Komissarov Artem E, Agranovich Olga E, Kuchinskaia Ianina A, Tkacheva Irina V, Bolshakova Olga I, Latypova Evgenia M, Batkin Sergey F, Sarantseva Svetlana V
Petersburg Nuclear Physics Institute Named by B.P. Konstantinov of National Research Centre "Kurchatov Institute", Gatchina 188300, Russia.
H. Turner National Medical Research Center for Children's Orthopedics and Trauma Surgery, Saint Petersburg 196603, Russia.
Int J Mol Sci. 2024 Dec 26;26(1):124. doi: 10.3390/ijms26010124.
Arthrogryposis, which represents a group of congenital disorders, includes various forms. One such form is amyoplasia, which most commonly presents in a sporadic form in addition to distal forms, among which hereditary cases may occur. This condition is characterized by limited joint mobility and muscle weakness, leading to limb deformities and various clinical manifestations. At present, the pathogenesis of this disease is not clearly understood, and its diagnosis is often complicated due to significant phenotypic diversity, which can result in delayed detection and, consequently, limited options for symptomatic treatment. In this study, a transcriptomic analysis of the affected muscles from patients diagnosed with amyoplasia was performed, and more than 2000 differentially expressed genes (DEGs) were identified. A functional analysis revealed disrupted biological processes, such as vacuole organization, cellular and aerobic respiration, regulation of mitochondrion organization, cellular adhesion, ATP synthesis, and others. The search for key nodes (hubs) in protein-protein interaction networks allowed for the identification of genes involved in mitochondrial processes.
先天性多发性关节挛缩症是一组先天性疾病,包括多种形式。其中一种形式是先天性肌发育不全,除了远端型外,最常见的是以散发性形式出现,其中可能会出现遗传性病例。这种疾病的特征是关节活动受限和肌肉无力,导致肢体畸形和各种临床表现。目前,这种疾病的发病机制尚不清楚,由于其显著的表型多样性,其诊断往往很复杂,这可能导致检测延迟,从而对症治疗的选择有限。在本研究中,对诊断为先天性肌发育不全的患者的受累肌肉进行了转录组分析,鉴定出2000多个差异表达基因(DEG)。功能分析揭示了生物过程的紊乱,如液泡组织、细胞和有氧呼吸、线粒体组织调节、细胞粘附、ATP合成等。在蛋白质-蛋白质相互作用网络中寻找关键节点(枢纽)有助于识别参与线粒体过程的基因。