{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,18]],"date-time":"2026-08-18T03:20:15Z","timestamp":1787023215231,"version":"build-2736575974"},"reference-count":14,"publisher":"SAGE Publications","issue":"6","license":[{"start":{"date-parts":[[2015,11,1]],"date-time":"2015-11-01T00:00:00Z","timestamp":1446336000000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/journals.sagepub.com\/page\/policies\/text-and-data-mining-license"}],"content-domain":{"domain":["journals.sagepub.com"],"crossmark-restriction":true},"short-container-title":["The Cleft Palate Craniofacial Journal"],"published-print":{"date-parts":[[2015,11]]},"abstract":"<jats:p>Syngnathia is a rare congenital disorder of jaw fusion with a paucity of literature from developed countries. We present a case of an infant noted to have multiple anomalies at birth including syngnathia, microcephaly with a variant of brain abnormality between holoprosencephaly and syntelencephaly, optic nerve hypoplasia, ear canal anomalies, hemi-vertebrae, and suspected hypomelanosis of Ito. To our knowledge, this patient with syngnathia and multiple anomalies is the first to be reported, but whether they are a coincidence, a pathogenetic association, or a new syndrome remains unknown. This case is discussed with a brief review of the literature.<\/jats:p>","DOI":"10.1597\/14-069","type":"journal-article","created":{"date-parts":[[2014,10,17]],"date-time":"2014-10-17T15:37:04Z","timestamp":1413560224000},"page":"201-204","update-policy":"https:\/\/doi.org\/10.1177\/sage-journals-update-policy","source":"Crossref","is-referenced-by-count":3,"title":["Postnatal Diagnosis of a Baby with Multiple Rare Congenital Anomalies Including Syngnathia, Brain Dysmorphism, and Skin Pigmentation"],"prefix":"10.1177","volume":"52","author":[{"given":"Linda","family":"Mahgoub","sequence":"first","affiliation":[{"name":"Department of Pediatrics, The Stollery Children's Hospital, Edmonton, Alberta, Canada."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Chloe","family":"Joynt","sequence":"additional","affiliation":[{"name":"Department of Pediatrics, University of Alberta, Edmonton, Alberta, Canada."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ravi","family":"Bhargava","sequence":"additional","affiliation":[{"name":"The Stollery Children's Hospital, Department of Radiology and Diagnostic Imaging, University of Alberta, Edmonton, Alberta, Canada."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Dawn","family":"Davies","sequence":"additional","affiliation":[{"name":"Department of Pediatrics, University of Alberta, and Medical Director, Pediatric Palliative Care Program, The Stollery Children's Hospital, Edmonton, Alberta, Canada."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hamdy","family":"El-Hakim","sequence":"additional","affiliation":[{"name":"Divisions of Otolaryngology Head & Neck Surgery, and Department of Surgery, The Stollery Children's Hospital, University of Alberta Hospitals, Edmonton, Alberta, Canada."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Walter","family":"Dobrovolsky","sequence":"additional","affiliation":[{"name":"Edmonton Zone, AHS; Executive Director, AOMSA; and Clinical Professor, OMFS, Faculty of Medicine and Dentistry, University of Alberta, Edmonton, Alberta, Canada."}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"179","published-online":{"date-parts":[[2018,1,3]]},"reference":[{"key":"bibr1-14-069","doi-asserted-by":"publisher","DOI":"10.1136\/jmedgenet-2012-100892"},{"key":"bibr2-14-069","doi-asserted-by":"publisher","DOI":"10.1597\/1545-1569_1997_034_0141_cbsapc_2.3.co_2"},{"key":"bibr3-14-069","doi-asserted-by":"publisher","DOI":"10.1002\/1096-8628(20001127)95:3<269::AID-AJMG14>3.0.CO;2-P"},{"key":"bibr4-14-069","doi-asserted-by":"publisher","DOI":"10.1016\/j.ijom.2010.04.003"},{"key":"bibr5-14-069","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.c.30238"},{"key":"bibr6-14-069","doi-asserted-by":"publisher","DOI":"10.1016\/j.ijom.2012.05.004"},{"key":"bibr7-14-069","first-page":"796","volume":"110","author":"Klepacek I.","year":"2009","journal-title":"Bratisl Med J."},{"key":"bibr8-14-069","doi-asserted-by":"publisher","DOI":"10.1054\/ijom.2000.0009"},{"key":"bibr9-14-069","doi-asserted-by":"publisher","DOI":"10.1597\/09-090"},{"key":"bibr10-14-069","doi-asserted-by":"publisher","DOI":"10.1016\/j.ijom.2010.03.027"},{"key":"bibr11-14-069","doi-asserted-by":"publisher","DOI":"10.1597\/10-032"},{"key":"bibr12-14-069","doi-asserted-by":"publisher","DOI":"10.1097\/01.sap.0000215265.68252.f0"},{"key":"bibr13-14-069","doi-asserted-by":"publisher","DOI":"10.1097\/00001665-200503000-00016"},{"key":"bibr14-14-069","doi-asserted-by":"publisher","DOI":"10.1002\/pd.2208"}],"container-title":["The Cleft Palate Craniofacial Journal"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/journals.sagepub.com\/doi\/pdf\/10.1597\/14-069","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/journals.sagepub.com\/doi\/full-xml\/10.1597\/14-069","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/journals.sagepub.com\/doi\/pdf\/10.1597\/14-069","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,4,28]],"date-time":"2026-04-28T16:18:38Z","timestamp":1777393118000},"score":1,"resource":{"primary":{"URL":"https:\/\/journals.sagepub.com\/doi\/10.1597\/14-069"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2015,11]]},"references-count":14,"journal-issue":{"issue":"6","published-print":{"date-parts":[[2015,11]]}},"alternative-id":["10.1597\/14-069"],"URL":"https:\/\/doi.org\/10.1597\/14-069","relation":{},"ISSN":["1055-6656","1545-1569"],"issn-type":[{"value":"1055-6656","type":"print"},{"value":"1545-1569","type":"electronic"}],"subject":[],"published":{"date-parts":[[2015,11]]}}}