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      Diastematomyelia with hemimyelomeningocele: An exceptional and complex spinal dysraphism

      case-report

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          Abstract

          Variations in split cord malformation (SCM) have been described earlier. However, a true hemimyelomeningocele (HMM) as only congenital malformation is extremely rare and is reported infrequently in published literature. We are reporting the case of a 3-month-old girl child who presented with a swelling on the lower back since birth. Magnetic resonance imaging revealed a type 1 SCM with right hemicord forming a HMM. Precise diagnosis and thorough anatomical detail of dysraphism is essential for optimal, individualized neurosurgical management.

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          Split cord malformation: Part I: A unified theory of embryogenesis for double spinal cord malformations.

          Kerith-Rae Dias, Raina Pang, M. Michael Barmada (1992)
          Much confusion still exists concerning the pathological definitions and clinical significance of double spinal cord malformations. Traditional terms used to describe the two main forms of these rare malformations, diastematomyelia and diplomyelia, add to the confusion by their inconsistent usage, ambiguities, and implications of their dissimilar embryogenesis. Based on the detailed radiographic and surgical findings of 39 cases of double cord malformations and the autopsy data on two other cases, this study endorses a new classification for double cord malformations and proposes a unified theory of embryogenesis for all their variant forms and features. The new classification recommends the term split cord malformation (SCM) for all double spinal cords. A Type I SCM consists of two hemicords, each contained within its own dural tube and separated by a dura-sheathed rigid osseocartilaginous median septum. A Type II SCM consists of two hemicords housed in a single dural tube separated by a nonrigid, fibrous median septum. These two essential features necessary for typing, the state of the dural tube and the nature of the median septum, do not ever overlap between the two main forms and can always be demonstrated by imaging studies so that accurate preoperative typing is always possible. All other associated structures in SCM such as paramedian nerve roots, myelomeningoceles manqué, and centromedian vascular structures frequently do overlap between types and are not reliable typing criteria. The unified theory of embryogenesis proposes that all variant types of SCMs have a common embryogenetic mechanism. Basic to this mechanism is the formation of adhesions between ecto- and endoderm, leading to an accessory neurenteric canal around which condenses an endomesenchymal tract that bisects the developing notochord and causes formation of two hemineural plates. The altered state of the emerging split neural tube and the subsequent ontogenetic fates of the constituent components of the endomesenchymal tract ultimately determine the configuration and orientation of the hemicords, the nature of the median septum, the coexistence of various vascular, lipomatous, neural, and fibrous oddities within the median cleft, the high association with open myelodysplastic and cutaneous lesions, and the seemingly unlikely relationship with fore and midgut anomalies. The multiple facets of this theory are presented in increasing complexity against the background of known embryological facts and theories; the validity of each facet is tested by comparing structures and phenomena predicted by the facet with actual radiographic, surgical, and histopathological findings of these 41 cases of SCM.
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            Split cord malformation associated with myelomeningocele.

            Saeed Ansari, Farideh Nejat, Shahrooz Yazdani … (2007)
            The coexistence of split cord malformations (SCMs) and myelomeningoceles (MMCs) can be misdiagnosed or ignored and can cause spinal cord tethering. The authors investigated cases involving the coexistence of SCMs and MMCs in patients from the Children's Hospital Medical Center in Tehran.
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              Mixed split cord malformation: are we missing something?

              Pravin Salunke, Priyamvadha Kovai, Vinod Malik … (2011)
              The new classification for split cord malformation, based on the unified theory of embryogenesis includes two types Type I and II defined on the basis of the nature of the medium septum and the state of the dural tube. According to this, these are the only two essential features needed for typing and there is never an overlap between the two main forms. We have described two cases that defy this theory.
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                Author and article information

                Journal
                Journal ID (nlm-ta): J Pediatr Neurosci
                Journal ID (iso-abbrev): J Pediatr Neurosci
                Journal ID (publisher-id): JPN
                Title: Journal of Pediatric Neurosciences
                Publisher: Medknow Publications & Media Pvt Ltd (India )
                ISSN (Print): 1817-1745
                ISSN (Electronic): 1998-3948
                Publication date (Print): Jul-Sep 2015
                Volume: 10
                Issue: 3
                Pages: 237-239
                Affiliations
                [1]Department of Radiodiagnosis and Imaging, King George's Medical University, Lucknow, Uttar Pradesh, India
                [1 ]Department of Neurosurgery, Dr. Ram Manohar Lohia Institute of Medical Sciences, Lucknow, Uttar Pradesh, India
                Author notes
                Address for correspondence: Dr. Deepak Kumar Singh, Department of Neurosurgery, Dr. Ram Manohar Lohia Institute of Medical Sciences, Lucknow - 226 010, Uttar Pradesh, India. E-mail: gkp.deepak@ 123456gmail.com
                Article
                Publisher ID: JPN-10-237
                DOI: 10.4103/1817-1745.165665
                PMC ID: 4611892
                PubMed ID: 26557164
                SO-VID: 89cd070e-332c-4b8d-ace9-ccf6981d217a
                Copyright © Copyright: © Journal of Pediatric Neurosciences
                License:

                This is an open access article distributed under the terms of the Creative Commons Attribution NonCommercial ShareAlike 3.0 License, which allows others to remix, tweak, and build upon the work non commercially, as long as the author is credited and the new creations are licensed under the identical terms.

                History
                Categories
                Subject: Case Report

                ScienceOpen disciplines: Neurosciences
                Keywords: diastematomyelia,hemimyelomeningocele,spinal dysraphism,split cord malformation type 1
                Data availability:
                ScienceOpen disciplines: Neurosciences
                Keywords: diastematomyelia, hemimyelomeningocele, spinal dysraphism, split cord malformation type 1

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