{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,19]],"date-time":"2026-08-19T19:59:00Z","timestamp":1787169540913,"version":"build-2736575974"},"reference-count":20,"publisher":"Ovid Technologies (Wolters Kluwer Health)","issue":"Suppl 1","content-domain":{"domain":["lww.com","ovid.com"],"crossmark-restriction":true},"short-container-title":[],"published-print":{"date-parts":[[2022,2]]},"abstract":"<jats:sec>\n                    <jats:title\/>\n                    <jats:p>\n                      Binder's syndrome, a rare congenital malformation of nasomaxillary complex, first described in 1962, has a hexad of characteristic clinical and radiographic features consisting of arhinoid face, intermaxillary hypoplasia with malocclusion, abnormal position of nasal bones, atrophy of nasal mucosa, reduced or absent anterior nasal spine and hypoplastic\/absent frontal sinus. The typical facies due to mid-face hypoplasia may also be accompanied by other midline malformations such as cleft palate, spinal, skeletal and cardiac abnormalities. It is usually sporadic, of unknown etiology although various environmental and genetic mechanisms are implicated due to few familial cases predominantly in the Swedish population. A case of inherited Binder's syndrome is presented in an Indian female patient with an unusual finding of ankyloglossia (AG). The development of the anterior nasal spine and AG are chronologically related as they both occur during the 5\n                      <jats:sup>th<\/jats:sup>\n                      \u20136\n                      <jats:sup>th<\/jats:sup>\n                      weeks of gestation. The possible etiopathogenetic mechanisms for this rare association are reviewed.\n                    <\/jats:p>\n                  <\/jats:sec>","DOI":"10.4103\/jomfp.jomfp_143_21","type":"journal-article","created":{"date-parts":[[2022,3,8]],"date-time":"2022-03-08T07:18:46Z","timestamp":1646723926000},"page":"S5-S11","update-policy":"https:\/\/doi.org\/10.1097\/lww.0000000000001000","source":"Crossref","is-referenced-by-count":0,"title":["Binder's phenotype with ankyloglossia"],"prefix":"10.4103","volume":"26","author":[{"given":"Shalini R.","family":"Gupta","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"B.","family":"Rajiv","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Anuradha","family":"Yadav","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sheetal","family":"Sharma","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"276","reference":[{"key":"R1-2-20240906","doi-asserted-by":"crossref","first-page":"145","DOI":"10.1093\/ejo\/7.2.145","article-title":"Maxillonasal dysostosis (Binder syndrome); a review of the literature and case reports","volume":"7","author":"Ferguson","year":"1985","journal-title":"Eur J Orthod"},{"key":"R2-2-20240906","doi-asserted-by":"crossref","first-page":"114","DOI":"10.1016\/0278-2391(87)90401-0","article-title":"Maxillonasal dysplasia (Binder's syndrome): A critical review and case study","volume":"45","author":"Horswell","year":"1987","journal-title":"J Oral Maxillofac Surg"},{"key":"R3-2-20240906","doi-asserted-by":"crossref","first-page":"314","DOI":"10.4103\/0970-0358.197237","article-title":"Nasal correction in nasomaxillary hypoplasia (Binder's syndrome): An optimised classification and treatment","volume":"49","author":"Yamani","year":"2016","journal-title":"Indian J Plast Surg"},{"key":"R4-2-20240906","doi-asserted-by":"crossref","first-page":"1339","DOI":"10.1097\/SCS.0b013e3181ef2b71","article-title":"Binder phenotype: Associated findings and etiologic mechanisms","volume":"21","author":"Keppler-Noreuil","year":"2010","journal-title":"J Craniofac Surg"},{"key":"R5-2-20240906","doi-asserted-by":"crossref","first-page":"559","DOI":"10.1097\/00006534-198611000-00001","article-title":"Clinical and pathologic features of maxillonasal dysplasia (Binder's syndrome): Significance of the prenasal fossa on etiology","volume":"78","author":"Holmstr\u00f6m","year":"1986","journal-title":"Plast Reconstr Surg"},{"key":"R6-2-20240906","first-page":"69","article-title":"An etiologic study of maxillonasal dysplasia--Binder's syndrome","volume":"96","author":"Olow-Nordenram","year":"1988","journal-title":"Scand J Dent Res"},{"key":"R7-2-20240906","doi-asserted-by":"crossref","first-page":"146","DOI":"10.1016\/S0007-1226(63)80097-1","article-title":"Hypoplasia of the middle third of the face associated with congenital absence of the anterior nasal spine, depression of the nasal bones, and angle class III malocclusion","volume":"16","author":"Hopkin","year":"1963","journal-title":"Br J Plast Surg"},{"key":"R8-2-20240906","first-page":"227","article-title":"Binder syndrome in a mother and her son","volume":"8","author":"Roy-Doray","year":"1997","journal-title":"Genet Couns"},{"key":"R9-2-20240906","doi-asserted-by":"crossref","first-page":"227","DOI":"10.1097\/00006534-195210000-00002","article-title":"A simple method of reconstruction in some cases of dish-face deformity","volume":"10","author":"Ragnell","year":"1952","journal-title":"Plast Reconstr Surg (1946"},{"key":"R10-2-20240906","doi-asserted-by":"crossref","first-page":"329","DOI":"10.5999\/aps.2012.39.4.329","article-title":"A study on the genetic inheritance of ankyloglossia based on pedigree analysis","volume":"39","author":"Han","year":"2012","journal-title":"Arch Plast Surg"},{"key":"R11-2-20240906","first-page":"185803","article-title":"Clinical guidelines and management of ankyloglossia with 1-year followup: Report of 3 cases","volume":"2013","author":"Bhattad","year":"2013","journal-title":"Case Rep Dent"},{"key":"R12-2-20240906","doi-asserted-by":"crossref","first-page":"353","DOI":"10.1177\/028418518402500501","article-title":"Maxillo-nasal dysplasia (Binder syndrome) and associated malformations of the cervical spine","volume":"25","author":"Olow-Nordenram","year":"1984","journal-title":"Acta Radiol Diagn (Stockh"},{"key":"R13-2-20240906","doi-asserted-by":"crossref","first-page":"160","DOI":"10.1016\/S0022-3476(82)80261-8","article-title":"Errors of morphogenesis: Concepts and terms: Recommendations of an international working group","volume":"100","author":"Spranger","year":"1982","journal-title":"J Pediatr"},{"key":"R14-2-20240906","doi-asserted-by":"crossref","first-page":"145749","DOI":"10.1155\/2012\/145749","article-title":"Dental approach to craniofacial syndromes: How can developmental fields show us a new way to understand pathogenesis?","volume":"2012","author":"Kj\u00e6r","year":"2012","journal-title":"Int J Dent"},{"key":"R15-2-20240906","doi-asserted-by":"crossref","first-page":"1","DOI":"10.1016\/j.odw.2011.10.001","article-title":"New diagnostics of the dentition on panoramic radiographs focusing on the peripheral nervous system as an important aetiological factor behind dental anomalies","volume":"71","author":"Kj\u00e6r","year":"2012","journal-title":"Orthod Waves"},{"key":"R16-2-20240906","doi-asserted-by":"crossref","first-page":"384","DOI":"10.1136\/jmg.27.6.384","article-title":"Maxillonasal dysplasia (Binder's syndrome)","volume":"27","author":"Quarrell","year":"1990","journal-title":"J Med Genet"},{"key":"R17-2-20240906","doi-asserted-by":"crossref","first-page":"31","DOI":"10.1016\/bs.ctdb.2015.07.023","article-title":"Mandible and tongue development","volume":"115","author":"Parada","year":"2015","journal-title":"Curr Top Dev Biol"},{"key":"R18-2-20240906","doi-asserted-by":"crossref","first-page":"270","DOI":"10.4103\/0972-124X.85673","article-title":"Ankyloglossia and its management","volume":"15","author":"Chaubal","year":"2011","journal-title":"J Indian Soc Periodontol"},{"key":"R19-2-20240906","doi-asserted-by":"crossref","first-page":"1614","DOI":"10.1002\/ajmg.a.38690","article-title":"Familial autosomal dominant severe ankyloglossia with tooth abnormalities","volume":"176","author":"Lenormand","year":"2018","journal-title":"Am J Med Genet A"},{"key":"R20-2-20240906","doi-asserted-by":"crossref","first-page":"1321","DOI":"10.1016\/j.ijporl.2007.05.018","article-title":"Familial ankyloglossia (tongue-tie)","volume":"71","author":"Klockars","year":"2007","journal-title":"Int J Pediatr Otorhinolaryngol"}],"container-title":["Journal of Oral and Maxillofacial Pathology"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/www.ovid.com\/10.4103\/jomfp.jomfp_143_21","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,8,19]],"date-time":"2026-08-19T19:08:48Z","timestamp":1787166528000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.ovid.com\/10.4103\/jomfp.jomfp_143_21"}},"subtitle":["Report of a rare inherited association in an Indian female"],"short-title":[],"issued":{"date-parts":[[2022,2]]},"references-count":20,"journal-issue":{"issue":"Suppl 1","published-print":{"date-parts":[[2022]]}},"URL":"https:\/\/doi.org\/10.4103\/jomfp.jomfp_143_21","relation":{},"ISSN":["0973-029X"],"issn-type":[{"value":"0973-029X","type":"print"}],"subject":[],"published":{"date-parts":[[2022,2]]}}}