Limited Dorsal Myeloschisis: Reconsideration of its Embryological Origin

Kim, Joo Whan; Wang, Kyu Chang; Chong, Sangjoon; Kim, Seung Ki; Lee, Ji Yeoun

doi:10.1093/neuros/nyy632

Cited by 18 publications

(20 citation statements)

References 13 publications

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“…In the present case, the morphology of the association between RMC and CDS differed from that of the typical association between LDM and CDS, and the C-LS of the RMC was extended to become an extradural stalk with CDS elements. In a study by Kim et al [22], 11 out of 28 LDMs were located below the S1–2 vertebral level, suggesting that it had been caused by errors in secondary neurulation. In the present case, the RMC may have terminated in an LDM stalk located below the bifid S1 level, with CDS elements during secondary neurulation.…”

Section: Discussionmentioning

confidence: 99%

Congenital Dermal Sinus Elements in Each Tethering Stalk of Coexisting Thoracic Limited Dorsal Myeloschisis and Retained Medullary Cord

et al. 2020

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Introduction: The embryogenesis of limited dorsal myeloschisis (LDM) likely involves impaired disjunction between the cutaneous and neural ectoderms during primary neurulation. Because LDM and congenital dermal sinus (CDS) have a shared origin in this regard, CDS elements can be found in the LDM stalk. Retained medullary cord (RMC) is a closed spinal dysraphism involving a robust, elongated, cord-like structure extending from the conus medullaris to the dural cul-de-sac. Because the RMC is assumed to be caused by impaired secondary neurulation, concurrent RMC and CDS cannot be explained embryologically. In the present article, we report a case in which CDS elements were noted in each tethering stalk of a coexisting LDM and RMC. Case Presentation: A 2.5-month-old boy with left clubfoot and frequent urinary and fecal leakage had 2 tethering tracts. The upper tract, which ran from the thoracic tail-like cutaneous appendage, had CDS elements in the extradural stalk and a tiny dermoid cyst in the intradural stalk immediately after the dural entry. In the lower tract, which ran from the lumbosacral dimple, the CDS as an extradural stalk continued to the RMC at the dural cul-de-sac. Both stalks were entirely resected through skip laminotomy/laminectomy at 1 stage to untether the cord and resect the CDS elements. Conclusion: Surgeons should be aware that CDS elements, in addition to LDM, may coexist with RMC that extends out to the extradural space.

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Section: Discussionmentioning

confidence: 99%

Congenital Dermal Sinus Elements in Each Tethering Stalk of Coexisting Thoracic Limited Dorsal Myeloschisis and Retained Medullary Cord

et al. 2020

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“…Recently, my team reported that LDM may be found in the area of secondary neurulation (Fig. 2) 11) . The continuation of the caudal cell mass with the surface-located primitive streak in the early phase of secondary neurulation gives the chance for non-disjunction to occur.…”

Section: Limited Dorsal Myeloschisis (Ldm)mentioning

confidence: 98%

Spinal Dysraphism in the Last Two Decades : What I Have Seen during the Era of Dynamic Advancement

Wang

2020

J Korean Neurosurg Soc

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Compared to any other decade, the last two decades have been the most dynamic period in terms of advances in the knowledge on spinal dysraphism. Among the several factors of rapid advancement, such as embryology during secondary neurulation and intraoperative neurophysiological monitoring, there is no doubt that Professor Dachling Pang stood high amidst the period. I review here the last two decades from my personal point of view on what has been achieved in the field of spinal dysraphism, focusing on occult tethered cord syndrome, lumbosacral lipomatous malformation, terminal myelocystocele, retained medullary cord, limited dorsal myeloschisis and junctional neural tube defect. There are still many issues to revise, add and extend. Profound knowledge of basic science is critical, as well as refined clinical analysis. I expect that young scholars who follow the footsteps of precedent giants will shed bright light on this topic in the future.

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“…Recently, through the methods mentioned above, our team reported that LDM may be found in the area of secondary neural tube [ 27 ] ( Fig. 10 ).…”

Section: Focal Limited Dorsal Neuro-cutaneous Nondisjunctionmentioning

confidence: 99%

Disorders of Secondary Neurulation : Mainly Focused on Pathoembryogenesis

Yang

Lee

Kim

et al. 2021

J Korean Neurosurg Soc

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Recent advancements in basic research on the process of secondary neurulation and increased clinical experience with caudal spinal anomalies with associated abnormalities in the surrounding and distal structures shed light on further understanding of the pathoembryogenesis of the lesions and led to the new classification of these dysraphic entities. We summarized the changing concepts of lesions developed from the disordered secondary neurulation shown during the last decade. In addition, we suggested our new pathoembryogenetic explanations for a few entities based on the literature and the data from our previous animal research. Disordered secondary neurulation at each phase of development may cause corresponding lesions, such as failed junction with the primary neural tube (junctional neural tube defect and segmental spinal dysgenesis), dysgenesis or duplication of the caudal cell mass associated with disturbed activity of caudal mesenchymal tissue (caudal agenesis and caudal duplication syndrome), failed ingression of the primitive streak to the caudal cell mass (myelomeningocele), focal limited dorsal neuro-cutaneous nondisjunction (limited dorsal myeloschisis and congenital dermal sinus), neuro-mesenchymal adhesion (lumbosacral lipomatous malformation), and regression failure spectrum of the medullary cord (thickened filum and filar cyst, low-lying conus, retained medullary cord, terminal myelocele and terminal myelocystocele). It seems that almost every anomalous entity of the primary neural tube may occur in the area of secondary neurulation. Furthermore, the close association with the activity of caudal mesenchymal tissue in secondary neurulation involves a wider range of surrounding structures than in primary neurulation. Although the majority of the data are from animals, not from humans and many theories are still conjectural, these changing concepts of normal and disordered secondary neurulation will provoke further advancements in our management strategies as well as in the pathoembryogenetic understanding of anomalous lesions in this area.

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Limited Dorsal Myeloschisis: Reconsideration of its Embryological Origin

Cited by 18 publications

References 13 publications

Congenital Dermal Sinus Elements in Each Tethering Stalk of Coexisting Thoracic Limited Dorsal Myeloschisis and Retained Medullary Cord

Congenital Dermal Sinus Elements in Each Tethering Stalk of Coexisting Thoracic Limited Dorsal Myeloschisis and Retained Medullary Cord

Spinal Dysraphism in the Last Two Decades : What I Have Seen during the Era of Dynamic Advancement

Disorders of Secondary Neurulation : Mainly Focused on Pathoembryogenesis

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