Elsberg syndrome (ES) is an infectious syndrome presenting with variable signs of acute lumbosacral radiculomyelitis, such as urinary retention and lumbosacral sensory symptoms [1]. The viral etiology was established and reactivation of herpes simplex virus type 2 in spinal ganglia is the most common cause of ES [2]. Other agents responsible for ES include varicella zoster virus (VZV), herpes simplex virus type 1, Epstein–Barr virus, cytomegalovirus, and Angiostrongylus cantonensis [3-7].

Acute urinary retention is common in elderly people. However, due to the low awareness and rare reports of ES, it is often misdiagnosed and treated incorrectly [8]. Radiological or etiological examination cannot be performed in time to facilitate a definitive diagnosis; therefore, ES is usually treated as an idiopathic inflammatory condition [9]. Consequently, treatment targeted at the infection is limited; corticosteroids have become the most frequently prescribed drugs [10]. Some studies have reported that patients with ES can develop neurological sequelae due to improper treatment [11]. In addition, conventional drugs may be contraindicated in special cases, like pregnancy. Thus, conventional drug treatments for ES are inadequate and it is necessary to improve interventions. Acupuncture is a commonly used non- pharmacologic therapy that is advantageous in promoting the recovery of neurological function [12-14].

In this study, we reported an elderly patient with ES secondary to VZV infection who had complete recovery with acupuncture treatment after ineffective conventional drug treatment. The article follows the CARE guidelines.

1. Chief complaints

A 74-year-old woman presented with skin rash and pain in the left sacral area 3 weeks ago, with bladder dysfunction and constipation for 1 week.

2. History of present illness

She visited a doctor in a local clinic complaining of intermittent discharge-like pain and a skin rash in the left sacral area 3 weeks prior to admission. She was diagnosed with herpes zoster (HZ) and was prescribed famciclovir (300 mg bid), prednisone (15 mg qd), indomethacin (25 mg tid), and mecobalamin (0.5 mg tid) for 1 week. After the pharmacologic intervention, pain intensity defined by the visual analogue scale (VAS) score, decreased from 8 to 6 and the skin rash did not extend further. A week before admission, she started to complain about numbness in the perianal area, incomplete bladder emptying, frequent urination, and constipation. She was admitted to the Third Affiliated Hospital of Sun Yat-Sen University.

3. History of past illness

Past medical history revealed hypertension and hyperlipidemia with administration of oral hypotensive, antiplatelet, and lipid-lowering drugs.

4. Personal and family history

Her family history was negative for any disorder.

5. Physical examination

On admission, vital signs were: blood pressure, 164/75 mmHg; heart rate, 65 beats/min; respiratory rate, 18 breaths/min; and temperature 36.4℃. Physical examination findings were normal except for hypoesthesia in the perianal area and multiple clusters of erythematous vesicles distributed in the left sacral region.

6. Laboratory examinations

Hematological analysis and serum biochemistry, including C-reactive protein, were normal. Urine test results showed white blood cells (++).

7. Imaging examinations

Lumbar spinal magnetic resonance imaging (MRI) detected no compression or lesion that would be responsible for the bladder and rectal dysfunction (Fig. 1). Urodynamic tests showed detrusor areflexia (Fig. 2A).

Figure 1. Magnetic resonance imaging of the patient. Sagittal T1-weighted and T2-weighted images showed a lumbar disc hernia. This was not responsible for the neurological dysfunction because the conus medullaris was not affected.

Figure 2. Urodynamic test of the patient before and after treatment. (A) Urodynamic test before electroacupuncture (EA) showed an intermittent tip-shaped curve, suggesting detrusor areflexia and assisted urination by abdominal pressure (maximum flow rate: 9.7 ml/s, average flow rate: 3.2 ml/s, flow time: 25.8 s, time to peak: 423.8 s). (B) Urodynamic test after EA treatment showed a dome-shaped curve, suggesting normal detrusor contractions (maximum flow rate: 11.7 ml/s, average flow rate: 6.4 ml/s, flow time: 19.4 s, time to peak: 2.9 s).

Based on the clinical manifestation, imaging, and urodynamic tests, she was diagnosed with ES.

On day 2 of admission, the patient accepted urethral catheterization and clyster therapy with the anti-infective agent (intravenous sulperazon, 3 g, q12h, 7 d), oral analgesics (pregabalin, 75 mg, qd; tramadol, 50 mg, bid), and oral laxatives (lactulose, 10 ml, tid). On day 9 of admission, the symptoms of constipation gradually improved and medication intervention was no longer required. She tried to remove the bladder catheter but failed. Subsequently, she started to accept electroacupuncture (EA) treatment. The needle was inserted to a depth of 25-35 mm at the location of the acupoints. After the patient indicated that they felt the de qi sensation, paired alligator clips from the EA apparatus were attached to the needle holders. EA stimulation lasted for 30 minutes with a low-frequency continuous wave of 2 Hz and a current intensity of 0.1-1 mA, depending on the participant’s comfort level. EA was performed for six sessions (qd in first three sessions, qod in remaining three sessions). In the first three sessions, acupoints included CV4 (Guan yuan), CV3 (Qu gu), GV28 (Shui dao), SP6 (San yin jiao), and SP9 (Yin ling quan). We chose GV2 (Yao shu), GV3 (Yao yang guan), BL32 (Ci liao), BL54 (Zhi bian) and BL28 (Pang guang shu) in the remaining three sessions. EA was performed by a certified acupuncturist with over five years’ experience.

After the first three EA treatments, pain in the sacral region was released (VAS score: 3). The bladder catheter was removed because the patient could urinate spontaneously. She was discharged from hospital and suspended all drug treatments. However, after discharge, she still suffered with difficulties voiding, numbness, and neuralgia in the sacral region, which prevented her from sitting on a chair without a cushion. Ultrasound revealed a residual urine volume of > 150 ml. Thus, she accepted three more session of EA treatment in an outpatient department of acupuncture. After six EA treatments, she recovered completely with no residual volume after urination, as shown by ultrasound. The numbness and pain in the sacral region disappeared (VAS score: 0). Urodynamic tests demonstrated normal detrusor contractions (Fig. 2B). All symptoms were relieved completely without recurrence at 3- and 12-months’ follow-up. No adverse effects or unanticipated events were observed in association with the treatment methods described.

We managed a 74-year-old woman with HZ in the sacral region, whose chief complaint was pain, numbness in sacral region, urinary retention and constipation. The final diagnosis was ES caused by VZV infection. HZ is typically manifested as rash and postherpetic neuralgia. It is rarely reported; therefore, clinicians often do not recognize that visceral motor dysfunction can be caused by VZV infection. The probable mechanism is latent virus in sensory ganglia reactivating, replicating, and spreading within the axons retrogradely. This leads to inflammation of the nerve root or conus medullaris in some conditions [15]. In populations with anogenital VZV infection, the prevalence of ES is about 3.5% [16,17].

Urinary retention in elderly men is most commonly attributed to prostate hypertrophy. This condition is more uncommon in childhood and women due to neurological causes. ES should be considered in the diagnosis of acute urinary retention for women and children. Viral detection in the cerebrospinal fluid (CSF) is the diagnostic gold standard. However, the low awareness of ES and low diagnostic accuracy of the virological test means that the viral CSF polymerase chain reaction or serum virological test is limited and imperfect. The time window also influences test positivity. One study has shown a negative predictive value of 82% and positive predictive value of 54% [18]. Savoldi et al. [8] have established the diagnostic and exclusionary criteria for ES. According to the diagnostic criteria, our case matched the clinically definite category due to the absence of an CSF virology test because the patient refused a lumbar puncture test. Despite the lack of direct etiological proof, a clinical diagnosis of ES was established because of the clinical symptoms and signs of cauda equina involvement, acute onset of symptoms, preceding rash of sacral herpes infection, and exclusion of other causes by MRI.

ES often leaves some degree of permanent neurological deficit and may occasionally lead to catastrophic results. A retrospective study of 30 patients with ES reported that only one recovered completely; most recovered with sequelae and one patient died [8].

There is no consensus for the treatment of ES. Intermittent urinary catheterization is one recommended treatment. Antiviral drugs can be effective but are rarely used because of the low viral detection rate. The benefit of corticosteroids for the treatment of ES remains uncertain. Some studies have reported an increased risk of ES in immunocompromised or elderly patients [19]. The early application of prednisone may be harmful because it can increase the risk of HZ developing into ES.

Acupuncture, a component of traditional Chinese medicine (TCM), is widely used as a non-pharmaceutical treatment to relieve pain and promote recovery in neurological dysfunction. High-quality research has shown that acupuncture effectively promotes gastrointestinal function, improves stress urinary incontinence, and relieves angina and migraine [20-23]. Previous studies have reported a positive effect of acupuncture on bladder dysfunction after spinal cord injury, postpartum, and after cauda equina injury [12-14]. We chose the acupoints used in this patient for the following reasons. First, the majority of the acupoints are distributed in the Conception Vessel, Governor Vessel, and Bladder Vessel. The TCM principles that guide the selection of acupoints state that treatment should be administered where the vessel passes through the location. We chose acupoints to regulate the Qi of the region. Second, the acupoints were located in the sacral and bladder areas. According to anatomical knowledge, EA at acupoints located in the abdomen stimulates the pelvic plexus nerve. By contrast, EA at acupoints located at sacral region stimulates the S2 sacral nerve roots, inferior rectal nerve, and pudendal nerve [13]. These nerve structures are important for the control of urination and bowel movements. EA at the sacral region has a similar mechanism to sacral neuromodulation via stimulation of the parasympathetic center at S2-S4 [14]. A previous study has reported that EA at Governor Vessel acupoints facilitates myelin and axonal regrowth by triggering the synthesis and secretion of neurotrophin-3 [24]. In addition, EA inhibits inflammation and has an analgesic effect [25]. Huang and colleagues have reported that low-frequency EA is superior to high-frequency EA at improving symptoms of neural functional defect [26].

This case report suggests that acupuncture can improve paralytic bladder, pain, and numbness secondary to VZV infection. Nevertheless, the possibility of spontaneous recovery cannot be excluded. Therefore, prospective studies with adequate sample sizes and an appropriate control group are warranted to verify the effect of acupuncture as a treatment for ES.

This report has some limitations. There was a lack of direct etiological proof. Despite complete recovery after acupuncture treatment, we cannot confirm whether this was due to acupuncture or spontaneous healing.

Acupuncture improved the neurological function in sacral radiculitis. This indicates that acupuncture is a safe and promising complementary therapy for the management of ES.

We thank Dr. Cheung Wen for English language polishing.

The authors received no financial support for the research, authorship, and/or publication of this article.

LSY provided the case, collected the clinical data, and drafted the manuscript. KZ and DFZ edited the manuscript. SZZ took charge of material supports. LSY and JZ performed the acupuncture treatment. All authors issued final approval for the version to be submitted.

Obtained.

The authors declare no conflict of interest.