{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,3,29]],"date-time":"2026-03-29T22:26:06Z","timestamp":1774823166615,"version":"3.50.1"},"reference-count":30,"publisher":"Public Library of Science (PLoS)","issue":"12","license":[{"start":{"date-parts":[[2021,12,2]],"date-time":"2021-12-02T00:00:00Z","timestamp":1638403200000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"DOI":"10.13039\/501100009780","name":"Region Sk\u00e5ne","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100009780","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100009389","name":"Stiftelsen Promobilia","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100009389","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100006075","name":"Greta och Johan Kocks stiftelser","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100006075","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["www.plosone.org"],"crossmark-restriction":false},"short-container-title":["PLoS ONE"],"abstract":"<jats:sec id=\"sec001\">\n<jats:title>Background<\/jats:title>\n<jats:p>This study aimed to estimate the birth prevalence of children born with isolated or non-isolated clubfoot in Sweden using a national clubfoot register. Secondarily we aimed to describe the clubfoot population with respect to sex, laterality, severity of deformity, comorbidity and geographic location.<\/jats:p>\n<\/jats:sec>\n<jats:sec id=\"sec002\">\n<jats:title>Methods<\/jats:title>\n<jats:p>A national register, the Swedish Pediatric Orthopedic Quality register, was used to extract data on newborn children with clubfoot. To calculate the birth prevalence of children with isolated or non-isolated clubfoot between 1<jats:sup>st<\/jats:sup> of January 2016 and 31<jats:sup>st<\/jats:sup> of December 2019, we used official reports of the total number of Swedish live births from the Swedish Board of Statistics. The Pirani score and predefined signs of atypical clubfoot were used to classify clubfoot severity at birth.<\/jats:p>\n<\/jats:sec>\n<jats:sec id=\"sec003\">\n<jats:title>Results<\/jats:title>\n<jats:p>In total 612 children with clubfoot were identified. Of these, 564 were children with isolated clubfoot, generating a birth prevalence of 1.24\/1000 live births (95% confidence interval 1.15\u20131.35). About 8% were children with non-isolated clubfoot, increasing the birth prevalence to 1.35\/1000 live births (95% confidence interval 1.25\u20131.46). Of the children with isolated clubfoot, 74% were boys and 47% had bilateral involvement. The children with non-isolated clubfoot had more severe foot deformities at birth and a greater proportion of clubfeet with atypical signs compared with children with isolated clubfoot.<\/jats:p>\n<\/jats:sec>\n<jats:sec id=\"sec004\">\n<jats:title>Conclusion<\/jats:title>\n<jats:p>We have established the birth prevalence of children born with isolated or non-isolated clubfoot in Sweden based on data from a national register. Moreover, we have estimated the number of children born with atypical clubfeet in instances of both isolated and non-isolated clubfoot. These numbers may serve as a baseline for expected birth prevalence when planning clubfoot treatment and when evaluating time trends of children born with clubfoot.<\/jats:p>\n<\/jats:sec>","DOI":"10.1371\/journal.pone.0260336","type":"journal-article","created":{"date-parts":[[2021,12,2]],"date-time":"2021-12-02T19:15:24Z","timestamp":1638472524000},"page":"e0260336","update-policy":"https:\/\/doi.org\/10.1371\/journal.pone.corrections_policy","source":"Crossref","is-referenced-by-count":23,"title":["Epidemiology of clubfoot in Sweden from 2016 to 2019: A national register study"],"prefix":"10.1371","volume":"16","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-6240-4674","authenticated-orcid":true,"given":"Anna-Clara","family":"Esbj\u00f6rnsson","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Arne","family":"Johansson","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Hanneke","family":"Andriesse","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Henrik","family":"Wallander","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"340","published-online":{"date-parts":[[2021,12,2]]},"reference":[{"issue":"4","key":"pone.0260336.ref001","doi-asserted-by":"crossref","first-page":"595","DOI":"10.1002\/ajmg.a.61067","article-title":"Congenital clubfoot in Europe: A population-based study","volume":"179","author":"H Wang","year":"2019","journal-title":"Am J Med Genet A"},{"issue":"15","key":"pone.0260336.ref002","doi-asserted-by":"crossref","first-page":"1249","DOI":"10.2106\/JBJS.M.01257","article-title":"Soft-tissue abnormalities associated with treatment-resistant and treatment-responsive clubfoot: Findings of MRI analysis","volume":"96","author":"DK Moon","year":"2014","journal-title":"J Bone Joint Surg Am"},{"issue":"1","key":"pone.0260336.ref003","doi-asserted-by":"crossref","first-page":"7","DOI":"10.1097\/BPB.0b013e328349927c","article-title":"Genetics of clubfoot","volume":"21","author":"MB Dobbs","year":"2012","journal-title":"J Pediatr Orthop B"},{"key":"pone.0260336.ref004","first-page":"120","article-title":"Descriptive epidemiology of clubfoot in Vietnam: A clinic-based study.","volume":"32","author":"MC Nguyen","year":"2012","journal-title":"Iowa Orthop J"},{"key":"pone.0260336.ref005","volume-title":"Congenital clubfoot. 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Severin","year":"1956","journal-title":"Nord Med"},{"key":"pone.0260336.ref012","first-page":"1","article-title":"Review of the literature and an analysis of a series of 135 treated clubfeet.","volume":"209","author":"Somppi E. Clubfoot","year":"1984","journal-title":"Acta Orthop Scand Suppl"},{"issue":"6","key":"pone.0260336.ref013","doi-asserted-by":"crossref","first-page":"847","DOI":"10.1080\/17453670610013123","article-title":"Incidence of congenital clubfoot in Sweden.","volume":"77","author":"H Wallander","year":"2006","journal-title":"Acta Orthop"},{"issue":"6","key":"pone.0260336.ref014","doi-asserted-by":"crossref","first-page":"839","DOI":"10.1080\/17453670610013114","article-title":"Increasing incidence of club foot with higher population density: Incidence and geographical variation in Denmark over a 16-year period\u2014An epidemiological study of 936,525 births.","volume":"77","author":"MR Krogsgaard","year":"2006","journal-title":"Acta Orthop"},{"key":"pone.0260336.ref015","unstructured":"spoq.registercentrum.se [Internet]. Svenskt Pediatriskt Ortopediskt Qualitetsregister; c2021 [cited 2021 June 7]. Available from: http:\/\/www.spoq.registercentrum.se\/."},{"issue":"1","key":"pone.0260336.ref016","doi-asserted-by":"crossref","first-page":"1","DOI":"10.2519\/jospt.1994.20.1.1","article-title":"The treatment of congenital clubfoot","volume":"20","author":"IV Ponseti","year":"1994","journal-title":"J Orthop Sports Phys Ther"},{"issue":"4","key":"pone.0260336.ref017","doi-asserted-by":"crossref","first-page":"366","DOI":"10.1097\/BPB.0b013e3283514183","article-title":"Interobserver reliability in Pirani clubfoot severity scoring between a paediatric orthopaedic surgeon and a physiotherapy assistant","volume":"21","author":"S Shaheen","year":"2012","journal-title":"J Pediatr Orthop B"},{"issue":"1","key":"pone.0260336.ref018","doi-asserted-by":"crossref","first-page":"81","DOI":"10.4103\/0019-5413.197551","article-title":"Interobserver variability in Pirani clubfoot severity scoring system between the orthopedic surgeons.","volume":"51","author":"S Jain","year":"2017","journal-title":"Indian J Orthop"},{"issue":"5","key":"pone.0260336.ref019","doi-asserted-by":"crossref","first-page":"478","DOI":"10.1302\/1863-2548.13.190010","article-title":"Interobserver reliability of Dimeglio and Pirani score and their subcomponents in the evaluation of idiopathic clubfoot in a clinical setting: a need for improved scoring systems.","volume":"13","author":"C Bettuzzi","year":"2019","journal-title":"J Child Orthop"},{"key":"pone.0260336.ref020","unstructured":"Epitools (Summarise categorical or continuous data). Calculate confidence limits for a sample proportion [software]; c2021 [cited 2021 June 7]. Available from: https:\/\/epitools.ausvet.com.au\/ciproportion?page=CIProportion"},{"key":"pone.0260336.ref021","unstructured":"scb.se\/en\/ [Internet]. Statistics Sweden; c2021 [cited 2021 June 7]. Available from: http:\/\/www.scb.se\/en\/."},{"issue":"4","key":"pone.0260336.ref022","doi-asserted-by":"crossref","first-page":"e0194986","DOI":"10.1371\/journal.pone.0194986","article-title":"Trends in congenital anomalies in Europe from 1980 to 2012.","volume":"13","author":"JK Morris","year":"2018","journal-title":"PLoS One"},{"issue":"7","key":"pone.0260336.ref023","doi-asserted-by":"crossref","first-page":"1695","DOI":"10.1002\/ajmg.a.33481","article-title":"Epidemiology of congenital idiopathic talipes equinovarus in Iowa, 1997\u20132005","volume":"152A","author":"V Kancherla","year":"2010","journal-title":"Am J Med Genet A"},{"issue":"18","key":"pone.0260336.ref024","doi-asserted-by":"crossref","first-page":"1420","DOI":"10.1002\/bdr2.1589","article-title":"National population-based estimates for major birth defects, 2010\u20132014.","volume":"111","author":"CT Mai","year":"2019","journal-title":"Birth Defects Res"},{"issue":"4","key":"pone.0260336.ref025","doi-asserted-by":"crossref","first-page":"e17895","DOI":"10.1371\/journal.pone.0017895","article-title":"Is there evidence for aetiologically distinct subgroups of idiopathic congenital talipes equinovarus? 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