Journal of Clinical Anesthesia 37 (2017) 4951

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Journal of Clinical Anesthesia

Case report

A patient with postpolio syndrome developed cauda equina syndrome

after neuraxial anesthesia: a case report
Wei-Cheng Tseng, MD (Resident) a, Zhi-Fu Wu, MD (Professor) a, Wen-Jinn Liaw, MD, PhD (Professor) b,
Su-Yang Hwa, MD (Attending Physician) c, Nan-Kai Hung, MD (Attending Physician) a,
a
Department of Anesthesiology, Tri-Service General Hospital and National Defense Medical Center, Taipei, Taiwan, ROC
b
Department of Anesthesiology, Tungs' Taichung MetroHarbor Hospital, Taichung, Taiwan, ROC
c
Department of Orthopaedics, Tri-Service General Hospital and National Defense Medical Center, Taipei, Taiwan, ROC

a r t i c l e i n f o a b s t r a c t

Article history: Combined spinal anesthesia and postoperative epidural analgesia is widely used in orthopedic surgery. Uncom-
Received 10 March 2016 mon but serious neurologic complications of neuraxial anesthesia (NA) include direct trauma during needle or
Received in revised form 8 September 2016 catheter insertion, central nervous system infections, and neurotoxicity of local anesthetics. Cauda equina syn-
Accepted 28 September 2016
drome (CES) is a rare complication after NA but can result in severe neurologic deterioration that may require
Available online xxxx
surgical intervention. We present a case of a 69-year-old woman with postpolio syndrome who developed CES
Keywords:
after combined spinal anesthesia and postoperative epidural analgesia. Perioperative observations and follow-
Caudal equina syndrome up examinations, including magnetic resonance imaging, revealed no evidence of direct needle- or catheter-
Neuraxial anesthesia induced trauma, spinal hematoma, spinal ischemia, intraneural anesthetic injection, or infection. We speculate
Spinal anesthesia that CES symptoms were observed because of enhanced sensitivity to a combination of regional anesthetic
Epidural analgesia techniquerelated microtrauma and neurotoxicity of bupivacaine and ropivacaine. Thus, practitioners should
Postpolio syndrome be aware that patients with preexisting neurologic diseases may be at increased risk for CES after NA.
2017 Elsevier Inc. All rights reserved.

1. Introduction multiple puncture attempts, direct toxicity of local anesthetics (LAs),

Neuraxial anesthesia (NA) is widely used in surgeries involving the
lower abdominal region, pelvic organs, and lower extremities and for ce-
sarean deliveries. It is generally considered effective and safe because NA
provides sufcient postoperative analgesia and is associated with fewer
cardiopulmonary complications, prevention of deep vein thrombosis,
and reduced postoperative mortality [1]. Therefore, spinal anesthesia
(SA), epidural anesthesia (EA), combined SA and EA, and postoperative
epidural analgesia were frequently used in orthopedic surgeries. The
technique is often regarded as intrinsically safe; however, the incidence
of neurologic decits after NA is between 1 in 1000 and 1 in 1000,000
[2-3]. Cauda equina syndrome (CES), a severe neurologic disorder, is a
rare neurologic complication of NA that is caused by the damage of the
conus medullaris or spinal nerve roots. The incidence of CES after NA can-
not be determined on the basis of the existing literature because there are
only a few denitive case reports. Possible etiologies of CES after NA in-
clude iatrogenic events such as direct or indirect trauma following
Funding: None.
Corresponding author at: Department of Anesthesiology, Tri-Service General Hospital
and National Defense Medical Center, No. 325, Section 2, Chenggong Road, Neihu District
114, Taipei, Taiwan, ROC. Tel.: +886 2 87927128; fax: +886 2 87927127.
E-mail address: rvolvo307@gmail.com (N.-K. Hung).
and compression of the spinal cord or nerve roots by hematoma [3-6]. It
can lead to varying degrees of bowel and bladder dysfunction, insensation
of perineal areas, and even paraplegia [3,4]. Here, we report the case of a
patient with postpolio syndrome (PPS) who developed CES after com-
bined SA and postoperative epidural analgesia for total hip arthroplasty.
2. Case report
The patient in this case provided informed consent to anonymized
reporting of clinical details. A 69-year-old woman (height, 150 cm;
weight, 60 kg; body mass index, 26.7 kg/m2) was classied as American
Society of Anesthesiologists physical status III because of poliovirus infec-
tion in childhood, which developed PPS with muscle atrophy of the left
lower extremity afterward. She was scheduled for total hip arthroplasty
because of osteoarthritis of the right hip joint. Vital signs and results of
general physical and systemic examinations were unremarkable except
for known decits. Routine blood parameters, including coagulation pro-
le, were within normal limits. Two epidural puncture attempts (Tuohy
epidural needle, 16 G 80 mm) at L4-5 were made before successful epi-
dural catheter placement. A test dose of 3 mL of 2% lidocaine 20 mg/mL
with epinephrine 5 g/mL was injected via epidural catheter to detect
the intrathecal misplacement. Then the patient received 15 mg 0.5% iso-
baric bupivacaine intrathecally (BD spinal needle, 26 G 3 1/2 in.) at

http://dx.doi.org/10.1016/j.jclinane.2016.09.032
0952-8180/ 2017 Elsevier Inc. All rights reserved.
50 W.-C. Tseng et al. / Journal of Clinical Anesthesia 37 (2017) 4951
L4-5. There was no ashing pain or paresthesia during needle placement,
catheter insertion, or drug injection. A urinary catheter was inserted be-
fore the surgery. Twenty minutes after bupivacaine administration, pin-
prick test revealed bilateral symmetric sensory block caudal to T8. The
150-minute surgical procedure was performed in the lateral decubitus
position and was uneventful. The patient did not receive additionally epi-
dural LA infusion during the surgery. Then she was transferred to
postanesthesia care unit (PACU) under stable vital signs and patient-
controlled epidural analgesia was initiated with a loading dose of 8 mL
of 1.6 mg/mL ropivacaine and 1.0 g/mL fentanyl followed by an infusion
rate of 2 mL/h. The patient demonstrated improved muscle power within
30 minutes after surgery in the PACU but complained of numbness and
tingling in her right lower extremity (L4/L5 and L5/S1 dermatome). Her
complaints of numbness and tingling sensation persisted after discharge
from PACU to the ward.
In the ward, the patient continued to receive patient-controlled epi-
dural analgesia infusion. On postoperative days 1 and 2, gradual recov-
ery of motor function and no severe pain from surgical wound were
noted; however, abnormal sensation of the right lower extremity
remained. Unexpectedly, urinary incontinence and progressive weak-
ness of the right lower extremity (muscle power grade 2) developed
within 1 hour after smooth withdrawal of the epidural and Foley cathe-
ters on postoperative day 3. Also, neurologic dysfunction of the left
lower limb was observed. Immediate neurologic consultation was re-
quested. Lumbar magnetic resonance imaging (MRI) revealed no evi-
dence of epidural hematoma but did show disk degeneration with a
posterior bulge at L1/2 and L2/3 levels, mild compression of the thecal
sac, and bilateral neuroforaminal impingement (Fig. 1). A nerve conduc-
tion study and electromyogram indicated L5 and S1 radiculopathies.
Urodynamic tests revealed hypoexic bladder, fair compliance, and
sphincter insufciency. CES was diagnosed, and the patient was imme-
diately treated with high-dose intravenous steroids. She was then re-
ferred for a rehabilitation program that included balance training,
endurance training, and oral vitamin B complex administration. During
regular outpatient follow-up, bladder sphincter dysfunction and
weakness of the bilateral lower limbs gradually improved over 4
months (to grade 3-4 motor power) with return of sensation.
Fig. 1. Postoperative magnetic resonance imaging shows no evidence of epidural
hematoma but clear disk degeneration with posterior bulge at L1/2 and L2/3 levels with
mild compression of the thecal sac and bilateral neuroforaminal impingement.
Fortunately, the patient did not remain apparently residual sequelae
in sensorimotor function after 1 year of medication and rehabilitation.
3. Discussion
The major complications of NA are divided into 3 categories, includ-
ing adverse physiological responses, needle puncture or catheter place-
ment injury, and neurotoxicity of LAs. In our case, no ashing pain or
paresthesia was noted during epidural needle puncture, epidural cathe-
ter placement, or intrathecal bupivacaine injection; therefore, needle-
induced trauma to the spinal cord and intraneural injection were
ruled out. However, the NA techniquerelated microtrauma still could
not rule out. Spinal cord compression by hematoma was excluded
using MRI. There was no evidence of infection, spinal cord ischemia, or
postoperative exacerbation of preexisting neurologic symptoms. More-
over, results of neurologic examination were inconsistent with neuro-
logic injury from improper patient positioning or unusual surgical
trauma. Therefore, we speculated that NA techniquerelated
microtrauma and neurotoxicity of LAs might be the causes of new
onset CES in our case.
The incidence of neurologic complications after NA is very low;
therefore, establishing denitive etiological links has proven difcult.
In 1991, CES after NA was rst described in a case series administered
continuous SA [7]. In that series, there were 4 cases with evidence of
focal sensory block in which doses of LAs were greater than usually ad-
ministered by single injection. In large retrospective surveys, Pollock [8]
reported that the incidence of persistent neurologic sequelae after intra-
thecal anesthesia has ranged between 0.01% and 0.7%. Arai and Hoka [9]
suggested that intrathecal neurotoxicity of LAs is closely associated with
the concentration and is higher with lidocaine than with bupivacaine [3,
10]. In addition to high concentrations, higher total doses may increase
CES risk [3]. In an animal study, the incidences of histologic damage and
functional impairment of nerve roots and spinal cord were higher after
SA than after EA because the concentration of LAs after EA is lower than
after intrathecal administration [11]. Therefore, EA might be safer than
SA in patients with preexisting CNS disorders.
In the past, NA was relatively contraindicated for patients with
preexisting CNS disorders [12], so many anesthesiologists hesitate to
practice regional anesthesia in patients with preexisting neuromuscular
decits due to the concern of exacerbating existing disease or difculty
evaluating complications. However, the proof that NA or analgesia in
patients with preexisting neurologic dysfunction can aggravate the ex-
tent of injury is absent [12,13]. No controlled prospective study has
identied NA as a signicant risk factor for neurologic injury in patients
with preexisting neurologic disease. A retrospective study by Hebl et al
[13] reported no new or worsening neurologic decits after NA in pa-
tients with preexisting CNS disease. Joseph et al [14] reported that
preexisting neurologic disorders, including PPS, amyotrophic lateral
sclerosis, and multiple sclerosis, could relapse or exhibit symptom exac-
erbation in response to perioperative stress, mechanical trauma, or drug
toxicity, but Joseph et al [14] suggested that LAs are rarely neurotoxic
when administered at recommended concentrations and doses, unless
accompanied by mechanical damage to connective tissue barriers
protecting the spinal cord or nerve bers (eg, during needle/catheter
placement) or when used with vasoconstrictors that decrease clearance
of LAs. However, Lambert et al [15] reported that patients with PPS have
fewer motor neurons compared with healthy adults, and motor neurons
in those with PPS may have residual dysfunction or increased metabolic
demand for supplying abnormally enlarged motor units, leading to
more sensitivity to drug effects.
The recommended concentrations and doses are 0.5% isobaric
bupivacaine and 10-20 mg for SA, 2% lidocaine with epinephrine and
500 mg maximum for EA, and the maximum dose of ropivacaine is
200 mg [12,16]. In our patient, the doses and concentration were in
the safety range. However, maldistribution or delayed elimination
should be also considered as possible mechanism in our patient by
 W.-C. Tseng et al. / Journal of Clinical Anesthesia 37 (2017) 4951
delayed recovery of motor and sensory function. Based on the uncertain
risk of new neurologic complications in patients with preexisting neu-
rologic dysfunction [17-19], we suggest that NA may contribute to CES
development through a combination of needle-induced microtrauma
and neurotoxicity of LAs to patients with preexisting neurologic disease
due to enhanced CNS vulnerability. Nevertheless, up to date, there are
no direct experimental data to conrm this concept.
Ultimately, the decision about whether to use regional anesthesia or
analgesia in an individual patient with preexisting CNS disease should
be made based on weighing the risks and benets. When signs and
symptoms of acute CES develop, the rst step should be emergent MRI
imaging or computed tomography myelography to identify the level
of compression and conrm anatomical location. If disk herniation, he-
matoma, infection near the spinal cord, or neoplasm are present, early
decompressive surgery is recommended [4,5].
In our case, we initially preferred to perform general anesthesia due
to her medical history of PPS, but there were some reasons making us
implement regional anesthesia as follows: rst, the patient had abnor-
mal 3-3-2 rules and Mallampati grade III about her airway assessment
implying difcult intubation; second, she had a problem of obstructive
sleep apnea according to her statement; third, most importantly, she re-
fused to receive general anesthesia. Next time, we should make more
careful risk-to-benet evaluation and inform the patients about the
risk of possible neurologic complications when facing similar cases
with preexisting neurologic disorders.
4. Conclusion
This case emphasizes that practitioners should be aware of possible
neurologic complications of NA or analgesia, particularly in patients
with preexisting neurologic disorders. Although there has been no evi-
dence of adverse effects due to NA in PPS patients, but this does not nec-
essarily mean that regional techniques are without risk. These patients
should be informed regarding the risk of possible neurologic complica-
tions, including progression of preoperative decits and new-onset
neurologic dysfunction. Preoperative neurologic status should be
completely documented. Minimizing needle trauma and intraneural in-
jection can decrease risk of neurologic injury. Furthermore, the appro-
priate concentration and dose of LA solutions must be considered.
Once neurologic injury occurs, early detection and treatment of compli-
cations are important to minimize the risk of adverse outcome.
51
Authors' contributions
Wei-Cheng Tseng, Zhi-Fu Wu, Wen-Jung Liaw, and Nan-Kai Hung
contributed in writing the manuscript. Su-Yang Hwa contributed to
clinical care. All authors have read and approved the nal manuscript.
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