{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,31]],"date-time":"2026-08-31T02:04:00Z","timestamp":1788141840857,"version":"build-2803163510"},"reference-count":34,"publisher":"MDPI AG","issue":"19","license":[{"start":{"date-parts":[[2023,9,27]],"date-time":"2023-09-27T00:00:00Z","timestamp":1695772800000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"name":"Prof. dr. Henkes Stichting","award":["111356"],"award-info":[{"award-number":["111356"]}]},{"name":"Prof. dr. Henkes Stichting","award":["HV\/AB\/B20210035"],"award-info":[{"award-number":["HV\/AB\/B20210035"]}]},{"name":"Prof. dr. Henkes Stichting","award":["112425"],"award-info":[{"award-number":["112425"]}]},{"name":"Prof. dr. Henkes Stichting","award":["72"],"award-info":[{"award-number":["72"]}]},{"name":"Rotterdamse Stichting Blindenbelangen","award":["111356"],"award-info":[{"award-number":["111356"]}]},{"name":"Rotterdamse Stichting Blindenbelangen","award":["HV\/AB\/B20210035"],"award-info":[{"award-number":["HV\/AB\/B20210035"]}]},{"name":"Rotterdamse Stichting Blindenbelangen","award":["112425"],"award-info":[{"award-number":["112425"]}]},{"name":"Rotterdamse Stichting Blindenbelangen","award":["72"],"award-info":[{"award-number":["72"]}]},{"name":"Stichting Lijf en Leven","award":["111356"],"award-info":[{"award-number":["111356"]}]},{"name":"Stichting Lijf en Leven","award":["HV\/AB\/B20210035"],"award-info":[{"award-number":["HV\/AB\/B20210035"]}]},{"name":"Stichting Lijf en Leven","award":["112425"],"award-info":[{"award-number":["112425"]}]},{"name":"Stichting Lijf en Leven","award":["72"],"award-info":[{"award-number":["72"]}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["JCM"],"abstract":"<jats:p>(1) Background: Non-syndromic unicoronal craniosynostosis (UCS) is associated with a high prevalence of ocular anomalies. Currently, the etiology of this association remains obscure, however, it is presumed to be primarily attributed to their orbital malformations and\/or secondary to craniofacial surgery. We assessed pre-operative ophthalmological examinations of non-syndromic UCS patients and compared them with their postoperative outcomes and long-term follow-up. (2) Methods: A retrospective case series was conducted on medical records of patients with non-syndromic UCS at Sophia Children\u2019s Hospital, Rotterdam. Ophthalmologic examinations were collected at different time periods: T1 (first visit), T2 (&lt;1 year after cranioplasty), and T3 (long-term follow-up at last visit). The McNemar\u2019s test was used for statistical analysis. (3) Results: A total of 101 patients were included, for whom examinations were available at T1 and T3. Patients had a mean age of 2.8 years (\u00b12.7) and 9.5 (\u00b14.9) at T1 and T3, respectively. At T1, 52 patients (51.5%) were diagnosed with strabismus, and 61 patients (60.4%) at T3. Vertical strabismus increased significantly from 23 patients (22.8%) at T1 to 36 patients (35.6%) at T3 (p = 0.011). Followed by astigmatism, which increased significantly from 38 (37.6%) at T1 to 59 (58.4%) patients at T3 (p = 0.001). T1 was available in 20 patients prior to fronto-orbital advancement (FOA), therefore, a sub-analysis was conducted on these patients, which was followed shortly after FOA at T2. Prior to FOA, strabismus was present in 11 patients (55.0%) and in 12 patients (60.0%) at T2. After FOA, strabismus worsened in two patients. (4) Conclusions: This study showed the high prevalence of ocular anomalies in patients with non-syndromic UCS before and after cranioplasty and at long-term follow-up. The findings of this study show that ophthalmic and orthoptic examinations are an important part of the optimal treatment of patients with non-syndromic UCS.<\/jats:p>","DOI":"10.3390\/jcm12196224","type":"journal-article","created":{"date-parts":[[2023,9,27]],"date-time":"2023-09-27T03:49:14Z","timestamp":1695786554000},"page":"6224","update-policy":"https:\/\/doi.org\/10.3390\/mdpi_crossmark_policy","source":"Crossref","is-referenced-by-count":5,"title":["Pre-Operative Ocular Findings and Long-Term Follow-Up in a Large Cohort of Non-Syndromic Unicoronal Craniosynostosis"],"prefix":"10.3390","volume":"12","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-3539-2716","authenticated-orcid":false,"given":"Parinaz","family":"Rostamzad","sequence":"first","affiliation":[{"name":"Department of Plastic and Reconstructive Surgery, Erasmus Medical Center, 3000 CA Rotterdam, The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yasmin S.","family":"Esser","sequence":"additional","affiliation":[{"name":"Department of Ophthalmology, Erasmus Medical Center, 3000 CA Rotterdam, The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Emily T. C.","family":"Tan","sequence":"additional","affiliation":[{"name":"Department of Ophthalmology, Erasmus Medical Center, 3000 CA Rotterdam, The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Marjolein H. G.","family":"Dremmen","sequence":"additional","affiliation":[{"name":"Department of Radiology and Nuclear Medicine, Erasmus Medical Center, 3000 CA Rotterdam, The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mieke M.","family":"Pleumeekers","sequence":"additional","affiliation":[{"name":"Department of Plastic and Reconstructive Surgery, Erasmus Medical Center, 3000 CA Rotterdam, The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sjoukje E.","family":"Loudon","sequence":"additional","affiliation":[{"name":"Department of Ophthalmology, Erasmus Medical Center, 3000 CA Rotterdam, The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"1968","published-online":{"date-parts":[[2023,9,27]]},"reference":[{"key":"ref_1","doi-asserted-by":"crossref","first-page":"1459","DOI":"10.1007\/s00381-019-04193-1","article-title":"The growth of the neurocranium: Literature review and implications in cranial repair","volume":"35","author":"Frassanito","year":"2019","journal-title":"Childs Nerv. Syst."},{"key":"ref_2","doi-asserted-by":"crossref","first-page":"789","DOI":"10.1016\/j.jcms.2016.03.010","article-title":"Titanium cranioplasty in children and adolescents","volume":"44","author":"Williams","year":"2016","journal-title":"J. Craniomaxillofac. Surg."},{"key":"ref_3","doi-asserted-by":"crossref","first-page":"341","DOI":"10.1016\/j.coms.2022.02.001","article-title":"Epidemiology, Genetics, and Pathophysiology of Craniosynostosis","volume":"34","author":"Blessing","year":"2022","journal-title":"Oral. Maxillofac. Surg. Clin. N. Am."},{"key":"ref_4","doi-asserted-by":"crossref","first-page":"359","DOI":"10.1016\/j.ajhg.2015.07.006","article-title":"A Genetic-Pathophysiological Framework for Craniosynostosis","volume":"97","author":"Twigg","year":"2015","journal-title":"Am. J. Hum. Genet."},{"key":"ref_5","doi-asserted-by":"crossref","first-page":"413","DOI":"10.1016\/j.wneu.2022.05.093","article-title":"Global Epidemiology of Craniosynostosis: A Systematic Review and Meta-Analysis","volume":"164","author":"Shlobin","year":"2022","journal-title":"World Neurosurg."},{"key":"ref_6","doi-asserted-by":"crossref","first-page":"302","DOI":"10.1038\/gim.2013.134","article-title":"Familial incidence and associated symptoms in a population of individuals with nonsyndromic craniosynostosis","volume":"16","author":"Greenwood","year":"2014","journal-title":"Genet. Med."},{"key":"ref_7","doi-asserted-by":"crossref","first-page":"150","DOI":"10.1016\/j.spen.2007.08.008","article-title":"Genetics of craniosynostosis","volume":"14","author":"Kimonis","year":"2007","journal-title":"Semin. Pediatr. Neurol."},{"key":"ref_8","doi-asserted-by":"crossref","first-page":"1245","DOI":"10.1097\/SCS.0b013e318252d893","article-title":"Changing epidemiology of nonsyndromic craniosynostosis and revisiting the risk factors","volume":"23","author":"Lee","year":"2012","journal-title":"J. Craniofac. Surg."},{"key":"ref_9","doi-asserted-by":"crossref","first-page":"125","DOI":"10.1097\/PRS.0b013e3182729ed7","article-title":"Bilateral orbital dysmorphology in unicoronal synostosis","volume":"131","author":"Beckett","year":"2013","journal-title":"Plast. Reconstr. Surg."},{"key":"ref_10","doi-asserted-by":"crossref","first-page":"1199","DOI":"10.1097\/SCS.0000000000004509","article-title":"Surgical Treatment of Nonsyndromic Unicoronal Craniosynostosis","volume":"29","author":"Alford","year":"2018","journal-title":"J. Craniofac Surg."},{"key":"ref_11","doi-asserted-by":"crossref","first-page":"371","DOI":"10.1097\/SCS.0000000000007035","article-title":"Updated Guideline on Treatment and Management of Craniosynostosis","volume":"32","author":"Mathijssen","year":"2021","journal-title":"J. Craniofac Surg."},{"key":"ref_12","doi-asserted-by":"crossref","first-page":"838","DOI":"10.1097\/PRS.0000000000005383","article-title":"Nonsyndromic Unilateral Coronal Synostosis: A Comparison of Fronto-Orbital Advancement and Endoscopic Suturectomy","volume":"143","author":"Isaac","year":"2019","journal-title":"Plast. Reconstr. Surg."},{"key":"ref_13","doi-asserted-by":"crossref","unstructured":"Rostamzad, P., Arslan, Z.F., Mathijssen, I.M.J., Koudstaal, M.J., Pleumeekers, M.M., Versnel, S.L., and Loudon, S.E. (2022). Prevalence of Ocular Anomalies in Craniosynostosis: A Systematic Review and Meta-Analysis. J. Clin. Med., 11.","DOI":"10.3390\/jcm11041060"},{"key":"ref_14","doi-asserted-by":"crossref","first-page":"1629","DOI":"10.1097\/SCS.0000000000003085","article-title":"Ophthalmologic Outcomes Following Fronto-Orbital Advancement for Unicoronal Craniosynostosis","volume":"27","author":"Gencarelli","year":"2016","journal-title":"J. Craniofac. Surg."},{"key":"ref_15","doi-asserted-by":"crossref","first-page":"76.e1","DOI":"10.1016\/j.jaapos.2020.10.013","article-title":"Long-term ophthalmic outcomes in 120 children with unilateral coronal synostosis: A 20-year retrospective analysis","volume":"25","author":"Elhusseiny","year":"2021","journal-title":"J. Am. Assoc. Pediatr. Ophthalmol. Strabismus"},{"key":"ref_16","doi-asserted-by":"crossref","unstructured":"Gaillard, L., Goverde, A., Weerts, M.J.A., de Klein, A., Mathijssen, I.M.J., and Van Dooren, M.F. (2023). Genetic diagnostic yield in an 11-year cohort of craniosynostosis patients. Eur. J. Med. Genet., 104843.","DOI":"10.1016\/j.ejmg.2023.104843"},{"key":"ref_17","doi-asserted-by":"crossref","first-page":"54","DOI":"10.1080\/09273972.2019.1604773","article-title":"Global and regional prevalence of strabismus: A comprehensive systematic review and meta-analysis","volume":"27","author":"Hashemi","year":"2019","journal-title":"Strabismus"},{"key":"ref_18","doi-asserted-by":"crossref","first-page":"3274","DOI":"10.1177\/11206721221077548","article-title":"Ophthalmological findings in children with unicoronal craniosynostosis","volume":"32","author":"Touze","year":"2022","journal-title":"Eur. J. Ophthalmol."},{"key":"ref_19","doi-asserted-by":"crossref","first-page":"3","DOI":"10.1016\/j.joco.2017.08.009","article-title":"Global and regional estimates of prevalence of refractive errors: Systematic review and meta-analysis","volume":"30","author":"Hashemi","year":"2018","journal-title":"J. Curr. Ophthalmol."},{"key":"ref_20","doi-asserted-by":"crossref","first-page":"79","DOI":"10.1097\/SCS.0000000000001238","article-title":"Ophthalmic findings in children with nonsyndromic craniosynostosis treated by expansion cranioplasty","volume":"26","author":"Chung","year":"2015","journal-title":"J. Craniofac. Surg."},{"key":"ref_21","unstructured":"Polling, J.R. (2021). Development and Management of Refractive Error in Childhood. [Ph.D. Thesis, Erasmus University Rotterdam]."},{"key":"ref_22","doi-asserted-by":"crossref","first-page":"416","DOI":"10.1016\/S0002-9394(14)74927-X","article-title":"Variations in extraocular muscle number and structure in craniofacial dysostosis","volume":"90","author":"Diamond","year":"1980","journal-title":"Am. J. Ophthalmol."},{"key":"ref_23","doi-asserted-by":"crossref","first-page":"550","DOI":"10.1016\/j.jaapos.2005.07.004","article-title":"Ocular overelevation in adduction in craniosynostosis: Is it the result of excyclorotation of the extraocular muscles?","volume":"9","author":"Tan","year":"2005","journal-title":"J. Am. Assoc. Pediatr. Ophthalmol. Strabismus"},{"key":"ref_24","doi-asserted-by":"crossref","first-page":"307","DOI":"10.1016\/S1091-8531(98)90090-8","article-title":"Absence of multiple extraocular muscles in craniosynostosis","volume":"2","author":"Greenberg","year":"1998","journal-title":"J. Am. Assoc. Pediatr. Ophthalmol. Strabismus"},{"key":"ref_25","doi-asserted-by":"crossref","first-page":"1156","DOI":"10.1136\/bjophthalmol-2019-314989","article-title":"Extraocular muscle positions in anterior plagiocephaly: V-pattern strabismus explained using geometric mophometrics","volume":"104","author":"Touze","year":"2020","journal-title":"Br. J. Ophthalmol."},{"key":"ref_26","doi-asserted-by":"crossref","first-page":"215","DOI":"10.1097\/IIO.0b013e3181695af6","article-title":"The etiology of V pattern strabismus in patients with craniosynostosis","volume":"48","author":"Ron","year":"2008","journal-title":"Int. Ophthalmol. Clin."},{"key":"ref_27","doi-asserted-by":"crossref","first-page":"155","DOI":"10.1016\/j.jaapos.2008.10.011","article-title":"Treatment of unilateral coronal synostosis by endoscopic strip craniectomy or fronto-orbital advancement: Ophthalmologic findings","volume":"13","author":"MacKinnon","year":"2009","journal-title":"J. Am. Assoc. Pediatr. Ophthalmol. Strabismus"},{"key":"ref_28","doi-asserted-by":"crossref","first-page":"1904","DOI":"10.1097\/SCS.0000000000002020","article-title":"Distraction Osteogenesis Versus Conventional Fronto-Orbital Advancement for the Treatment of Unilateral Coronal Synostosis: A Comparison of Perioperative Morbidity and Short-Term Outcomes","volume":"26","author":"Tahiri","year":"2015","journal-title":"J. Craniofac. Surg."},{"key":"ref_29","doi-asserted-by":"crossref","first-page":"881","DOI":"10.1097\/00006534-199604001-00001","article-title":"A prospective study of the relationship between strabismus and head posture in patients with frontal plagiocephaly","volume":"97","author":"Gosain","year":"1996","journal-title":"Plast. Reconstr. Surg."},{"key":"ref_30","doi-asserted-by":"crossref","first-page":"367","DOI":"10.1016\/j.jaapos.2007.02.017","article-title":"Astigmatism in unilateral coronal synostosis: Incidence and laterality","volume":"11","author":"Levy","year":"2007","journal-title":"J. Am. Assoc. Pediatr. Ophthalmol. Strabismus"},{"key":"ref_31","doi-asserted-by":"crossref","first-page":"210","DOI":"10.1007\/BF00301156","article-title":"Refractive error and ocular motility in plagiocephaly","volume":"10","author":"Denis","year":"1994","journal-title":"Childs Nerv. Syst."},{"key":"ref_32","doi-asserted-by":"crossref","first-page":"5","DOI":"10.1111\/j.1444-0938.2007.00112.x","article-title":"A review of astigmatism and its possible genesis","volume":"90","author":"Read","year":"2007","journal-title":"Clin. Exp. Optom."},{"key":"ref_33","doi-asserted-by":"crossref","first-page":"24","DOI":"10.1111\/aos.13898","article-title":"High rate of failed visual-acuity measurements with the Amsterdam Picture Chart in screening at the age of 36 months","volume":"97","author":"Telleman","year":"2019","journal-title":"Acta Ophthalmol."},{"key":"ref_34","doi-asserted-by":"crossref","first-page":"126","DOI":"10.1016\/j.ydbio.2022.07.010","article-title":"TWIST1, a gene associated with Saethre-Chotzen syndrome, regulates extraocular muscle organization in mouse","volume":"490","author":"Whitman","year":"2022","journal-title":"Dev. Biol."}],"container-title":["Journal of Clinical Medicine"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/www.mdpi.com\/2077-0383\/12\/19\/6224\/pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,10,10]],"date-time":"2025-10-10T20:59:16Z","timestamp":1760129956000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.mdpi.com\/2077-0383\/12\/19\/6224"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2023,9,27]]},"references-count":34,"journal-issue":{"issue":"19","published-online":{"date-parts":[[2023,10]]}},"alternative-id":["jcm12196224"],"URL":"https:\/\/doi.org\/10.3390\/jcm12196224","relation":{},"ISSN":["2077-0383"],"issn-type":[{"value":"2077-0383","type":"electronic"}],"subject":[],"published":{"date-parts":[[2023,9,27]]}}}