Short-term spinal cord stimulation for refractory Elsberg syndrome in diabetic neuropathy: A case report

Abstract

BACKGROUND

Elsberg syndrome is a type of postinfectious lumbosacral radiculitis typically triggered by neurotropic viruses and manifests as bladder/bowel dysfunction, saddle sensory disturbances (including hypoesthesia, hyperesthesia, or dysesthesia), and variable neurological deficits. Typically self-limiting, it often responds to antiviral and neurotropic therapies. However, in patients with comorbidities that confer susceptibility to peripheral neuropathy (e.g., diabetes mellitus), timely escalation to neuromodulation strategies, such as spinal cord stimulation, may be warranted to optimize functional outcomes when conservative measures are inadequate.

CASE SUMMARY

A 60-year-old male with diabetes mellitus presented with severe bladder and bowel dysfunction persisting for more than two months, followed by left gluteal and perianal (saddle area) herpes zoster eruption that was accompanied by significant neuropathic pain. Following a suboptimal response to conservative therapy, the patient underwent implantation of a short-term spinal cord stimulation. Following a 10-day trial of continuous tonic stimulation, the percutaneous electrode lead was removed. The patients experienced no surgical complications, and after the procedure, the patient achieved complete restoration of bladder and bowel function and significant pain alleviation. Two-month follow-up confirmed sustained full recovery.

CONCLUSION

Early implementation of short-term spinal cord stimulation represents a promising therapeutic approach for promoting neurological recovery in patients with Elsberg syndrome refractory to conservative management, especially those with predisposing comorbidities such as diabetes mellitus.

Key Words: Elsberg syndrome; Herpes zoster; Postherpetic neuralgia; Short-term spinal cord stimulation; Treatment; Case report

Core Tip: Elsberg syndrome is a type of sacral radiculitis caused by various viruses. We report a rare case of a diabetic patient with refractory Elsberg syndrome secondary to varicella-zoster virus sacral neuropathy. A suboptimal response to conservative therapy was observed, likely compounded by underlying diabetic polyneuropathy, and short-term spinal cord stimulation therapy led to complete neurological recovery and pain resolution, which was sustained at the two-month follow-up.

INTRODUCTION

Elsberg syndrome, also known as lumbosacral radiculomyelitis, is commonly caused by neurotropic viruses such as herpes simplex virus type 2 and varicella-zoster virus (VZV), which affect the sacral nerve roots[1]. Clinical manifestations include bladder/bowel dysfunction, sensory disturbances (including anesthesia, dysesthesia, or hyperesthesia) in the saddle distribution and lower extremities, and flaccid paralysis. Although Elsberg syndrome typically follows a self-limiting course or resolves with antiviral therapy, persistent refractory manifestations documented in clinical observations necessitate exploration of innovative therapies. We report a refractory case of VZV-associated Elsberg syndrome characterized by urinary retention, severe constipation, and debilitating neuropathic pain, in which short-term spinal cord stimulation (st-SCS) was employed successfully.

CASE PRESENTATION

Chief complaints

A 60-year-old male presented with acute-onset bladder and bowel dysfunction persisting for more than two months, followed by left gluteal and perianal herpes zoster eruption with severe neuropathic pain within two days.

History of present illness

The patient was referred to the pain management clinic for severe dysuria, constipation, and left gluteal/perianal vesicular rash accompanied by debilitating pain (> 2 months). Previously diagnosed with herpes zoster-related neuralgia in the Dermatology Department, he received valacyclovir (1 g ter in die × 7 days), pregabalin (up to 300 mg/day), B-complex vitamins, laxatives, and tamsulosin with a suboptimal response. Despite intermittent urethral catheterization and physical therapy, he required intermittent self-catheterization and digital rectal stimulation, hot compresses, and laxatives to facilitate bladder/bowel evacuation. Persistent perineal pain significantly impaired his quality of life.

History of past illness

His medical history included hypertension diagnosed > 10 years prior and type 2 diabetes mellitus diagnosed > 1 year prior.

Personal and family history

There was no significant personal and family history.

Physical examination

The vital signs on admission were as follows: Temperature, 36.1 °C; heart rate, 69 beats per minute; respiratory rate, 20/minutes; and blood pressure, 145/92 mmHg. The patient exhibited an anxious demeanor with hyperpigmented cutaneous lesions distributed across the left S2-S4 dermatomes, encompassing the gluteal and perianal regions (Figure 1). Neurological assessment revealed cutaneous allodynia to light touch and dermatomal hypoesthesia in affected territories, accompanied by reduced anal sphincter tone (Modified Oxford Scale 2/5). No motor deficits or pathological reflexes were detected. Pain intensity was quantified as 6/11 on the visual analog scale.

Figure 1 Dermatomal distribution of herpes zoster eruption. A: Acute-phase clustered erythematosus lesions over left S2-S4 dermatomes at initial presentation (day 3); B: Convalescent-phase residual hyperpigmentation without active inflammation upon admission (> 60 days).

Laboratory examinations

The laboratory results revealed glycosuria (4+; reference range: Negative) and a positive fecal occult blood test. Complete blood count, renal/hepatic function tests, electrolyte panel, glycated hemoglobin level, tumor markers (including carcinoembryonic antigen, carbohydrate antigen 19-9, and alpha-fetoprotein levels), coagulation profile, and blood-borne pathogen screening (human immunodeficiency virus, hepatitis B virus, hepatitis C virus, and syphilis) were within normal limits.

Imaging examinations

Lumbosacral magnetic resonance imaging revealed diffuse degenerative changes accompanied by mild disc bulging spanning from the L2 to S4 vertebral levels. Concurrent urinary tract ultrasonography revealed benign prostatic hyperplasia with a prostate volume of 38.6 mL (normal < 20 mL), accompanied by a postvoid residual urine volume measuring 104 mL.

FINAL DIAGNOSIS

The patient was diagnosed with the following conditions: (1) VZV-induced Elsberg syndrome; (2) Herpes zoster-related neuralgia; (3) Stage 3 hypertension (very high-risk group); (4) Type 2 diabetes mellitus; and (5) Benign prostatic hyperplasia.

TREATMENT

Initial management with pregabalin (150 mg twice a day), mecobalamin (0.5 mg ter in die), tamsulosin (0.2 mg quaque die), and finasteride (5 mg quaque die) provided minimal symptom relief. Ultrasound-guided sacral canal blockade (20 mL of 1% lidocaine) resulted in transient perineal pruritus/pain reduction and immediate restoration of voluntary micturition (approximately 800 mL of urine output), but bowel dysfunction remained. st-SCS was subsequently performed under digital subtraction angiography guidance. A percutaneous electrode lead (PINS Medical TL3213-65, Beijing) was advanced into the epidural space via the L3-L4 interlaminar approach, with the tip positioned at the inferior T12 level (Figure 2). The following stimulation parameters were delivered via an implantable pulse generator: Contact configuration 5+6-7-8-, amplitude 1.0 mA (sensory threshold: 0.8 mA), pulse width 320 microseconds, frequency 60 Hz. Paresthesia coverage encompassed the left saddle area without lower extremity involvement. Immediate resolution of pain/pruritus was reported. Spontaneous bladder and bowel evacuation resumed on the operative day.

Figure 2 Intraoperative fluoroscopy of short-term spinal cord stimulation lead. A: Anteroposterior view at T12; B: Lateral view showing ventral epidural placement.

OUTCOME AND FOLLOW-UP

The st-SCS system remained unadjusted for 10 days before explantation. At discharge, normal genitourinary/gastrointestinal function was documented, with residual mild cutaneous hypoesthesia and no pain. At the 2-month postdischarge follow-up conducted via telephone, the patient reported spontaneous voiding and defecation without difficulty, resolution of pain in the herpetic region, and residual mild superficial hypoesthesia localized to the affected dermatomes.

DISCUSSION

First described by Elsberg in 1913[2], Elsberg syndrome (lumbosacral radiculomyelitis) is characterized by bladder/bowel dysfunction, saddle-distribution sensory abnormalities (anesthesia/hyperesthesia), and flaccid lower extremity paralysis. VZV and herpes simplex virus type 2 are the predominant pathogens[1]. The incidence of herpes zoster in mainland China ranges from 1.9 to 5.8 per 1000 person-years[3], with approximately 4% of cases exhibiting voiding difficulties[4]. A gold-standard diagnostic framework for Elsberg syndrome remains undefined in current clinical practice; in this case, the diagnosis of Elsberg syndrome was established on the basis of the following: (1) Severe bladder/bowel dysfunction; (2) Classic herpes zoster eruption in S2-S4 dermatomes; and (3) Subacute neuropathic pain (> 2 months in duration). Although lumbosacral magnetic resonance imaging revealed no myelitis and cerebrospinal fluid (CSF) virological testing was not performed due to the chronic phase, the constellation of symptoms fulfilled the diagnostic criteria for viral radiculitis.

Pathophysiologically, VZV reactivation in the sacral ganglia damages sensory neurons and autonomic fibers innervating the bladder/rectum. Parasympathetic impairment causes detrusor hypoactivity and reduced rectal motility, whereas sympathetic dysfunction contributes to sphincter dyssynergia. Crucially, pain-induced central sensitization and reflexive pelvic floor muscle spasm further inhibit voiding/defecation reflexes, establishing a self-perpetuating “pain-dysfunction cycle”[5].

Herpes zoster-associated neuralgia progresses through three phases: Acute herpetic neuralgia (< 30 days), subacute herpetic neuralgia (30-90 days), and postherpetic neuralgia (> 90 days). First-line therapy for acute herpetic neuralgia includes antivirals, tricyclic antidepressants, vitamin B complex, and physical therapy. A literature review (PubMed/EMBASE: 2000-2023; keywords: Elsberg syndrome, VZV radiculitis) identified 10 reported cases of VZV-associated Elsberg syndrome (Table 1)[6-15]. Six patients achieved complete recovery with acute-phase antiviral therapy, while 3 developed chronic sequelae (including 2 requiring permanent catheterization). Our patient’s refractory symptoms despite > 2 months of conventional treatment may be attributed to diabetic neuropathy[12], which exacerbates neural damage and impairs regeneration.

Table 1 Summary of published Elsberg syndrome cases identified through database search.

Ref.	Region	Sex/age (years)	Disease course	Comorbidities	Clinical manifestations	Treatment	Outcome	Follow-up
Nishiyama et al[6], 2023	Japan	Female/77	10 days	None	Urinary retention, genital rash	Acyclovir, acetaminophen, urinary catheter	Persistent postherpetic neuralgia	6 months
Yang et al[7], 2022	China	Female/74	3 weeks	Hypertension and hyperlipidemia	Urinary retention, constipation, sacral numbness	Famciclovir, prednisone, indomethacin, mecobalamin; electroacupuncture	Complete resolution	1 year
Desai et al[8], 2022	United States	Male/45	12 days	Acute ischemic stroke; smoldering myeloma	Urinary retention, fecal incontinence, lumbosacral vesicular rash, right leg paresthesia	Acyclovir, dexamethasone	Required self-catheterization	1 year
Bhagat et al[9], 2021	United States	Male/56	4 days	AIDs; cerebrovascular event	Fever, painful rash, bilateral limb weakness, bowel/bladder dysfunction	Acyclovir; Biktarvy®	Complete resolution	1 month
Santos et al[10], 2021	Brazil	Male/52	2 months	Crohn’s disease; AIDs	Urinary retention and loss of sensation of the genital and left lower limb	Corticosteroid pulse and acyclovir	Incomplete improvement	-
Shah et al[11], 2021	United Kingdom	Male/63	2 weeks	-	Low back pain, perineal and genital numbness, together with bilateral lower limb paresthesia and urinary retention	Acyclovir	Death	-
Saito et al[12], 2018	Japan	Female/68	10 days	Type 2 diabetes mellitus; hypertension and dyslipidemia	Urinary retention and constipation; rash in the left hip	IV acyclovir, catheterization and methylprednisolone pulse	Complete resolution	6 months
Abe et al[13], 2015	Japan	Male/57	7 days	None	Fever with chills, headache with nausea and vomiting and intention tremors of the bilateral upper limbs, constipation and urinary retention	Acyclovir, bladder catheter	Persistent symptoms	1 year
Fujii et al[14], 2015	Japan	Male/76	8 days	Right testicular hydrocele	Anal pain and voiding difficulty, urinary retention, and incontinence of feces	Acyclovir, methylprednisolone and immunoglobulin	Asymptomatic at discharge	-
Matsumoto et al[15], 2012	Japan	Female/55	4 days	Rectal ulcer	Herpes zoster, urinary retention, and constipation	Intravenous acyclovir and oral purgatives urethral catheter	Asymptomatic at discharge	-

AIDs: Acquired immune deficiency syndrome; IV: Intravenous injection.

While pulsed radiofrequency ablation targets isolated ganglia, its application to multisegmental sacral radiculitis (S2-S4) is anatomically challenging owing to osseous constraints. Although sacral neuromodulation has demonstrated efficacy in neurogenic bladder/bowel disorders[16,17], its utility in VZV-induced sacral plexopathy remains unestablished. Given the complex presentation of neuropathic pain with autonomic dysfunction in the patient, st-SCS at the conus medullaris level was selected. The proposed mechanisms involved in the treatment method include: (1) The activation of dorsal column promotes neuroplasticity, reestablishing neural continuity and preventing neurofunctional loss[18]; and (2) The classic gate control theory, which states that spinal cord stimulation selectively activates large-diameter Aβ sensory fibers, thereby inhibiting the synaptic transmission of nociceptive signals from small-diameter Aδ and C fibers in the dorsal horn. This “closes the gate” to pain signal transmission to higher central regions[19]. This dual mechanism underpins sustained pain relief and neurological recovery. Given the patient’s subacute presentation, a st-SCS was implanted at the left conus medullaris level (T12-L1 vertebral segments). This cost-effective trial strategy allowed therapeutic efficacy assessment prior to considering permanent implantation. To our knowledge, this represents the first documented case of refractory Elsberg syndrome successfully managed with st-SCS. At the 2-month follow-up, the patient demonstrated complete restoration of bladder and bowel function and resumed baseline activities of daily living. This outcome underscores that early st-SCS intervention during the subacute phase (30-90 days after onset) may interrupt the pathophysiological cascade, preventing irreversible axonal degeneration and the transition to refractory neuropathic pain.

Notwithstanding the constraints inherent in this single-center case report including: (1) Absence of CSF polymerase chain reaction for virological confirmation due to delayed patient presentation; (2) Reliance on subjective symptom reporting without objective urodynamic profiling (e.g., detrusor pressure/flow rates) or quantitative anal sphincter assessment; (3) Lack of neurophysiological validation for sacral autonomic involvement; and (4) Limited initial follow-up period (2 months) precluding long-term therapeutic durability assessment - our findings warrant further investigation. Multicenter collaborations should prioritize: (1) Establishing cohort databases for similar patients to define evidence-based neuromodulation timing criteria, preventing irreversible neural injury; and (2) Implementing standardized objective metrics including etiological confirmation (e.g., CSF polymerase chain reaction), urodynamic studies, quantitative anal sphincter tone assessment, and autonomic function testing.

CONCLUSION

This report presents a novel therapeutic approach for refractory VZV-associated Elsberg syndrome, in which st-SCS results in complete symptomatic resolution after failure of conventional therapy. To our knowledge, this represents the first documented case of successful st-SCS management of this condition, suggesting its potential neuroregenerative properties beyond pain modulation.