Accepted Manuscript A rare complication of spine surgery: Guillain–Barré syndrome Sheng-Li Huang, Hua-Guang Qi, Jing-Jie Liu, Ya-Juan Huang, Li Xiang PII:
S1878-8750(15)00474-X
DOI:
10.1016/j.wneu.2015.04.045
Reference:
WNEU 2870
To appear in:
World Neurosurgery
Received Date: 4 March 2015 Revised Date:
20 April 2015
Accepted Date: 22 April 2015
Please cite this article as: Huang S-L, Qi H-G, Liu J-J, Huang Y-J, Xiang L, A rare complication of spine surgery: Guillain–Barré syndrome, World Neurosurgery (2015), doi: 10.1016/j.wneu.2015.04.045. This is a PDF file of an unedited manuscript that has been accepted for publication. As a service to our customers we are providing this early version of the manuscript. The manuscript will undergo copyediting, typesetting, and review of the resulting proof before it is published in its final form. Please note that during the production process errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain.
ACCEPTED MANUSCRIPT A rare complication of spine surgery: Guillain–Barré syndrome Sheng-Li Huang1, Hua-Guang Qi2, Jing-Jie Liu3, Ya-Juan Huang1, Li Xiang3 1
Department of Orthopaedics, the Second Affiliated Hospital, Xi'an Jiaotong University
College of Medicine, Xi'an, China Department of Function Examination, Hong Hui Hospital, Xi'an Jiaotong University College
RI PT
2
of Medicine, Xi’an, China 3
Department of Neurology, the Second Affiliated Hospital, Xi'an Jiaotong University College
SC
of Medicine, Xi'an, China
M AN U
To whom correspondence should be addressed: Li Xiang, No.157 Xiwulu, Xi'an 710004, China (Tel: +86-29-87679228; Fax:﹢8629 8767 8634; E-mail: [email protected])
ABSTRACT
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OBJECTIVE: Since little has been reported about Guillain–Barré syndrome (GBS) following spine surgery, this study demonstrates some of the clinical features and the management of the condition.
EP
METHODS: A total of 4 cases of GBS following spine surgery were included. The medical charts of the patients were reviewed to obtain demographic and clinical details. All the
AC C
patients underwent neurological and electrophysiologic examinations, and were followed up after surgery.
RESULTS: The onset of symptoms about GBS was 2-7 days following the operation. Neurological evaluation showed weaknesses of upper and lower extremities and repeal of tendon reflexes. The patients exhibited typical clinical symptoms and signs of GBS, and electromyographic findings. Lumbar puncture was performed in two cases, and cerebrospinal fluid examination showed albumino-cytological dissociation. All the four patients were diagnosed as GBS based on typical clinical, laboratory and electrophysiological findings.
1
ACCEPTED MANUSCRIPT Intravenous immunoglobulin was instituted. At follow-up, one patient needed ventilator support; one patient could transfer from bed to chair; one walked with assistive devices; and the rest one had residual minor neurologic deficits. CONCLUSIONS: These cases warn surgeons to be alert to the association of GBS and spine
patients with paralysis after spine surgery.
RI PT
surgery. Based on our experience, we recommend consideration of this rare diagnosis in
Key words: Spine surgery; Guillain–Barré syndrome; Complication; clinical features;
SC
neuropathology
M AN U
Abbreviations and Acronyms: Guillain–Barré syndrome (GBS); Cerebrospinal fluid (CSF)
INTRODUCTION
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Spine surgery procedures may result in various complications. The onset of complications ranges from 4% to 19% (1), among which the overall incidence of neurologic complications is less than 1% (2). Guillain–Barré syndrome (GBS), one type of neurologic complications
EP
following spinal operation, has rarely been reported (3-7).
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GBS is a rare acute polyradiculoneuropathy that manifests with weakness and diminished reflexes, and may also involve impairment of sensation, cranial nerves, or bulbar muscles. The incidence of GBS is 1 to 2 per 100,000 annually worldwide (8). However, as surgeons may be unfamiliar with it, weakness and paralysis after spine surgery is often attributed to other postoperative complication. Not much about GBS after spine surgery has been published. Most of the original articles are case reports (9-18). Since this condition is rare, it is possible that patients would be
2
ACCEPTED MANUSCRIPT misdiagnosed and subsequently treated incorrectly. To the best of our knowledge, no systematic research has been published so far discussing its clinical characteristics and management strategies. To address this relative lack of information, we performed this
RI PT
retrospective analysis. MATERIALS AND METHODS
From January 2013 to June 2014, a total of 7770 patients were received spine surgery, and 4
SC
of them were diagnosed as GBS after operation. We retrieved the clinical data of the patients,
neurological status, and ancillary tests.
M AN U
including age, gender, history of past illness, surgical procedures, clinical presentation,
The 4 male patients were aged 50 to 69 years with an average of 57.5 years (Table 1). One patient (Case 2) was operated with anterior cervical discectomy and fusion one year before
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this hospitalization, others had no surgical history. They also had neither a recent illness (for example respiratory infection or gastroenteritis) nor any vaccinations in the few months prior to the spine surgery. There was no history of the use of neuromuscular blocking agents,
EP
catecholamines, and aminoglycoside antibiotics in any of these patients. The surgery was
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performed under general anaesthesia. One patient (Case 3) received blood transfusion. No complications occurred during the procedure. All patients received corticosteroids (dexamethasone or prednisolone) after operation. The post-operative recovery was uneventful
until the onset of symptoms of GBS. All patients were followed up. RESULTS On postoperative day 3, two patients (Case 2 and Case 4) developed leg weakness with unstable gait that worsened over several days. On day 2 and 7, two patients (Case 3 and Case
3
ACCEPTED MANUSCRIPT 1) showed weaknesses in upper and lower extremities respectively. In these cases, GBS developed from 2 to 7 days after surgery, and the symptoms progressed rapidly within 1 week. The delay between the onset and diagnosis of GBS ranged from 4 to 19 days (average of
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12.75 days). The principal clinical manifestation was progressive symmetric motor weakness, with choking while drinking. Stiff neck and other signs of meningeal irritation were not
bilateral facial paralysis in Case 1 and ptosis in Case 2.
SC
present. There was no pathologic reflex. And they were afebrile. Especially there were
M AN U
Case 1 underwent reoperation for exploration 1 day after the aggravated neurological deficit, whereas nothing (such as spinal epidural hematoma) was seen at operation, and
postoperative neurologic deficit was progressively exacerbated. Three of the patients (Case 1, 2 and 3) were moved to the intensive care unit, intubated and supported by mechanical
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ventilation.
Nerve conduction examination in all the patients showed slowed conduction velocity, prolonged terminal latencies in the major peripheral nerves, marked reduction in the
EP
amplitude of muscle action potential, and even an absent response (Table 2 and 3). And
AC C
F-wave was either absent or markedly decreased in number. Electromyography demonstrated fibrillations in Case 2 and 4; unfortunately, at the bedside, fibrillation potentials were not completed in Case 1 and 3 for environmental intervention in the intensive care unit. Therefore, electrophysiological examination revealed peripheral nerve damage. Cerebrospinal fluid (CSF) examination was performed only in Case 2 and Case 4, because Case 3 had postoperative back wound and Case 1 refused to take the examination. The results of the examination presented albumino-cytological dissociation. Once GBS was
4
ACCEPTED MANUSCRIPT confirmed, treatment with high doses of corticosteroids and human immunoglobulin were performed. Intravenous human immunoglobulin was performed at a dose of 0.4 g/kg for 5 days.
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The follow-up period for these patients ranged from 7 to 22 months (average 15.5 months). One patient could walk with the help of a cane to help walk; one patient could not walk but could transfer from bed to chair; another patient was severely disabled and needed mechanical
SC
support for breathing; and the rest one had completely resolved, except for minor weakness of
M AN U
the intrinsic muscles of the hands, the neurological deficits. DISCUSSION
GBS is a rare but severe neurologic complication after operation. During the past decades, there have been a number of reports presenting GBS following different types of surgery (3-7,
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9-18), but only 5 of the cases (3-7) developed the complication 8 to 22 days after spine surgery (Table 4). The 4 post-spinal-surgery GBS we observed were out of a total of 7770 cases; the incidence of GBS after spine surgery is 1 per 2,000 (4/7770). The onset of the
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complication in these patients was within 1 week after surgery, earlier than that in the patients
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reported previously.
GBS is a rapidly progressive demyelinating, monophasic polyneuropathy. It can present
with cranial nerve deficits and respiratory compromise. Typical symptoms of GBS are muscle weakness in the limbs and sensory disturbances such as pain and numbness. The symptoms and signs of GBS are similar to those of spinal cord injury in clinical performance. GBS mimics an acute spinal cord compression injury or nerve root compression syndrome in its initial appearance (sudden postoperative paralysis). Being an unusual postoperative
5
ACCEPTED MANUSCRIPT complication, GBS has rarely been described. This makes it difficult for most surgeons to diagnose GBS or to distinguish it from other complications in postoperative settings. The four cases we report here underwent extensive neurological evaluations and exhibited
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the typical clinical symptoms and signs of GBS. The patients had progressively aggravating muscular weakness and paraplegia. The weakness developed acutely by hours or days after surgery. Choking while drinking was developed in all the patients; this cranial nerve
SC
involvement in particular led to the consideration of GBS. The facial nerve was less often
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affected. Physical examination revealed hyporeflexia or areflexia of the extremities, with the weakness more severe in the distal part than in the proximal part. It can be seen that our patients have many similarities to these previous case reports.
Despite these features, GBS in the setting of surgery remains a diagnostic challenge in
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regard to differentiating it from other complications following operation due to the significant overlap in symptoms. However, when postsurgical spinal cord injury cannot explain the bulbar symptoms or facial weakness in patients, surgeons should take GBS as a consideration.
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The diagnosis of GBS involves a combination of clinical presentation and ancillary tests, such
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as CSF analysis and electromyography. Although spine magnetic resonance imaging can be a choice of diagnostic modality, it remains inconclusive in early postoperative stages. Electromyography is crucial in arriving at the diagnosis. So, we advocate electromyography rather than imaging.
When a patient is found to aggravate in symptoms and signs after spine surgery, corresponding measures should be taken to relieve the condition. For example, the postsurgical neurological deficits in patients with space-occupying lesions such as extradural
6
ACCEPTED MANUSCRIPT hematoma and displaced operative implant could be improved after reoperation. However, the situation might worsen if handled inappropriately, such as in our Case 1. Therefore, a precise diagnosis is crucial in deciding and adopting appropriate treatment. The differential diagnosis
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of GBS includes hemorrhage, displaced implants, bony instability, infection, medullar ischemia (vascular insults) and toxicity due to drugs. Cranial nerve involvement, which is associated with choking while drinking or facial palsy, supports the diagnosis. Certainly,
SC
making a diagnosis of GBS is difficult for most surgeons because they are not familiar with
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this disease. It often takes some time for surgeons to recognize and confirm the condition, and the therapeutic measures specific to the condition are often delayed. For instance, in the four patients we report here, GBS was diagnosed averagely 12.75 days after its onset. This delay might lead to poor prognosis. To avoid such delay, we recommend neurological examinations
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to assist with the diagnosis and management when there is unexplainable progressive postoperative muscle weakness.
GBS is typically caused by an autoimmune attack of peripheral nerve. Infection and
EP
vaccination have been considered as associated with its occurrence (19). Although the
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condition also occurs after surgery, the mechanisms that link GBS with surgery remain unclear. In the five postsurgical GBS cases reported previously, no other risk factors were identified except surgery. The four patients we discuss here also had not encountered any other incidents; the only clearly identified precipitating event was the spinal operation. However, considering the low incidence of GBS after spine surgery, the operation should not be regarded as the primary inducement of GBS. The limited number of case reports in literature could not support a conclusion that there is a causal relationship between GBS and
7
ACCEPTED MANUSCRIPT surgery. However, it is not easy to affirm whether it is mere coincidence. Regular treatment of GBS consists of a 5-day course of intravenous immunoglobulin at a dose of 0.4 g/kg and/or plasma exchange. It has been shown to improve prognosis. So an
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empiric course of intravenous immunoglobulin may be warranted if GBS is a reasonable possibility diagnosis after spinal operation. Supportive care, such as ventilator support, during and following hospitalization is also crucial.
SC
Recovery is dependent on multiple factors, such as acuity of the onset of symptoms and
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need for ventilator support. If not identified early and treated promptly, this neurological disorder will result in adverse consequences such as permanent neurological impairments. The sequela may worsen with delays in management. Besides, when patients need ventilator support, clinical improvement is slow and residual disability is pronounced.
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CONCLUSION
GBS is a rare neurological complication after spine surgery. When progressive neurological deterioration occurs during the early postoperative period, it is extremely important to be
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aware of occurrence of GBS. The characteristic presentation is rapidly aggravating symmetric
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motor weakness and areflexia, peripheral facial palsy, choking while drinking, or respiratory difficulty. Intravenous immunoglobulin and plasma exchange are efficacious treatments. REFERENCE 1.
Dekutoski MB, Norvell DC, Dettori JR, Fehlings MG, Chapman JR: Surgeon perceptions and reported complications in spine surgery. Spine (Phila Pa 1976) 35:S9-S21, 2010.
2.
Willson MC, Ross JS. Postoperative Spine Complications. Neuroimag Clin N Am
8
ACCEPTED MANUSCRIPT 24:305-326, 2014. 3.
Stambough JL, Quinlan JG, Swanson JD: Guillain-Barré syndrome following spinal fusion for adult scoliosis. Spine (Phila Pa 1976) 15:45-46, 1990. Riebel GD, Heller JG, Hopkins LC: Guillain-Barré syndrome after an operation on the
RI PT
4.
spine. A case report. J Bone Joint Surg Am 77:1565-1567, 1995. 5.
Son DW, Song GS, Sung SK, Kim SH: Guillain-Barre syndrome following spinal fusion
Cheng J, Kahn DE, Wang MY: The acute motor-sensory axonal neuropathy variant of
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6.
SC
for thoracic vertebral fracture. J Korean Neurosurg Soc 50:464-467, 2011.
Guillain-Barré syndrome after thoracic spine surgery. J Neurosurg Spine 15:605-609, 2011. 7.
Battaglia F, Sevy A, Moyse E, Roche PH: Guillain-Barre´ syndrome following severe
8.
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head trauma and spine surgery. Rev Neurol (Paris) 169:166-168, 2013. Sejvar JJ, Baughman AL, Wise M, Morgan OW: Population incidence of Guillain-Barré
2011. Hogan
JC,
Briggs
TP,
Oldershaw
PJ:
Guillain-Barré
syndrome
following
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9.
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syndrome: a systematic review and meta-analysis. Neuroepidemiology 36:123-133,
cardiopulmonary bypass. Int J Cardiol 35:427-428, 1992.
10. Rodriguez V, Kuehnle I, Heslop HE, Khan S, Krance RA: Guillain-Barré syndrome after allogeneic hematopoietic stem cell transplantation. Bone marrow transplant 29:515-517, 2002. 11. Koc M, Ozalp N, Zulfikaroglu B: Major surgery with Guillain-Barré syndrome: a case report. J Int Med Res 30:601-604, 2002.
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ACCEPTED MANUSCRIPT 12. Foubert-Samier A, Penchet G, Yekhlef F, Lemasson G, Sibon I: Guillain-Barré syndrome secondary to cranial surgery: direct or fortuitous relationship? Neurochirurgie 51:604-606, 2005.
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13. Kuok CH, Tsai PS, Hsu YW, Ko YP, Huang CJ, Chen CC: Postoperative delayed respiratory failure caused by Guillain-Barré Syndrome -- a case report. Acta Anaesthesiol Taiwan 45:43-46, 2007.
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fixation. J Trauma 67:E132-E135, 2009.
SC
14. Lee MC, Campbell R, Born C: Guillain-Barré syndrome after failed pelvic fracture
15. Heyworth BE, Fabricant PD, Pizzurro MM, Beksac B, Salvati EA: Guillain–Barré syndrome mimicking nerve injury after total hip arthroplasty. HSS J 7:286-289, 2011. 16. Steger CM, Antretter H, Höfer D: Guillain-Barré syndrome due to CMV reactivation
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after cardiac transplantation. Case Rep Cardiol 2012:506290, 2012. 17. Cingoz F, Tavlasoglu M, Kurkluoglu M, Sahin MA: Guillain–Barré syndrome after coronary artery bypass surgery. Interact Cardiovasc Thorac Surg 15:918-919, 2012.
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18. Masajtis-Zagajewska A, Muras K, Mochecka-Thoelke A, Kurnatowska I, Nowicki M:
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Guillain-Barré syndrome in the course of EBV infection after kidney transplantation--a case report. Ann Transplant 17:133-137, 2012.
19. Israeli E, Agmon-Levin N, Blank M, Chapman J, Shoenfeld Y: Guillain–Barré syndrome--a classical autoimmune disease triggered by infection or vaccination. Clinic Rev Allergy Immunol 42:121-130, 2012.
10
ACCEPTED MANUSCRIPT
TABLE 1 Clinical summary of the patient's information
M/50
2
M/53
3
M/69
4
M/58
Congenital occipitocervical malformations and Cervical disc heriation Cervical spinal stenosis Degenerative lumbar scoliosis Cervical disc heriation
Operation Occipitocervical fusion (Occipital-C2) and Anterior cervical discectomy and fusion (C5-6) Laminoplasty (C3-6) Instrumented posterior-approach lumbar interbody fusion (T10-L5) Anterior cervical discectomy and fusion (C4-7)
Onset time of post-surgery (ds) 7
Delayed time of diagnosis (ds) 16
3 2
3
Muscle power Time of GBS diagnosis 0/5
+
Cerebrospinal fluid: protein (mg/L)/ white blood cell count Missing
Yes
Immunoglobulin
4/5
19
+
3053/8
Yes
Immunoglobulin
4/5
1/5
3-4/5
12
+
Missing
Yes
Immunoglobulin
4/5
2/5
2/5
4
+
500/5
No
Immunoglobulin
4/5
0/5
5/5
Choking while drinking
Ventilator support
Treatment
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Abbreviation: M: male; yrs: years; mns: months; ds: days; C: cervical; T: thoracic; L: lumbar；BI: Barthel index; ADL: activities of daily living
Before operation
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1
Illness
SC
Sex/ Age (ys)
M AN U
Case No
Time of follow-u p 4/5
Category of ADL by BI
Follow-up (mns)
Prognosis
Low dependency
walk with a cane
20
Moderate dependency Total dependency
To transfer from bed to chair To need ventilator support Minor weakness of the intrinsic muscles of the hands
20
Independent
9
5
ACCEPTED MANUSCRIPT
Conduction velocity (m/s) No response Missing 34.0 62.5 >50
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EP
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SC
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TABLE 2 Electrophysiological findings of the median nerve CMAP SNAP Terminal Amplitude Conduction Terminal Amplitude latency (ms) (mV) velocity (m/s) latency (ms) (mV) Case 1 No response No response No response No response No response Case 2 3.2 1.1 52.2 Missing Missing Case 3 4.4 0.5 42.6 3.5 1.3 Case 4 5.1 1.7 54.8 2.4 18.0 Control 5 >50 10 Abbreviation: CMAP: compound motor action potential; SNAP: sensory nerve action potential
F wave responses (%) No response No response 10 5 >75
ACCEPTED MANUSCRIPT
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F wave responses (%) No response No response 5 No response >75
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EP
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SC
TABLE 3 Electrophysiological findings of the posterior tibial nerve CMAP Terminal latency (ms) Amplitude (mV) Conduction velocity (m/s) Case 1 No response No response No response Case 2 6.0 0.6 26.4 Case 3 5.5 1.1 32.1 Case 4 12.0 0.3 29.6 Control 5 >40 Abbreviation: CMAP: compound motor action potential
ACCEPTED MANUSCRIPT
TABLE 4 Summary of previously reported Guillain–Barré syndrome after spine surgery Delayed time of diagnosis (ds) 14
Ventilator support
Electrophysiological examination
Stambough/ 1990[3]
F/33
Idiopathic scoliosis
Riebel/ 1995[4]
F/62
Degenerative lumbar scoliosis and spinal stenosis
Son/ 2011[5]
M/50
Thoracic vertebrae fracture with dislocation
Posterior spinal fusion with Cotrel-Dubousset instrumentation Lumbar decompression with segmental spinal instrumentation and arthrodesis Spinal canal decompression and spinal fusion
Yes
+
22
3
No
+
10
Missing
Cheng/ 2011[6]
F/59
Thoracic meningioma
Laminectomy and tumor resected
8
Missing
Battaglia/ 2013[7]
F/73
Lumbar vertebrae fracture and bilateral temporal and frontal haematomas
Sextant and Kyphoplasty
14
Missing
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Abbreviation: M: male; F: female; yrs: years; mns: months; ds: days
Cerebrospinal fluid analyses
Treatment
Prognosis
Follow-up (mns)
Missing
High-dose decadron
Without neurologic deficits
24
+
High doses of corticosteroids, human immunoglobulin, plasmapheresis Human immunoglobulin
Minor weakness of the intrinsic muscles of the hands Minor weakness of the intrinsic muscles of the hands To transfer from bed to chair Left peripheral facial palsy
8
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Onset time of post-surgery (ds) 10
SC
Operation
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Illness
Yes
+
Missing
No
+
+
Immunoglobulin
No
+
+
Immunoglobulin
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Sex/Age (ys)
EP
Author/year
2
7
4
ACCEPTED MANUSCRIPT
AC C
EP
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SC
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Abbreviations and Acronyms: Guillain–Barré syndrome (GBS); Cerebrospinal fluid (CSF)
ACCEPTED MANUSCRIPT This study demonstrates the clinical features and the management of Guillain–Barré syndrome.
This study warns surgeons to be alert to the association of GBS and spine surgery.
The presentation of GBS after spine surgery is characteristic.
AC C
EP
TE D
M AN U
SC
RI PT
S1878-8750(15)00474-X
DOI:
10.1016/j.wneu.2015.04.045
Reference:
WNEU 2870
To appear in:
World Neurosurgery
Received Date: 4 March 2015 Revised Date:
20 April 2015
Accepted Date: 22 April 2015
Please cite this article as: Huang S-L, Qi H-G, Liu J-J, Huang Y-J, Xiang L, A rare complication of spine surgery: Guillain–Barré syndrome, World Neurosurgery (2015), doi: 10.1016/j.wneu.2015.04.045. This is a PDF file of an unedited manuscript that has been accepted for publication. As a service to our customers we are providing this early version of the manuscript. The manuscript will undergo copyediting, typesetting, and review of the resulting proof before it is published in its final form. Please note that during the production process errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain.
ACCEPTED MANUSCRIPT A rare complication of spine surgery: Guillain–Barré syndrome Sheng-Li Huang1, Hua-Guang Qi2, Jing-Jie Liu3, Ya-Juan Huang1, Li Xiang3 1
Department of Orthopaedics, the Second Affiliated Hospital, Xi'an Jiaotong University
College of Medicine, Xi'an, China Department of Function Examination, Hong Hui Hospital, Xi'an Jiaotong University College
RI PT
2
of Medicine, Xi’an, China 3
Department of Neurology, the Second Affiliated Hospital, Xi'an Jiaotong University College
SC
of Medicine, Xi'an, China
M AN U
To whom correspondence should be addressed: Li Xiang, No.157 Xiwulu, Xi'an 710004, China (Tel: +86-29-87679228; Fax:﹢8629 8767 8634; E-mail: [email protected])
ABSTRACT
TE D
OBJECTIVE: Since little has been reported about Guillain–Barré syndrome (GBS) following spine surgery, this study demonstrates some of the clinical features and the management of the condition.
EP
METHODS: A total of 4 cases of GBS following spine surgery were included. The medical charts of the patients were reviewed to obtain demographic and clinical details. All the
AC C
patients underwent neurological and electrophysiologic examinations, and were followed up after surgery.
RESULTS: The onset of symptoms about GBS was 2-7 days following the operation. Neurological evaluation showed weaknesses of upper and lower extremities and repeal of tendon reflexes. The patients exhibited typical clinical symptoms and signs of GBS, and electromyographic findings. Lumbar puncture was performed in two cases, and cerebrospinal fluid examination showed albumino-cytological dissociation. All the four patients were diagnosed as GBS based on typical clinical, laboratory and electrophysiological findings.
1
ACCEPTED MANUSCRIPT Intravenous immunoglobulin was instituted. At follow-up, one patient needed ventilator support; one patient could transfer from bed to chair; one walked with assistive devices; and the rest one had residual minor neurologic deficits. CONCLUSIONS: These cases warn surgeons to be alert to the association of GBS and spine
patients with paralysis after spine surgery.
RI PT
surgery. Based on our experience, we recommend consideration of this rare diagnosis in
Key words: Spine surgery; Guillain–Barré syndrome; Complication; clinical features;
SC
neuropathology
M AN U
Abbreviations and Acronyms: Guillain–Barré syndrome (GBS); Cerebrospinal fluid (CSF)
INTRODUCTION
TE D
Spine surgery procedures may result in various complications. The onset of complications ranges from 4% to 19% (1), among which the overall incidence of neurologic complications is less than 1% (2). Guillain–Barré syndrome (GBS), one type of neurologic complications
EP
following spinal operation, has rarely been reported (3-7).
AC C
GBS is a rare acute polyradiculoneuropathy that manifests with weakness and diminished reflexes, and may also involve impairment of sensation, cranial nerves, or bulbar muscles. The incidence of GBS is 1 to 2 per 100,000 annually worldwide (8). However, as surgeons may be unfamiliar with it, weakness and paralysis after spine surgery is often attributed to other postoperative complication. Not much about GBS after spine surgery has been published. Most of the original articles are case reports (9-18). Since this condition is rare, it is possible that patients would be
2
ACCEPTED MANUSCRIPT misdiagnosed and subsequently treated incorrectly. To the best of our knowledge, no systematic research has been published so far discussing its clinical characteristics and management strategies. To address this relative lack of information, we performed this
RI PT
retrospective analysis. MATERIALS AND METHODS
From January 2013 to June 2014, a total of 7770 patients were received spine surgery, and 4
SC
of them were diagnosed as GBS after operation. We retrieved the clinical data of the patients,
neurological status, and ancillary tests.
M AN U
including age, gender, history of past illness, surgical procedures, clinical presentation,
The 4 male patients were aged 50 to 69 years with an average of 57.5 years (Table 1). One patient (Case 2) was operated with anterior cervical discectomy and fusion one year before
TE D
this hospitalization, others had no surgical history. They also had neither a recent illness (for example respiratory infection or gastroenteritis) nor any vaccinations in the few months prior to the spine surgery. There was no history of the use of neuromuscular blocking agents,
EP
catecholamines, and aminoglycoside antibiotics in any of these patients. The surgery was
AC C
performed under general anaesthesia. One patient (Case 3) received blood transfusion. No complications occurred during the procedure. All patients received corticosteroids (dexamethasone or prednisolone) after operation. The post-operative recovery was uneventful
until the onset of symptoms of GBS. All patients were followed up. RESULTS On postoperative day 3, two patients (Case 2 and Case 4) developed leg weakness with unstable gait that worsened over several days. On day 2 and 7, two patients (Case 3 and Case
3
ACCEPTED MANUSCRIPT 1) showed weaknesses in upper and lower extremities respectively. In these cases, GBS developed from 2 to 7 days after surgery, and the symptoms progressed rapidly within 1 week. The delay between the onset and diagnosis of GBS ranged from 4 to 19 days (average of
RI PT
12.75 days). The principal clinical manifestation was progressive symmetric motor weakness, with choking while drinking. Stiff neck and other signs of meningeal irritation were not
bilateral facial paralysis in Case 1 and ptosis in Case 2.
SC
present. There was no pathologic reflex. And they were afebrile. Especially there were
M AN U
Case 1 underwent reoperation for exploration 1 day after the aggravated neurological deficit, whereas nothing (such as spinal epidural hematoma) was seen at operation, and
postoperative neurologic deficit was progressively exacerbated. Three of the patients (Case 1, 2 and 3) were moved to the intensive care unit, intubated and supported by mechanical
TE D
ventilation.
Nerve conduction examination in all the patients showed slowed conduction velocity, prolonged terminal latencies in the major peripheral nerves, marked reduction in the
EP
amplitude of muscle action potential, and even an absent response (Table 2 and 3). And
AC C
F-wave was either absent or markedly decreased in number. Electromyography demonstrated fibrillations in Case 2 and 4; unfortunately, at the bedside, fibrillation potentials were not completed in Case 1 and 3 for environmental intervention in the intensive care unit. Therefore, electrophysiological examination revealed peripheral nerve damage. Cerebrospinal fluid (CSF) examination was performed only in Case 2 and Case 4, because Case 3 had postoperative back wound and Case 1 refused to take the examination. The results of the examination presented albumino-cytological dissociation. Once GBS was
4
ACCEPTED MANUSCRIPT confirmed, treatment with high doses of corticosteroids and human immunoglobulin were performed. Intravenous human immunoglobulin was performed at a dose of 0.4 g/kg for 5 days.
RI PT
The follow-up period for these patients ranged from 7 to 22 months (average 15.5 months). One patient could walk with the help of a cane to help walk; one patient could not walk but could transfer from bed to chair; another patient was severely disabled and needed mechanical
SC
support for breathing; and the rest one had completely resolved, except for minor weakness of
M AN U
the intrinsic muscles of the hands, the neurological deficits. DISCUSSION
GBS is a rare but severe neurologic complication after operation. During the past decades, there have been a number of reports presenting GBS following different types of surgery (3-7,
TE D
9-18), but only 5 of the cases (3-7) developed the complication 8 to 22 days after spine surgery (Table 4). The 4 post-spinal-surgery GBS we observed were out of a total of 7770 cases; the incidence of GBS after spine surgery is 1 per 2,000 (4/7770). The onset of the
EP
complication in these patients was within 1 week after surgery, earlier than that in the patients
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reported previously.
GBS is a rapidly progressive demyelinating, monophasic polyneuropathy. It can present
with cranial nerve deficits and respiratory compromise. Typical symptoms of GBS are muscle weakness in the limbs and sensory disturbances such as pain and numbness. The symptoms and signs of GBS are similar to those of spinal cord injury in clinical performance. GBS mimics an acute spinal cord compression injury or nerve root compression syndrome in its initial appearance (sudden postoperative paralysis). Being an unusual postoperative
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ACCEPTED MANUSCRIPT complication, GBS has rarely been described. This makes it difficult for most surgeons to diagnose GBS or to distinguish it from other complications in postoperative settings. The four cases we report here underwent extensive neurological evaluations and exhibited
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the typical clinical symptoms and signs of GBS. The patients had progressively aggravating muscular weakness and paraplegia. The weakness developed acutely by hours or days after surgery. Choking while drinking was developed in all the patients; this cranial nerve
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involvement in particular led to the consideration of GBS. The facial nerve was less often
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affected. Physical examination revealed hyporeflexia or areflexia of the extremities, with the weakness more severe in the distal part than in the proximal part. It can be seen that our patients have many similarities to these previous case reports.
Despite these features, GBS in the setting of surgery remains a diagnostic challenge in
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regard to differentiating it from other complications following operation due to the significant overlap in symptoms. However, when postsurgical spinal cord injury cannot explain the bulbar symptoms or facial weakness in patients, surgeons should take GBS as a consideration.
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The diagnosis of GBS involves a combination of clinical presentation and ancillary tests, such
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as CSF analysis and electromyography. Although spine magnetic resonance imaging can be a choice of diagnostic modality, it remains inconclusive in early postoperative stages. Electromyography is crucial in arriving at the diagnosis. So, we advocate electromyography rather than imaging.
When a patient is found to aggravate in symptoms and signs after spine surgery, corresponding measures should be taken to relieve the condition. For example, the postsurgical neurological deficits in patients with space-occupying lesions such as extradural
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ACCEPTED MANUSCRIPT hematoma and displaced operative implant could be improved after reoperation. However, the situation might worsen if handled inappropriately, such as in our Case 1. Therefore, a precise diagnosis is crucial in deciding and adopting appropriate treatment. The differential diagnosis
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of GBS includes hemorrhage, displaced implants, bony instability, infection, medullar ischemia (vascular insults) and toxicity due to drugs. Cranial nerve involvement, which is associated with choking while drinking or facial palsy, supports the diagnosis. Certainly,
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making a diagnosis of GBS is difficult for most surgeons because they are not familiar with
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this disease. It often takes some time for surgeons to recognize and confirm the condition, and the therapeutic measures specific to the condition are often delayed. For instance, in the four patients we report here, GBS was diagnosed averagely 12.75 days after its onset. This delay might lead to poor prognosis. To avoid such delay, we recommend neurological examinations
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to assist with the diagnosis and management when there is unexplainable progressive postoperative muscle weakness.
GBS is typically caused by an autoimmune attack of peripheral nerve. Infection and
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vaccination have been considered as associated with its occurrence (19). Although the
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condition also occurs after surgery, the mechanisms that link GBS with surgery remain unclear. In the five postsurgical GBS cases reported previously, no other risk factors were identified except surgery. The four patients we discuss here also had not encountered any other incidents; the only clearly identified precipitating event was the spinal operation. However, considering the low incidence of GBS after spine surgery, the operation should not be regarded as the primary inducement of GBS. The limited number of case reports in literature could not support a conclusion that there is a causal relationship between GBS and
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ACCEPTED MANUSCRIPT surgery. However, it is not easy to affirm whether it is mere coincidence. Regular treatment of GBS consists of a 5-day course of intravenous immunoglobulin at a dose of 0.4 g/kg and/or plasma exchange. It has been shown to improve prognosis. So an
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empiric course of intravenous immunoglobulin may be warranted if GBS is a reasonable possibility diagnosis after spinal operation. Supportive care, such as ventilator support, during and following hospitalization is also crucial.
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Recovery is dependent on multiple factors, such as acuity of the onset of symptoms and
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need for ventilator support. If not identified early and treated promptly, this neurological disorder will result in adverse consequences such as permanent neurological impairments. The sequela may worsen with delays in management. Besides, when patients need ventilator support, clinical improvement is slow and residual disability is pronounced.
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CONCLUSION
GBS is a rare neurological complication after spine surgery. When progressive neurological deterioration occurs during the early postoperative period, it is extremely important to be
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aware of occurrence of GBS. The characteristic presentation is rapidly aggravating symmetric
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motor weakness and areflexia, peripheral facial palsy, choking while drinking, or respiratory difficulty. Intravenous immunoglobulin and plasma exchange are efficacious treatments. REFERENCE 1.
Dekutoski MB, Norvell DC, Dettori JR, Fehlings MG, Chapman JR: Surgeon perceptions and reported complications in spine surgery. Spine (Phila Pa 1976) 35:S9-S21, 2010.
2.
Willson MC, Ross JS. Postoperative Spine Complications. Neuroimag Clin N Am
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ACCEPTED MANUSCRIPT 24:305-326, 2014. 3.
Stambough JL, Quinlan JG, Swanson JD: Guillain-Barré syndrome following spinal fusion for adult scoliosis. Spine (Phila Pa 1976) 15:45-46, 1990. Riebel GD, Heller JG, Hopkins LC: Guillain-Barré syndrome after an operation on the
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spine. A case report. J Bone Joint Surg Am 77:1565-1567, 1995. 5.
Son DW, Song GS, Sung SK, Kim SH: Guillain-Barre syndrome following spinal fusion
Cheng J, Kahn DE, Wang MY: The acute motor-sensory axonal neuropathy variant of
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for thoracic vertebral fracture. J Korean Neurosurg Soc 50:464-467, 2011.
Guillain-Barré syndrome after thoracic spine surgery. J Neurosurg Spine 15:605-609, 2011. 7.
Battaglia F, Sevy A, Moyse E, Roche PH: Guillain-Barre´ syndrome following severe
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head trauma and spine surgery. Rev Neurol (Paris) 169:166-168, 2013. Sejvar JJ, Baughman AL, Wise M, Morgan OW: Population incidence of Guillain-Barré
2011. Hogan
JC,
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PJ:
Guillain-Barré
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syndrome: a systematic review and meta-analysis. Neuroepidemiology 36:123-133,
cardiopulmonary bypass. Int J Cardiol 35:427-428, 1992.
10. Rodriguez V, Kuehnle I, Heslop HE, Khan S, Krance RA: Guillain-Barré syndrome after allogeneic hematopoietic stem cell transplantation. Bone marrow transplant 29:515-517, 2002. 11. Koc M, Ozalp N, Zulfikaroglu B: Major surgery with Guillain-Barré syndrome: a case report. J Int Med Res 30:601-604, 2002.
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ACCEPTED MANUSCRIPT 12. Foubert-Samier A, Penchet G, Yekhlef F, Lemasson G, Sibon I: Guillain-Barré syndrome secondary to cranial surgery: direct or fortuitous relationship? Neurochirurgie 51:604-606, 2005.
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13. Kuok CH, Tsai PS, Hsu YW, Ko YP, Huang CJ, Chen CC: Postoperative delayed respiratory failure caused by Guillain-Barré Syndrome -- a case report. Acta Anaesthesiol Taiwan 45:43-46, 2007.
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fixation. J Trauma 67:E132-E135, 2009.
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14. Lee MC, Campbell R, Born C: Guillain-Barré syndrome after failed pelvic fracture
15. Heyworth BE, Fabricant PD, Pizzurro MM, Beksac B, Salvati EA: Guillain–Barré syndrome mimicking nerve injury after total hip arthroplasty. HSS J 7:286-289, 2011. 16. Steger CM, Antretter H, Höfer D: Guillain-Barré syndrome due to CMV reactivation
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after cardiac transplantation. Case Rep Cardiol 2012:506290, 2012. 17. Cingoz F, Tavlasoglu M, Kurkluoglu M, Sahin MA: Guillain–Barré syndrome after coronary artery bypass surgery. Interact Cardiovasc Thorac Surg 15:918-919, 2012.
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18. Masajtis-Zagajewska A, Muras K, Mochecka-Thoelke A, Kurnatowska I, Nowicki M:
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Guillain-Barré syndrome in the course of EBV infection after kidney transplantation--a case report. Ann Transplant 17:133-137, 2012.
19. Israeli E, Agmon-Levin N, Blank M, Chapman J, Shoenfeld Y: Guillain–Barré syndrome--a classical autoimmune disease triggered by infection or vaccination. Clinic Rev Allergy Immunol 42:121-130, 2012.
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TABLE 1 Clinical summary of the patient's information
M/50
2
M/53
3
M/69
4
M/58
Congenital occipitocervical malformations and Cervical disc heriation Cervical spinal stenosis Degenerative lumbar scoliosis Cervical disc heriation
Operation Occipitocervical fusion (Occipital-C2) and Anterior cervical discectomy and fusion (C5-6) Laminoplasty (C3-6) Instrumented posterior-approach lumbar interbody fusion (T10-L5) Anterior cervical discectomy and fusion (C4-7)
Onset time of post-surgery (ds) 7
Delayed time of diagnosis (ds) 16
3 2
3
Muscle power Time of GBS diagnosis 0/5
+
Cerebrospinal fluid: protein (mg/L)/ white blood cell count Missing
Yes
Immunoglobulin
4/5
19
+
3053/8
Yes
Immunoglobulin
4/5
1/5
3-4/5
12
+
Missing
Yes
Immunoglobulin
4/5
2/5
2/5
4
+
500/5
No
Immunoglobulin
4/5
0/5
5/5
Choking while drinking
Ventilator support
Treatment
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Abbreviation: M: male; yrs: years; mns: months; ds: days; C: cervical; T: thoracic; L: lumbar；BI: Barthel index; ADL: activities of daily living
Before operation
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1
Illness
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Sex/ Age (ys)
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Case No
Time of follow-u p 4/5
Category of ADL by BI
Follow-up (mns)
Prognosis
Low dependency
walk with a cane
20
Moderate dependency Total dependency
To transfer from bed to chair To need ventilator support Minor weakness of the intrinsic muscles of the hands
20
Independent
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Conduction velocity (m/s) No response Missing 34.0 62.5 >50
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TABLE 2 Electrophysiological findings of the median nerve CMAP SNAP Terminal Amplitude Conduction Terminal Amplitude latency (ms) (mV) velocity (m/s) latency (ms) (mV) Case 1 No response No response No response No response No response Case 2 3.2 1.1 52.2 Missing Missing Case 3 4.4 0.5 42.6 3.5 1.3 Case 4 5.1 1.7 54.8 2.4 18.0 Control 5 >50 10 Abbreviation: CMAP: compound motor action potential; SNAP: sensory nerve action potential
F wave responses (%) No response No response 10 5 >75
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F wave responses (%) No response No response 5 No response >75
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TABLE 3 Electrophysiological findings of the posterior tibial nerve CMAP Terminal latency (ms) Amplitude (mV) Conduction velocity (m/s) Case 1 No response No response No response Case 2 6.0 0.6 26.4 Case 3 5.5 1.1 32.1 Case 4 12.0 0.3 29.6 Control 5 >40 Abbreviation: CMAP: compound motor action potential
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TABLE 4 Summary of previously reported Guillain–Barré syndrome after spine surgery Delayed time of diagnosis (ds) 14
Ventilator support
Electrophysiological examination
Stambough/ 1990[3]
F/33
Idiopathic scoliosis
Riebel/ 1995[4]
F/62
Degenerative lumbar scoliosis and spinal stenosis
Son/ 2011[5]
M/50
Thoracic vertebrae fracture with dislocation
Posterior spinal fusion with Cotrel-Dubousset instrumentation Lumbar decompression with segmental spinal instrumentation and arthrodesis Spinal canal decompression and spinal fusion
Yes
+
22
3
No
+
10
Missing
Cheng/ 2011[6]
F/59
Thoracic meningioma
Laminectomy and tumor resected
8
Missing
Battaglia/ 2013[7]
F/73
Lumbar vertebrae fracture and bilateral temporal and frontal haematomas
Sextant and Kyphoplasty
14
Missing
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Abbreviation: M: male; F: female; yrs: years; mns: months; ds: days
Cerebrospinal fluid analyses
Treatment
Prognosis
Follow-up (mns)
Missing
High-dose decadron
Without neurologic deficits
24
+
High doses of corticosteroids, human immunoglobulin, plasmapheresis Human immunoglobulin
Minor weakness of the intrinsic muscles of the hands Minor weakness of the intrinsic muscles of the hands To transfer from bed to chair Left peripheral facial palsy
8
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Onset time of post-surgery (ds) 10
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Operation
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Illness
Yes
+
Missing
No
+
+
Immunoglobulin
No
+
+
Immunoglobulin
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Sex/Age (ys)
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Author/year
2
7
4
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Abbreviations and Acronyms: Guillain–Barré syndrome (GBS); Cerebrospinal fluid (CSF)
ACCEPTED MANUSCRIPT This study demonstrates the clinical features and the management of Guillain–Barré syndrome.
This study warns surgeons to be alert to the association of GBS and spine surgery.
The presentation of GBS after spine surgery is characteristic.
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