{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,30]],"date-time":"2026-09-30T00:55:30Z","timestamp":1790729730290,"version":"4.1.0"},"reference-count":13,"publisher":"Journal of Neurosurgery Publishing Group (JNSPG)","issue":"13","license":[{"start":{"date-parts":[[2026,9,28]],"date-time":"2026-09-28T00:00:00Z","timestamp":1790553600000},"content-version":"unspecified","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["J Neurosurg Case Lessons"],"published-print":{"date-parts":[[2026,9,28]]},"abstract":"<jats:sec>\n                    <jats:title>BACKGROUND<\/jats:title>\n                    <jats:p>Split cord malformation (SCM) and myelocystocele (MC) are rare forms of spinal dysraphism arising from aberrant embryogenesis at distinct developmental stages. Although both lesions share embryological origins, their concomitant occurrence is exceedingly uncommon.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>OBSERVATIONS<\/jats:title>\n                    <jats:p>The authors report the first case of a type II SCM coexisting with an MC in an infant. The patient underwent microsurgical detethering and repair of the MC. Postnatal postoperative MRI revealed a type II SCM of the thoracolumbar spine. At 17 months, the patient demonstrated neurological symptoms consistent with spinal cord tethering and was brought back to the operating room for a complex detethering procedure with the assistance of the plastic surgery team and intraoperative neurophysiological monitoring. At the 2-year follow-up, the patient demonstrated stable spinal anatomy and improved functional outcomes. Genetic testing identified a variant in the GLI2 gene, suggesting a potential role for midline developmental gene perturbations in complex dysraphic syndromes.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>LESSONS<\/jats:title>\n                    <jats:p>This case expands the spectrum of closed spinal dysraphism, highlighting that errors in gastrulation and secondary neurulation can co-occur. Comprehensive preoperative imaging, high-resolution postnatal MRI, multidisciplinary coordination, and consideration of genetic evaluation are essential when managing complex spinal dysraphism. Awareness of such rare lesion combinations informs both surgical planning and developmental understanding of spinal cord malformations.<\/jats:p>\n                    <jats:p>\n                      <jats:ext-link xmlns:xlink=\"http:\/\/www.w3.org\/1999\/xlink\" ext-link-type=\"uri\" xlink:href=\"https:\/\/thejns.org\/doi\/10.3171\/CASE26552\">https:\/\/thejns.org\/doi\/10.3171\/CASE26552<\/jats:ext-link>\n                    <\/jats:p>\n                  <\/jats:sec>","DOI":"10.3171\/case26552","type":"journal-article","created":{"date-parts":[[2026,9,23]],"date-time":"2026-09-23T00:03:17Z","timestamp":1790121797000},"source":"Crossref","is-referenced-by-count":0,"title":["A rare case of split cord malformation type II with concomitant myelocystocele: illustrative case"],"prefix":"10.3171","volume":"12","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-0058-9500","authenticated-orcid":true,"given":"Bryce S.","family":"Owen","sequence":"first","affiliation":[{"name":"Department of Neurosurgery, University of Cincinnati College of Medicine, Cincinnati"},{"name":"Division of Pediatric Neurosurgery, Cincinnati Children\u2019s Hospital Medical Center, Cincinnati"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Natalie A.","family":"Ivey","sequence":"additional","affiliation":[{"name":"Department of Neurosurgery, University of Cincinnati College of Medicine, Cincinnati"},{"name":"Division of Pediatric Neurosurgery, Cincinnati Children\u2019s Hospital Medical Center, Cincinnati"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Karin S.","family":"Bierbrauer","sequence":"additional","affiliation":[{"name":"Department of Neurosurgery, University of Cincinnati College of Medicine, Cincinnati"},{"name":"Division of Pediatric Neurosurgery, Cincinnati Children\u2019s Hospital Medical Center, Cincinnati"},{"name":"Fetal Care Center, Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-8251-7481","authenticated-orcid":true,"given":"Usha D.","family":"Nagaraj","sequence":"additional","affiliation":[{"name":"Department of Radiology, University of Cincinnati College of Medicine, Cincinnati"},{"name":"Department of Radiology and Medical Imaging, Cincinnati Children\u2019s Hospital Medical Center, Cincinnati"},{"name":"Fetal Care Center, Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-9026-0660","authenticated-orcid":true,"given":"Smruti K.","family":"Patel","sequence":"additional","affiliation":[{"name":"Department of Neurosurgery, University of Cincinnati College of Medicine, Cincinnati"},{"name":"Division of Pediatric Neurosurgery, Cincinnati Children\u2019s Hospital Medical Center, Cincinnati"},{"name":"Fetal Care Center, Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"1754","reference":[{"issue":"3","key":"bib1","series-title":"Neurosurgery","first-page":"451","article-title":"Split cord malformation: part I: a unified theory of embryogenesis for double spinal cord malformations","volume":"31","author":"Pang","year":"1992","unstructured":"Pang DDias MSAhab-Barmada M.Split cord malformation: part I: a unified theory of embryogenesis for double spinal cord malformations. Neurosurgery. 1992;31(3):451-480. doi:10.1227\/00006123-199209000-00010"},{"key":"bib2","series-title":"World Neurosurg","first-page":"e601","article-title":"Split cord malformation: presentation, management, and surgical outcome","volume":"136","author":"Alnefaie","year":"2020","unstructured":"Alnefaie NAlharbi AAlamer OBSplit cord malformation: presentation, management, and surgical outcome. World Neurosurg. 2020;136:e601-e607. doi:10.1016\/j.wneu.2020.01.092"},{"issue":"6","key":"bib3","series-title":"Neurosurgery","first-page":"1383","article-title":"Terminal myelocystocele: surgical observations and theory of embryogenesis","volume":"70","author":"Pang","year":"2012","unstructured":"Pang DZovickian JLee JYMoes GSWang KC.Terminal myelocystocele: surgical observations and theory of embryogenesis. Neurosurgery. 2012;70(6):1383-1404. doi:10.1227\/NEU.0b013e31824c02c0"},{"issue":"3","key":"bib4","series-title":"Neurosurgery","first-page":"509","article-title":"Spectrum of nonterminal myelocystoceles","volume":"58","author":"Rossi","year":"2006","unstructured":"Rossi APiatelli GGandolfo CSpectrum of nonterminal myelocystoceles. Neurosurgery. 2006;58(3):509-509. doi:10.1227\/01.NEU.0000197122.92954.82"},{"issue":"1","key":"bib5","series-title":"J Pediatr Neurosci","first-page":"58","article-title":"An unusual case of 4 level spinal dysraphism: multiple composite type 1 and type 2 split cord malformation, dorsal myelocystocele and hydrocephalous","volume":"6","author":"Khandelwal","year":"2011","unstructured":"Khandelwal ATandon VMahapatra AK.An unusual case of 4 level spinal dysraphism: multiple composite type 1 and type 2 split cord malformation, dorsal myelocystocele and hydrocephalous. J Pediatr Neurosci. 2011;6(1):58-61. doi:10.4103\/1817-1745.84411"},{"issue":"1","key":"bib6","series-title":"J Neurosci Rural Pract","first-page":"87","article-title":"A rare case of thoracic myelocystocele associated with type 1 split cord malformation with low lying tethered cord, dorsal syrinx and sacral agenesis: pentad finding","volume":"6","author":"Mankotia","year":"2015","unstructured":"Mankotia DSSatyarthee GDSharma BS.A rare case of thoracic myelocystocele associated with type 1 split cord malformation with low lying tethered cord, dorsal syrinx and sacral agenesis: pentad finding. J Neurosci Rural Pract. 2015;6(1):87-90. doi:10.4103\/0976-3147.143209"},{"issue":"6","key":"bib7","series-title":"Brain Dev","first-page":"469","article-title":"Unusual split of the spinal cord in a caudal regression syndrome with myelocystocele","volume":"4","author":"Schmitt","year":"1982","unstructured":"Schmitt HPKawakami M.Unusual split of the spinal cord in a caudal regression syndrome with myelocystocele. Brain Dev. 1982;4(6):469-474. doi:10.1016\/S0387-7604(82)80075-2"},{"issue":"9","key":"bib8","series-title":"Childs Nerv Syst","first-page":"1527","article-title":"Complex forms of spinal dysraphism","volume":"29","author":"Patnaik","year":"2013","unstructured":"Patnaik AMahapatra AK.Complex forms of spinal dysraphism. Childs Nerv Syst. 2013;29(9):1527-1532. doi:10.1007\/s00381-013-2161-1"},{"issue":"5-6","key":"bib9","series-title":"Childs Nerv Syst","first-page":"376","article-title":"Multiple coexistent dysraphic pathologies","volume":"19","author":"Solanki","year":"2003","unstructured":"Solanki GAEvans JCopp AThompson DNP.Multiple coexistent dysraphic pathologies. Childs Nerv Syst. 2003;19(5-6):376-379. doi:10.1007\/s00381-003-0728-y"},{"issue":"1","key":"bib10","series-title":"Clin Imaging","first-page":"41","article-title":"Diastematomyelia with terminal lipomyelocystocele arising from one hemicord: case report","volume":"27","author":"Parmar","year":"2003","unstructured":"Parmar HPatkar DShah JMaheshwari M.Diastematomyelia with terminal lipomyelocystocele arising from one hemicord: case report. Clin Imaging. 2003;27(1):(41-43). doi:10.1016\/s0899-7071(02)00522-3"},{"issue":"3","key":"bib11","series-title":"J Clin Res Pediatr Endocrinol","first-page":"319","article-title":"Ectopic posterior pituitary, polydactyly, midfacial hypoplasia and multiple pituitary hormone deficiency due to a novel heterozygous IVS11-2A>C(c.1957-2A>C) mutation in the GLI2 gene","volume":"12","author":"Demiral","year":"2020","unstructured":"Demiral MDemirbilek HUnal EDurmaz CDCeylaner S\u00d6zbek MN.Ectopic posterior pituitary, polydactyly, midfacial hypoplasia and multiple pituitary hormone deficiency due to a novel heterozygous IVS11-2A>C(c.1957-2A>C) mutation in the GLI2 gene. J Clin Res Pediatr Endocrinol. 2020;12(3):319-328. doi:10.4274\/jcrpe.galenos.2019.2019.0142"},{"issue":"11","key":"bib12","series-title":"J Clin Endocrinol Metab","first-page":"E384","article-title":"Novel heterozygous nonsense GLI2 mutations in patients with hypopituitarism and ectopic posterior pituitary lobe without holoprosencephaly","volume":"95","author":"Fran\u00e7a","year":"2010","unstructured":"Fran\u00e7a MMJorge AALCarvalho LRSNovel heterozygous nonsense GLI2 mutations in patients with hypopituitarism and ectopic posterior pituitary lobe without holoprosencephaly. J Clin Endocrinol Metab. 2010;95(11):E384-E391. doi:10.1210\/jc.2010-1050"},{"issue":"2","key":"bib13","series-title":"J Pediatr Surg","first-page":"381","article-title":"Murine models of VACTERL syndrome: role of sonic hedgehog signaling pathway","volume":"36","author":"Kim","year":"2001","unstructured":"Kim PCWMo RHui Cc C.Murine models of VACTERL syndrome: role of sonic hedgehog signaling pathway. J Pediatr Surg. 2001;36(2):381-384. doi:10.1053\/jpsu.2001.20722"}],"container-title":["Journal of Neurosurgery: Case Lessons"],"original-title":[],"link":[{"URL":"https:\/\/thejns.org\/view\/journals\/j-neurosurg-case-lessons\/12\/13\/article-CASE26552.xml","content-type":"text\/html","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/thejns.org\/downloadpdf\/journals\/j-neurosurg-case-lessons\/12\/13\/article-CASE26552.xml","content-type":"text\/html","content-version":"vor","intended-application":"syndication"},{"URL":"https:\/\/thejns.org\/downloadpdf\/journals\/j-neurosurg-case-lessons\/12\/13\/article-CASE26552.xml","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,9,30]],"date-time":"2026-09-30T00:01:19Z","timestamp":1790726479000},"score":1,"resource":{"primary":{"URL":"https:\/\/thejns.org\/view\/journals\/j-neurosurg-case-lessons\/12\/13\/article-CASE26552.xml"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2026,9,28]]},"references-count":13,"journal-issue":{"issue":"13"},"URL":"https:\/\/doi.org\/10.3171\/case26552","relation":{},"ISSN":["2694-1902"],"issn-type":[{"value":"2694-1902","type":"electronic"}],"subject":[],"published":{"date-parts":[[2026,9,28]]}}}