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CASE REPORT
Very rare incidence of ascending paralysis in a patient of
traumatic spinal cord injury: a case report
1✉
Anurug Biswas , Sanjay Kumar Pandey1, Anil Kumar Gupta 2
, Jyoti Pandey1 and Srutarshi Ghosh1

© The Author(s), under exclusive licence to International Spinal Cord Society 2022

INTRODUCTION: After spinal cord injury, further neurological deterioration up to one to two neurological levels is not uncommon.
Late neurological deterioration can occur after two months, mainly due to the syrinx formation. In a rare case like in sub-acute post-
traumatic ascending myelopathy, the neurological level may ascend more than four levels from the initial level of injury and it
usually starts within a few weeks after injury.
CASE PRESENTATION: Our case was diagnosed as a case of traumatic spinal cord injury having a lower thoracic neurological level
of injury initially, which rapidly progressed over a few weeks into a higher thoracic neurological level. He was operated with pedicle
screw fixation of the spine before admission to rehabilitation unit. He was having progressive ascending neurological deterioration,
starting a few days after surgery, which was evident by the progression of neurological level by more than four segments clinically.
Cerebrospinal fluid(CSF) study showed no significant abnormality. Magnetic resonance imaging (MRI) study showed involvement of
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the spinal cord at the upper thoracic region. Patient was monitored to note any further worsening. Rehabilitation and supportive
measures were provided according to standard protocol.
DISCUSSION: Very few cases of ascending paralysis of more than four levels have been reported globally. It results in increased
morbidity and mortality in spinal cord injury patients. In our case few possible reasons are ruled out but the actual underlying
reason was not clear. Various hypotheses have been proposed as the cause in previous published literatures. Management is mostly
supportive.
Spinal Cord Series and Cases (2022)8:69 ; https://doi.org/10.1038/s41394-022-00536-4

INTRODUCTION CASE PRESENTATION


Spinal cord injury (SCI) is a dreaded condition causing morbidity Our case was a 22-year-old male, diagnosed as a case of traumatic
and mortality. In a developing country like India, fall from height spinal cord injury with the initial neurological level of injury (NLI)
comprises the majority of the SCI cases [1]. Following spinal cord at the 12th thoracic level (T12) level and clinically belonging to
injury, 5–10% of the patients have acute neurological deteriora- American Spinal Injury Association (ASIA) impairment scale A. He
tion in which the neurological level progresses one to two had a history of fall from a height of 10 feet one month ago which
segments above the initial level [2, 3]. Late neurological resulted in the fracture of the 1st lumbar (L1) vertebra. There was
deterioration usually occurs two months after the initial injury, no concomitant vertebral injury along with L1 fracture. No steroid
among which syrinx formation and myelomalacia comprise the was given post injury at the primary health center from where he
majority of the cases [4, 5]. Sub-acute posttraumatic ascending received primary treatment. Patient was brought to tertiary care
myelopathy (SPAM) is a rare complication that occurs following center six days after injury. Seven days post-injury, he was
traumatic spinal cord injury where neurological level ascends operated on with pedicle screw fixation for his affected vertebra.
more than four segments above the level of initial injury within a During initial clinical evaluation before and after surgery, NLI was
few weeks [6, 7]. It significantly increases morbidity and a few at the 12th thoracic (T12) level as mentioned in his previous
cases might need ventilator support due to diaphragmatic medical records (Fig. 1). He was given intravenous antibiotics and
weakness [7]. Frankel first time reported a case where neurological steroid as post-operative medication. Steroid was added mainly
level ascended 12 segments above within 17 days [8]. SPAM for better post-operative pain and local edema management [10].
occurs in 0.42% to 1% of SCI patients. The exact pathophysiolo- Two weeks after surgery, the patient noticed a progression of his
gical mechanism of SPAM is not known clearly. Different theories sensory loss and gradual weakness of his abdominal muscles. In
have been suggested to explain the underlying mechanism [9]. total, 22 days post-surgery the NLI was T4 during admission to the
Very few cases of SPAM were reported to date globally and most rehabilitation unit. The NLI progressed to T2 level within three
of those are mainly case reports. Considering the rarity of the days of admission. Cerebrospinal fluid (CSF) study was normal and
incident, we decided to report a case of post-traumatic ascending nerve conduction study (NCS) did not show any demyelinating
myelopathy. etiology but T2 weighted spine magnetic resonance imaging (MRI)

1
Department of Physical Medicine and Rehabilitation, All India Institute of Medical Sciences, Patna 801507 Bihar, India. 2Department of Physical Medicine and Rehabilitation, King
George’s Medical University, Lucknow 226003 Uttar Pradesh, India. ✉email: anurugbiswas@gmail.com

Received: 6 February 2022 Revised: 8 July 2022 Accepted: 17 July 2022


A. Biswas et al.
2

Fig. 2 MRI spine (3 week post surgery). Different sections (A–C) of


MRI of the spine in the sagittal plane showing patchy areas of short
segment hyper intensity (red arrow) in the upper thoracic spinal
cord (A); diffuse hyper intensity and thickening involving (yellow
arrow) lower thoracic and lumbar spinal cord (B, C). The spinal cord
from T12 to L2 could not be visualized due to the presence of the
metallic implant (blue arrow).

successful rehabilitation. The patient was rehabilitated and


discharged as wheelchair ambulatory with the advice of close
monitoring of danger signs at home. Regular follow-ups were
done at monthly interval for first 6 months and then he was
Fig. 1 Post injury MRI spine (pre operative). Multiple sections of advised to visit at 6 monthly intervals.
MRI at axial (A–D) and sagittal (E) plane showing burst fracture of
the L1 vertebra (yellow arrow) with retropulsion of bony fragment
causing cord compression and diffuse cord edema (red arrows)
extending from T10 to L1 level. DISCUSSION
After traumatic spinal cord injury, the neurological level of injury
(NLI) is generally lower than the vertebral level as per
revealed long segment diffuse hyper intensity extending from T6 correspondence of the vertebral body with exiting nerve roots
level to conus medullaris, affecting almost the entire section of of the spinal cord [11]. Moreover, due to initial inflammation and
cord with mild thickening of the affected cord and patchy areas of local edema, the probable neurological level may vary in different
hyper intensity in upper thoracic level (Fig. 2). He was given patients with the same vertebral level of injury. In many cases of
intravenous methylprednisolone 500 milligram infusion for five spinal cord injury, there can be a progression of the neurological
days starting from the day of admission to rehabilitation unit till level by one or two-level from the initial level of injury [2, 3].
the MRI report is received. Along with it other rehabilitation However, progression of NLI for four or more levels is not seen
measures like upper limb and core strengthening, lower limb commonly and it is a striking feature of sub-acute post-traumatic
passive range of motion exercises, incentive spirometry, mat ascending myelopathy (SPAM). SPAM is more common in the third
activity, tilt table training, transfer and activity of daily living decade and it can occur with any level of NLI. Though, it is more
training, bladder, bowel care, training for self-intermittent common with injuries of the dorso-lumbar vertebra [9].
catheterization and wheel chair ambulation training were In many instances of SPAM, neurological deterioration is
provided. The patient was closely monitored for any further preceded by low-grade fever [7, 12].
worsening of NLI and the emergence of any new complication but The patient may also have complaints of neck, back or arm pain
NLI remained the same for the further course of management before the appearance of ascending sensory and motor involve-
after initial worsening. His functional independence measure score ment [7, 13, 14]. In our case, the patient was having a low-grade
was 66 on admission which changed to 98 at discharge after fever for two days, before the onset of ascending weakness and

Spinal Cord Series and Cases (2022)8:69


A. Biswas et al.
3
sensory loss. In our case, the CSF study did not reveal any better understanding of risk factors and pathological mechanisms.
significant abnormal finding. A repeat MRI study showed In a populous country like India, prevalence of spinal cord injury is
involvement of different levels of the spinal cord which was not very high, yet very less number of SPAM is reported, showing lack
present in earlier MRI. There was also a mild elevation of white of awareness among physicians regarding SPAM. Available
blood cell count, but that might be due to associated infected literatures are primarily case series and reports and associated
sacral pressure injury. After initial worsening, there was no further features of SPAM are yet to be established. To obtain more
deterioration. We infused intravenous steroid after consulting with information about a rare disease like this each case report like ours
the Neurology team. However, it is not clear whether the stoppage counts.
of progression of the condition is due to steroid or not.
We monitored the patient and followed him up monthly for six
months after discharge from the hospital but there was no further DATA AVAILABILITY
deterioration. Data regarding this case report can be made available with maintaining patient’s
Frankel first described this ascending type of paralysis in the privacy by requesting the corresponding author through provided e-mail.
year 1969 [8]. Multiple case reports have been described later with
various hypotheses explaining this type of difference in the initial
and present neurological level of injury. Few associated factors REFERENCES
have also been identified, such as complete injury, asymptomatic 1. Mathur N, Jain S, Kumar N, Srivastava A, Purohit N, Patni A, et al. Spinal cord
low blood pressure, early postoperative mobilization, nonsurgical injury: scenario in an Indian state. Spinal Cord. 2015;53:349–52. https://doi.org/
treatment [3, 9]. In a developing country like India, majority of the 10.1038/sc.2014.153
patients of spinal cord injury comes from remote areas where 2. Harrop JS, Sharan AD, Vaccaro AR, Przybylski GJ. The cause of neurologic dete-
facility for emergency surgery is not available. Even in urban areas, rioration after acute cervical spinal cord injury. Spine. 2001;26:340–6. https://
most hospitals are not equipped for spinal surgery. Moreover, doi.org/10.1097/00007632-200102150-00008
3. Yablon IG, Ordia J, Mortara R, Reed J, Spatz E. Acute ascending myelopathy of the
majority of patients have financial issues leading to delay in
spine. Spine. 1989;14:1084–9. https://doi.org/10.1097/00007632-198910000-00010
visiting well equipped hospitals and delay in surgery. Our case had 4. Fehlings MG, Austin JW. Posttraumatic syringomyelia. J Neurosurg Spine.
initial delay before surgery and we know SPAM is more common 2011;14:570–2. https://doi.org/10.3171/2010.4.SPINE1047
in patients with non-surgical treatment and early postoperative 5. Lee TT, Arias JM, Andrus HL, Quencer RM, Falcone SF, Green BA, et al. Progressive
mobilization. So, the initial delay in surgery might have mimicked posttraumatic myelomalacic myelopathy: treatment with untethering and expansive
the similar condition leading to initiation of the mechanism duraplasty. J Neurosurg. 1997;86:624–8. https://doi.org/10.3171/jns.1997.86.4.0624
responsible for increased incidence of SPAM in conservatively 6. Belanger E, Picard C, Lacerte D, Lavallee P, Levi AD. Subacute posttraumatic
managed traumatic spinal cord injury. ascending myelopathy after spinal cord injury. Report of three cases. J Neurosurg.
Ghatany et al. even performed an autopsy in a case of post- 2000;93:294–9. https://doi.org/10.3171/spi.2000.93.2.0294
7. Planner AC, Pretorius PM, Graham A, Meagher TM. Subacute progressive
traumatic ascending myelopathy; and found infarction and edema
ascending myelopathy following spinal cord injury: MRI appearances and clinical
of the spinal cord above the level of initial injury [14]. Vascular presentation. Spinal Cord. 2008;46:140–4. https://doi.org/10.1038/sj.sc.3102056
thrombosis, autoimmune disease, infection, obstruction of CSF 8. Frankel H. Ascending cord lesion in the early stages following spinal injury. Spinal
flow, hypotensive ischemia, etc. are described as the probable Cord. 1969;7:111–8. https://doi.org/10.1038/sc.1969.21
cause behind this phenomenon in a literature review by Zhang 9. Zhang J, Wang G. Subacute posttraumatic ascending myelopathy: a literature
et al. in 2017 [9]. review. Spinal cord. 2017;55:644–50. https://doi.org/10.1038/sc.2016.175
However, our patient had lower motor neuron type features, 10. Fletcher ND, Ruska T, Austin TM, Guisse NF, Murphy JS, Bruce RW Jr., et al.
like flaccid lower limbs, atonic bladder, absent plantar reflex, etc., Postoperative dexamethasone following posterior spinal fusion for adolescent
which is not commonly seen in a patient with high thoracic NLI idiopathic scoliosis. J Bone Jt Surg Am. 2020;102:1807–13. https://doi.org/
10.2106/JBJS.20.00259.
[15]. It is not common for patient to be in spinal shock for this long
11. Canbay S, Gürer B, Bozkurt M, Comert A, Izci Y, Başkaya MK, et al. Anatomical
and these findings might be due to the involvement of many relationship and positions of the lumbar and sacral segments of the spinal cord
alpha motor neurons. There was no significant rise or fall in blood according to the vertebral bodies and the spinal roots. Clin Anat. 2014;27:227–33.
pressure from baseline in our patient. Vascular studies were not https://doi.org/10.1002/ca.22253
done in our case. We also considered the possibility of post- 12. Kumar A, Kumar J, Garg M, Farooque K, Gamanagatti S, Sharma V. Posttraumatic
traumatic Guillain-barre syndrome (GBS) [16, 17]. Though, clinical subacute ascending myelopathy in a 24-year-old male patient. Emerg Radiol.
features, CSF study, and nerve conduction study (NCS) study did 2010;17:249–52. https://doi.org/10.1007/s10140-009-0832-8
not show anything significant in favor of the GBS in our case. The 13. Schmidt BJ. Subacute delayed ascending myelopathy after low spine injury: case
exact underlying reason behind the findings in our case is still report and evidence of a vascular mechanism. Spinal Cord. 2006;44:322–5.
https://doi.org/10.1038/sj.sc.3101801
not clear.
14. Al-Ghatany M, Al-Shraim M, Levi AD, Midha R. Pathological features including
Different medications such as anticoagulation therapy, steroid, apoptosis in subacute posttraumatic ascending myelopathy. Case report and
antibiotics, etc. have been tried as conservative options in past review of the literature. J Neurosurg Spine. 2005;2:619–23. https://doi.org/
[6, 9, 12, 13]. Surgery like decompression laminectomy, untether- 10.3171/spi.2005.2.5.0619
ing of the cord have also been described as the management 15. Doherty JG, Burns AS, O’Ferrall DM, Ditunno JF Jr. Prevalence of upper motor
options in a few cases [5, 8]. However, for most cases, manage- neuron vs lower motor neuron lesions in complete lower thoracic and lumbar
ment is usually supportive with close monitoring of vitals, NLI, and spinal cord injuries. J Spinal Cord Med. 2002;25:289–92. https://doi.org/10.1080/
the new onset of a complication. Institution-based rehabilitation 10790268.2002.11753630
protocol was followed for the patient along with close monitoring 16. Gensicke H, Datta AN, Dill P, Schindler C, Fischer D. Increased incidence of
Guillain-Barré syndrome after surgery. Eur J Neurol. 2012;19:1239–44. https://
of vitals and NLI. Features of our case was similar to the sub-acute
doi.org/10.1111/j.1468-1331.2012.03730.x
post-traumatic ascending myelopathy (SPAM) described by 17. Al-Hashel JY, John JK, Vembu P. Unusual presentation of Guillain-Barré syndrome
different authors previously. In our case, there were no fatal following traumatic bone injuries: report of two cases. Med Princ Pract.
complications, fortunately. Though we ruled out a few causes, the 2013;22:597–9. https://doi.org/10.1159/000348797
exact reason behind the findings of our case was not clear.
Management of SPAM is mostly supportive and close monitoring
is required to detect it early. As SPAM is associated with increased ACKNOWLEDGEMENTS
morbidity and mortality, awareness for regular monitoring and We thank to all faculty members and residents of Department of Physical Medicine
clinical assessment is necessary for all spinal cord injury patients. and Rehabilitation, AIIMS, Patna for their support in care of the patient and data
This rare condition needs to be reported and studied further for a acquisition.

Spinal Cord Series and Cases (2022)8:69


A. Biswas et al.
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AUTHOR CONTRIBUTIONS ADDITIONAL INFORMATION
AB identified the case and particular findings, conceptualized the manuscript, Supplementary information The online version contains supplementary material
searched literature and wrote the manuscript; SKP reviewed the examination and available at https://doi.org/10.1038/s41394-022-00536-4.
investigations, determined management protocol and responsible for reviewing the
manuscript; AKG helped in patient care, conceptualized the manuscript, helped in Correspondence and requests for materials should be addressed to Anurug Biswas.
writing discussion and gave feedback for modifications of manuscript; JP examined
the patient and acquired data and photographs; SG examined the patient and Reprints and permission information is available at http://www.nature.com/
acquired data and helped in writing the manuscript. reprints

Publisher’s note Springer Nature remains neutral with regard to jurisdictional claims
COMPETING INTERESTS in published maps and institutional affiliations.
The authors declare no competing interests.

Spinal Cord Series and Cases (2022)8:69

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