Climate warming in the mid- to high-latitudes and high-elevation mountainous regions is occurring more rapidly than anywhere else on Earth, causing extensive loss of glaciers and snowpack. However, little is known about the effects of climate change on alpine stream biota, especially invertebrates. Here, we show a strong linkage between regional climate change and the fundamental niche of a rare aquatic invertebrate—themeltwater stonefly Lednia tumana—endemic toWaterton- Glacier International Peace Park, Canada and USA. L. tumana has been petitioned for listing under the U.S. Endangered Species Act due to climate-change-induced glacier loss, yet little is known on specifically how climate impacts may threaten this rare species and many other enigmatic alpine aquatic species worldwide. During 14 years of research, we documented that L. tumana inhabits a narrow distribution, restricted to short sections (∼500 m) of cold, alpine streams directly below glaciers, permanent snowfields, and springs. Our simulation models suggest that climate change threatens the potential future distribution of these sensitive habitats and persistence of L. tumana through the loss of glaciers and snowfields. Mountaintop aquatic invertebrates are ideal early warning indicators of climate warming in mountain ecosystems. Research on alpine invertebrates is urgently needed to avoid extinctions and ecosystem change.