Clinical and epidemiological features of 7 human immunodeficiency virus–negative Peruvian patients coinfected with human T-lymphotropic virus type 1 (HTLV-1) and cryptococcosis (2006–2017) were studied. Most cases had meningeal involvement, were male, and originated from Peru’s jungle. Patients with cryptococcosis should be tested for HTLV-1 in endemic areas of this retrovirus. © The Author(s) 2024.