BurstDR spinal cord stimulation for chronic regional pain syndrome secondary to Castleman's disease treatment: A case report.

A 32-year-old nulligravid female was referred to pain management for evaluation of 6 months of non-cyclical right lower quadrant abdominal pain radiating to the right posterior thigh, leg, and ankle. Given her history of endometriosis, a computed tomography scan of the abdomen/pelvis was obtained that revealed a 3.4 cm diameter simple, right ovarian cyst and a 4.6 ×3.2 cm dense ovoid structure in the right pelvis, thought to be an endometriotic implant. The non-contrast pelvic magnetic resonance (MR) revealed a well-circumscribed, T1-hypointense, T2-intermediate oval mass (measuring 3.2 × 4.5 × 4.6 cm) located in the deep right retroperitoneum, along the right pelvic sidewall; the differential diagnoses included desmoid tumor, neurogenic tumor, extra-ovarian fibroma, and an enlarged lymph node. She underwent an exploratory laparoscopy with excision of endometriotic lesions, right ovarian cystectomy, bilateral ureterolysis, and right pelvic neurolysis involving the obturator nerve and sacral nerve roots to the Frankenhäuser plexus. The histopathologic and immunochemistry analyses were diagnostic for unicentric hyaline vascular type of Castleman’s disease. The analyses revealed that the right retroperitoneal mass was an enlarged lymph node with prominent mantle zones that had an “onion-skin” appearance. Immunohistochemistry showed cluster of differentiation (CD) 21 positivity within the follicular dendritic cell meshwork and mantle zone cells, and CD138 positivity in scattered polyclonal plasma cells with kappa and lambda in situ hybridization. Postoperatively, although the abdominal pain improved, she developed new, progressively worsening neuropathic pain in the right lower extremity. Pregabalin 100 mg daily and physical therapy were of no benefit. The lumbar MR imaging revealed a mild right-sided L5-S1 disc herniation. She was referred to pain management and was diagnosed with CRPS type 2, likely secondary to intraoperative neurolysis. The patient underwent a percutaneous trial of a burst-mode SCS (Abbott, Plano, TX) using two leads placed at the T8 superior endplate; this resulted in 80% reduction in groin and right lower-extremity pain. She then underwent implantation of a 5-column Penta paddle lead placed through T9 laminectomy, with stimulation parameters set to an intra-burst frequency of 500 Hz, overall frequency of 40 Hz, and 1 ms pulse width [ Figure 1 ]. The patient had an uncomplicated postoperative recovery, and 3 months later, reported sustained, significant improvement in pain/functional status, and no longer required pregabalin therapy. Intraoperative anterior–posterior X-ray with the paddle lead spinal cord stimulator centered at the level of T8.

Castleman’s disease is a rare lymphoproliferative disorder that can cause chronic pain.[ 1 ] (She opted for this with mass resection and neurolysis, which damaged other nerves and caused a complex regional pain syndrome (CRPS) type 2. Ultimately, with CRPS type 2, she achieved pain relief with the placement of a spinal cord stimulator (SCS).

Multiple studies [ Table 1 ] document the efficacy of SCS management of neuropathic pain and CRPS type 2 syndromes. In Molina-Gil et al .’s studies,[ 5 ] a patient developed left lower extremity neuropathic pain/CRPS after laparoscopic gynecologic surgery requiring adhesiolysis involving the rectosigmoid colon, the left uterosacral ligament, and the uterine cervix; it was medically managed for 3 months.[ 5 ] Possover et al .[ 6 ] demonstrated in 21 patients with endometriosis infiltrating the lateral pelvic wall that laparoscopic neurolysis of pelvic somatic nerves, including the sacral plexus and sciatic nerve, was effective.[ 6 ] Similarly, Roviglione et al .[ 7 ] reported 402 consecutive patients with infiltrating endometriosis with recurrent sciatica/anogenital pain treated with surgical neural decompression or neurolysis of sacral roots and pelvic somatic nerves (i.e., 313 patients (77.9%) partial neurolysis and 89 (22.1%) complete neurolysis). Although a significant improvement in neurological symptoms was documented at 6- postoperative months, 78 patients (19.4%) developed postoperative neuritis, necessitating treatment with steroids, antiepileptics, or opioids.[ 7 ] Here, our 40-year-old female, who developed CRPS type 2 neuropathic pain secondary to surgical intervention for Castleman’s disease, was successfully managed with SCS, a modality that would likely be effective for comparable patients in the future. Summary of studies that have demonstrated the efficacy of SCS for neuropathic pain.

A 32-year-old female with Castleman’s disease developed CRPS type 2 secondary to neurolysis that was effectively treated with Burst SCS.