Literature DB >> 26267577

Distinguishing Goldenhar Syndrome from Craniofacial Microsomia.

Jorien Tuin1, Youssef Tahiri, James T Paliga, Jesse A Taylor, Scott P Bartlett.   

Abstract

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.

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Year:  2015        PMID: 26267577     DOI: 10.1097/SCS.0000000000002017

Source DB:  PubMed          Journal:  J Craniofac Surg        ISSN: 1049-2275            Impact factor:   1.046


  4 in total

1.  Phenotypic characterization of epibulbar dermoids.

Authors:  Brian A Walker; Babette S Saltzman; Erin P Herlihy; Daniela V Luquetti
Journal:  Int Ophthalmol       Date:  2016-07-12       Impact factor: 2.031

Review 2.  Three-dimensional imaging of soft and hard facial tissues in patients with craniofacial syndromes: a systematic review of methodological quality.

Authors:  Arianne Lewyllie; Maria Cadenas De Llano-Pérula; Anna Verdonck; Guy Willems
Journal:  Dentomaxillofac Radiol       Date:  2017-12-18       Impact factor: 2.419

3.  Three-dimensional characterization of mandibular asymmetry in craniofacial microsomia.

Authors:  Yun-Fang Chen; Frank Baan; Robin Bruggink; Ewald Bronkhorst; Yu-Fang Liao; Edwin Ongkosuwito
Journal:  Clin Oral Investig       Date:  2020-05-07       Impact factor: 3.573

4.  Distribution and phenotypes of hemifacial microsomia and its association with other anomalies.

Authors:  Il-Hyung Yang; Jee Hyeok Chung; Sunjin Yim; Il-Sik Cho; Seung-Weon Lim; Kikap Kim; Sukwha Kim; Jin-Young Choi; Jong-Ho Lee; Myung-Jin Kim; Seung-Hak Baek
Journal:  Korean J Orthod       Date:  2020-01-22       Impact factor: 1.372

  4 in total

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