The management of neuropathic pain in children poses an enormous challenge for pediatricians and pain management physicians. Current treatment options include physical therapy and medication management. Peripheral nerve stimulation/neuromodulation is a novel and minimally invasive treatment that can be initiated and monitored in an outpatient setting. This new technology can be a useful tool for treating pain secondary to pediatric neuropathy and chronic refractory pain after conservative management. Our case report describes the use of a peripheral nerve stimulator in the successful management of pediatric neuropathic leg pain.

Chronic neuropathic pain is caused by pathology of the somatosensory system that persists beyond 3 months. It can have multiple deleterious effects in children on growth, sleep, and quality of life because of debilitating pain.1  Epidemiologic studies report that 5% to 15% of children suffer from chronic pain–related debility.2  Despite great progress in pediatric pain research and management, many children continue to suffer because of insufficient treatment of pain. The medical community continues to place a greater weight on pain management in adult populations. Pediatric pain is too frequently misconstrued, underdiagnosed, and undertreated.1,2 

Peripheral nerve stimulation (PNS) has been used in adults for the treatment of several conditions when neuropathic pain continues despite adequate use of conventional therapies such as physical therapy, medications, transcutaneous electrical stimulations, and nerve blocks.3  Some of these conditions include pain from postherpetic neuralgia, complex regional pain syndrome, cranial neuralgia, arthritic back pain, quadriceps tendon rupture, and lumbar radiculopathy.410  Its use in children, however, has not been systematically studied or anecdotally reported. Here, we present the management of a 15-year-old boy with chronic and refractory neuropathic leg pain with a saphenous PNS.

A 15-year-old adolescent male presented to the pain clinic for management of chronic left thigh and knee pain. He had no significant medical history. The pain started 2 years after he was involved in a motor vehicle accident, during which he sustained impact to the lateral aspect of his left knee while riding his bike. He initially presented to an emergency department for evaluation; radiographs were unremarkable. He continued to have pain and followed up with an orthopedic surgeon who ordered magnetic resonance imaging. Imaging showed possible edema around the tibial tubercle; there were no other overt signs of injury. No surgical treatment was offered, and he was referred to our pain clinic. His pain at the time of presentation was in the anterior thigh with radiation to the anteromedial aspect of the left knee. The pain persisted despite conservative therapies that included 6 months of physical therapy, mobility exercises, and the constant use of a sleeve/brace. Additionally, he was taking 1 to 2 ibuprofen tablets daily. These therapies did not decrease the pain or improve the functionality of the limb. He described the pain as constant, sharp, and throbbing. He also reported numbness and tingling in the anterior thigh and medial part of the knee. His pain intensity at presentation, using the Numerical Rating Scale, was 4/10 at rest that increased to 8/10 with activity. His pain limited him from walking more than 20 feet. He did not report any alleviating factors and denied any additional associated symptoms including bowel or bladder changes, saddle anesthesia, recent fever/chills, and weight loss.

The LEEDS assessment of neuropathic symptoms and signs pain scale was performed in the clinic11  (Table 1).

TABLE 1

Leeds Assessment of Neuropathic Symptoms and Signs Pain Scale

A. Pain questionnaire: Think about how your pain has felt over the last week.
Question Patient Response (bold) Score 
1. Does your pain feel like strange, unpleasant sensations in your skin? Words like prickling, tingling, and pins-and-needles might describe these sensations. a. “No, my pain doesn’t really feel like this.” (No = 0)
b. “Yes, I get these sensations quite often” (Yes = 5) 
2. Does your pain make the skin in the painful area look different from normal? Words like mottled or looking more red or pink might describe the appearance. a. “No, my pain does not affect the color of the skin.” (No = 0)
b. “Yes, I’ve noticed that the pain does make my skin look different from normal.” (Yes = 5) 
3. Does your pain make the affected skin abnormally sensitive to touch? Getting unpleasant sensations when lightly stroking the skin or getting pain when wearing tight clothes might describe the abnormal sensitivity. a. “No, my pain doesn’t make my skin abnormally sensitive in that area.” (No = 0)
b. “Yes, my skin seems abnormally sensitive to touch in that area.” (Yes = 3) 
4. Does your pain come on suddenly and in bursts for no apparent reason when you’re still? Words like electric shocks, jumping, and bursting describe these sensations. a. “No, my pain doesn’t really feel like this.” (No = 0)
b. “Yes, I get these sensations quite often.” (Yes = 2) 
5. Does your pain feel as if the skin temperature in the painful area has changed abnormally? Words like hot and burning describe these sensations. a. “No, I don’t really get these sensations.” (No = 0)
b. “Yes, I get these sensations quite often.” (Yes = 1) 
B. Sensory testing 
Skin sensitivity can be examined by comparing the painful area with the contralateral or adjacent nonpainful area for the presence of allodynia and an altered pinprick threshold (PPT). 
Examination Response Score 
1. Allodynia. Examine the response to lightly stroking cotton wool across the nonpainful area and then the painful area. If normal sensations are experienced in the nonpainful site, but pain or unpleasant sensations (tingling, nausea) are experienced in the painful area when stroking, allodynia is present. a. No, normal sensation in both areas. (No = 0)
b. Yes, allodynia in the painful area only. (Yes = 5) 
2. Altered PPT. Determine the pinprick threshold by comparing the response to a 23-gauge (blue) needle mounted inside a 2-mL syringe barrel placed gently onto the skin in nonpainful and then painful areas. If a sharp pinprick is felt in the nonpainful area, but a different sensation is experienced in the painful area (eg, none/blunt only [raised PPT] or very painful sensation [lowered PPT]), an altered PPT is present. If a pinprick is not felt in either area, mount the syringe onto the needle to increase the weight and repeat. a. No, equal sensation in both areas. (No = 0)
b. Yes, altered PPT in the painful area. (Yes = 3) 
Scoring: Add values for sensory description and examination findings to obtain an overall score. total score (maximum 24) 15 
A. Pain questionnaire: Think about how your pain has felt over the last week.
Question Patient Response (bold) Score 
1. Does your pain feel like strange, unpleasant sensations in your skin? Words like prickling, tingling, and pins-and-needles might describe these sensations. a. “No, my pain doesn’t really feel like this.” (No = 0)
b. “Yes, I get these sensations quite often” (Yes = 5) 
2. Does your pain make the skin in the painful area look different from normal? Words like mottled or looking more red or pink might describe the appearance. a. “No, my pain does not affect the color of the skin.” (No = 0)
b. “Yes, I’ve noticed that the pain does make my skin look different from normal.” (Yes = 5) 
3. Does your pain make the affected skin abnormally sensitive to touch? Getting unpleasant sensations when lightly stroking the skin or getting pain when wearing tight clothes might describe the abnormal sensitivity. a. “No, my pain doesn’t make my skin abnormally sensitive in that area.” (No = 0)
b. “Yes, my skin seems abnormally sensitive to touch in that area.” (Yes = 3) 
4. Does your pain come on suddenly and in bursts for no apparent reason when you’re still? Words like electric shocks, jumping, and bursting describe these sensations. a. “No, my pain doesn’t really feel like this.” (No = 0)
b. “Yes, I get these sensations quite often.” (Yes = 2) 
5. Does your pain feel as if the skin temperature in the painful area has changed abnormally? Words like hot and burning describe these sensations. a. “No, I don’t really get these sensations.” (No = 0)
b. “Yes, I get these sensations quite often.” (Yes = 1) 
B. Sensory testing 
Skin sensitivity can be examined by comparing the painful area with the contralateral or adjacent nonpainful area for the presence of allodynia and an altered pinprick threshold (PPT). 
Examination Response Score 
1. Allodynia. Examine the response to lightly stroking cotton wool across the nonpainful area and then the painful area. If normal sensations are experienced in the nonpainful site, but pain or unpleasant sensations (tingling, nausea) are experienced in the painful area when stroking, allodynia is present. a. No, normal sensation in both areas. (No = 0)
b. Yes, allodynia in the painful area only. (Yes = 5) 
2. Altered PPT. Determine the pinprick threshold by comparing the response to a 23-gauge (blue) needle mounted inside a 2-mL syringe barrel placed gently onto the skin in nonpainful and then painful areas. If a sharp pinprick is felt in the nonpainful area, but a different sensation is experienced in the painful area (eg, none/blunt only [raised PPT] or very painful sensation [lowered PPT]), an altered PPT is present. If a pinprick is not felt in either area, mount the syringe onto the needle to increase the weight and repeat. a. No, equal sensation in both areas. (No = 0)
b. Yes, altered PPT in the painful area. (Yes = 3) 
Scoring: Add values for sensory description and examination findings to obtain an overall score. total score (maximum 24) 15 

If the score <12, neuropathic mechanisms are unlikely to be contributing to the patient’s pain. If the score >12, neuropathic mechanisms are likely to be contributing to the patient’s pain.

On physical examination, we identified no swelling, ecchymosis, or deformity over the left knee and thigh, but noted tenderness along the mid-third of the tibial crest and diffusely over the medial and anterior aspects of the knee. We detected no tenderness over the fibular head or the medial and lateral joint lines of the knee. He demonstrated full active range of motion with knee flexion and extension without eliciting his typical pain. There was increased pain with tapping over the saphenous nerve at the mid-thigh. Strength testing was 5 of 5 for knee flexion and extension bilaterally. Sensation was intact to light touch throughout the left lower extremity. Further sensory testing was as described in the LEEDs questionnaire. Physical examinations also included performance of McMurray test, Lachman test, valgus/varus stress tests, and anteroposterior drawer tests, all of which were negative.

The patient then underwent a saphenous nerve block with steroids (10 mg dexamethasone and 5 mL 0.25% bupivacaine), which provided 2 weeks of 100% pain relief. Using ultrasound guidance, a peripheral nerve stimulator (SPRINT, SPR Therapeutics, Inc., Cleveland, OH) was subsequently implanted on the left saphenous nerve for 60 days (Fig 14). The patient tolerated the procedure well and was given instructions to cover the lead exit site with a plastic dressing when showering to minimize accidental lead dislodgement. At his 2-month follow-up, the PNS leads were removed. He reported significant pain relief and functional improvement at that time. Specifically, he described being able to participate in family activities, ride his bike, and run with friends, all of which were hindered before initiation of PNS. His updated Numerical Rating Scale (NRS) pain score had fallen to 1/10 at rest and 3-4/10 with activity. The pediatric pain disability index was then performed12,13  (Table 2).

FIGURE 1

Ultrasound image using a linear array transducer demonstrating the anatomy of the saphenous nerve and final stimulating electrode position. FA, femoral artery; FV, femoral vein; LAT, lateral; MED, medial; SN, saphenous nerve; SR, sartorius muscle; VM, vastus medialis. *Final position of the stimulating electrode.

FIGURE 1

Ultrasound image using a linear array transducer demonstrating the anatomy of the saphenous nerve and final stimulating electrode position. FA, femoral artery; FV, femoral vein; LAT, lateral; MED, medial; SN, saphenous nerve; SR, sartorius muscle; VM, vastus medialis. *Final position of the stimulating electrode.

Close modal
FIGURE 2

Image demonstrating the size of fine thread coiled lead.

FIGURE 2

Image demonstrating the size of fine thread coiled lead.

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FIGURE 3

Image demonstrating the implanted lead at the skin after being trimmed and secured.

FIGURE 3

Image demonstrating the implanted lead at the skin after being trimmed and secured.

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FIGURE 4

Image demonstrating the pulse generator mounted on the body (side of the hip) using an adhesive hydrogel pad.

FIGURE 4

Image demonstrating the pulse generator mounted on the body (side of the hip) using an adhesive hydrogel pad.

Close modal
TABLE 2

Pediatric Pain Disability Index

If in pain, how often does it bother you in the following activities?
(1-never, 2-not often, 3-sometimes, 4-often, 5-all the time)
Activities Pre Post 
1. Family time 
2. Eating 
3. Meeting friends 
4. Sports 
5. Sleep 
6. Television 
7. Reading 
8. Homework 
9. School 
10. Going to movies 
11. Doing something you like (riding a bike, playing with a friend) 
12. Doing something you don’t like (gym) 
If in pain, how often does it bother you in the following activities?
(1-never, 2-not often, 3-sometimes, 4-often, 5-all the time)
Activities Pre Post 
1. Family time 
2. Eating 
3. Meeting friends 
4. Sports 
5. Sleep 
6. Television 
7. Reading 
8. Homework 
9. School 
10. Going to movies 
11. Doing something you like (riding a bike, playing with a friend) 
12. Doing something you don’t like (gym) 

He had a follow-up visit 1 year after initiation of PNS. His pain score continued to be 1-2/10 at rest and 3/10 with activity. He was able to return to physical activity including running, biking, and attending gym classes; running beyond 30 minutes and squatting caused mild discomfort but was tolerable. In addition, he took up wrestling 5 months after the procedure. He discontinued the use of ibuprofen and reported no complications from PNS.

Our case report presents the successful management of a pediatric patient with peripheral neuropathy with neuromodulation/PNS. Pediatric chronic pain provides significant challenges to physicians for both diagnosis and treatment. In addition, patient communication barriers and psychosocial factors of a family dynamic dealing with their child in pain can further complicate this process.

The assessment and diagnosis of neuropathic pain in children are often based on the design and guidelines created for adults. No specific pediatric guidelines are available.14  Diagnosis of neuropathic pain is based on history and clinical examination; specific laboratory tests are unavailable; therefore, very young children who may have difficulty with verbalization make this diagnosis a challenge. However, evaluations should include character; intensity; temporal aspects of pain including frequency, aggravating, and relieving factors; and response to treatment in conjunction with a thorough physical examination to delineate specific nerve injury. Screening questionnaire tools, such as LEEDS assessment of neuropathic symptoms and signs and Neuropathic Pain Questionnaire, are also useful in the diagnosis of neuropathic pain.11,15  The additive effect of frequent steroid injections for pediatric peripheral nerve injury has not been studied. In our patient, a repeat saphenous nerve block would not have provided additional benefit. The initial block provided significant short-term relief, so further diagnostic information was not necessary. A small amount of steroid is added in the block in anticipation of long-lasting pain relief, but the extended relief period was only 2 weeks in duration and therefore was also not a reasonable therapeutic option. Electroneuromyography, skin biopsy, and neuroimaging modalities may help in acute injury but if the diagnosis is highly probable with history and examination, these modalities are rarely required for clinical decision-making in pediatric chronic pain.16 

Pain management physicians prefer a multimodal treatment approach for pediatric neuropathic pain using a combination of physical therapy, oral medications, and in rare conditions local anesthetic/steroid blocks.17  Oral medications including anti-inflammatories, muscle relaxants, antidepressants, anticonvulsants, and opioids may help in initial management. Drug therapy is often unsatisfactory because the underlying mechanism in the pediatric population is rarely well understood. PNS involves the application of electrical stimulation via stimulating electrodes placed near the affected peripheral nerves. Studies have demonstrated that the PNS system acts via both peripheral and central analgesic mechanisms and has a role in modulating the autonomic nervous system.18  PNS was initially thought to provide an analgesic effect based on the gate-control theory where the electrode stimulates the A-β nerve fibers preferentially, which induces an inhibitory response on the transmission of a signal from pain processing A-δ and C nerve fibers.19  Recent studies have also shown its involvement in modulating the signals in cortical and subcortical areas of the brain.20,21  Neuroinflammatory marker modulation has also been reported, including decreases in proinflammatory cytokines interleukin-1β, interleukin-6, and tumor necrosis factor-α after initiation of PNS.22 

PNS is a minimally invasive treatment of pain that can be initiated and monitored in an outpatient setting. It does not involve medication and therefore potential harmful interactions with other medications are limited. PNS can also be used in combination with physical therapy, psychological support, and medication management, making it a versatile treatment option. There are rare limitations to using PNS. However, implantation of the PNS device has potential risks of infection, bleeding, lead breakage, and incomplete coverage. The duration of the PNS device placement can vary depending on the needs of the patient. It can range from 60 days (as was done with the SPRINT PNS used in our case) to a more permanent implant. We were not able to identify previous published reports of treating pediatric neuropathic pain with PNS; hence, our case provides new information for pediatricians and pain specialists about this subject matter.

In conclusion, PNS is a promising treatment option for pediatric neuropathic pain. However, PNS is not suitable for every child, and the decision to use this treatment should be based on a comprehensive evaluation of the child’s pain levels and response to other treatments. PNS typically should be well tolerated by adolescent children, and the device can be easily removed if the child experiences any adverse reactions. Cognitively delayed children may not have the ability to describe their neuropathic pain or adequately report any adverse effects of nerve stimulation treatment. Young children may not be able to adequately handle and care for the device. Therefore, PNS is not recommended for developmentally/cognitively delayed children and children younger than 10 years of age. Further research is needed to understand the long-term effects of PNS and its applicability in the pediatric population younger than age 10 years. Our case study shows its effectiveness in reducing pain and disability, improving function, and enhancing the quality of life in an adolescent child suffering from neuropathic pain.

Dr Gargya performed the procedure, contributed to the conception and design, collected and interpreted data, drafted the initial manuscript, and critically reviewed and revised the manuscript; Drs Zats and Lake made substantial contributions to the conception and design, acquisition of data, analysis and interpretation of data, helped draft the initial manuscript, and critically reviewed and revised the manuscript; and all authors approved the final manuscript as submitted and agreed to be accountable for all aspects of the work.

Our institution does not require parental or patient approval for reporting individual cases or case series. We obtained written informed consent from the patient’s guardian to include anonymized information in this case report.

FUNDING: No external funding.

CONFLICT OF INTEREST DISCLOSURES: The authors have indicated they have no potential conflicts of interest to disclose.

PNS

peripheral nerve stimulation

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Supplementary data