Patient with oculo-facio-cardio-dental syndrome treated with surgical orthodontics
Patient with oculo-facio-cardio-dental syndrome treated with surgical orthodontics
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DOI：
10.1016/j.ajodo.2011.10.020
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发表时间：
2012-04-01
影响因子：
3
通讯作者：
Miyawaki, Shouichi
中科院分区：
文献类型：
--
作者：
Sakaguchi, Katsuyoshi;Yagi, Takakazu;Miyawaki, Shouichi
眼-面-心-牙综合征(OFCD)是一种罕见的综合征，以眼部、面部、心脏和牙科疾病为特征。到目前为止，仅报告了大约20例病例。该综合征的各种特征中最突出的是犬神经根肥大。其他特征包括又长又窄的脸，高高的鼻梁，宽而尖的鼻子，鼻裂，耳朵畸形，腭裂或粘膜下裂，上颌发育迟缓，性交角度大，牙尖开放，延迟萌出，持续的乳牙，极度覆盖和收缩的上颌。正畸和正畸治疗已经报道了一些患者，但手术-正畸治疗OFCD尚未见报道。采用方丝弓矫治器结合正颌外科手术治疗1例18岁骨性安氏III类错牙合并OFCD综合征患者。我们在治疗过程中施加了轻微的力，以免引起关节僵硬。在手术和正畸治疗结束时，功能性咬合和面部轮廓得到改善。保留期结束后，口腔颌面功能得到改善。治疗结果表明，手术-正畸治疗是改善OFCD患者骨骼不协调、面部侧貌、咬合和口腔功能的有效方法。(Am J Orthod DentofaceOrthop 2012；141：S159-70)
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. (Am J Orthod Dentofacial Orthop 2012;141:S159-70)