Anesth Analg 2007;104:1078-1080
© 2007 International Anesthesia Research Society
doi: 10.1213/01.ane.0000260563.39299.9c
PEDIATRIC ANESTHESIA
Intrathecal Local Anesthetic Infusion as a Treatment for Complex Regional Pain Syndrome in a Child
Ibrahim S. Farid, MD, and
Elizabeth J. Heiner, MSN, RN, CNS
From the Division of Pain Medicine, Akron Children's Hospital, Akron, Ohio.
Address correspondence and reprints requests to Dr. Ibrahim Farid, Pain Center, Director, Children's Hospital Medical Center of Akron, 1 Perkin's Square, Akron, Ohio 44308. Address e-mail to ifarid{at}chmca.org.
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Abstract
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Diagnosis and treatment of complex regional pain syndrome in children is one of the most challenging clinical pain problems encountered in the chronic pain setting. Despite the intense and debilitating nature of the pain, referral to appropriate specialists often comes too late, thereby prolonging an already arduous (and controversial) course of treatment and risking long-term disability. On the contrary, full recovery can be expected with early diagnosis and prompt treatment. The following case study describes a novel and successful use of intrathecal catheter administration of ropivacaine in a child with early, rapidly progressing, and debilitating complex regional pain syndrome.
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Introduction
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Complex regional pain syndrome (CRPS), formerly known as reflex sympathetic dystrophy and causalgia, is a debilitating neuropathic pain syndrome composed of sensory, autonomic, and motor changes, which are disproportionate to the expected response of an inciting, often minor, injury (1,2,9). Clinical presentation includes severe pain, allodynia, edema, regional temperature changes, and disuse atrophy. Trophic changes in skin color and texture, hair and nail growth are often observed (3,15). There is considerable variability inpatient presentation, intensity of symptoms, and disease progression. Response to therapy also varies, probably because the etiology and pathophysiology of the syndrome is not well understood (3–5,13), and the response to therapies is also variable. This variable response is likely due to a lack of understanding of the etiology and pathophysiology of the syndrome (3–5).
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CASE REPORT
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An otherwise healthy 8-yr-old, 31 kg girl was referred by an orthopedic surgeon to the Pain Center at Akron Children's Hospital for evaluation and treatment of left foot pain of 6 wk duration. Her pain began without any recalled trauma, accident, or inciting event. She awoke from sleep with bilateral foot pain, which spontaneously resolved in the right foot but progressed in the ankle, sole, and dorsum of the entire left foot. The pain was described as burning in nature, moderate to severe, continuous, and exacerbated with any touch or movement. The pain awakened the child from sleep and restricted her from using her foot in any kind of activity. She used crutches or was pulled in a wagon. Associated signs and symptoms included edema, color and temperature changes. She was noted by the orthopedic surgeon to have some small blisters on the medial and plantar surface of the foot. A history of intermittent "low-grade temperatures" was reported by the mother. Plain radiographs showed diffuse disuse osteopenia. Bone scan revealed decreased bloodflow to the left ankle with decreased tracer in the growth plates on the blood pool, and mild tracer accumulation in the distal left tibia in the metaphyseal and adjacent diaphyseal region. Magnetic resonance imaging showed mild dorsal soft tissue edema, which was thought to be developmental. Her 6-wk treatment history included use of a pneumatic walker, and pharmacologic treatment with Tylenol, ibuprofen, and Tylenol with codeine, none of which provided relief.
On presentation to the Pain Center, the patient was alert and cooperative. She answered all questions appropriately regarding her pain, and did not appear overtly anxious or depressed. General examination was unremarkable, and she had a fully normal neurological evaluation of the upper extremities and the right lower extremity. The left lower extremity demonstrated sympathetically mediated pain signs, including loss of hair, glistening skin, excessive dryness, purple discoloration, excessive sweating of the sole, as well as edema encircling the entire left foot (Fig. 1). She also had allodynia of the left foot and lower leg with very pronounced guarding of the extremity. Based on history and examination, the diagnosis of CRPS Type 1 was established.
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Figure 1. An 8-yr-old child with diagnosis of complex regional pain syndrome (CRPS) on presentation to the pediatric pain center. Sympathetically mediated signs of CRPS include hair loss, glistening of the skin, excessive dryness, purple discoloration and edema, some of which may be seen in the above photo.
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The child was started on nortriptyline, titrating to 25 mg at hs, and later changed to amitriptyline 30 mg at hs due to adverse side effects. Because of absolute touch avoidance due to extreme pain, she was unable to tolerate even minimal physical therapy. This, in addition to the severity and rapid progression of symptoms at presentation, prompted the scheduling of an epidural catheter for infusion of local anesthetic for the following week in order to allow for twice daily physical therapy and daily massage therapy over the course of a week's admission to the hospital. Cognitive-behavioral intervention was also initiated as a part of a multidisciplinary approach to treatment.
Between the initial evaluation and the planned hospitalization, the patient's mother called to report pain developing spontaneously in the right foot, essentially immobilizing her. She had mottling, dryness, and edema of both feet. Oxycodone 0.1 mg/kg/dose was prescribed in the interim, but did not impact the patient's pain.
Under procedural sedation an epidural catheter was placed at the L5–S1 space using loss of resistance technique. An infusion of 0.2% Ropivacaine with fentanyl 2 mcg/mL was started at 0.4 mg/kg/h of Ropivacaine. She continued to complain of severe pain in both feet, though she was "numb" from knee to thigh on the right side. IV morphine, and 5 cc epidural bolus of 0.5% Ropivacaine, did not provide any relief, and the physical therapist remained unable to touch the child's feet. The epidural catheter was replaced at the L4–5 space for improved bilateral anesthetic coverage. This provided complete pain relief to the right foot, but the left foot remained unchanged. After detailed discussion with the patient's parents regarding risks versus benefits, a decision was made to place an indwelling 20 gauge intrathecal catheter at the L4–5 spinal space using an 18 gauge Touhy needle. The catheter was placed by a single needle puncture without the aid of fluoroscopy. Strict aseptic precautions were used, occlusive dressing was applied, and the site was checked at least every 8 h. A dense block to T10 was achieved with 0.5% Ropivacaine, which receded to provide complete analgesia to bilateral feet with intact motor function and strength. Infusion of 0.2% Ropivacaine at 0.03 mg/kg/h was continued without complication for 7 days. She received twice daily physical therapy in the gym, massotherapy daily, and ambulated about the hospital with the aid of a walker. She was also started on gabapentin, which was titrated to 300 mg tid. She was cared for on the general nursing care unit with strict monitoring guidelines and protocols for care of the intrathecal infusion. She had no complications from the procedure or the continuous infusion, and she was discharged home after removal of the catheter after 7 d of treatment. Her pain at discharge was rated 0–3/10, experienced primarily with ambulation, though she could bear her own weight with the assistance of a walker.
After discharge, the patient continued to receive physical therapy three times per week. Over the next 2-mo period, she regained nearly full range of motion, strength, and sensation. She finished school, has returned to her usual level of active play. She has a left-sided limp, but only when she runs. Pain scores are 2/10 in the left foot, 0/10 in the right foot per patient report. Physical therapy continues twice a week; psychotherapy has been discontinued. Amitriptyline and gabapentin will be weaned after approximately 6 mo (Fig. 2).
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Figure 2. Outpatient at the first outpatient follow-up appointment 2 wk after intrathecal infusion of local anesthetic (May 3, 2006). Note the lack of sympathetically mediated signs of complex regional pain syndrome.
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DISCUSSION
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CRPS has a progressive course, with pain and inactivity creating a vicious circle of exaggerated and inappropriate signals sent to the brain and spinal cord, resulting in increasing pain and disuse of the affected extremity (2). Symptoms spread contiguously in all patients, independently (to distant sites) in most patients, and in a mirror-image fashion in a few (1,5). Early diagnosis and treatment are critical for full recovery, whereas delayed diagnosis and treatment may result in prolonged pain, functional impairments, complications, and, potentially, lifelong disability (2,3,11). Associated symptoms of psychological distress result from pain, lack of sleep, and limitations in activity (1,2,6,7). Some researchers have suggested progression through distinct stages (early, dystrophic, and atrophic) (1–3,5,6,14) and whether or not these stages are accurate, staging or subtyping implies a critical period for targeting effective treatment for full recovery (8). In children, there are often delays in treatment because the syndrome is relatively rare and not well understood (2).
Therapeutic modalities for CRPS are multidisciplinary, involving physiotherapeutic, pharmacotherapeutic, and psychotherapeutic strategies, with the goal of pain relief, recovery of function, and relief of psychological suffering (1,3,5,10,16,18). However, a review of the literature reveals that there is considerable variability in approach to treatment.
Clinical features of CRPS in the pediatric population differ from adult CRPS in location, gender, and prognosis. Children are affected in the lower extremities versus upper extremities at a ratio of 6:1, probably because their injuries are most often due to sports and play rather than work-related or due to overuse, as seen in adults. In adults, upper extremities are affected more than the lower extremity by a 2:1 ratio. In children, females are more affected than males (ratio of 7:1), while in adults, the female to male ratio is of 2:1. Finally, the prognosis for complete recovery is much more likely in children with CRPS, whereas long-term disability is relatively common in adults CRPS (2,4,17).
Our purpose in presenting this case study is to promote recognition of CRPS in children and to present a previously unreported and successful treatment option.
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CONCLUSIONS
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This case study offers a novel approach to the treatment of CRPS in a child for whom the standard treatment was not successful. Because of the severity and rapid progression of her presenting symptoms, it was deemed imperative to use an interventional technique immediately, rather than beginning with the more conservative approach of medications and physical therapy. Epidural infusion, though normally successful in providing analgesia for physical therapy, was not effective in this case. The debilitating nature of CRPS and the severity of the pain and resulting immobility mandated that a more aggressive approach be attempted. The intrathecal infusion was successful and this child's CRPS has almost completely resolved.
We were not able to identify any previous reports of intrathecal local anesthetic management of CRPS in children. It is our hope that use of this modality, in conjunction with physical therapy, psychotherapy, and adjuvant drugs may benefit other patients with CRPS who are resistant to standard treatment. Clearly, this technique must be done in a setting with pain management specialists and specifically trained nursing staff to ensure patient safety. Early, comprehensive, and individualized multidisciplinary treatment offers children with CRPS the best opportunity for complete recovery.
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Abstract
Introduction
