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骨骼疾病的遗传学

Genetics of Skeletal Disorders.

作者信息

Hannan Fadil M, Newey Paul J, Whyte Michael P, Thakker Rajesh V

机构信息

Nuffield Department of Women's and Reproductive Health, University of Oxford, Oxford, UK.

Division of Molecular and Clinical Medicine, Ninewells Hospital and Medical School, University of Dundee, Dundee, UK.

出版信息

Handb Exp Pharmacol. 2020;262:325-351. doi: 10.1007/164_2020_350.

Abstract

Bone and mineral diseases encompass a variety of conditions that involve altered skeletal homeostasis and are frequently associated with changes in circulating calcium, phosphate, or vitamin D metabolites. These disorders often have a genetic etiology and comprise monogenic disorders caused by a single-gene mutation, which may be germline or somatic, or an oligogenic or polygenic condition involving multiple genetic variants. Single-gene mutations causing Mendelian diseases are usually highly penetrant, whereas the gene variants contributing to oligogenic or polygenic disorders are each associated with smaller effects with additional contributions from environmental factors. The detection of monogenic disorders is clinically important and facilitates timely assessment and management of the patient and their affected relatives. The diagnosis of monogenic metabolic bone disorders requires detailed clinical assessment of the wide variety of symptoms and signs associated with these diseases. Thus, clinicians should undertake a systematic approach commencing with careful history taking and physical examination, followed by appropriate laboratory and skeletal imaging investigations. Finally, clinicians should be familiar with the range of molecular genetic tests available to ensure their appropriate use and interpretation. These considerations are reviewed in this chapter.

摘要

骨与矿物质疾病涵盖了多种涉及骨骼内环境稳态改变的病症,并且常常与循环钙、磷或维生素D代谢产物的变化相关。这些疾病通常具有遗传病因,包括由单基因突变引起的单基因疾病,该突变可能是种系突变或体细胞突变,或者是涉及多个基因变异的寡基因或多基因病症。导致孟德尔疾病的单基因突变通常具有高度的外显率,而导致寡基因或多基因疾病的基因变异各自的影响较小,且受环境因素的额外影响。单基因疾病的检测在临床上很重要,有助于及时评估和管理患者及其受影响的亲属。单基因代谢性骨病的诊断需要对与这些疾病相关的各种各样的症状和体征进行详细的临床评估。因此,临床医生应采取系统的方法,首先仔细询问病史和进行体格检查,然后进行适当的实验室和骨骼影像学检查。最后,临床医生应熟悉可用的分子遗传学检测范围,以确保其正确使用和解读。本章将对这些注意事项进行综述。

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