Climate warming poses a serious threat to alpine-restricted species worldwide, yet few studies have empirically documented climate-induced changes in distributions. The rare stonefly, Zapada glacier (Baumann and Gaufin), endemic to alpine streams of Glacier National Park (GNP), Montana, was recently petitioned for listing under the US Endangered Species Act because of climate-change-induced glacier loss, yet little was known about its current status and distribution. We resampled streams throughout the historical distribution of Z. glacier to investigate trends in occurrence associated with changes in temperature and glacial extent. The current geographic distribution of the species was assessed using morphological characteristics of adults and DNA barcoding of nymphs. Bayesian phylogenetic analysis of mtDNA data revealed 8 distinct clades of the genus corresponding with 7 known species from GNP, and one potentially cryptic species. Climate model simulations indicate that average summer air temperature increased (0.67–1.00°C) during the study period (1960–2012), and glacial surface area decreased by ∼35% from 1966 to 2005. We detected Z. glacier in only 1 of the 6 historically occupied streams and at 2 new locations in GNP. These results suggest that an extremely restricted historical distribution of Z. glacierin GNP has been further reduced over the past several decades by an upstream retreat to higher, cooler sites as water temperatures increased and glacial masses decreased. More research is urgently needed to determine the status, distribution, and vulnerability of Z. glacier and other alpine stream invertebrates threatened by climate change in mountainous ecosystems.