Tuin Jorien, Tahiri Youssef, Paliga James T, Taylor Jesse A, Bartlett Scott P
*Division of Plastic Surgery, Children's Hospital of Philadelphia, The Perelman School of Medicine at the University of Pennsylvania, PA †Division of Plastic Surgery, Riley Hospital for Children, Indiana University, IN.
J Craniofac Surg. 2015 Sep;26(6):1887-92. doi: 10.1097/SCS.0000000000002017.
Goldenhar syndrome is characterized by the typical features of craniofacial microsomia (CFM) with the addition of epibulbar dermoids and vertebral anomalies. The aim of this study is to examine the objective differences between patients carrying a diagnosis of Goldenhar syndrome to those diagnosed with CFM. Thus, we performed an Institutional Review Board-approved retrospective chart review on all patients who presented with a diagnosis of CFM or Goldenhar syndrome from January 1990 to December 2012. Demographic, diagnosis, OMENS+ classification, accompanying diagnoses, and radiographic data were collected. For subjective analysis, subgroups were designed based on the diagnosis Goldenhar syndrome or CFM per history. For objective analysis, subgroups were designed based on the presence of epibulbar dermoids and/or vertebral anomalies. The cohorts were compared with respect to associated medical abnormalities and severity of CFM features. One hundred thirty eight patients met inclusion criteria. Epibulbar dermoids and vertebral anomalies were seen in 17% and 34% of the patients, respectively. Only 10 patients (7.2%) had both epibulbar dermoids and vertebral anomalies. The subjective "Goldenhar" group (N = 44, 32%) was found to have a higher percentage of bilaterally affected patients (P = 0.001), a more severe mandibular deformity (P = <0.001), a more severe soft tissue deformity (P = 0.01), and a higher incidence of macrostomia (P = 0.003). In the objective subgroup analysis, the only significant difference was found in the degree of soft tissue deficiency (P = 0.049). The diagnostic criteria of Goldenhar syndrome remain unclear, thereby making clinical use of the term "Goldenhar" inconsequential. Goldenhar syndrome is over diagnosed subjectively in patients who show more severe CFM features.
Goldenhar综合征的特征是颅面短小畸形(CFM)的典型特征,外加眼球皮样囊肿和脊柱异常。本研究的目的是检查诊断为Goldenhar综合征的患者与诊断为CFM的患者之间的客观差异。因此,我们对1990年1月至2012年12月期间所有诊断为CFM或Goldenhar综合征的患者进行了一项经机构审查委员会批准的回顾性病历审查。收集了人口统计学、诊断、OMENS+分类、伴随诊断和影像学数据。为进行主观分析,根据既往诊断为Goldenhar综合征或CFM设计亚组。为进行客观分析,根据眼球皮样囊肿和/或脊柱异常的存在设计亚组。比较各队列相关的医学异常情况和CFM特征的严重程度。138例患者符合纳入标准。分别有17%和34%的患者出现眼球皮样囊肿和脊柱异常。只有10例患者(7.2%)同时有眼球皮样囊肿和脊柱异常。主观“Goldenhar”组(N = 44,32%)中双侧受累患者的比例更高(P = 0.001),下颌畸形更严重(P = <0.001),软组织畸形更严重(P = 0.01),大口畸形的发生率更高(P = 0.003)。在客观亚组分析中,仅在软组织缺损程度方面发现显著差异(P = 0.049)。Goldenhar综合征的诊断标准仍不明确,因此“Goldenhar”一词在临床应用中无实际意义。在表现出更严重CFM特征的患者中,Goldenhar综合征被主观过度诊断。