{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,11,14]],"date-time":"2025-11-14T21:25:55Z","timestamp":1763155555984,"version":"3.40.5"},"reference-count":10,"publisher":"The Angle Orthodontist (EH Angle Education & Research Foundation)","issue":"5","content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2007,9,1]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:p>Stickler syndrome (MIM 108300, 604841, 184840) is an autosomal dominant disease characterized by midfacial flattening and variable disorders of vision, hearing and articulation. There are three types of the syndrome caused by mutations in different genes (type 1, COL2A1; type 2, COL11A1; and type 3, COL11A2). About 20% of type 1 patients have cleft palate or bifid uvula, but there have been no case reports of orthodontic treatment of this syndrome so far. The Japanese female patient presented here with Stickler syndrome was characterized by a flat midface and had high myopia, sensorineural hearing loss, enlarged joints, and cleft of the soft palate. She had fairly small SNA and SNB angles and a steep mandibular plane with an enlarged gonial angle. The incisors of both arches were retroclined, and a large overjet and overbite were noted. Orthodontic treatment was initiated at 11 years of age using a lingual arch appliance followed by an edgewise multibracket appliance. Stable functional occlusion was obtained after the treatment. Most of the other seven Stickler syndrome patients exhibited pretreatment characteristics of small SNA and SNB angles, steep mandibular planes, enlarged gonial angles, and retroclined incisors of both arches, demonstrating the characteristic skeletal and occlusal features of this syndrome.<\/jats:p>","DOI":"10.2319\/082406-347","type":"journal-article","created":{"date-parts":[[2007,5,9]],"date-time":"2007-05-09T17:32:04Z","timestamp":1178731924000},"page":"931-939","source":"Crossref","is-referenced-by-count":6,"title":["Orthodontic Treatment of a Patient with Stickler Syndrome"],"prefix":"10.2319","volume":"77","author":[{"given":"Naoto","family":"Suda","sequence":"first","affiliation":[{"name":"a Lecturer, Maxillofacial Orthognathics, Department of Maxillofacial Reconstruction and Function, Division of Maxillofacial\/ Neck Reconstruction, Graduate School, Tokyo Medical and Dental University, Tokyo, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Sachiko","family":"Handa","sequence":"first","affiliation":[{"name":"b Clinical Fellow, Maxillofacial Orthognathics, Department of Maxillofacial Reconstruction and Function, Division of Maxillofacial\/Neck Reconstruction, Graduate School, Tokyo Medical and Dental University, Tokyo, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Norihisa","family":"Higashihori","sequence":"first","affiliation":[{"name":"c Dental Resident, Maxillofacial Orthognathics, Department of Maxillofacial Reconstruction and Function, Division of Maxillofacial\/Neck Reconstruction, Graduate School, Tokyo Medical and Dental University, Tokyo, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Takuya","family":"Ogawa","sequence":"first","affiliation":[{"name":"c Dental Resident, Maxillofacial Orthognathics, Department of Maxillofacial Reconstruction and Function, Division of Maxillofacial\/Neck Reconstruction, Graduate School, Tokyo Medical and Dental University, Tokyo, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Michiko","family":"Tsuji","sequence":"first","affiliation":[{"name":"d Assistant Professor, Maxillofacial Orthognathics, Department of Maxillofacial Reconstruction and Function, Division of Maxillofacial\/Neck Reconstruction, Graduate School, Tokyo Medical and Dental University, Tokyo, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Kimie","family":"Ohyama","sequence":"first","affiliation":[{"name":"e Ex-Professor, Maxillofacial Orthognathics, Department of Maxillofacial Reconstruction and Function, Division of Maxillofacial\/Neck Reconstruction, Graduate School, Tokyo Medical and Dental University, Tokyo, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"1164","reference":[{"key":"2020042117470983500_i0003-3219-077-05-0931-b1","first-page":"495","article-title":"Hereditary progressive arthroophthalmopathy. II. Additional observation on vertebral anomalies, a hearing defect and a report of a similar case.","volume":"42","author":"Stickler","year":"1967","journal-title":"Mayo Clin Proc"},{"key":"2020042117470983500_i0003-3219-077-05-0931-b2","doi-asserted-by":"crossref","first-page":"331","DOI":"10.1111\/j.1749-6632.1996.tb56300.x","article-title":"Stickler syndrome type 2 and linkage to the COL11A1 gene.","volume":"785","author":"Snead","year":"1996","journal-title":"Ann N Y Acad Sci"},{"article-title":"Syndromes affecting bone; other skeletal dysplasia.","author":"Gorlin","key":"2020042117470983500_i0003-3219-077-05-0931-b3","doi-asserted-by":"crossref","DOI":"10.1093\/oso\/9780195118612.003.0009"},{"key":"2020042117470983500_i0003-3219-077-05-0931-b4","doi-asserted-by":"crossref","first-page":"6624","DOI":"10.1073\/pnas.88.15.6624","article-title":"Related. Stop codon in the procollagen II gene (COL2A1) in a family with the Stickler syndrome (arthro-ophthalmopathy).","volume":"88","author":"Ahmad","year":"1991","journal-title":"Proc Natl Acad Sci U S A"},{"key":"2020042117470983500_i0003-3219-077-05-0931-b5","doi-asserted-by":"crossref","first-page":"807","DOI":"10.1038\/sj.ejhg.5200377","article-title":"Stickler syndrome: further mutations in COL11A1 and evidence for additional locus heterogeneity.","volume":"7","author":"Martin","year":"1999","journal-title":"Eur J Hum Genet"},{"key":"2020042117470983500_i0003-3219-077-05-0931-b6","doi-asserted-by":"crossref","first-page":"368","DOI":"10.1016\/S0022-3476(98)70466-4","article-title":"Stickler syndrome without eye involvement is caused by mutations in COL11A2, the gene encoding the alpha2 (XI) chain of type XI collagen.","volume":"132","author":"Sirko-Osadsa","year":"1998","journal-title":"J Pediatr"},{"key":"2020042117470983500_i0003-3219-077-05-0931-b7","first-page":"18","article-title":"Roentgencephalometric analysis of craniofacial growth in Japanese children.","volume":"25","author":"Iizuka","year":"1958","journal-title":"J Stomatol Soc Jpn"},{"key":"2020042117470983500_i0003-3219-077-05-0931-b8","first-page":"1","article-title":"A study on the developmental changes of dentofacial complex of the Japanese with special reference to sella turcica.","volume":"18","author":"Sakamoto","year":"1959","journal-title":"J Jpn Orthod Soc"},{"key":"2020042117470983500_i0003-3219-077-05-0931-b9","first-page":"373","article-title":"Stickler syndrome and Marshall syndrome among the cleft palate patients.","volume":"18","author":"Ohyama","year":"1993","journal-title":"J Jpn Cleft Palate Assoc"},{"key":"2020042117470983500_i0003-3219-077-05-0931-b10","first-page":"896","article-title":"Skeletal characteristics and treatment outcome of five cases with Robin sequence.","volume":"76","author":"Matsuda","year":"2006","journal-title":"Angle Orthod"}],"container-title":["The Angle Orthodontist"],"original-title":[],"language":"en","link":[{"URL":"http:\/\/meridian.allenpress.com\/angle-orthodontist\/article-pdf\/77\/5\/931\/1374029\/082406-347.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"syndication"},{"URL":"http:\/\/meridian.allenpress.com\/angle-orthodontist\/article-pdf\/77\/5\/931\/1374029\/082406-347.pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,5,7]],"date-time":"2025-05-07T16:04:38Z","timestamp":1746633878000},"score":1,"resource":{"primary":{"URL":"https:\/\/angle-orthodontist.kglmeridian.com\/view\/journals\/angl\/77\/5\/article-p931.xml"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2007,9,1]]},"references-count":10,"journal-issue":{"issue":"5","published-print":{"date-parts":[[2007,9,1]]}},"URL":"https:\/\/doi.org\/10.2319\/082406-347","relation":{},"ISSN":["0003-3219","1945-7103"],"issn-type":[{"type":"print","value":"0003-3219"},{"type":"electronic","value":"1945-7103"}],"subject":[],"published":{"date-parts":[[2007,9,1]]}}}