Isolated abducens nerve palsy following lumbar puncture is a rare complication. In scientific literature, it has been mostly described after performing spinal anesthesia in anesthesiology literature rather than in neurological literature. Isolated abducens nerve palsy usually occurs from one to three weeks after performing lumbar puncture and it is important to connect it with this procedure and exclude other possible etiologies. The actual incidence of this complication is unknown. We describe a 32-year-old male patient who presented with unspecific headache attributed to cavernous malformation and two developmental venous anomalies. While searching for headache etiology, lumbar puncture was performed. Two days after lumbar puncture, the patient presented with isolated abducens palsy of the right eye. Our planned diagnostic evaluation included measuring intracranial pressure by noninvasive optic sheath nerve sonography and repeating brain magnetic resonance imaging. We also planned to treat the patient with epidural blood patch, which is usually successful when performed as soon as the diagnosis is established. Unfortunately, our patient refused diagnostic and therapeutic proposal, so it was impossible to know final outcome. In conclusion, we want to highlight these complications, especially to young doctors and specialists who might see it rarely.