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RAS opathy 患者的突变和表型谱。

Mutation and Phenotypic Spectrum of Patients With RASopathies.

机构信息

Institute of Medical Genetics and Genomics, Sir Ganga Ram Hospital, New Delhi, India.

Department of Medical Genetics, Sanjay Gandhi Postgraduate Institute of Medical Sciences, Lucknow, India.

出版信息

Indian Pediatr. 2021 Jan 15;58(1):30-33.

Abstract

OBJECTIVE

To examine the common and specific clinical features, mutation spectrum and genotype-phenotype correlation in Noonan syndrome and related RASopathies.

PARTICIPANTS

Records of 30 patients with clinical diagnosis of Noonan syndrome and related RASopathies presenting over a six-year period at a tertiary care medical genetics centre were reviewed. Detailed clinical phenotype evaluation and genetic testing (PTPN11 sequencing or next generation sequencing) was done. The genetic results were used to classify the patients.

RESULTS

Noonan syndrome was confirmed in 22 patients, 5 had cardiofaciocutaneous syndrome and 3 had Noonan syndrome like disorder with loose anagen hair. The molecular diagnosis was confirmed in 27 patients. Mutations in PTPN11 gene were confirmed in 57.8 % patients. Developmental delay, cardiac defects, ectodermal abnormalities and coarse face was the predominant phenotype. Noonan syndrome like disorder with loose anagen hair was clinically identifiable by the sparse, slow growing hair and caused by one recurrent SHOC2, c.4A>G mutation.

CONCLUSION

Noonan syndrome and other RASopathies should be suspected in patients with short stature, cardiac defects, typical facial dysmorphism with or without ectodermal involvement.

摘要

目的

研究 Noonan 综合征及相关 RAS 通路疾病的常见和特有临床特征、突变谱及基因型-表型相关性。

参与者

对一家三级医疗遗传中心在 6 年期间诊治的 30 例临床诊断为 Noonan 综合征及相关 RAS 通路疾病的患者的病历进行了回顾性分析。对患者进行了详细的临床表型评估和基因检测(PTPN11 测序或下一代测序)。利用基因检测结果对患者进行了分类。

结果

22 例患者确诊为 Noonan 综合征,5 例为心面皮肤综合征,3 例为具有粗面型的 Noonan 综合征样疾病,其中 27 例患者的分子诊断得到了确认。PTPN11 基因突变在 57.8%的患者中得到了确认。发育迟缓、心脏缺陷、外胚层异常和粗面型是主要的表型特征。具有稀疏、生长缓慢的毛发的具有粗面型的 Noonan 综合征样疾病可通过临床识别,该疾病由一个反复出现的 SHOC2 基因 c.4A>G 突变引起。

结论

存在身材矮小、心脏缺陷、典型的面部畸形伴或不伴外胚层受累的患者应怀疑为 Noonan 综合征及其他 RAS 通路疾病。

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