Introduction: Complex regional pain syndrome (CRPS) is a disorder characterized by pain as well as a myriad of sensory, autonomic, and motor disturbances. We are reporting a case of child diagnosed with CRPS and successfully treated with supraclavicular brachial plexus catheter infusion of local anesthetic. Case Report: An eight-year-old male underwent a left thoracotomy, repair of esophageal vascular ring, and translocation of the left subclavian to the left carotid artery. Post-operative course was relevant for severe intractable left shoulder and left arm pain associated with allodynia and hyperalgesia. A supraclavicular catheter was inserted, and an infusion of ropivacaine 0.2% was started. The child was sent for physical therapy as he gradually regained all functions of his left arm and resumed his regular activities. Conclusion: Continuous supraclavicular brachial plexus catheter infusion of local anesthetic is a valuable method of reducing pain in severe cases of upper extremity pediatric CRPS and may be safer and more effective than other invasive measures such as sympathetic blocks and epidural catheterization. Further research surrounding the diagnosis and treatment of pediatric CRPS is needed to allow early diagnosis and treatment and to improve outcome.
Complex regional pain syndrome (CRPS) is a disorder characterized by pain, as well as a myriad of sensory, autonomic, and motor disturbances, and represents a significant medical entity [
Management of CPRS in children is multidisciplinary and should aim to provide adequate pain control and appropriate physical therapy. Conservative treatment modalities include physical therapy, behavioral therapy and management with opioids and membrane stabilizers. More invasive approaches such as transcutaneous electrical nerve stimulation (TENS), sympathetic blocks, tunneled epidural catheters, regional nerve blocks, as well as more aggressive surgical approaches may be necessary in recalcitrant cases. In this case we report the use of supraclavicular brachial plexus catheter continuous infusion for the treatment of upper extremity CRPS in an 8-year-old child. This case study details how continuous supraclavicular brachial plexus catheter infusion of local anesthetic for 7 days helped control the child’s pain and allowed for the initiation and maintenance of physical therapy with the eventual restoration of the patient’s normal activity level.
Eight-year-old male underwent a left thoracotomy, repair of esophageal vascular ring, and translocation of the left Subclavian to the left carotid artery. The child sustained a stable intra-operative course. Post-operatively, the patient complained of continuous burning 10/10 pain over his left shoulder with marked limitation of arm movement. The pain progressed to involve the whole left arm with associated allodynia and hyperalgesia on exam. The pain could not be controlled with oral pain medications, membrane stabilizers and IV opioids. X-rays and MRI of the shoulder failed to reveal any pathology of the brachial plexus or the shoulder itself and subsequently the diagnosis of CRPs was made. This was based on clinical finding of allodynia and hyperlagesia, and the presence of severe pain out of proportion that cannot be explained by radiological and EMG findings. The decision was made to perform a left supraclavicular brachial plexus block and catheter insertion for continuous infusion of local anesthetic. Under general anesthesia, using ultrasound and nerve stimulator guidance, a supraclavicular catheter was inserted. An initial bolus of 12 cc mepivacaine 1.5% was injected with adequate spread around the nerve. The catheter was then tunneled and secured. The patient experienced dramatic pain relief after the block, however the next day, the catheter dislodged. At this time, the decision was made to place a tunneled cervical epidural catheter under general anesthesia using fluoroscopy guidance. This procedure was ineffective at controlling the pain. The patient continued to experience significant burning pain in his left arm and shoulder. On account of the significant pain reduction experienced after the initial supraclavicular block, another supraclavicular brachial plexus catheter was inserted under ultrasound guidance. Ropivacaine 0.2% infusion was used with 5 cc basal rate, 3 cc bolus, and 60 minutes lockout intervals. The patient was sent to rehabilitation with the catheter still in place. The catheter was kept in place for seven days with considerable pain relief as the patient started his rehabilitation program.
The child was followed later in surgery clinic after finishing successful rehabilitation program. He gradually regained all functions of his left arm and resumed his regular activities.
The occurrence of CRPS in children was questioned for a long time however several studies and case reports have since come to describe the syndrome and lend more credence to its existence as a clinical entity. Unlike adults, involved children are generally female and develop CRPS following minor trauma. CRPS in the pediatric population, in contrast to adults, mainly involves the lower extremity, especially the foot [
Conservative management via physical therapy and psychotherapy alone (and possibly oral analgesia) may suffice for a majority of patients; however this will not be curative in all cases. Low describes a study of 20 children diagnosed with CRPS at a major children’s hospital over a 4-year period. Although a large percentage of them had resolution of symptoms following intense physiotherapy and psychotherapy, almost 70% of these patients required adjuvant medication to enable them to participate in physiotherapy, 40% required in patient hospital treatment, and 20% had a relapse episode [
A recent study found that pain induced persistent reorganization and hyper-connectivity of the cortical, limbic, and basal ganglia pain circuits in pediatric CRPS patients even after treatment and symptom resolution [
The prognosis and responses to the same therapy vary among children suffering from CRPS [
Sympathetic blocks, commonly stellate ganglion blocks for upper extremity pathology or lumbar sympathetic blocks for lower extremity pathology, are often used in refractory cases of CRPS in adults, and some clinicians have utilized these in recalcitrant pediatric cases as well [
Continuous brachial plexus supraclavicular nerve catheter placement may be a great, cost effective alternative for pain management in recalcitrant upper extremity CRPS. A retrospective study of 25 patients, 17 of whom were diagnosed with CRPS, showed significant improvement in pain and ROM following Bupivacaine brachial plexus injections [
Certainly, there is a need for further research surrounding the diagnosis and treatment of pediatric CRPS. Delays in diagnosis, higher reoccurrence rates in pediatric cases of CRPS than in adults, instances of conservative measure failure, as well as possible biological mechanisms linking inadequate pain control and reoccurrence, all highlight the need for change. It follows that better diagnostic criteria for pediatric CRPS must be developed and that a more comprehensive and at times invasive approach to treating pediatric CRPS may be necessary. This case study illustrates that pediatric CRPS is not a mild clinical entity and that invasive measures may be useful to expedite physical therapy and recovery.
Continuous supraclavicular brachial plexus catheter infusion of local anesthetic is a valuable method of reducing pain in severe cases of upper extremity pediatric CRPS and may be safer and more effective than other invasive measures, such as sympathetic blocks and epidural catheterization. Further research surrounding the diagnosis and treatment of pediatric CRPS is needed to allow early diagnosis and treatment and to improve outcome.