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Herein, we report a case of Down syndrome complicated by congenital atlanto-occipital dislocation. The patient presented with severe cervical myelopathy at 13\u00a0years of age after a 10-year follow-up. Radiography and computed tomography revealed os odontoideum protruding into the foramen magnum and congenital anterior atlanto-occipital dislocation. Additionally, a bifurcated internal occipital crest with a thinned central portion of the occipital bone was noted. Magnetic resonance imaging revealed kyphotic alignment of the spinal cord with severe compression at the foramen magnum level. As the neurological impairment was partially improved by halo vest immobilization, we performed in situ O-C2 fusion with an iliac autograft and decompression of the foramen magnum and posterior arch of C1. An improvement was observed immediately after surgery. Two years after surgery, radiography and computed tomography showed solid O-C2 segment fusion. The accumulation of similar cases is essential for determining the prognosis or optimal treatment for this rare congenital condition.<\/jats:p>","DOI":"10.1007\/s00256-023-04297-5","type":"journal-article","created":{"date-parts":[[2023,2,11]],"date-time":"2023-02-11T14:16:28Z","timestamp":1676124988000},"page":"1785-1789","update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":2,"title":["Congenital atlanto-occipital dislocation in a patient with Down syndrome: a case report"],"prefix":"10.1007","volume":"52","author":[{"given":"Ryoko","family":"Onodera","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Ryuji","family":"Sakamoto","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-2329-123X","authenticated-orcid":false,"given":"Yuki","family":"Taniguchi","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Shima","family":"Hirai","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Yoshitaka","family":"Matsubayashi","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"So","family":"Kato","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Yasushi","family":"Oshima","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Sakae","family":"Tanaka","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"297","published-online":{"date-parts":[[2023,2,11]]},"reference":[{"issue":"1","key":"4297_CR1","doi-asserted-by":"publisher","first-page":"e7","DOI":"10.1097\/BPB.0000000000000865","volume":"31","author":"Y Fujimoto","year":"2022","unstructured":"Fujimoto Y, Miyoshi K, Oshima Y, et al. The relationship between atlas hypoplasia and os odontoideum in children with Down syndrome: a preliminary case report. J Pediatr Orthop B. 2022;31(1):e7\u201310.","journal-title":"J Pediatr Orthop B"},{"issue":"10","key":"4297_CR2","doi-asserted-by":"publisher","first-page":"1165","DOI":"10.1007\/s00381-008-0603-y","volume":"24","author":"AH Menezes","year":"2008","unstructured":"Menezes AH. Specific entities affecting the craniocervical region: Down\u2019s syndrome. Childs Nerv Syst. 2008;24(10):1165\u20138.","journal-title":"Childs Nerv Syst"},{"issue":"1","key":"4297_CR3","doi-asserted-by":"publisher","first-page":"30","DOI":"10.1097\/MOP.0b013e3282f35f19","volume":"20","author":"G Mik","year":"2008","unstructured":"Mik G, Gholve PA, Scher DM, Widmann RF, Green DW. Down syndrome: orthopedic issues. 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