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DOI: 10.1016/j.ijep.2015.02.003
A case of paroxysmal nocturnal dystonia responsive to cyproheptadine: Is it almost always epileptic?
Publikationsverlauf
Received: 16. Oktober 2014
Accepted: 15. Februar 2015
Publikationsdatum:
05. Mai 2018 (online)
![](https://www.thieme-connect.de/media/10.1055-s-00037682/201501/lookinside/thumbnails/10-1016-j-ijep-2015-02-003-1.jpg)
Abstract
Nocturnal 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.
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References
- 1 Hirsch E, Sellal F, Maton B, Rumbach L, Marescaux C. Nocturnal paroxysmal dystonia: a clinical form of focal epilepsy. Neurophysiol Clin 24 1994; 207-217
- 2 Provini F, Plazzi G, Lugaresi E. From nocturnal paroxysmal dystonia to nocturnal frontal lobe epilepsy. Clin Neurophysiol 111 Suppl. (02) 2000; S2-S8
- 3 Meierkord H, Fish DR, Smith SJM, Scott CA, Shorvon SD, Marsden CD. Is nocturnal paroxysmal dystonia a form of frontal lobe epilepsy?. Mov Disord 7 1992; 38-42
- 4 Williams DM. Paroxysmal hypnogenic dyskinesia responsive to doxylamine: a case report. Case Rep Neurol Med 2012 2012; 484689
- 5 Lugaresi E, Cirignotta F, Montagna P. Nocturnal paroxysmal dystonia. J Neurol Neurosurg Psychiatry 49 1986; 375-380
- 6 Haas HL, Sergeeva OL, Selbach O. Histamine in the nervous system. Physiol Rev 88 2008; 1183-1241