluated.
Methods:
Neurological examination data were obtained from two samples of children with CRPS (cohort #1; N = 24 and cohort #2; N = 14). The structure among CRPS signs was analyzed on pooled data from cohorts #1 and 2 using hierarchical clustering. A phenotype score was quantified for patients and then submitted to unsupervised k-means clustering. Patients in cohort #2 were enrolled in a 4-6-week treatment program consisting of psychological, physical, and occupational therapy. Questionnaires about pain and mental and physical health were evaluated at treatment admission and discharge.
Results:
A two-cluster structure of CRPS signs could be replicated in the pediatric population. Cluster #1 includes skin color, temperature, and edema resembling peripheral inflammation ("peripheral" phenotype). Cluster #2 grouped together motor disorders, minor injury, glove/stocking-like sensory deficits, and allodynia ("central" phenotype). K-means clustering also showed three phenotypes, "peripheral" (N = 12), "central" (N = 10), and "mixed" (N = 17), similar to adults. The impact of treatment was revealed for pain affect, pain behavior, psychological distress, depression, pain catastrophizing levels, as well as physical activity and strength (Cohen's d = [0.8-1.3]).
Conclusions:
CRPS phenotypes reflecting major pathophysiological mechanisms of peripheral inflammation and central reorganization are identifiable in adult and pediatric patients, suggesting phenomenological features of the disease. An intensive interdisciplinary pain treatment approach was effective in reducing CRPS severity and restoring function in pediatric CRPS.
