The use of botulinum neurotoxins (BoNTs) for therapeutic purposes in neuromuscular disorders and peripheral hypercholinergic conditions as well as in aesthetic medicine is widespread and common. BoNTs are also able to block the release of a wide range of transmitters from presynaptic boutons. Therefore, application of BoNTs directly in the central nervous system (CNS) is currently under study with respect to basic research and potentially as a new therapeutic strategy of neurological diseases. Investigations concentrate on effects of intracerebral and intraspinal application of BoNTs in rodents on the impact on spinal, nuclear, limbic and cortical neuronal circuits. In animal model first promising BoNT-induced therapeutical benefit has been shown in the treatment of pain, epilepsy, stroke and Parkinson's disease.