Neurological Sciences

, Volume 39, Issue 12, pp 2217–2220 | Cite as

A case of concurrent diurnal and nocturnal paroxysmal dystonia

  • Shenghua Li
  • Xiaomin Pang
  • Lan Chen
  • Chunyong Chen
  • Jingli LiuEmail author
Letter to the Editor

Dear Editor,

Nocturnal paroxysmal dystonia (NPD), first described by Lugaresi and Cirignotta in 1981, is interpreted as a special short form of sleep-related hypermotor epilepsy and is characterized by abnormal dyskinetic movements occur mostly during NREM sleep and with no ictal EEG activity or a diffuse ictal pattern [1, 2, 3]. To the best of our knowledge, there have been very few reports of NPD with attacks occur mainly after awakening from noon sleep. We experienced a peculiar case of NPD presenting with a series of diurnal dystonic episodes and some nocturnal attacks. This case shows that NPD is still a diagnostic challenge for clinicians.

A 41-year-old right-handed Chinese man with a 36-year history of episodes of involuntary movements was admitted to the Department of Neurology, The First Affiliated Hospital of Guangxi Medical University. He was born at term after an uncomplicated pregnancy and delivery, with an uneventful neonatal course. His early developmental milestones...


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Conflict of interest

The authors declare that they have no conflict of interest.

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  1. 1.
    Lugaresi E, Cirignotta F, Montagna P (1986) Nocturnal paroxysmal dystonia. J Neurol Neurosurg Psychiatry 49(4):375–380CrossRefPubMedPubMedCentralGoogle Scholar
  2. 2.
    Tinuper P, Bisulli F, Provini F, Lugaresi E (2010) Familial frontal lobe epilepsy and its relationship with other nocturnal paroxysmal events. Epilepsia 51(Suppl 1):51–53. CrossRefGoogle Scholar
  3. 3.
    Tinuper P, Bisulli F, Cross JH, Hesdorffer D, Kahane P, Nobili L, Provini F, Scheffer IE, Tassi L, Vignatelli L, Bassetti C, Cirignotta F, Derry C, Gambardella A, Guerrini R, Halasz P, Licchetta L, Mahowald M, Manni R, Marini C, Mostacci B, Naldi I, Parrino L, Picard F, Pugliatti M, Ryvlin P, Vigevano F, Zucconi M, Berkovic S, Ottman R (2016) Definition and diagnostic criteria of sleep-related hypermotor epilepsy. Neurology 86(19):1834–1842. CrossRefPubMedPubMedCentralGoogle Scholar
  4. 4.
    Kim HJ, Yoon JH (2017) A case of Wilson’s disease presenting with paroxysmal dystonia. Neurol Sci 38(10):1881–1882. CrossRefPubMedGoogle Scholar
  5. 5.
    Gu C, Li J, Zhu L, Lu Z, Huang H (2016) Analysis of catechol-O-methyltransferase gene mutation and identification of new pathogenic gene for paroxysmal kinesigenic dyskinesia. Neurol Sci 37(3):377–383. CrossRefPubMedGoogle Scholar
  6. 6.
    Bruno MK, Hallett M, Gwinn-Hardy K, Sorensen B, Considine E, Tucker S, Lynch DR, Mathews KD, Swoboda KJ, Harris J, Soong BW, Ashizawa T, Jankovic J, Renner D, Fu YH, Ptacek LJ (2004) Clinical evaluation of idiopathic paroxysmal kinesigenic dyskinesia: new diagnostic criteria. Neurology 63(12):2280–2287CrossRefPubMedGoogle Scholar
  7. 7.
    Montagna P, Cirignotta F, Giovanardi Rossi P, Lugaresi E (1992) Dystonic attacks related to sleep and exercise. Eur Neurol 32(4):185–189. CrossRefPubMedGoogle Scholar
  8. 8.
    Lugaresi E, Cirignotta F, Montagna P (1991) Nocturnal paroxysmal dystonia. Epilepsy Res Suppl 2:137–140PubMedGoogle Scholar
  9. 9.
    Hirsch E, Sellal F, Maton B, Rumbach L, Marescaux C (1994) Nocturnal paroxysmal dystonia: a clinical form of focal epilepsy. Neurophysiol Clin 24(3):207–217CrossRefPubMedGoogle Scholar

Copyright information

© Springer-Verlag Italia S.r.l., part of Springer Nature 2018

Authors and Affiliations

  1. 1.Department of NeurologyThe First Affiliated Hospital of Guangxi Medical UniversityNanningChina
  2. 2.Department of Internal MedicineThe Second Affiliated Hospital of Guangxi Medical UniversityNanningChina

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