Dipartimento di Biologia, Università Federico II, 80126 Napoli, Italy.
Istituto di Chimica Biomolecolare, Consiglio Nazionale delle Ricerche, Comprensorio Olivetti, Edificio 70, Via Campi Flegrei 34, 80078 Pozzuoli, Italy.
Int J Mol Sci. 2017 Sep 24;18(10):2049. doi: 10.3390/ijms18102049.
Rare diseases represent a challenge for physicians because patients are rarely seen, and they can manifest with symptoms similar to those of common diseases. In this work, genetic confirmation of diagnosis is derived from DNA sequencing. We present a tutorial for the molecular analysis of a rare disease using Fabry disease as an example.
An exonic sequence derived from a hypothetical male patient was matched against human reference data using a genome browser. The missense mutation was identified by running BlastX, and information on the affected protein was retrieved from the database UniProt. The pathogenic nature of the mutation was assessed with PolyPhen-2. Disease-specific databases were used to assess whether the missense mutation led to a severe phenotype, and whether pharmacological therapy was an option.
An inexpensive bioinformatics approach is presented to get the reader acquainted with the diagnosis of Fabry disease. The reader is introduced to the field of pharmacological chaperones, a therapeutic approach that can be applied only to certain Fabry genotypes.
The principle underlying the analysis of exome sequencing can be explained in simple terms using web applications and databases which facilitate diagnosis and therapeutic choices.
罕见病对医生来说是一个挑战,因为罕见病患者很少见,而且它们的症状可能与常见疾病相似。在这项工作中,通过 DNA 测序来确定诊断的基因确认。我们以法布瑞氏病为例,提供了一个罕见病分子分析的教程。
使用基因组浏览器将源自假设男性患者的外显子序列与人类参考数据进行匹配。通过运行 BlastX 来识别错义突变,并从数据库 UniProt 中检索受影响蛋白的信息。使用 PolyPhen-2 评估突变的致病性。使用特定于疾病的数据库来评估错义突变是否导致严重表型,以及是否有药物治疗的选择。
提出了一种廉价的生物信息学方法,使读者熟悉法布瑞氏病的诊断。向读者介绍了药理学伴侣这一治疗方法,该方法仅适用于某些法布瑞氏基因型。
可以使用网络应用程序和数据库来解释外显子组测序分析的原理,从而方便诊断和治疗选择。