{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,10]],"date-time":"2026-10-10T03:51:22Z","timestamp":1791604282122,"version":"4.3.4"},"reference-count":19,"publisher":"Wiley","issue":"1","license":[{"start":{"date-parts":[[2026,3,23]],"date-time":"2026-03-23T00:00:00Z","timestamp":1774224000000},"content-version":"vor","delay-in-days":81,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"},{"start":{"date-parts":[[2026,1,1]],"date-time":"2026-01-01T00:00:00Z","timestamp":1767225600000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/doi.wiley.com\/10.1002\/tdm_license_1.1"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Congenital Anomalies"],"published-print":{"date-parts":[[2026,1]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n                  <jats:p>\n                    Currarino syndrome (CS) is a congenital disorder caused by\n                    <jats:italic>MNX1<\/jats:italic>\n                    gene mutation, resulting in abnormal caudal cell mass development. Clinical presentation varies from severe to mild forms. Classic triad of CS includes sacral agenesis, presacral mass, and anorectal malformation (ARM). We report a pair of monozygotic twins, both with the\n                    <jats:italic>MNX1<\/jats:italic>\n                    : c.780C&gt;G mutation, presenting with different clinical phenotypes of Currarino syndrome (CS). Both were suspected of anorectal atresia in utero, and postnatal diagnosis confirmed CS with tethered cord syndrome (TCS). They underwent staged surgeries, with normal growth at 10\u2009months follow\u2010up. Additionally, a retrospective analysis of 95 CS cases diagnosed between April 2010 and April 2024 at Shanghai Xinhua Hospital was performed. Clinical, imaging, and pathological data were collected, and binary logistic regression was used to analyze the correlation between clinical factors and TCS. A literature review of twin CS cases was also conducted. Of the 95 patients, 23.2% had complete CS, and 76.8% had incomplete CS. TCS was present in 55.8% of cases. Logistic regression showed no significant correlation between age, sex, sacral deformity severity, ARM type, presacral mass, and TCS. Two previous twin CS cases were identified, both with differing phenotypes. CS exhibits phenotypic variability, and monozygotic twins with the same\n                    <jats:italic>MNX1<\/jats:italic>\n                    mutation may present with different severities. The\n                    <jats:italic>MNX1<\/jats:italic>\n                    : c.780C&gt;G mutation broadens the genetic spectrum of CS. TCS is common, emphasizing the need for multidisciplinary management. Surgical approaches should be personalized based on presentation.\n                  <\/jats:p>","DOI":"10.1002\/cga.70046","type":"journal-article","created":{"date-parts":[[2026,3,24]],"date-time":"2026-03-24T00:14:26Z","timestamp":1774311266000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["Clinical Diversity in Currarino Syndrome: Insights From Monozygotic Twins and a Single\u2010Center Retrospective Study"],"prefix":"10.1002","volume":"66","author":[{"ORCID":"https:\/\/orcid.org\/0009-0003-1359-0548","authenticated-orcid":false,"given":"Xinyao","family":"Wang","sequence":"first","affiliation":[{"name":"Department of Pediatric Neurosurgery Xinhua Hospital Affiliated to Shanghai Jiao Tong University School of Medicine  Shanghai China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lingzhao","family":"Min","sequence":"additional","affiliation":[{"name":"Department of Pediatric Neurosurgery Xinhua Hospital Affiliated to Shanghai Jiao Tong University School of Medicine  Shanghai China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lei","family":"Bao","sequence":"additional","affiliation":[{"name":"Department of Imaging Xinhua Hospital Affiliated to Shanghai Jiao Tong University School of Medicine  Shanghai China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jia","family":"Wei","sequence":"additional","affiliation":[{"name":"Department of Pediatric Neurosurgery Xinhua Hospital Affiliated to Shanghai Jiao Tong University School of Medicine  Shanghai China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-1825-0895","authenticated-orcid":false,"given":"Xiaoqiang","family":"Wang","sequence":"additional","affiliation":[{"name":"Department of Pediatric Neurosurgery Xinhua Hospital Affiliated to Shanghai Jiao Tong University School of Medicine  Shanghai China"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2026,3,23]]},"reference":[{"key":"e_1_2_12_2_1","doi-asserted-by":"publisher","DOI":"10.2214\/ajr.137.2.395"},{"key":"e_1_2_12_3_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jpedsurg.2004.05.003"},{"key":"e_1_2_12_4_1","doi-asserted-by":"publisher","DOI":"10.1038\/3828"},{"key":"e_1_2_12_5_1","doi-asserted-by":"publisher","DOI":"10.1111\/ped.14622"},{"key":"e_1_2_12_6_1","doi-asserted-by":"publisher","DOI":"10.1136\/bcr-2014-204276"},{"key":"e_1_2_12_7_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jpedsurg.2005.08.002"},{"key":"e_1_2_12_8_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ejmg.2013.09.011"},{"key":"e_1_2_12_9_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.carj.2016.05.007"},{"key":"e_1_2_12_10_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jpedsurg.2016.12.010"},{"key":"e_1_2_12_11_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.clineuro.2022.107470"},{"key":"e_1_2_12_12_1","doi-asserted-by":"publisher","DOI":"10.1007\/s003300050846"},{"issue":"2","key":"e_1_2_12_13_1","doi-asserted-by":"crossref","first-page":"114","DOI":"10.1055\/s-2008-1066487","article-title":"The Currarino Syndrome\u2014Hereditary Transmitted Syndrome of Anorectal, Sacral and Presacral Anomalies. Case Report and Review of the Literature","volume":"6","author":"K\u00f6chling J.","year":"1996","journal-title":"European Journal of Pediatric Surgery: Official Journal of Austrian Association of Pediatric Surgery"},{"key":"e_1_2_12_14_1","doi-asserted-by":"publisher","DOI":"10.1186\/s13052-018-0500-2"},{"key":"e_1_2_12_15_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.juro.2012.01.128"},{"key":"e_1_2_12_16_1","doi-asserted-by":"publisher","DOI":"10.3171\/2018.5.PEDS17582"},{"key":"e_1_2_12_17_1","doi-asserted-by":"publisher","DOI":"10.1007\/s003830050615"},{"key":"e_1_2_12_18_1","doi-asserted-by":"publisher","DOI":"10.1002\/humu.20718"},{"key":"e_1_2_12_19_1","doi-asserted-by":"publisher","DOI":"10.1007\/PL00007296"},{"issue":"2","key":"e_1_2_12_20_1","first-page":"138","article-title":"\u201cLike Mother, Like Son\u201d: Hereditary Anterior Sacral Meningocele. Case Report and Review of the Literature","volume":"104","author":"Gardner P. A.","year":"2006","journal-title":"Journal of Neurosurgery"}],"container-title":["Congenital Anomalies"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/cga.70046","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/full-xml\/10.1002\/cga.70046","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/cga.70046","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,10,10]],"date-time":"2026-10-10T02:58:18Z","timestamp":1791601098000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1002\/cga.70046"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2026,1]]},"references-count":19,"journal-issue":{"issue":"1","published-print":{"date-parts":[[2026,1]]}},"alternative-id":["10.1002\/cga.70046"],"URL":"https:\/\/doi.org\/10.1002\/cga.70046","archive":["Portico"],"relation":{},"ISSN":["0914-3505","1741-4520"],"issn-type":[{"value":"0914-3505","type":"print"},{"value":"1741-4520","type":"electronic"}],"subject":[],"published":{"date-parts":[[2026,1]]},"assertion":[{"value":"2025-12-30","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2026-02-18","order":2,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2026-03-23","order":3,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}],"article-number":"e70046"}}