Background: Obsessive-compulsive disorder (OCD) is characterized by intrusive and irrational thoughts as well as repetitive behaviours. OCD-like behaviours have been described in a wide range of neurological disorders. In cerebrovascular accidents, the semiology arises mostly from lesions to the basal ganglia – though cortical regions may also be involved. In the past few years, the mechanisms underlying OCD in psychiatric patients have been re- examined, in particular the functional relationship between anxiety, obsessions and compulsions. Traditionally, obsessions are viewed as a trigger for compulsive behaviour that represents an attempt to reduce anxiety. By contrast, other models place compulsions – as a manifestation of an imbalance between goal-directed action and automatic habits that leads to maladaptive habit learning – at the core of OCD. CASE: We show neurological evidence of pure compulsions without obsession in a patient following stroke in the left subcortical regions. Furthermore, we present comprehensive neuropsychological findings that identify specific alterations across executive and emotional domains. Finally, MRI analyses reveal that the subcortical stroke had resulted in a strong decrease of connectivity suggestive of large network alterations. CONCLUSIONS: Our case provides direct information on how brain structure and function relate in an OCD patient, highlighting the central role of compulsions in the pathology.