Introduction: A syndrome of headache with neurologic deficits and cerebrospinal fluid (CSF) lymphocytosis is uncommon and clinicians should be aware of this entity.
Exegesis: We report a 28-year-old man without previous medical history of migraine, who presented with severe headache and temporary focal, neurological deficits. Lumbar puncture revealed aseptic lymphocytic pleiocytosis. The patient completely recovered within one month. This condition was suggestive of a transient syndrome of headache with neurologic deficits and lymphocytosis. The main characteristics and the pathophysiology of this uncommon disorder, generally with a benign course, are discussed.
Conclusion: Such syndrome of headache, neurologic deficits and CSF lymphocytosis should be included in the differential diagnosis of meningo-encephalitis. The constant benign course of this affection should be emphasized.