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Interv Pain Med
Interv Pain Med
Interventional Pain Medicine
2772-5944
Elsevier

S2772-5944(22)00107-8
10.1016/j.inpm.2022.100111
100111
Letters to the Editor
Spinal Cord Stimulation as an approach to treating Parsonage Turner Syndrome
Seaton Christopher christophercseaton@gmail.com
∗
Yale-New Haven Hospital, USA
Bushey David david.bushey@yale.edu

Yale-New Haven Hospital, USA
Haddad Daniel daniel.haddad@yale.edu

Yale-New Haven Hospital, USA
Diluna Michael michael.diluna@yale.edu

Yale University, USA
Chow Robert robert.chow@yale.edu

Yale University, USA
∗ Corresponding author. Yale-New Haven Hospital, Department of Anesthesiology, 333 Cedar Street, PO Box 208051, New Haven, CT, 06520-8051, USA christophercseaton@gmail.com
16 6 2022
9 2022
16 6 2022
1 3 1001119 3 2022
24 5 2022
26 5 2022
© 2022 The Authors
2022
https://creativecommons.org/licenses/by-nc-nd/4.0/ This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).
Keywords

Parsonage turner syndrome
Neuralgic amyotrophy
Brachial plexopathy
Spinal cord stimulation
Chronic pain
Neuropathic pain syndrome
Abbreviations

PTS Parsonage Turner Syndrome

NRS Numerical Rating Scale

MMT Manuel Muscle Testing

SCS Spinal Cord Stimulation

TENS Transcutaneous Electrical Nerve Stimulation
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pmcDear Editor

Parsonage Turner Syndrome (PTS) is an uncommon neuritis affecting the brachial plexus characterized by the abrupt onset of severe [numerical rating scale (NRS) ​> ​7], neurogenic pain in the shoulder and proximal upper extremities. The acute pain classically persists for days to weeks and is often followed by atrophic muscle weakness over subsequent weeks to months with rare progression to flaccid paresis. The syndrome was first reported in the literature in 1897 by Feinburg and more widely and eponymously by Parsonage and Turner, who in 1948 described “shoulder-girdle syndrome” in a case series of 136 patients [1,2]. The etiology remains unknown, although most cases are speculated to be idiopathic and immune-mediated with antecedent viral illness, immunization, surgery, or heavy exercise present in roughly 50% of cases [[3], [4], [5]]. The diagnosis of PTS is clinical and supported by electrodiagnostic testing showing asymmetric denervation as well as imaging results to exclude differential pathologies of the cervical spine and brachial plexus [[6], [7], [8]]. The pain classically resolves over days to weeks with recovery of motor function occurring more gradually over the course of months to years; however, recent literature suggests that morbidity from PTS may be more prolonged, underscored by a high rate of recurrence despite initial therapy as well as progression to chronic pain in up to one third of patients [[9], [10], [11]]. While early-stage treatment is largely supportive, there exists no consensus therapy for the subset of patients who progress to chronic pain. Comparative studies are lacking, and current literature is limited to case reports and case series. Spinal cord stimulation (SCS) has been utilized with positive results for the treatment of brachial plexus pathologies in a small number of case reports, however, there exists only a single published description of the use of SCS in treating PTS [[12], [13], [14], [15], [16]]. We present a second case of a patient with chronic pain from PTS responsive to SCS.

A 27-year-old female with a four-year history of PTS following small bowel resection was referred to Pain Management by Neurology for chronic, intractable arm and neck pain. Her postoperative course had been complicated by coma requiring 3-month Neuro ICU stay from which she recalled awakening with severe, debilitating pain and marked weakness of her bilateral upper extremities. She complained of persistent, severe (NRS 9/10) neck pain radiating to the bilateral upper extremities and refractory to gabapentin 300mg TID, pregabalin 50mg TID, and methocarbamol 500mg TID prn (dose-limited by the patient's intolerance of medication side effects) as well as multiple trials of physical therapy and bilateral lower cervical and upper thoracic sympathetic blocks performed during the prior 18 months. Electromyography (EMG) revealed patchy brachial neuritis notable for absent motor and sensory responses in the radial nerves and mildly reduced recruitment of the biceps brachii, brachioradialis, and first dorsal interosseus muscles bilaterally, while serial MRIs demonstrated edema of the cervical nerve roots on T2-weighted imaging without evidence of compressive pathology. Her exam was notable for mild motor weakness [manual muscle testing (MMT) 4/5] bilaterally as well as reduced light touch sensation over the anterolateral forearms and hyperalgesia over the proximal upper extremities, trapezius, and deltoid muscles. Having failed prior interventions as well as ongoing medical therapy with duloxetine 60mg QD, nortriptyline 30mg QHS, and tizanidine 2mg Q8H, she was determined to be a candidate for a trial of cervical SCS. Pre-procedural psychiatric screening was performed, and the patient was cleared for the stimulator trial. The risks and benefits were discussed, and all questions were answered. Informed consent was obtained, and the patient was scheduled for the procedure.

Using 14-gauge insertion needles to access the epidural space at T1-2, 8-contact leads were advanced to the C2-3 interspace on the left and the C3-4 interspace on the right under fluoroscopic guidance using AP, lateral, and oblique views (Fig. 1, Fig. 2). Subsequent testing verified that all the patient's areas of pain were covered during paresthesia mapping, utilizing an amplitude of 1.7 mA, pulse width of 280 ​ms, and frequency of 50 Hz with optimal coverage between C3-4. Our patient reported a subjective 70–80% reduction in her pain post-procedurally as well as on her follow-up appointment 5 days later for percutaneous lead removal. She subsequently underwent bilateral partial laminectomy at C5 and placement of spinal cord stimulator paddle electrodes covering C2-5 (Fig. 3, Fig. 4, Fig. 5) with a consistent 50% reduction in her pain scores (NRS 4–5/10) utilizing traditional paresthesia as well as sub-perception programming for her SCS over a 12-month follow-up period during which time she reported subjectively improved functional capacity owing to reduced sedative side effects after discontinuation of duloxetine and 67% reduction in nortriptyline dosage. There was no change in the patient's upper extremity weakness (MMT 4/5) during the course of her SCS therapy (see Fig. 6).Fig. 1 Cervical percutaneous leads under fluoroscopy during spinal cord stimulation (SCS) trial, AP View.

Fig. 1

Fig. 2 Cervical Percutaneous Leads under Fluoroscopy during Spinal Cord Stimulation (SCS) Trial, Lateral View. The left lead is positioned in the lateral epidural space while the right lead is in the far lateral epidural space.

Fig. 2

Fig. 3 Cervical paddle leads under fluoroscopy during surgical implantation, lateral View.

Fig. 3

Fig. 4 XR Cervical Spine showing C5 Laminectomy and Paddle Leads in the Posterior Epidural Space, Lateral View.

Fig. 4

Fig. 5 XR Cervical and Thoracic Spine, AP View.

Fig. 5

Fig. 6 XR Thoracic Spine showing the Implantable Pulse Generator overlying the Right T10-T11 Posterior Paraspinal Region, AP View.

Fig. 6

PTS is classically self-limited, with pain spontaneously resolving within days to weeks, and an estimated 89% of patients making a complete functional recovery at 3 years [5,17]. Results from more recent literature, however, suggest that morbidity may be more prolonged and underappreciated, as longitudinal studies by van Alfen document a 26% recurrence rate despite initial treatment as well as progression to chronic pain in one third of patients – often with comorbid fatigue, psychological distress, and functional impairment [[9], [10], [11]]. While early-stage treatment remains largely supportive, there exists no consensus therapy for the cohort of PTS patients who progress to chronic pain. Controlled studies are lacking due to the low incidence of the condition in the general population (1.64–3 per 100,000/year), and as mentioned previously, the available literature is limited to case reports and small-number case series [18]. Corticosteroids and neuromodulating medications have not consistently demonstrated efficacy beyond the acute phase, nor does the literature support physical therapy, massage, or transcutaneous electrical nerve stimulation (TENS) as effective in hastening functional recovery [17,19]. Nerve grafting and tendon transfers have been trialed with some recovery of function but without improvement in pain scores [20]. Studies of nerve blocks are likewise mixed, with a 2019 report by Stair et al. documenting a positive response to an interscalene block performed in a patient with PTS, while two earlier case reports describe PTS paradoxically arising after interscalene blocks performed for shoulder surgery [21,22].

The efficacy of SCS for treatment of medically refractory neuropathic pain is well-established in settings of complex regional pain syndrome, failed back surgery syndrome, and peripheral neuropathy; however, reports on the use of SCS for treatment of brachial plexus pathologies remain limited [[12], [13], [14], [15]], and there exists only a single published account of its application in PTS – a 2015 case report by Kim describing a consistent 50% reduction in pain scores with SCS over a 12-month follow-up period accompanied by decreased analgesic requirements and modest functional improvement in a single patient [16]. We present a second case of SCS utilized with positive results for treatment of chronic PTS pain, suggesting that SCS may represent an effective therapy for patients with severe, refractory pain from this condition. It is notable, however, that follow up of our subject was restricted to 12 months after paddle implantation due to her untimely demise from unrelated pathology, and the limited follow-up duration in both cases represents a barrier to assessing the long-term clinical and cost-effectiveness of SCS for the treatment of this disease entity. Furthermore, programming in our patient's case involved only paresthesia waveforms, and hence we did not evaluate burst or high-frequency SCS that have been favorably described, particularly in patients with suboptimal responses to tonic stimulation [13].

In conclusion, PTS is a rare brachial plexopathy with no defined therapy. We present a case of PTS pain responsive to SCS, raising the possibility that the efficacy of SCS for neuropathic pain conditions may extend to PTS, although further studies and lengthier follow-up remain prerequisites to defining the role of neuromodulation in treating brachial plexus pathologies.

FUNDING

This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.

Declaration of competing interests

The authors declare that they have no known competing financial interests or personal relationships that could have appeared to influence the work reported in this paper.
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