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骨骼发育不良分类学的变化。

Changes in skeletal dysplasia nosology.

机构信息

Department of Genetics, Carol Davila University of Medicine and Pharmacy, Bucharest, Romania;

出版信息

Rom J Morphol Embryol. 2021 Jul-Sep;62(3):689-696. doi: 10.47162/RJME.62.3.05.

DOI:10.47162/RJME.62.3.05
PMID:35263396
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC9019670/
Abstract

Skeletal dysplasia (SD), also called osteochondrodysplasia (OCD), is a large group of skeletal disorders (over 400 distinct entities) caused by abnormalities in bone development and growth. SDs varies according to different natural histories, prognoses, hereditary patterns to etiopathogenetic mechanisms. At birth, the incidence is low, reported at the level of each entity, but taken collectively; the incidence is estimated at 1:5000 births. Nosology is a branch of medical science. It deals with the systematic classification of diseases and disorders. Thus, combining information about the catalogue of clinically distinct disorders, pending molecular explanations, and genotype-phenotype correlations, the classification of SDs will be more accurate. This is extremely useful for diagnosing patients with genetic skeletal diseases, especially given the expected flow of information with new sequencing technologies. Over the years, various terms and classifications of SD have been used and have attempted to order and classify this group of genetic diseases according to clinical, radiological, and molecular criteria. In 2019, the Nosology Committee of the International Skeletal Dysplasia Society (ISDS) updated the classification of SD. This new classification divides SD into 42 large groups that include 461 entities. Advances in next-generation sequencing techniques have revolutionized the entire field of genetics, with 437 different genes are currently identified in 426 (92.4%) of SDs. Nosology is a real help for the clinician in establishing a diagnosis as accurately as possible, for the recognition of new diseases while serving as a guide for the interpretation of new genetic variants.

摘要

骨发育不良(SD),也称为骨软骨发育不良(OCD),是一大类骨骼疾病(超过 400 种不同的实体),由骨骼发育和生长的异常引起。SD 根据不同的自然病史、预后、遗传模式和病因发病机制而有所不同。在出生时,发病率较低,每种实体的报道发病率都在 1/5000 左右,但综合来看,发病率估计为 1/5000 出生。命名法是医学科学的一个分支。它涉及疾病和障碍的系统分类。因此,结合临床上不同疾病的目录信息、分子解释和基因型-表型相关性,SD 的分类将更加准确。这对于诊断遗传骨骼疾病的患者非常有用,特别是考虑到新测序技术带来的信息流动。多年来,已经使用了各种术语和 SD 分类,并试图根据临床、放射学和分子标准对这组遗传疾病进行排序和分类。2019 年,国际骨骼发育不良学会(ISDS)的命名法委员会更新了 SD 的分类。这种新的分类将 SD 分为 42 个大组,其中包括 461 个实体。下一代测序技术的进步彻底改变了整个遗传学领域,目前在 426 种(92.4%)SD 中已经鉴定出 437 个不同的基因。命名法为临床医生尽可能准确地建立诊断提供了真正的帮助,有助于识别新疾病,并为解释新的遗传变异提供指导。