Composite Type of Split Cord Malformation: Rare and Difficult to Explain

İzci, Yusuf; Kural, Cahit

doi:10.1159/000339209

Cited by 11 publications

(4 citation statements)

References 14 publications

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“…Tethered cord syndrome (TCS) is the clinical manifestation of an abnormal stretch of the spinal cord and is presumed to cause mechanical injury, a compromised blood supply, and altered spinal cord metabolism. TCS is associated with neurological deficits in the lower limbs, leg numbness, lower back pain, foot deformities, and bowel and bladder disturbances [ 1 , 2 ]. It is unlikely that symptoms will sufficiently improve if surgery is performed after the onset of neurological symptoms associated with TCS.…”

Section: Introductionmentioning

confidence: 99%

Surgical Outcomes of Pediatric Patients with Asymptomatic Tethered Cord Syndrome

et al. 2018

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Study DesignA retrospective cohort study.Purpose To examine the validity of prophylactic surgery for children with tethered cord syndrome (TCS).Overview of LiteratureProphylactic surgery for pediatric patients with TCS remains controversial.Methods We retrospectively analyzed the surgical outcomes of 14 children (nine boys and five girls) with asymptomatic TCS who were surgically treated at Hokkaido University Hospital between 1989 and 2015.ResultsThe median age at the time of initial surgery for asymptomatic TCS was 28.6 months (range, 0–66 months). The median final follow-up period was 142 months (range, 7–232 months). Of the 14 children with asymptomatic TCS, 12 had lumbosacral lipoma and two had meningocele. According to the classification of spinal lipoma, two children had dorsal type, four had caudal type, two had transitional type, and four had filar type. There were no children with lipomyelomeningocele. All children were free of neurological symptoms until 94 months after the initial surgery. Subsequently, one child exhibited delayed neurological deficits and underwent a second surgery because of motor and sensory disturbances; slight sensory disturbance was noted at the final follow-up examination. Another child later showed bowel and bladder dysfunction. However, a second surgery was not performed for this child because his motor and sensory functions were normal; hence, we chose to avoid nerve injury in the case of dissecting adhesion.ConclusionsAll 14 children with asymptomatic TCS were free of neurological symptoms until 94 months after the initial surgery. However, two children exhibited delayed neurological deficits at 94 months and 177 months. We believe that prophylactic surgery for asymptomatic TCS is effective for a certain period. However, because the natural history of TCS is poorly understood, strict follow-up after surgery is necessary.

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Section: Introductionmentioning

confidence: 99%

Surgical Outcomes of Pediatric Patients with Asymptomatic Tethered Cord Syndrome

et al. 2018

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“…There is a lack of consensus on the terminology for this combined SCM, whose terminology includes mixed SCM 18) , intermediate SCM 11) , and composite SCM 16) . Recently, Meena et al 15) proposed type 1.5 SCM to define this unusual variation.…”

Section: Discussionmentioning

confidence: 99%

“…In type II SCM (diplomyelia), the hemicords are divided by a thin sagittal fibrous septum within a single dural sac 16) . However, some authors have reported a few cases demonstrating characteristics of both type I and type II SCM, that is, mixed, intermediate, or composite type SCM 2,11,18) . Recently, Meena et al 15) suggested type 1.5 SCM to define these unusual cases.…”

Section: Introductionmentioning

confidence: 99%

Type 1.5 Split Cord Malformation : A New Theory of Pathogenesis

Sun

Tao

Luo³

et al. 2022

J Korean Neurosurg Soc

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To report two cases of type 1.5 split cord malformation (SCM), a subtype of SCM with combined characteristics of types I and II and to review the relevant literature and propose a new possible pathogenetic theory for type 1.5 SCM. A 52-year-old woman had hemicords within a single dural sac with a dorsal bony septum at the L5 level. A 9-year-old boy had hemicords within a single dural sac with a ventral bony septum and fibrous extension at the L3 level. Both patients underwent microsurgical treatments for removing the bony septum, detethering the spinal cord, and sectioning the filum terminale. The surgical procedure revealed an extradural partial bony septum and hemicords within an intact single dural sac in each patient. Both patients were discharged from the hospital without de novo nerve dysfunction. Published cases have validated that types I and II SCM can overlap. We recommend recent type 1.5 SCM as a normative terminology for this overlapping SCM and report two rare cases of this SCM. We propose an associated pathogenesis consisting of uneven distribution and regression to explain type 1.5 SCM. Furthermore, we postulate that the amount of condensing meninx primitiva might determine whether the left bony septum has fibrous extensions to the opposite dura in type 1.5 SCM.

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“…Reviewing the literature, we found that different authors have proposed various terms to name this special subtype of SCM [1][2][3] , but there is still no acknowledged nomenclature. We recommend the use of "type 1.5 SCM" because this subtype contains both characteristics of type 1 and 2 SCM, that is a bony septum of type 1 and a single dura sac of type 2, and we consider that this term can directly reflect its morphological features.…”

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confidence: 99%