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http://dx.doi.org/10.3340/jkns.2020.0360

Type 1.5 Split Cord Malformation : A New Theory of Pathogenesis  

Sun, Mengchun (Department of Neurosurgery, the First Medical Center of Chinese PLA General Hospital)
Tao, Benzhang (Department of Neurosurgery, the First Medical Center of Chinese PLA General Hospital)
Luo, Tianbao (Department of Neurosurgery, Yuquan Hospital)
Gao, Gan (Department of Neurosurgery, the First Medical Center of Chinese PLA General Hospital)
Shang, Aijia (Department of Neurosurgery, the First Medical Center of Chinese PLA General Hospital)
Publication Information
Journal of Korean Neurosurgical Society / v.65, no.1, 2022 , pp. 138-144 More about this Journal
Abstract
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.
Keywords
Split cord malformation; Pathogenesis; Embryogenesis; Meninx; Regression;
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1 Akay KM, Izci Y, Baysefer A, Timurkaynak E : Composite type of split cord malformation: two different types at three different levels: case report. J Neurosurg 102(4 Suppl) : 436-438, 2005
2 Chandra PS, Kamal R, Mahapatra AK : An unusual case of dorsally situated bony spur in a lumbar split cord malformation. Pediatr Neurosurg 31 : 49-52, 1999   DOI
3 Ersahin Y : An unusual split cord malformation. Pediatr Neurosurg 32 : 109, 2000   DOI
4 Herren RY, Edwards JE : Diplomyelia (duplication of the spinal cord). Arch Pathol 30 : 1203-1213, 1940
5 Hertwig O : Urmund und spina bifida. Archiv f mikrosk Anatomie 39 : 353-502, 1892   DOI
6 Marr GE, Uihlein A : Diplomyelia and compression of the spinal cord and not of the cauda equina, by a congenital anomaly of the third lumbar vertebra. Surg Clin North Am 24 : 963-977, 1944
7 Meena RK, Doddamani RS, Gurjar HK, Kumar A, Chandra PS : Type 1.5 split cord malformations: an uncommon entity. World Neurosurg 133 : 142-149, 2020   DOI
8 Singh PK, Khandelwal A, Singh A, Ailawadhi P, Gupta D, Mahapatra AK : Long-segment type 1 split cord malformation with two-level split cord malformation and a single dural sac at the lower split. Pediatr Neurosurg 47 : 227-229, 2011   DOI
9 Van Aalst J, Beuls EA, Vles JS, Cornips EM, van Straaten HW : The intermediate type split cord malformation: hypothesis and case report. Childs Nerv Syst 21 : 1020-1024, 2005   DOI
10 Lichtenstein BW : Spinal dysraphism: spina bifida and myelodysplasia. Arch Neur Psych 44 : 792-810, 1940   DOI
11 Izci Y, Kural C : Composite type of split cord malformation: rare and difficult to explain. Pediatr Neurosurg 47 : 461, 2011   DOI
12 Salunke P, Kovai P, Malik V, Sharma M : Mixed split cord malformation: are we missing something? Clin Neurol Neurosurg 113 : 774-778, 2011   DOI
13 Xu X, Li C, Takahashi K, Slavkin HC, Shum L, Deng CX : Murine fibroblast growth factor receptor 1alpha isoforms mediate node regression and are essential for posterior mesoderm development. Dev Biol 208 : 293-306, 1999   DOI
14 Akay KM, Izci Y, Baysefer A : Dorsal bony septum: a split cord malformation variant. Pediatr Neurosurg 36 : 225-228, 2002   DOI
15 Basak M, Ozel A, Erturk M : An unusual case of diastematomyelia. Presence of one dural sheath associated with a bony spur. Acta Radiol 43 : 626, 2002   DOI
16 Bruce A, M'Donald S, Pirie JHH : A second case of partial doubling of the spinal cord. Rev Neurol Psychiatr 4 : 6, 1906
17 Colas JF, Schoenwolf GC : Towards a cellular and molecular understanding of neurulation. Dev Dyn 221 : 117-145, 2001   DOI
18 Garg K, Mahapatra AK, Tandon V : A rare case of type 1 C split cord malformation with single dural sheath. Asian J Neurosurg 10 : 226-228, 2015   DOI
19 Maxwell HP, Bucy PC : Diastematomyelia; report of a clinical case. J Neuropathol Exp Neurol 5 : 165-167, 1946   DOI
20 Pickles W : Duplication of the spinal cord; an account of a clinical example with a consideration of other reports. J Neurosurg 6 : 324-331, 1949   DOI
21 Vaishya S, Kumarjain P : Split cord malformation: three unusual cases of composite split cord malformation. Childs Nerv Syst 17 : 528-530, 2001   DOI
22 Pang D, Dias MS, Ahab-Barmada M : Split cord malformation: part I: a unified theory of embryogenesis for double spinal cord malformations. Neurosurgery 31 : 451-480, 1992   DOI