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\title{Spontaneous Spinal Cord Herniation with Post-operative paraplegia- A
case report with 10-year follow-up}



\author[1]{Syed Ifthekar}%
\author[2]{Sang-Ha Shin}%
\author[2]{Sang-Ho Lee}%
\author[2]{Junseok Bae}%
\affil[1]{All India Institute of Medical Sciences - Bibinagar}%
\affil[2]{Wooridul Hospital}%


\vspace{-1em}



  
  \date{June 18, 2024}


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\textbf{Spontaneous Spinal Cord Herniation with Post-operative
paraplegia- A case report with 10-year follow-up}

\textbf{Abstract:}

\textbf{Background:} Spinal cord herniation is an uncommon diagnosis in
the field of spine surgery. The usual presentation of spontaneous spinal
cord herniation is in the form of progressive Brown-Sequard syndrome. We
describe a case of a 37-year-old male with progressive back pain and
sensory deficits due to spinal cord herniation and a post-operative
complication associated with reduction of the hernia.

\textbf{Case description:} A 37-year-old male presented with insidious
onset upper back pain and altered sensations of pain and temperature
over the right half of the body below the nipple 2 months before the
examination. The patient did not have motor weakness of lower limbs,
abnormal/ involuntary movements, or loss of control over the bowel and
bladder. MRI of the thoracic spine showed an anterolateral defect(left)
at the level of the T2-T3 vertebra. A posterior approach was chosen and
the cord with roots was reduced into the dura. The defect was covered by
a dural graft (Lyodura) and the wound was closed with a drain insitu. On
the 3\textsuperscript{rd} post-operative day, patient developed
paraplegia. Patient was treated by exploration and decompression of the
hematoma that compressed spinal cord. The deficits were completely
recovered at one-month follow-up.

\textbf{Conclusion:} Patients with spinal cord herniation and neurologic
deficits when treated timely with reduction of the hernia, have good
outcomes. The drain should be removed only when the treating team is
satisfied regarding the lack of ongoing hemorrhage. The recovery was
maintained till the last follow-up at 10 years.

\textbf{Key words:} spontaneous spinal cord herniation, spinal cord
defect, paraplegia, Brown-Sequard syndrome

\textbf{Background:}

Spinal cord herniation is an uncommon diagnosis in the field of spine
surgery{[}1{]}. The etiology is unknown but few theories are attributed
to spontaneous herniation like congenital defects in the dura, pressure
erosion of the dura, duplication of anterior dura, and traumatic
etiology{[}1, 2{]}. The usual presentation of spontaneous spinal cord
herniation is in the form of progressive Brown-Sequard syndrome{[}3{]}.
Neurological deficits are thought to occur as a result of tethering of
herniated cord to the surrounding structures{[}4{]}. The tethering
causes a longitudinal stretch and impairs the neuronal oxidative
metabolism resulting in a wide spectrum of neurological deficits{[}4{]}.
The neurological deficits stabilise or recover when the herniation is
reduced surgically and the dural defect is closed{[}5{]}. We describe a
case of a 37-year-old male with progressive back pain and sensory
deficits due to spinal cord herniation and a post-operative complication
associated with reduction of the hernia.

\textbf{Case report:}

A 37-year-old male patient visited the outpatient clinic with insidious
onset of upper back pain and altered sensations of pain and temperature
over the right half of the body below the nipple 2 months before the
examination. The patient did not have motor weakness of lower limbs,
abnormal/ involuntary movements, or loss of control over the bowel and
bladder. There was no history of injury to the back, fever, weight loss,
or any other constitutional symptoms. There was no history of Diabetes
Mellitus, hypertension, or other co-morbidities. There was no specific
tenderness or visible deformity of the spine. The complete neurological
examination is described in table 1.

The radiologic examination was done in the form of X-rays of the
thoracic spine in anteroposterior and lateral views. There were no
significant findings on the x-ray of the thoracic spine. Magnetic
Resonance Imaging (MRI) of the thoracic spine showed an anterolateral
defect(left) in the dura at the level of the T2-T3 vertebra (Fig 1A\&B).
Computed Tomography(CT) scan of the thoracic spine also showed a defect
of the spinal cord at the T2-T3 vertebral level(2A-C). A differential
diagnosis of spontaneous spinal cord herniation and arachnoid cysts was
considered. Surgical intervention was planned as the patient had
neurological deficits.

A posterior approach was chosen to repair the spinal cord defect. After
laminectomy, the dural defect was assessed and the diagnosis of
spontaneous spinal cord herniation was confirmed (Fig 3A). The cord with
roots was reduced into the dura. The defect was covered by a dural graft
(Lyodura) and the wound was closed with a drain insitu (Fig 3B). The
patient's sensory deficits improved on the first postoperative day. The
output from the drain was 100 ml on the first postoperative day and 50
ml on the second postoperative day. The drain was removed in the evening
of second postoperative day. In the early morning of the third
post-operative day, the patient complained of pain over the operated
site and inability to move bilateral lower limbs. On examination, there
was a marked swelling over the operated site and paraplegia. The motor
weakness was also associated with sensory loss below the nipples
bilaterally. However, bowel and bladder control was not lost. The
patient was immediately taken into the operating room as there was
swelling and tenderness over the wound and a hematoma compressing the
spinal cord was suspected. The stitches were removed and the wound was
explored. There was a hematoma at the operated site which was removed
(Fig 4). The bleeders were identified and cauterised. Repair of the
defect was re-examined and was found to be satisfactory. Layer-wise
closure was done. The check MRI showed good closure of the defect and
compression over spinal cord(Fig 5A \& 5B). The motor weakness recovered
but the sensory paraesthesia persisted till the ninth postoperative day.
The patient was discharged on the ninth postoperative day with residual
sensory deficits and was kept under regular follow-up. The deficits were
completely recovered at one-month follow-up. The recovery was maintained
till the last follow-up at 10 years.

\textbf{Discussion:}

Thoracic spinal cord herniation is a rarely reported diagnosis in the
literature. The rarity may partly be due to the difficulty in arriving
at the diagnosis due to inconsistent clinical findings{[}5{]}. The first
case was reported by wortzman G et al when high resolution MRI was not
available for diagnosis{[}6{]}. The study group identified a progressive
Brown-Sequard-like syndrome which was not explained by the usual
etiology{[}6{]}. The majority of the spontaneous thoracic cord
herniations reported in the literature are in the form of single case
reports and we found less than 220 cases in the entire literature to
date{[}1{]}. Most of the cases of herniations are localised between the
thoracic third and seventh segments in the middle-aged
individuals{[}7{]}. The herniation of the spinal cord through the dura
usually occurs through the anterior or anterolateral part of the dural
sheath{[}1{]}. The dorsal herniation is very rare{[}4{]}. There are few
postulated theories for the herniation of the spinal cord which include
congenital dural deficiency with a preexisting anterior meningocele,
traumatic etiology, duplication of anterior dura, and microscopic
erosion of anterior dura during repeated flexion and extension but none
of these etiologies are proven in literature{[}1, 2{]}. So, this
condition is often referred to as an idiopathic condition{[}2{]}.

The cases of spinal cord herniation show a wide spectrum of symptoms.
The usual presenting symptom is progressive Brown-Sequard type of
syndrome with contralateral altered pain and temperature with
ipsilateral hemiparesis{[}3{]}. The neurological deficits may or may not
follow the dermatomal/myotomal distribution. The adhesion of the spinal
cord to the surrounding structures and resultant vascular compromise are
thought to cause neurological deficits{[}4{]}. Due to these non-standard
symptoms, many cases of spinal cord herniations were misdiagnosed as
medical causes of paraplegia like multiple sclerosis, demyelinating
neuropathy, and sub-acute combined degeneration of spinal cord{[}8{]}.
With the wide-spread use of MRI for the diagnosis of spinal conditions,
many cases of spinal cord herniations are being reported in the recent
past. The MRI sagittal view would show focal anterior displacement of
the spinal cord and the axial view shows a lateral defect in the dura
and herniation of the spinal cord{[}9{]}. Care must be taken not to miss
the arachnoid cysts if they are present with spinal cord
herniations{[}9{]}. The nerve roots are visible in the periphery of the
arachnoid cysts whereas the nerve roots are visible in the dorsal
subarachnoid region in the case of spinal cord herniations without
arachnoid cysts{[}1, 9{]}.

The usual treatment in patients presenting with neurological deficits
and with a diagnosis of spinal cord herniation is surgical repair of the
herniation{[}10{]}. The repair is focused on reducing the cord contents
into the dura and closing the defect. Reducing the cord into the dura
has been shown to improve neurological symptoms{[}11{]}. There are few
cases where the symptoms have deteriorated even after surgical
intervention. The cause could be late retethering or the development of
hematoma causing compression. In our case, although the neurological
deficits improved after surgery, there was a sudden deterioration of the
neurology on the third day. This is usually due to hematoma causing
mechanical compression or occlusion of venous drainage. In our case, the
etiology was hematoma and removal of hematoma satisfactorily treated the
symptoms. Ammar et al reported spontaneous retethering in patients with
spinal cord herniation after treatment{[}12{]}. The patient should be
kept in constant follow-up and the retethering symptoms should be
explained in detail at discharge.

The evidence available in the literature for treating spontaneous spinal
cord herniations is limited to case reports and few case series. As
there are reports stating spontaneous reduction of the hernia and
deterioration of symptoms after surgery, strong guidelines for the
management of spinal cord herniations are lacking{[}1{]}. With the
extensive use of MRI, the number of cases diagnosed as spinal cord
herniation is increasing and strong evidence-based guidelines for which
patients to be treated surgically are the need of the hour.

\textbf{Conclusion:}

Patients with spinal cord herniation and neurologic deficits when
treated timely with reduction of the hernia, have good outcomes. The
drain should be removed only when the treating team is satisfied
regarding the lack of ongoing hemorrhage. The surgical approach should
be considered as a treatment in cases of spinal cord herniation.

\textbf{References:}

1. Samuel N, Goldstein CL, Santaguida C, Fehlings MG (2015) Spontaneous
resolution of idiopathic thoracic spinal cord herniation: case report. J
Neurosurg Spine 23:306--308. https://doi.org/10.3171/2014.12.SPINE14950

2. Morley S, Naidoo P, Robertson A, Chong W (2006) Thoracic ventral
dural defect: idiopathic spinal cord herniation. Australas Radiol
50:168--170. https://doi.org/10.1111/j.1440-1673.2006.01547.x

3. Cellerini M, Bayon S, Scazzeri F, et al (2002) Idiopatic spinal cord
herniation: a treatable cause of Brown-S\selectlanguage{ngerman}équard syndrome. Acta Neurochir
(Wien) 144:321--325. https://doi.org/10.1007/s007010200044

4. Le TC, Grunch BH, Karikari IO, et al (2012) Dorsal thoracic spinal
cord herniation: report of an unusual case and review of the literature.
The Spine Journal 12:e9--e12.
https://doi.org/10.1016/j.spinee.2012.09.039

5. Jeong K-H, Lee H-W, Kwon Y-M (2016) Spontaneous Anterior Thoracic
Spinal Cord Herniation through Dura Defect: A Case Report. Korean J
Spine 13:77--79. https://doi.org/10.14245/kjs.2016.13.2.77

6. Wortzman G, Tasker RR, Rewcastle NB, et al (1974) Spontaneous
incarcerated herniation of the spinal cord into a vertebral body: a
unique cause of paraplegia. Case report. J Neurosurg 41:631--635.
https://doi.org/10.3171/jns.1974.41.5.0631

7. Groen RJM, Middel B, Meilof JF, et al (2009) Operative treatment of
anterior thoracic spinal cord herniation: three new cases and an
individual patient data meta-analysis of 126 case reports. Neurosurgery
64:ons145-159; discussion ons159-160.
https://doi.org/10.1227/01.NEU.0000327686.99072.E7

8. Hassler W, Al-Kahlout E, Schick U (2008) Spontaneous herniation of
the spinal cord: operative technique and follow-up in 10 cases: Clinical
article. SPI 9:438--443. https://doi.org/10.3171/SPI.2008.9.11.438

9. Sioutos P, Arbit E, Tsairis P, Gargan R (1996) Spontaneous thoracic
spinal cord herniation. A case report. Spine (Phila Pa 1976)
21:1710--1713. https://doi.org/10.1097/00007632-199607150-00019

10. Kawachi I, Nozaki H, Watanabe M, et al (2001) Spontaneous spinal
cord herniation. Neurology 56:977. https://doi.org/10.1212/wnl.56.7.977

11. Eguchi T, Yokota H, Nikaido Y, et al (2001) Spontaneous thoracic
spinal cord herniation--case report. Neurol Med Chir (Tokyo)
41:508--512. https://doi.org/10.2176/nmc.41.508

12. Ammar KN, Pritchard PR, Matz PG, Hadley MN (2005) Spontaneous
thoracic spinal cord herniation: three cases with long-term follow-up.
Neurosurgery 57:E1067; discussion E1067.
https://doi.org/10.1227/01.neu.0000180016.69507.e0\selectlanguage{english}
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