A case of paroxysmal nocturnal dystonia responsive to cyproheptadine: Is it almost always epileptic?

International Journal of Epilepsy - Tập 2 - Trang 49-50 - 2015
Mohammad Sayadnasiri1
1Department of Clinical Sciences, Razi Hospital, University of Social Welfare and Rehabilitation Sciences, Tehran, Iran

Tóm tắt

AbstractNocturnal Paroxysmal Dystonia (NPD) is characterized by abnormal dyskinetic movements occur mostly during non-REM sleep. From introducing NPD in early 1980s, most authors have been in favour of an epileptic origin for these sleep-related episodes. Clinical characteristics of NPD including abrupt hypermotor behaviours, automatisms and vocalizations, abnormal EEG findings in some patients and therapeutic response to anti-epileptic drugs persuade clinicians to accept NPD as sleep-related epilepsy with frontal lobe origin but it seems this conclusion may not be true in all cases. We present a young adult patient with refractory NPD who responded to antihistamine cyproheptadine and propose an alternative theory to describe NPD according to the basal ganglia dysregulation. So, in such patients, other therapeutic approach should be reasonably sought.

Tài liệu tham khảo

Hirsch, 1994, Nocturnal paroxysmal dystonia: a clinical form of focal epilepsy, Neurophysiol Clin, 24, 207, 10.1016/S0987-7053(05)80185-6 Provini, 2000, From nocturnal paroxysmal dystonia to nocturnal frontal lobe epilepsy, Clin Neurophysiol, 111, S2, 10.1016/S1388-2457(00)00396-5 Meierkord, 1992, Is nocturnal paroxysmal dystonia a form of frontal lobe epilepsy?, Mov Disord, 7, 38, 10.1002/mds.870070107 Williams, 2012, Paroxysmal hypnogenic dyskinesia responsive to doxylamine: a case report, Case Rep Neurol Med, 2012 Lugaresi, 1986, Nocturnal paroxysmal dystonia, J Neurol Neurosurg Psychiatry, 49, 375, 10.1136/jnnp.49.4.375 Haas, 2008, Histamine in the nervous system, Physiol Rev, 88, 1183, 10.1152/physrev.00043.2007