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Published on: January 21, 2020
Plasticity of complex regional pain syndrome (CRPS) in children
1Pain Management Department, Center for Neurological Restoration, Consulting Staff, Children's Hospital CCF Shaker Campus, Pediatric Pain Rehabilitation Program, Cleveland Clinic, Cleveland, Ohio 44195, USA. stantom@ccf.org
Insights
Complex regional pain syndrome I (CRPS I) in children, predominantly affecting young females, shows high remission rates with exercise and behavioral therapy. Diagnosis delays are common, but treatment is effective.
Area of Science:
- Pediatric Pain Management
- Neurology
- Rehabilitation Medicine
Background:
- Complex regional pain syndrome I (CRPS I) is characterized by disproportionate pain, sensory, autonomic, and motor dysfunction, typically after minor trauma.
- While well-documented in adults, CRPS I in children is less reported, with recent data indicating a predominance in females aged 8-16 years.
- Diagnostic delays for pediatric CRPS I can extend up to four months.
Purpose of the Study:
- To review the characteristics, diagnosis, and treatment of Complex regional pain syndrome I (CRPS I) in the pediatric population.
- To highlight the differences and similarities in pathophysiology and clinical presentation between pediatric and adult CRPS I.
- To emphasize the high remission rates achievable with specific therapeutic interventions in children.
Main Methods:
- Literature review focusing on pediatric CRPS I prevalence, clinical presentation, and treatment outcomes.
- Analysis of recent studies comparing pediatric and adult CRPS I pathophysiology.
- Evaluation of therapeutic strategies including exercise, behavioral management, and interventional procedures.
Main Results:
- Pediatric CRPS I predominantly affects females (90%) aged 8-16 years, with a significant diagnostic delay.
- Treatment, particularly exercise therapy combined with behavioral management, achieves a high remission rate of approximately 97% in children.
- Pathophysiology likely involves peripheral and central nervous system mechanisms, with endocrine, behavioral, developmental, and environmental factors influencing presentation.
Conclusions:
- Pediatric CRPS I, while sharing underlying pathophysiology with adult cases, presents distinct clinical features influenced by developmental factors.
- Early diagnosis and a multidisciplinary approach involving exercise therapy and behavioral management are crucial for achieving high remission rates.
- Interventional procedures and continuous analgesia techniques may be necessary for refractory cases or to facilitate initial therapy.
Abstract:
Complex regional pain syndrome I (CRPS I) is defined by the International Association for the Study of Pain (IASP) criteria to include pain that is disproportionate to the inciting event, sensory disturbances such as allodynia/ hyperalgesia, autonomic dysfunction, and motor dysfunction that usually occurs after trauma that is frequently trivial and generally expressed in an extremity. These symptoms are well described in the adult population, but there are relatively few data or reports of its prevalence in the pediatric population. Recent studies have demonstrated that unlike the adult population, about 90% of the cases reported are females in a range of 8 to 16 years, the youngest being 3 years old. There tends to be delay in recognizing the diagnosis, which may be as long as 4 months. In contrast to adults, the response to treatment, particularly exercise therapy with behavioral management will achieve almost 97% remission. While the pathophysiology is poorly understood, many features, particularly the neurologic abnormalities, suggest both peripheral and central nervous system involvement. Peripheral small fiber neuropathy as an etiology and inflammation involving small nerve fibers (neurogenic inflammatory pain) has been suggested. A tissue inflammatory etiology has been investigated over the past 25 years. However, these inflammatory aspects differ from those seen in other conditions involving tissue inflammation. The suggestion that CRPS in children is a different clinical entity than that seen in the adult, is probably incorrect, as recent evidence would suggest that the pathophysiology is most likely identical involving endocrine, behavioral, developmental, and environmental factors that distinguish clinical presentation in children from the adult. Behavioral management is a mandatory accompaniment of any program of exercise therapy and the sometimes extreme sensory disturbances and parental enmeshment do distinguish the clinical presentation from that in the adult. Interventional procedures may be required in the face of extreme allodynia preventing exercise therapy, and in occasional cases interruption of the sympathetic nerves may reverse this symptom in a few children. Occasionally, continuous analgesia techniques such as that which can be delivered by tunneled epidural catheter or an externalized neurostimulator (spinal cord stimulation) for short periods of time are effective.
