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眼面心牙综合征患者经外科正牙治疗。

Patient with oculo-facio-cardio-dental syndrome treated with surgical orthodontics.

机构信息

Department of Orthodontics, Field of Developmental Medicine, Health Research Course, Graduate School of Medical and Dental Sciences, Kagoshima University, Kagoshima, Japan.

出版信息

Am J Orthod Dentofacial Orthop. 2012 Apr;141(4 Suppl):S159-70. doi: 10.1016/j.ajodo.2011.10.020.

DOI:10.1016/j.ajodo.2011.10.020
PMID:22449596
Abstract

Oculo-facio-cardio-dental (OFCD) syndrome is a rare syndrome characterized by ocular, facial, cardiac, and dental disorders. Only about 20 cases have been reported to date. The most prominent of the various features of this syndrome is canine radiculomegaly. Other features include a long and narrow face, a high nasal bridge, a broad and pointed nose, a bifid nose, ear deformity, cleft palate or submucous cleft palate, maxillary growth retardation, a large gonial angle, open apices, delayed eruption, persistent deciduous teeth, extreme overbite, and constricted maxilla. Orthodontic and prosthodontic treatment has been reported for several patients, but surgical-orthodontic treatment for OFCD has not been reported. An 18-year-old woman with skeletal Class III malocclusion and OFCD syndrome was treated with edgewise appliance therapy combined with orthognathic surgery. We applied a light force during the treatment so as not to induce ankylosis. At the end of the surgical and orthodontic treatments, functional occlusion and an improved facial profile were achieved. After the retention period, stomatognathic function was improved. The results of this treatment suggest that surgical-orthodontic treatment is an effective method for improving skeletal disharmony, facial profile, occlusion, and stomatognathic function in patients with OFCD.

摘要

眼面心牙(OFCD)综合征是一种罕见的综合征,其特征为眼部、面部、心脏和牙齿异常。迄今为止,仅报道了约 20 例病例。该综合征的各种特征中最突出的是犬齿根肥大。其他特征包括长而窄的脸、高鼻梁、宽而尖的鼻子、分叉的鼻子、耳部畸形、腭裂或黏膜下腭裂、上颌生长迟缓、大下颌角、开颌、萌出延迟、乳牙滞留、严重的深覆颌,以及上颌骨狭窄。已有报道对一些患者进行了正畸和修复治疗,但尚未报道 OFCD 的手术-正畸治疗。我们对一名 18 岁的骨骼 III 类错颌伴 OFCD 综合征的女性患者进行了方丝弓矫治器治疗联合正颌手术。我们在治疗过程中施加轻力,以免导致关节强直。在手术和正畸治疗结束时,获得了功能咬合和改善的面部轮廓。保持期结束后,口腔颌面部功能得到改善。该治疗结果表明,手术-正畸治疗是改善 OFCD 患者骨骼不协调、面部轮廓、咬合和口腔颌面部功能的有效方法。

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Case Rep Dent. 2022 Mar 29;2022:3548370. doi: 10.1155/2022/3548370. eCollection 2022.
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New radiological findings and radiculomegaly in oculofaciocardiodental syndrome with a novel BCOR mutation: A case report.伴有新型BCOR突变的眼面心脏牙齿综合征的新影像学发现及神经根肿大:一例报告
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