Department of Clinical Genetics, Hospital of Rehabilitation of Craniofacial Anomalies (HRAC), University of São Paulo, Bauru, SP, Brazil.
Am J Med Genet A. 2010 Jul;152A(7):1838-40. doi: 10.1002/ajmg.a.33477.
We describe a patient with a phenotype characterized by mandibulofacial dysostosis with severe lower eyelid coloboma, cleft palate, abnormal ears, alopecia, delayed eruption and crowded teeth, and sensorioneural hearing loss. The karyotype and the screening for mutations in the coding region of TCOF1 gene were normal. The clinical signs of our case overlap the new mandibulofacial dysostosis described by Stevenson et al. [2007] and the case with Johnson-McMillin syndrome described by Cushman et al. [2005]. The similar clinical signs, mainly, the severe facial involvement observed in these cases suggest that they can represent a new distinct form of mandibulofacial dysostosis or the end of the spectrum of Johnson-McMillin syndrome.
我们描述了一位患者的表型特征为下颌面骨发育不全,伴有严重的下眼睑裂、腭裂、耳朵异常、脱发、出牙延迟和牙齿拥挤,以及感觉神经性听力损失。核型和 TCOF1 基因突变编码区的筛查均正常。我们的病例的临床特征与 Stevenson 等人描述的新的下颌面骨发育不全[2007]和 Cushman 等人描述的 Johnson-McMillin 综合征病例重叠[2005]。这些病例的主要相似临床特征,即严重的面部受累,表明它们可能代表一种新的下颌面骨发育不全的独特形式或 Johnson-McMillin 综合征的表现范围的末端。