Gomal Centre of Biochemistry and Biotechnology, Gomal University, Indus Highway, Multan Road, Dera Ismail Khan, 29050, Khyber Pakhtunkhwa, Pakistan.
Translational Research Institute & Qatar Metabolic Institute, Hamad Medical Corporation, Doha, Qatar.
Mol Genet Genomics. 2022 Sep;297(5):1195-1214. doi: 10.1007/s00438-022-01930-1. Epub 2022 Jul 30.
Disorders that result from de-arrangement of growth, development and/or differentiation of the appendages (limbs and digit) are collectively called as inherited abnormalities of human appendicular skeleton. The bones of appendicular skeleton have central role in locomotion and movement. The different types of appendicular skeletal abnormalities are well described in the report of "Nosology and Classification of Genetic skeletal disorders: 2019 Revision". In the current article, we intend to present the embryology, developmental pathways, disorders and the molecular genetics of the appendicular skeletal malformations. We mainly focused on the polydactyly, syndactyly, brachydactyly, split-hand-foot malformation and clubfoot disorders. To our knowledge, only nine genes of polydactyly, five genes of split-hand-foot malformation, nine genes for syndactyly, eight genes for brachydactyly and only single gene for clubfoot have been identified to be involved in disease pathophysiology. The current molecular genetic data will help life sciences researchers working on the rare skeletal disorders. Moreover, the aim of present systematic review is to gather the published knowledge on molecular genetics of appendicular skeleton, which would help in genetic counseling and molecular diagnosis.
由于附属物(四肢和手指)的生长、发育和/或分化失调而导致的疾病统称为人类附属骨骼遗传异常。附属骨骼的骨骼在运动和运动中起着核心作用。在“遗传骨骼疾病的命名法和分类:2019 年修订版”的报告中,详细描述了不同类型的附属骨骼异常。在本文中,我们旨在介绍附属骨骼畸形的胚胎发生、发育途径、疾病和分子遗传学。我们主要关注多指畸形、并指畸形、短指畸形、分裂手足畸形和马蹄内翻足畸形。据我们所知,只有九个多指畸形基因、五个分裂手足畸形基因、九个并指畸形基因、八个短指畸形基因和一个马蹄内翻足畸形基因被确定与疾病发病机制有关。目前的分子遗传学数据将帮助从事罕见骨骼疾病研究的生命科学研究人员。此外,本系统评价的目的是收集关于附属骨骼分子遗传学的已发表知识,这将有助于遗传咨询和分子诊断。