Background
Spinal cord injury without radiographic abnormality (SCIWORA) was defined as a myelopathy with no radiographic or computed tomographic features of spinal fracture or instability [
1]. This disorder is more common in children, accounting for 6 to 19% of all pediatric spinal cord injuries [
2]. SCIWORA is more often associated with high-energy injuries in younger children [
3]. In older children, especially adolescents, SCIWORA is most commonly associated with athletic competition or automobile accidents. The mechanism of pediatric SCIWORA following minor trauma is still unclear. The trauma factors of pediatric SCIWORA include hyperextension, flexion, distraction, direct crush injury or combinations and ischemia spinal cord injuries due to the unique and inherent anatomic malleability of the pediatric spine [
4].
Tethered cord syndrome (TCS) refers to a group of signs and symptoms of motor and sensory neuron dysfunction attributable to spinal cord traction [
5]. TCS is a component of occult spinal dysraphism, such as tight filum terminale (TFT) [
6]. TFT has been well studied in the literature, but the information regarding the predisposing factor for SCIWORA is still being defined. We report such 3 cases of thoracic and lumber SCIWORA with TFT. To the best of our knowledge, this is the first report of SCIWORA with TFT. TFT was found during operation. We suggest that TFT and chronic spinal cord traction play an important role in the mechanism of SCIWORA following minor trauma.
Discussion and conclusion
SCIWORA is relatively common in children under 8 years old [
3]. The severity of SCIWORA was related to the age of onset [
4]. Thoracic cord injury accounts for 9.61% of pediatric SCIWORA cases and lumbar cord injury accounts for 1.48% [
7]. TCS is a stretch-induced functional disorder of the spinal cor d[
8].The filum terminale has clinical significance in its contribution to TCS, a form of occult spinal dysraphis m[
5,
9,
10].TFT is not an uncommon condition, but is often overlooked and left untreated due to insufficient diagnostic options [
11,
12].
Considering the trauma, it was potentially mild in all patients, but resulted in catastrophic damage of the cord. All three SCIWORA patients presented delayed onset flaccid paraparesis, sensory disturbance below the thoracic dermatome, and bladder and bowel dysfunction after the injury. All of the patients exhibited lower thoracic and lumber spinal injuries and the prognosis was poor. In the first case, TFT with fibrous degeneration, contusion, and laceration in the conus medullary were found in the operation. In the second case, contusion in the conus medullary and TFT with fibrolipoma were found in the operation. In the third case, swelling in the conus medullary, tortuous dilation of proximal cone vessels and 4 mm in diameter TFT with fibrolipoma were found in the operation.
Considering the preexisting spinal or neurological abnormality, this is the first report of SCIWORA with TFT. Preoperative MRI showed fibrolipoma of the filum terminale in the third case. Terminal filum with fibrous degeneration was found in the first case and terminal filum fibrolipoma was found in the second case and the third case. TFT was found in all cases during operation. The basic underlying problem in TFT is abnormal longitudinal stretch on the conus by the filum. In this case a subclinical degree of spinal cord traction is present. The exact pathophysiological mechanisms are not yet completely elucidated. Yamada et al. reported that the longitudinal stretch precipitates metabolic derangements equivalent to ischemic injury [
13]. Experimental models studies show that mild to moderate stretch causes transient reductions in metabolism and severe stretch precipitates persistent metabolic derangements that may not recover [
14]. The pediatric spine is vulnerable to external forces and allows for significant intersegmental movement [
15].. Tani et al. reported that stretch of the terminal filum produces maximum elongation of the lumbo-sacral enlargements in the spinal cor d[
16].The mechanism of the trauma could have been related to the flexion–extension movement, which may cause “additional tugging” in congenital tight conus and damage the cord [
17‐
19]. Despite the small number of patients in this report, we inferred that pre-existing TFT might be a predisposing factor for SCIWORA following minor trauma.
In conclusion, we suggest that TFT might be a predisposing factor for SCIWORA and chronic spinal cord traction play an important role in the mechanism of pediatric thoracic and lumber SCIWORA following minor trauma. However, future studies are needed to test its validity. Patients who never undergo treatment for TFT likely have an elevated risk of developing SCIWORA following minor trauma.
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