Division of Infectious Diseases and Geographic Medicine, Department of Medicine.
Division of Infectious Diseases, Department of Pediatrics.
J Clin Invest. 2022 May 2;132(9). doi: 10.1172/JCI157983.
BACKGROUNDHyaluronan (HA), an extracellular matrix glycosaminoglycan, has been implicated in the pathophysiology of COVID-19 infection, pulmonary hypertension, pulmonary fibrosis, and other diseases, but is not targeted by any approved drugs. We asked whether hymecromone (4-methylumbelliferone [4-MU]), an oral drug approved in Europe for biliary spasm treatment that also inhibits HA in vitro and in animal models, could be repurposed as an inhibitor of HA synthesis in humans.METHODSWe conducted an open-label, single-center, dose-response study of hymecromone in healthy adults. Subjects received hymecromone at 1200 (n = 8), 2400 (n = 9), or 3600 (n = 9) mg/d divided into 3 doses daily, administered orally for 4 days. We assessed safety and tolerability of hymecromone and analyzed HA, 4-MU, and 4-methylumbelliferyl glucuronide (4-MUG; the main metabolite of 4-MU) concentrations in sputum and serum.RESULTSHymecromone was well tolerated up to doses of 3600 mg/d. Both sputum and serum drug concentrations increased in a dose-dependent manner, indicating that higher doses lead to greater exposures. Across all dose arms combined, we observed a significant decrease in sputum HA from baseline after 4 days of treatment. We also observed a decrease in serum HA. Additionally, higher baseline sputum HA levels were associated with a greater decrease in sputum HA.CONCLUSIONAfter 4 days of exposure to oral hymecromone, healthy human subjects experienced a significant reduction in sputum HA levels, indicating this oral therapy may have potential in pulmonary diseases where HA is implicated in pathogenesis.TRIAL REGISTRATIONClinicalTrials.gov NCT02780752.FUNDINGStanford Medicine Catalyst, Stanford SPARK, Stanford Innovative Medicines Accelerator program, NIH training grants 5T32AI052073-14 and T32HL129970.
透明质酸(HA)是细胞外基质糖胺聚糖的一种,它与 COVID-19 感染、肺动脉高压、肺纤维化和其他疾病的病理生理学有关,但没有任何已批准的药物针对它。我们想知道 hymecromone(4-甲基伞形酮[4-MU])是否可以被重新用于人类 HA 合成的抑制剂。方法:我们在健康成年人中进行了 hymecromone 的开放性、单中心、剂量反应研究。受试者每天口服 3 次,每次 1200(n = 8)、2400(n = 9)或 3600(n = 9)mg/d,连续 4 天。我们评估了 hymecromone 的安全性和耐受性,并分析了痰和血清中的 HA、4-MU 和 4-甲基伞形基葡萄糖醛酸(4-MUG;4-MU 的主要代谢物)浓度。结果:hymecromone 最高剂量达 3600mg/d 时耐受性良好。痰和血清中的药物浓度均呈剂量依赖性增加,表明高剂量会导致更高的暴露量。在所有剂量组中,我们观察到治疗 4 天后痰 HA 从基线显著下降。我们还观察到血清 HA 下降。此外,较高的基线痰 HA 水平与痰 HA 下降幅度更大相关。结论:健康受试者在接受 hymecromone 口服治疗 4 天后,痰 HA 水平显著降低,表明这种口服疗法可能对涉及 HA 发病机制的肺部疾病具有潜在疗效。试验注册:ClinicalTrials.gov NCT02780752。资金:斯坦福医学催化剂、斯坦福 SPARK、斯坦福创新药物加速计划、NIH 培训补助金 5T32AI052073-14 和 T32HL129970。