Letters to the Editor
5. Ip VH, Tsui BC. Continuous interscalene block: the good, the bad and the refined spread. Acta Anaesthesiol Scand. 2012;56:526–530. 6. Tsui BC, Malherbe S, Koller J, Aronyk K. Reversal of an unintentional spinal anesthetic by cerebrospinal lavage. Anesth Analg. 2004;98:434–436.
Management of Neuropathic Pain in an Adolescent With Parsonage-Turner Brachial Plexitis Accepted for Publication: November 29, 2013. To the Editor:
W
e present the case of an adolescent who developed postinfectious Parsonage-Turner brachial plexitis and responded favorably to an antineuropathic regimen and rehabilitation. The patient’s legal guardian has given permission to publish this report. A 15-year-old previously healthy female athlete developed left shoulder/arm pain with paresthesias after a febrile upper respiratory tract infection. Pain intensified and expanded to include her entire left upper extremity. Within 10 days, she developed weakness and near immobility of this limb. Physical examination was negative for allodynia, vasomotor changes, or sudomotor changes. Shoulder mobility was limited, winging of the scapula was noted, and triceps and bicep reflexes were diminished. Magnetic resonance imaging was normal, and electromyography suggested progressive denervation, demonstrating prolonged sensory nerve action potential peak latencies and evidence of long thoracic nerve dysfunction. She was diagnosed as having idiopathic ParsonageTurner syndrome (PTS) and enrolled in physical therapy (PT). Pain was unresponsive to over-the-counter analgesics or opioids, limiting her ability to participate in PT. She was then prescribed pregabalin 25 mg daily and amitriptyline 10 mg nightly. A brachial plexus catheter was
176
Regional Anesthesia and Pain Medicine • Volume 39, Number 2, March-April 2014
considered for analgesia and to facilitate PT1 but was not required because of significant rapid improvement on the antineuropathic regimen. Pregabalin and amitriptyline doses were increased to 25 mg twice daily and 12.5 mg nightly, respectively, with further escalation limited by somnolence. At 2 weeks, the patient showed marked improvement, with a 75% reduction in pain scores; at 3 months, she was able to resume competitive sports. The patient reported no pain and complete resolution of sensory and motor neurologic symptoms at 6 months, with recovery patterns seen on electromyography. Parsonage-Turner syndrome, or neuralgic amyotrophy, presents with abrupt onset of unilateral upper extremity pain and progressive focal neurologic deficits. Symptoms include debilitating neuropathic pain with progression to unilateral limb dysfunction caused by motor weakness. The pathophysiology of idiopathic PTS remains poorly elucidated, although a multifactorial model has been proposed involving infectious, inflammatory, traumatic, and autoimmune factors.2,3 A hereditary form of PTS may be associated with the SEPT9 gene, which seems to be involved in cell division and development of autoimmune disease.4 Parsonage-Turner syndrome is rare in the pediatric population. This patient likely developed PTS because of repetitive impact injury with possible contribution from an autoimmune mechanism after her viral upper respiratory tract infection. Literature review focuses on the genetic, pathophysiologic, diagnostic, and rehabilitative aspects of this disease, with minimal attention given to treatment of pain. We are not aware of articles reporting the efficacy of antineuropathic medications to assist in the management of PTS. Oral corticosteroids may have some benefit in the early phase of this disease, but evidence is inconsistent.5 Diagnosis is usually made several months after the onset of pain, outside the period of corticosteroid efficacy, after evaluation for more common forms of upper extremity pain yields no other diagnosis. Evidence suggests that up to one third of PTS patients may continue to have neuro-
pathic pain and functional limitation for years after onset of symptoms.6 As with other pain syndromes, inadequately treated acute pain is a risk factor for the development of chronic pain and may significantly affect functional capacity. Long-term effects of inadequately treated pain can influence social and family dynamics well after resolution of symptoms. The impressive response of this patient to pregabalin and amitriptyline was apparent within 2 weeks and significantly improved her ability to return to PT and school. Although the response of all adolescents with PTS may not be as favorable, a trial of antineuropathic agents should be considered in the treatment plan. Jenna Helmer Sobey, MD Andrew Franklin, MD Department of Anesthesiology Pediatric Pain Services Monroe Carell Jr. Children’s Hospital at Vanderbilt Vanderbilt University Nashville, TN
The authors declare no conflict of interest. REFERENCES 1. Franklin A, Austin T. The use of a continuous brachial plexus catheter to facilitate inpatient rehabilitation in a pediatric patient with refractory upper extremity complex regional pain syndrome. Pain Pract. 2013;13:109–113. 2. Feinberg J, Radecki J. Parsonage-Turner syndrome. Hss J. 2010;6:199–205. 3. van Alfen N, van Engelen BG, Hughes RA. Treatment for idiopathic and hereditary neuralgic amyotrophy (brachial neuritis). Cochrane Database Syst Rev. 2009;8: CD006976. 4. vanAlfen N. Clinical and pathophysiological concepts of neuralgic amyotrophy. Nat Rev Neurol. 2011;7:315–322. 5. Yamada K, Mano T, Toribe Y, Yanagihara K, Suzuki Y. MRI findings and steroid therapy for neuralgic amyotrophy in children. Pediatr Neurol. 2011;45:200–202. 6. Stutz CM. Neuralgic amyotrophy: Parsonage-Turner syndrome. J Hand Surg Am. 2010;35:2104–2106.
© 2014 American Society of Regional Anesthesia and Pain Medicine
Copyright © 2014 American Society of Regional Anesthesia and Pain Medicine. Unauthorized reproduction of this article is prohibited.
5. Ip VH, Tsui BC. Continuous interscalene block: the good, the bad and the refined spread. Acta Anaesthesiol Scand. 2012;56:526–530. 6. Tsui BC, Malherbe S, Koller J, Aronyk K. Reversal of an unintentional spinal anesthetic by cerebrospinal lavage. Anesth Analg. 2004;98:434–436.
Management of Neuropathic Pain in an Adolescent With Parsonage-Turner Brachial Plexitis Accepted for Publication: November 29, 2013. To the Editor:
W
e present the case of an adolescent who developed postinfectious Parsonage-Turner brachial plexitis and responded favorably to an antineuropathic regimen and rehabilitation. The patient’s legal guardian has given permission to publish this report. A 15-year-old previously healthy female athlete developed left shoulder/arm pain with paresthesias after a febrile upper respiratory tract infection. Pain intensified and expanded to include her entire left upper extremity. Within 10 days, she developed weakness and near immobility of this limb. Physical examination was negative for allodynia, vasomotor changes, or sudomotor changes. Shoulder mobility was limited, winging of the scapula was noted, and triceps and bicep reflexes were diminished. Magnetic resonance imaging was normal, and electromyography suggested progressive denervation, demonstrating prolonged sensory nerve action potential peak latencies and evidence of long thoracic nerve dysfunction. She was diagnosed as having idiopathic ParsonageTurner syndrome (PTS) and enrolled in physical therapy (PT). Pain was unresponsive to over-the-counter analgesics or opioids, limiting her ability to participate in PT. She was then prescribed pregabalin 25 mg daily and amitriptyline 10 mg nightly. A brachial plexus catheter was
176
Regional Anesthesia and Pain Medicine • Volume 39, Number 2, March-April 2014
considered for analgesia and to facilitate PT1 but was not required because of significant rapid improvement on the antineuropathic regimen. Pregabalin and amitriptyline doses were increased to 25 mg twice daily and 12.5 mg nightly, respectively, with further escalation limited by somnolence. At 2 weeks, the patient showed marked improvement, with a 75% reduction in pain scores; at 3 months, she was able to resume competitive sports. The patient reported no pain and complete resolution of sensory and motor neurologic symptoms at 6 months, with recovery patterns seen on electromyography. Parsonage-Turner syndrome, or neuralgic amyotrophy, presents with abrupt onset of unilateral upper extremity pain and progressive focal neurologic deficits. Symptoms include debilitating neuropathic pain with progression to unilateral limb dysfunction caused by motor weakness. The pathophysiology of idiopathic PTS remains poorly elucidated, although a multifactorial model has been proposed involving infectious, inflammatory, traumatic, and autoimmune factors.2,3 A hereditary form of PTS may be associated with the SEPT9 gene, which seems to be involved in cell division and development of autoimmune disease.4 Parsonage-Turner syndrome is rare in the pediatric population. This patient likely developed PTS because of repetitive impact injury with possible contribution from an autoimmune mechanism after her viral upper respiratory tract infection. Literature review focuses on the genetic, pathophysiologic, diagnostic, and rehabilitative aspects of this disease, with minimal attention given to treatment of pain. We are not aware of articles reporting the efficacy of antineuropathic medications to assist in the management of PTS. Oral corticosteroids may have some benefit in the early phase of this disease, but evidence is inconsistent.5 Diagnosis is usually made several months after the onset of pain, outside the period of corticosteroid efficacy, after evaluation for more common forms of upper extremity pain yields no other diagnosis. Evidence suggests that up to one third of PTS patients may continue to have neuro-
pathic pain and functional limitation for years after onset of symptoms.6 As with other pain syndromes, inadequately treated acute pain is a risk factor for the development of chronic pain and may significantly affect functional capacity. Long-term effects of inadequately treated pain can influence social and family dynamics well after resolution of symptoms. The impressive response of this patient to pregabalin and amitriptyline was apparent within 2 weeks and significantly improved her ability to return to PT and school. Although the response of all adolescents with PTS may not be as favorable, a trial of antineuropathic agents should be considered in the treatment plan. Jenna Helmer Sobey, MD Andrew Franklin, MD Department of Anesthesiology Pediatric Pain Services Monroe Carell Jr. Children’s Hospital at Vanderbilt Vanderbilt University Nashville, TN
The authors declare no conflict of interest. REFERENCES 1. Franklin A, Austin T. The use of a continuous brachial plexus catheter to facilitate inpatient rehabilitation in a pediatric patient with refractory upper extremity complex regional pain syndrome. Pain Pract. 2013;13:109–113. 2. Feinberg J, Radecki J. Parsonage-Turner syndrome. Hss J. 2010;6:199–205. 3. van Alfen N, van Engelen BG, Hughes RA. Treatment for idiopathic and hereditary neuralgic amyotrophy (brachial neuritis). Cochrane Database Syst Rev. 2009;8: CD006976. 4. vanAlfen N. Clinical and pathophysiological concepts of neuralgic amyotrophy. Nat Rev Neurol. 2011;7:315–322. 5. Yamada K, Mano T, Toribe Y, Yanagihara K, Suzuki Y. MRI findings and steroid therapy for neuralgic amyotrophy in children. Pediatr Neurol. 2011;45:200–202. 6. Stutz CM. Neuralgic amyotrophy: Parsonage-Turner syndrome. J Hand Surg Am. 2010;35:2104–2106.
© 2014 American Society of Regional Anesthesia and Pain Medicine
Copyright © 2014 American Society of Regional Anesthesia and Pain Medicine. Unauthorized reproduction of this article is prohibited.
