{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,26]],"date-time":"2026-08-26T14:44:34Z","timestamp":1787755474778,"version":"build-2784847793"},"reference-count":12,"publisher":"Ovid Technologies (Wolters Kluwer Health)","issue":"3","content-domain":{"domain":["lww.com","ovid.com"],"crossmark-restriction":true},"short-container-title":[],"abstract":"<jats:sec>\n                    <jats:title>Case:<\/jats:title>\n                    <jats:p>Two patients (ages 5 and 14 years) with Larsen syndrome and severe cervical kyphosis (patient 1) and spondyloptosis (patient 2), as well as complete anterior-posterior dissociation of the cervical spine, presented with severe, progressive myelopathy. Both patients underwent preoperative halo-gravity traction to slowly reduce the deformity followed by staged anterior-posterior decompression and fusion. Both patients demonstrated complete return of neurologic function.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusion:<\/jats:title>\n                    <jats:p>Patients with Larsen syndrome should be screened for cervical deformity at the time of diagnosis. Proper screening will facilitate early detection and treatment of significant deformities to prevent neurologic deterioration, which can be catastrophic in these patients.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.2106\/jbjs.cc.21.00236","type":"journal-article","created":{"date-parts":[[2022,1,31]],"date-time":"2022-01-31T23:01:38Z","timestamp":1643670098000},"update-policy":"https:\/\/doi.org\/10.1097\/lww.0000000000001000","source":"Crossref","is-referenced-by-count":3,"title":["Severe Cervical Kyphosis and Spondyloptosis with Myelopathy in Larsen Syndrome"],"prefix":"10.2106","volume":"11","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-6181-3684","authenticated-orcid":false,"given":"Robert K.","family":"Merrill","sequence":"first","affiliation":[{"name":"Department of Orthopaedic Surgery, Einstein Medical Center Philadelphia, Philadelphia, Pennsylvania"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-8475-6822","authenticated-orcid":false,"given":"Terrence G.","family":"Ishmael","sequence":"additional","affiliation":[{"name":"Department of Orthopaedic Surgery, Robert Wood Johnson University Hospital, New Brunswick, New Jersey"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-4206-610X","authenticated-orcid":false,"given":"Amer F.","family":"Samdani","sequence":"additional","affiliation":[{"name":"Shriners Hospitals for Children-Philadelphia, Philadelphia, Pennsylvania"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-0832-3683","authenticated-orcid":false,"given":"Steven W.","family":"Hwang","sequence":"additional","affiliation":[{"name":"Shriners Hospitals for Children-Philadelphia, Philadelphia, Pennsylvania"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-8965-354X","authenticated-orcid":false,"given":"Joshua M.","family":"Pahys","sequence":"additional","affiliation":[{"name":"Shriners Hospitals for Children-Philadelphia, Philadelphia, Pennsylvania"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"276","published-online":{"date-parts":[[2021,7]]},"reference":[{"issue":"4","key":"bib1-20240809","doi-asserted-by":"crossref","first-page":"574","DOI":"10.1016\/S0022-3476(50)80268-8","article-title":"Multiple congenital dislocations associated with characteristic facial abnormality","volume":"37","author":"Larsen","year":"1950","journal-title":"J Pediatr."},{"issue":"7","key":"bib2-20240809","doi-asserted-by":"crossref","first-page":"335","DOI":"10.1016\/j.jgg.2017.04.007","article-title":"The next hotspot in skeletal research?","volume":"44","author":"Xu","year":"2017","journal-title":"J Genet Genomics."},{"issue":"197","key":"bib3-20240809","first-page":"159","article-title":"Spinal deformities in Larsen's syndrome","author":"Bowen","year":"1985 Jul-Aug","journal-title":"Clin Orthop Relat Res."},{"issue":"12","key":"bib4-20240809","doi-asserted-by":"crossref","first-page":"E296","DOI":"10.1097\/00007632-200206150-00023","article-title":"Congenital anteroposterior spinal dissociation in Larsen's Syndrome: report on two operated cases with long-term follow-up","volume":"27","author":"Luk","year":"2002","journal-title":"Spine (Phila Pa 1976)."},{"issue":"4","key":"bib5-20240809","doi-asserted-by":"crossref","first-page":"429","DOI":"10.1097\/01.bpo.0000161091.85350.54","article-title":"Cervical kyphosis associated with anteroposterior dissociation and quadriparesis in Larsen's syndrome","volume":"25","author":"Katz","year":"2005","journal-title":"J Pediatr Orthop."},{"issue":"1","key":"bib6-20240809","doi-asserted-by":"crossref","first-page":"E43","DOI":"10.1097\/BRS.0b013e318278e59d","article-title":"Larsen syndrome with C3-C4 spondyloptosis and atlantoaxial dislocation in an adult","volume":"38","author":"Roopesh Kumar","year":"2013","journal-title":"Spine (Phila Pa 1976)."},{"issue":"4","key":"bib7-20240809","doi-asserted-by":"crossref","first-page":"562","DOI":"10.2106\/00004623-197658040-00024","article-title":"Spinal instability in Larsen's syndrome: report of three cases","volume":"58","author":"Micheli","year":"1976","journal-title":"J Bone Joint Surg Am."},{"issue":"1","key":"bib8-20240809","doi-asserted-by":"crossref","first-page":"199","DOI":"10.1542\/peds.111.1.199","article-title":"Cervical spine involvement in Larsen's syndrome: a case illustration","volume":"111","author":"Banks","year":"2003","journal-title":"Pediatrics."},{"issue":"4","key":"bib9-20240809","doi-asserted-by":"crossref","first-page":"320","DOI":"10.3171\/PED\/2008\/1\/4\/320","article-title":"Management of severe cervical kyphosis in a patient with Larsen syndrome. Case report","volume":"1","author":"Madera","year":"2008","journal-title":"J Neurosurg Pediatr."},{"issue":"2","key":"bib10-20240809","doi-asserted-by":"crossref","first-page":"262","DOI":"10.4103\/0028-3886.96438","article-title":"Management of upper cervical kyphosis in an adolescent with Larsen's syndrome","volume":"60","author":"Mohindra","year":"2012","journal-title":"Neurol India."},{"issue":"4","key":"bib11-20240809","doi-asserted-by":"crossref","first-page":"538","DOI":"10.2106\/00004623-199604000-00007","article-title":"Cervical kyphosis in patients who have Larsen syndrome","volume":"78","author":"Johnston","year":"1996","journal-title":"J Bone Joint Surg Am."},{"issue":"1","key":"bib12-20240809","doi-asserted-by":"crossref","first-page":"E39","DOI":"10.1097\/01.brs.0000250103.88392.8e","article-title":"Surgical treatment of cervical kyphosis in Larsen syndrome: report of 3 cases and review of the literature","volume":"32","author":"Sakaura","year":"2007","journal-title":"Spine (Phila Pa 1976)."}],"container-title":["JBJS Case Connector"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/www.ovid.com\/10.2106\/JBJS.CC.21.00236","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,8,26]],"date-time":"2026-08-26T13:43:15Z","timestamp":1787751795000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.ovid.com\/10.2106\/JBJS.CC.21.00236"}},"subtitle":["A Report of 2 Cases"],"short-title":[],"issued":{"date-parts":[[2021,7]]},"references-count":12,"journal-issue":{"issue":"3","published-online":{"date-parts":[[2021]]}},"URL":"https:\/\/doi.org\/10.2106\/jbjs.cc.21.00236","relation":{},"ISSN":["2160-3251"],"issn-type":[{"value":"2160-3251","type":"electronic"}],"subject":[],"published":{"date-parts":[[2021,7]]}}}