Division of Human Genetics, Department of Pediatrics, University of Maryland School of Medicine, Baltimore, Maryland, USA.
Department of Pathology, Stanford University Medical Center, Stanford, California, USA.
Genet Med. 2017 Oct;19(10). doi: 10.1038/gim.2017.84. Epub 2017 Jul 5.
Disclaimer: These ACMG Standards and Guidelines are intended as an educational resource for clinical laboratory geneticists to help them provide quality clinical laboratory genetic services. Adherence to these Standards and Guidelines is voluntary and does not necessarily assure a successful medical outcome. These Standards and Guidelines should not be considered inclusive of all proper procedures and tests or exclusive of others that are reasonably directed to obtaining the same results. In determining the propriety of any specific procedure or test, clinical laboratory geneticists should apply their professional judgment to the specific circumstances presented by the patient or specimen. Clinical laboratory scientists and geneticists are encouraged to document in the patient's record the rationale for the use of a particular procedure or test, whether or not it is in conformance with these Standards and Guidelines. They also are advised to take notice of the date any particular guideline was adopted, and to consider other relevant medical and scientific information that becomes available after that date. It also would be prudent to consider whether intellectual property interests may restrict the performance of certain tests and other procedures.Biotinidase deficiency is an autosomal recessively inherited disorder of biotin recycling that is associated with neurologic and cutaneous consequences if untreated. Fortunately, the clinical features of the disorder can be ameliorated or prevented by administering pharmacological doses of the vitamin biotin. Newborn screening and confirmatory diagnosis of biotinidase deficiency encompasses both enzymatic and molecular testing approaches. These guidelines were developed to define and standardize laboratory procedures for enzymatic biotinidase testing, to delineate situations for which follow-up molecular testing is warranted, and to characterize variables that can influence test performance and interpretation of results.
这些 ACMG 标准和指南旨在为临床实验室遗传学家提供教育资源,帮助他们提供高质量的临床实验室遗传服务。遵守这些标准和指南是自愿的,并不一定保证医疗结果的成功。这些标准和指南不应被视为包括所有适当的程序和测试,也不应排除其他合理指向获得相同结果的测试。在确定任何特定程序或测试的适当性时,临床实验室遗传学家应将其专业判断应用于患者或标本提出的具体情况。鼓励临床实验室科学家和遗传学家在患者记录中记录使用特定程序或测试的理由,无论其是否符合这些标准和指南。他们还应注意到任何特定指南的采用日期,并考虑在该日期之后获得的其他相关医学和科学信息。考虑知识产权权益是否可能限制某些测试和其他程序的执行也是明智的。生物素酶缺乏症是一种常染色体隐性遗传的生物素再循环障碍,如果不治疗,会导致神经和皮肤后果。幸运的是,通过给予药理剂量的维生素生物素,可以改善或预防该疾病的临床特征。生物素酶缺乏症的新生儿筛查和确认诊断包括酶学和分子检测方法。这些指南旨在定义和标准化酶学生物素酶检测的实验室程序,描述需要进行后续分子检测的情况,并描述可能影响测试性能和结果解释的变量。