Hughes Alysia N, Li Xing, Lehman Julia S, Nelson Steven A, DiCaudo David J, Mudappathi Rekha, Hwang Angelina, Kechter Jacob, Pittelkow Mark R, Mangold Aaron R, Sekulic Aleksandar
Department of Dermatology, Mayo Clinic, Scottsdale, Arizona, USA.
Department of Dermatology, Mayo Clinic, Rochester, Minnesota, USA.
JID Innov. 2024 Jun 26;4(6):100296. doi: 10.1016/j.xjidi.2024.100296. eCollection 2024 Nov.
Drug repurposing is an attractive strategy for therapy development, particularly in rare diseases where traditional drug development approaches may be challenging owing to high cost and small numbers of patients. In this study, we used a drug identification and repurposing pipeline to identify candidate targetable drivers of disease and corresponding therapies through application of causal reasoning using a combination of open-access resources and transcriptomics data. We optimized our approach on psoriasis as a disease model, demonstrating the ability to identify known and, to date, unrecognized molecular drivers of psoriasis and link them to current and emerging therapies. Application of our approach to a cohort of tissue samples of necrobiosis lipoidica (an unrelated; rare; and, to date, molecularly poorly characterized cutaneous inflammatory disorder) identified a unique set of upstream regulators, particularly highlighting the role of IFNG and the Jak-signal transducer and activator of transcription pathway as a likely driver of disease pathogenesis and linked it to Jak inhibitors as potential therapy. Analysis of an independent cohort of necrobiosis lipoidica samples validated these findings, with the overall agreement of drug-matched upstream regulators above 96%. These data highlight the utility of our approach in rare diseases and offer an opportunity for drug discovery in other rare diseases in dermatology and beyond.
药物重新利用是一种颇具吸引力的治疗开发策略,尤其在罕见病领域,由于成本高昂且患者数量较少,传统药物开发方法可能面临挑战。在本研究中,我们使用了一种药物识别和重新利用流程,通过结合开放获取资源和转录组学数据应用因果推理,来识别疾病的候选可靶向驱动因素及相应疗法。我们以银屑病作为疾病模型对方法进行了优化,证明了能够识别银屑病已知的以及迄今未被认识的分子驱动因素,并将它们与现有和新兴疗法联系起来。将我们的方法应用于一组类脂质渐进性坏死组织样本(一种不相关的、罕见的且迄今分子特征尚不明确的皮肤炎症性疾病),确定了一组独特的上游调节因子,特别突出了IFNG以及Jak信号转导子和转录激活子通路作为疾病发病机制可能驱动因素的作用,并将其与Jak抑制剂作为潜在疗法联系起来。对另一组独立的类脂质渐进性坏死样本的分析验证了这些发现,药物匹配的上游调节因子总体一致性超过96%。这些数据凸显了我们的方法在罕见病中的实用性,并为皮肤病学及其他领域的其他罕见病药物发现提供了机会。