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CLINICAL REPORT The Use of a Continuous Brachial Plexus Catheter to Facilitate Inpatient Rehabilitation in a Pediatric Patient with Refractory Upper Extremity Complex Regional Pain Syndrome Andrew Franklin, MD; Thomas Austin, MD Department of Anesthesiology, Division of Pediatric Anesthesiology, Vanderbilt University Medical Center, Nashville, Tennessee, U.S.A. n Abstract Background: The goal of interventional management of refractory pediatric complex regional pain syndrome is to facilitate early restoration of function to the affected extremity. These interventions are more complicated in chil- dren, as most do not tolerate these procedures without sedation. Case report: We report the first detailed description of a pediatric patient with complex regional pain syndrome refractory to medical management who had complete reso- lution of symptoms after brief inpatient rehabilitation involving continuous brachial plexus blockade and a multi- disciplinary approach. Conclusion: Repeated interventional therapy for refrac- tory, severe complex regional pain syndrome may not be feasible in children owing to the requirement for deep sedation or general anesthesia. A multidisciplinary appro- ach of brief inpatient rehabilitation and continuous block- ade via an indwelling pain catheter may provide a safer, more cost-effective means of restoring function in children with advanced disease. n Key Words: pediatric, complex regional pain syndrome, complex regional pain syndrome, continuous brachial plexus blockade, perineural clonidine, inpatient rehabilitation INTRODUCTION Complex regional pain syndrome (CRPS) has histori- cally been underdiagnosed and undertreated in the pediatric population although clinical interest in this important type of neuropathic pain has increased in recent years. The pathophysiology and clinical mani- festations of CRPS in children and adults are quite similar with some key differences such as coexisting Address correspondence and reprints requests to: Andrew Franklin, MD, Department of Anesthesiology, Division of Pediatric Anesthesiology, Monroe Carell Jr. Children’s Hospital at Vanderbilt, 2200 Children’s Way, Suite 3115 Nashville, TN 37232, U.S.A. E-mail: andrew.franklin@ vanderbilt.edu. Submitted: February 13, 2012; Revision accepted: March 25, 2012 DOI. 10.1111/j.1533-2500.2012.00561.x Ó 2012 The Authors Pain Practice Ó 2012 World Institute of Pain, 1530-7085/12/$15.00 Pain Practice, Volume ••, Issue , 2012 ••••

The Use of a Continuous Brachial Plexus Catheter to Facilitate Inpatient Rehabilitation in a Pediatric Patient with Refractory Upper Extremity Complex Regional Pain Syndrome

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CLINICAL REPORT

The Use of a Continuous Brachial

Plexus Catheter to Facilitate

Inpatient Rehabilitation in a

Pediatric Patient with Refractory

Upper Extremity Complex

Regional Pain Syndrome

Andrew Franklin, MD; Thomas Austin, MD

Department of Anesthesiology, Division of Pediatric Anesthesiology, Vanderbilt UniversityMedical Center, Nashville, Tennessee, U.S.A.

n Abstract

Background: The goal of interventional management of

refractory pediatric complex regional pain syndrome is to

facilitate early restoration of function to the affected

extremity. These interventions are more complicated in chil-

dren, as most do not tolerate these procedures without

sedation.

Case report: We report the first detailed description of a

pediatric patient with complex regional pain syndrome

refractory to medical management who had complete reso-

lution of symptoms after brief inpatient rehabilitation

involving continuous brachial plexus blockade and a multi-

disciplinary approach.

Conclusion: Repeated interventional therapy for refrac-

tory, severe complex regional pain syndrome may not be

feasible in children owing to the requirement for deep

sedation or general anesthesia. A multidisciplinary appro-

ach of brief inpatient rehabilitation and continuous block-

ade via an indwelling pain catheter may provide a safer,

more cost-effective means of restoring function in children

with advanced disease. n

Key Words: pediatric, complex regional pain syndrome,

complex regional pain syndrome, continuous brachial

plexus blockade, perineural clonidine, inpatient

rehabilitation

INTRODUCTION

Complex regional pain syndrome (CRPS) has histori-

cally been underdiagnosed and undertreated in the

pediatric population although clinical interest in this

important type of neuropathic pain has increased in

recent years. The pathophysiology and clinical mani-

festations of CRPS in children and adults are quite

similar with some key differences such as coexisting

Address correspondence and reprints requests to: Andrew Franklin,MD, Department of Anesthesiology, Division of Pediatric Anesthesiology,Monroe Carell Jr. Children’s Hospital at Vanderbilt, 2200 Children’s Way,Suite 3115 Nashville, TN 37232, U.S.A. E-mail: [email protected].

Submitted: February 13, 2012; Revision accepted: March 25, 2012DOI. 10.1111/j.1533-2500.2012.00561.x

� 2012 The Authors

Pain Practice � 2012 World Institute of Pain, 1530-7085/12/$15.00

Pain Practice, Volume ••, Issue •, 2012 ••–••

psychological factors1–4 and response to treatment.5,6

Although physical therapy is the cornerstone of multi-

disciplinary treatment of CRPS in both the adult and

pediatric population, the ability of consistent physical

therapy regimens to restore function in children

appears to be greater, possibly due to the plasticity of

their central and peripheral nervous systems.7 The

overall goal in the management of pediatric CRPS is to

restore function, and the pediatric pain physician is

often called upon to provide sufficient analgesia to

facilitate rehabilitation and physical therapy.7 We

present the case of an adolescent girl with refractory

stage 2, type 1 upper extremity CRPS who was suc-

cessfully treated with continuous brachial plexus

blockade and aggressive inpatient rehabilitation. This

process has been briefly mentioned as effective in the

pediatric population,6–8 particularly to facilitate outpa-

tient management.9 Continuous peripheral nerve

blockade has also been used with success in adult

patients with CRPS of the shoulder.10 We provide the

first detailed report of this technique in the pediatric

population and provide confirmation that this

treatment modality is useful in the management of

pediatric CRPS associated with significant functional

impairment.

CASE REPORT

A twelve-year-old, 50-kg, right-handed girl with no

significant medical history sustained a nonoperative

torus mid-shaft right radius fracture during a fall and

was placed in a cast for 6 weeks. After the cast was

removed, the child reported exquisite pain with light

skin touch and the extremity appeared cool, mottled,

and pale. This was initially managed with ibuprofen;

however, the disease progressed rapidly over the next

6 weeks and the child developed right-hand contrac-

tures, hair loss over the forearm, intermittent cyano-

sis, and brittle fingernails. She was prescribed

pregabalin 25 mg once daily and physical therapy.

Debilitating pain and increasing immobility of the

arm caused withdrawal from physical therapy after

only one session. The child was also withdrawn from

school because of frequent reports of excruciating

pain and the inability to write or perform other types

of schoolwork.

On initial evaluation in our pediatric pain clinic,

the child was wearing a long sleeve shirt with the

right shirtsleeve rolled up past her elbow and dis-

played significant guarding during attempts to exam-

ine the arm. There was significant static mechanical

allodynia throughout the right forearm and hand.

This extremity was cool, pale, and mottled with some

cyanosis noted distally with hair loss and brittle, but

intact, fingernails. The pregabalin was sequentially

increased to 50 mg twice daily, and nortriptyline was

eventually titrated to 50 mg once at night. A com-

pounded formulation of topical lidocaine, clonidine,

gabapentin, and ketamine was used three times daily

to provide cutaneous desensitization therapy. Despite

these efforts, the child’s pain remained severe enough

to prevent participation in physical therapy, although

sudomotor and vasomotor changes decreased, and so

the decision was made to proceed with sympathetic

blockade. There were significant socioeconomic limi-

tations, and the child lived over four hours away

from our institution. As a result of the severity of dis-

ease, we felt it unlikely that sustained relief to allow

the patient to return to physical therapy and school

would be attained with single blockade. We felt that

the best plan was to admit the child for 3 days of

inpatient rehabilitation with continuous brachial

plexus blockade.

The child was brought to the operating room, and a

separate anesthesia team administered general anesthe-

sia. An ultrasound-guided, fluoroscopically confirmed

stellate ganglion block was placed easily using a

2-inch, 22-gauge B. Braun Stimuplex D insulated nee-

dle (Figures 1 and 2). The injectate was 7 mL of 0.1%

ropivicaine with clonidine 50 mcg.11 A supraclavicular

brachial plexus catheter was also placed easily using a

Figure 1. Stellate ganglion block, ultrasound image.

2 • FRANKLIN AND AUSTIN

2-inch, 18-gauge B. Braun Contiplex insulated stimu-

lating Tuohy tip needle (Figure 3). No local anesthetic

was used during placement of the brachial plexus cath-

eter, and dextrose-50 was used to dilate the nerve

sheath to facilitate passage of the catheter.12 The child

displayed a mild right ptosis, miosis, and relative

hyperemia of the right upper extremity in the recovery

room and was pain-free. The child was admitted to the

ward under a pediatric hospitalist service. We adminis-

tered a bolus dose of 10 mL of 0.1% ropivicaine

through the catheter, and the child was placed on an

infusion of 0.1% ropivicaine with 0.5 mcg/mL cloni-

dine13 at a rate of 7 mL/hour as well as intravenous

ketorolac every 6 hours. The child was started on

twice daily physical and occupational therapy and was

instructed on self-therapy every three hours while

awake. Oral pregabalin and nortriptyline were contin-

ued during the admission. On the second admission

day, the infusion was further reduced to 0.05% ropivi-

caine with 0.5 mcg/mL clonidine at a rate of 5 mL/

hour, and this continued to provide enough analgesia

to participate in therapy without any sensory or motor

blockade. Consultations were made to the pediatric

psychiatry service and child life specialists to assist

with nonpharmacological, complementary techniques

of pain management including biofeedback, distrac-

tion, music therapy, guided imagery, and relaxation.

The patient was pain-free throughout the hospital

course and was discharged to home on the evening of

hospital day three after removal of the nerve catheter.

The child was immediately able to return to school.

She continued with another 6 weeks of physical ther-

apy with daily self-therapy at home. Six months after

discharge, she was weaned off of her nortriptyline and

pregabalin. She maintains full function of her right

upper extremity and continues to be asymptomatic at

the time of this writing.

DISCUSSION

Successful treatment of pediatric CRPS usually requires

a multidisciplinary approach with the primary objec-

tive being early restoration of function.7,8 Providing

adequate analgesia for the child to participate in reha-

bilitation is often the most difficult, but likely the most

important aspect to management as physical therapy is

often the treatment modality that may lead to resolu-

tion of symptoms. Indeed, some practitioners report

cure rates > 90% when aggressive physical therapy

programs, as much as 6 hours daily, are used as the

sole treatment strategy.14–16 Although each child may

respond differently to different modalities, a balance of

physical therapy, medications, cognitive-behavioral

therapy,17 complementary medicine,18–20 and interven-

tional procedures for refractory cases is likely the best

way to approach this disease. While adults often

require a series of closely spaced sympathetic blocks

for effective treatment, this strategy poses obvious

socioeconomic and safety concerns in the pediatric

population owing to the requirement of sedation or

general anesthesia for the safe performance of inter-

ventions. Although we concede that all children may

not respond as favorably as in the above presentation,

we propose that refractory, severe cases of pediatric

CRPS requiring interventions may be better served

Figure 3. Brachial plexus catheter, ultrasound image.

Figure 2. Stellate ganglion block, fluoroscopic image.

Inpatient Rehabilitation Pediatric CRPS • 3

with inpatient admission for rehabilitation instead of

multiple repeated sedated interventions over the course

of several days to weeks. Although this will vary based

on institution and clinical situation, this technique may

be more cost-effective when factors such as repeated

operating room costs and repeated anesthesia charges

are taken into consideration for children requiring

multiple interventions to restore function. This tech-

nique also reduces parental time off from work and

missed school days, which are important consider-

ations when managing pediatric CRPS. When consid-

ering an indwelling catheter to provide analgesia

during rehabilitation, we assert that upper extremity

continuous peripheral nerve blockade is superior to

continuous cervical or upper thoracic neuraxial block-

ade. Placement of a continuous brachial plexus cathe-

ter allows for unilateral blockade of the affected

extremity, avoids the risk of trauma to the neuraxis,

and is useful when the goal is to provide analgesia to

participate in physical therapy.21 In addition, the

increased safety that ultrasound provides possibly

reduces the risk of placing stellate or brachial plexus

blocks.22–24 As a continuous catheter is often intended

to be left in place for several days to weeks during

rehabilitation, a tunneled peripheral nerve catheter

placed under strict aseptic technique25 may allow for

earlier recognition of localized infection and poses less

potential devastating infectious complications, such as

epidural abscess, when compared with a continuous

neuraxial technique.

CONCLUSION

Refractory pediatric CRPS is often difficult to manage

without interventional therapy. The technique of pair-

ing a diagnostic/therapeutic stellate ganglion block

with an indwelling continuous brachial plexus catheter

to provide sufficient analgesia to participate in inpa-

tient rehabilitation may be a safe and cost-effective

option for this patient population.

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Inpatient Rehabilitation Pediatric CRPS • 5