Botia-Arciniegas Valentina, Jimenez-Cardozo Natalia, Lores Juliana
Department of Basic Sciences, Faculty of Health Sciences.
Grupo de Investigación en Ciencias Básicas y Clínicas de la Salud, Pontificia Universidad Javeriana Cali, Colombia.
Pharmacogenet Genomics. 2025 Aug 1;35(6):173-182. doi: 10.1097/FPC.0000000000000569. Epub 2025 Jun 4.
RASopathies are rare genetic disorders caused by germline mutations in genes that regulate the RAS-mitogen-activated protein kinase (MAPK) pathway, a critical pathway involved in various cellular processes. Disruption of this pathway leads to multisystemic manifestations, including cardiomyopathies, a cause of high morbi-mortality. In response to the urgent need to improve survival in patients with RASopathies, alternative therapies, such as MAPK inhibitors traditionally used in cancer treatment, have been explored. This article reviews the current evidence on the use of these medications in treating cardiomyopathies associated with RASopathies. The search was conducted in the PubMed, Scopus, and Embase databases identifying nine studies reporting a total of 14 cases (nine with Noonan syndrome and five with Costello syndrome) where patients were successfully treated with trametinib, a MEK inhibitor. This therapeutic alternative broadens the horizons of opportunity for patients who often face limited options for enhancing their quality of life. Therefore, it is important to prioritize ongoing research in this field, focusing not only on further investigation on trametinib, but also exploring other potential therapeutic approaches.
RAS 病是由调节 RAS-丝裂原活化蛋白激酶(MAPK)途径的基因种系突变引起的罕见遗传疾病,该途径是参与各种细胞过程的关键途径。该途径的破坏会导致多系统表现,包括心肌病,这是高病亡率的一个原因。为了满足改善 RAS 病患者生存率的迫切需求,人们探索了替代疗法,例如传统上用于癌症治疗的 MAPK 抑制剂。本文综述了目前关于使用这些药物治疗与 RAS 病相关的心肌病的证据。在 PubMed、Scopus 和 Embase 数据库中进行了检索,确定了 9 项研究,共报告了 14 例病例(9 例努南综合征和 5 例科斯特洛综合征),这些患者使用 MEK 抑制剂曲美替尼成功治疗。这种治疗选择为那些常常在提高生活质量方面面临有限选择的患者拓宽了机会视野。因此,重要的是要优先开展该领域的 ongoing 研究,不仅要进一步研究曲美替尼,还要探索其他潜在的治疗方法。