Division of Plastic Surgery, Michael E. DeBakey Department of Surgery, Baylor College of Medicine, Houston, Texas, USA.
Division of Plastic Surgery, Texas Children's Hospital, Houston, Texas, USA.
Am J Med Genet A. 2021 Dec;185(12):3606-3612. doi: 10.1002/ajmg.a.61986. Epub 2020 Nov 25.
Robinow syndrome is characterized by mesomelic limb shortening, hemivertebrae, and genital hypoplasia. Due to low prevalence and considerable phenotypic variability, it has been challenging to definitively characterize features of Robinow syndrome. While craniofacial abnormalities associated with Robinow syndrome have been broadly described, there is a lack of detailed descriptions of genotype-specific phenotypic craniofacial features. Patients with Robinow syndrome were invited for a multidisciplinary evaluation conducted by specialist physicians at our institution. A focused assessment of the craniofacial manifestations was performed by a single expert examiner using clinical examination and standard photographic images. A total of 13 patients with clinical and molecular diagnoses consistent with either dominant Robinow syndrome (DRS) or recessive Robinow syndrome (RRS) were evaluated. On craniofacial examination, gingival hyperplasia was nearly ubiquitous in all patients. Orbital hypertelorism, a short nose with anteverted and flared nares, a triangular mouth with a long philtrum, cleft palate, macrocephaly, and frontal bossing were not observed in all individuals but affected individuals with both DRS and RRS. Other anomalies were more selective in their distribution in this patient cohort. We present a comprehensive analysis of the craniofacial findings in patients with Robinow Syndrome, describing associated morphological features and correlating phenotypic manifestations to underlying genotype in a manner relevant for early recognition and focused evaluation of these patients.
罗宾诺综合征的特征为中胚层肢体缩短、半椎体和生殖器发育不全。由于发病率低且表型变异较大,因此明确罗宾诺综合征的特征一直具有挑战性。虽然与罗宾诺综合征相关的颅面异常已被广泛描述,但缺乏对特定基因型表型颅面特征的详细描述。罗宾诺综合征患者应邀在我们机构接受由专科医生进行的多学科评估。由一位专家检查者使用临床检查和标准摄影图像对颅面表现进行了集中评估。共评估了 13 名具有明确临床和分子诊断的患者,这些患者符合显性罗宾诺综合征(DRS)或隐性罗宾诺综合征(RRS)。在颅面检查中,所有患者几乎都存在牙龈增生。眶距增宽、短鼻伴前鼻孔倾斜和外展、三角口伴长人中、腭裂、大头畸形和额骨突出并非所有患者都存在,但 DRS 和 RRS 患者都存在这些特征。其他异常在该患者队列中的分布更具选择性。我们对罗宾诺综合征患者的颅面发现进行了全面分析,描述了相关的形态特征,并将表型表现与潜在的基因型相关联,这对于早期识别和重点评估这些患者具有重要意义。