Improvement of post-hypoxic action myoclonus with levetiracetam add-on therapy: A case report

Ksenija Božić; Ksenija Gebauer-Bukurov; Lorand Sakalaš; Ivana Divjak; Aleksandar Ješić

doi:10.2298/1343

Ksenija Božić Clinic for Neurology, Clinical Center of Vojvodina, Novi Sad, Serbia
Ksenija Gebauer-Bukurov Clinic for Neurology, Clinical Center of Vojvodina, Novi Sad, Serbia
Lorand Sakalaš Clinic for Neurology, Clinical Center of Vojvodina, Novi Sad, Serbia
Ivana Divjak Clinic for Neurology, Clinical Center of Vojvodina, Novi Sad, Serbia
Aleksandar Ješić Clinic for Neurology, Clinical Center of Vojvodina, Novi Sad, Serbia

DOI: https://doi.org/10.2298/1343

Keywords: myoclonus, anoxia, syndrome, diagnosis, drug therapy, drug resistance, treatment outcome,

Abstract

Introduction. Chronic post-anoxic myoclonus, also known as Lance-Adams syndrome, may develop following hypoxic brain injury, and is resistant to pharmacological therapy. Case report. The patient we presented developed post-anoxic action myoclonus with severe, completely incapacitating myoclonic jerks. Myoclonus did not respond to the treatment with commonly used agents, i.e. valproate and clonazepam alone or in combination. Improvement of the action myoclonus was observed only after adding levetiracetam. Conclusion. Although Lance-Adams syndrome may not be fully curable at this point, levetiracetam appears to be a promising agent that can significantly improve functional level and overall quality of life of patients with this disorder.

References

Lance JW, Adams RD. The syndrome of intention or action myoclonus as a sequel to hypoxic encephalopathy. Brain 1963; 86: 111−36.

Werhahn KJ, Brown P, Thompson PD, Marsden CD. The clinical features and prognosis of chronic posthypoxic myoclonus. Mov Disord 1997; 12(2): 216−20.

Borg M. Symptomatic myoclonus. Neurophysiol Clin 2006; 36(5−6): 309−18.

Frucht S, Fahn S. The clinical spectrum of posthypoxic myoc-lonus. Mov Disord 2000; 15(Suppl 1): 2−7.

Hallett M, Chadwick D, Adams J, Marsden CD. Reticular reflex myoclonus: a physiological type of human post-hypoxic myoclonus. J Neurol Neurosurg Psychiatry 1977; 40(3): 253−64.

Frucht SJ. The clinical challenge of posthypoxic myoclonus. Adv Neurol 2002; 89: 85–8.

Lance JW, Adams RD. Negative myoclonus in post-hypoxic pa-tients: historical note. Mov Disor 2001; 16(1): 162−3.

Truong DD, Kirby M, Kanthasamy A, Matsumoto RR. Posthypoxic myoclonus animal models. Adv Neurol 2002; 89: 295−306.

Matsumoto RR, Truong DD, Nguyen KD, Dang AT, Hoang TT, Vo PQ, et al. Involvement of GABA(A) receptors in myoclonus. Mov Disord 2000; 15 (Suppl 1): 47−52.

Tai KK, Truong DD. Post-hypoxic myoclonus induces Fos ex-pression in the reticualr thalamic nuclei and neurons in brains-tem. Brain Res 2005; 1059(2): 122−8.

Frucht SJ, Trost M, May Y, Eidelberg D. The metabolic topogra-phy of posthypoxic myoclonus. Neurology 2004; 62(10): 1879−81.

Witte OW, Niedermeyer E, Arendt G, Freund HJ. Post-hypoxic ac-tion (intention) myoclonus: a clinico-electroencephalographic study. J Neurol 1988; 235(4): 214−8.

Rollinson RD, Gilligan BS. Postanoxic action myoclonus (Lance Adams syndrome) responding to valproate. Arch Neurol 1979; 36(1): 44−5.

Frucht SJ. Myoclonus. Curr Treat Options Neurol 2000; 2(3): 231−42.

Polesin A, Stern M. Post-anoxic myoclonus: A case presentation and review of management in the rehabilitation setting. Brain Inj 2006; 20(2): 213−7.

Terwinghe G, Daumerie J, Nicaise C, Rosillon O. Therapeutic effect of piracetam in a case of posthypoxic action myoclonus. Acta Neurol Belg 1978; 8(1): 30−6. (French)

Obeso JA, Artieda J, Quinn N, Rothwell JC, Luquin MR, Vaamonde J, et al. Piracetam in the treatment of different types of myoc-lonus. Clin Neuropharmacol 1988; 11(6): 529−36.

Genton P, Guerrini R, Remy C. Piracetam in the treatment of cor-tical myoclonus. Pharmacopsychiatry 1999; 32(Suppl 1): 49−53.

Janszky J, Holló A, Halász P. Treatment and long term follow-up of post-anoxic myoclonus. Orv Hetil 2001; 142(38): 2091−3. (Hungarian)

Genton P, Gelisse P. Antimyoclonic effect of levetiracetam. Epi-leptic Disord 2000; 2(4): 209−12.

Lim LL, Ahmed A. Limited efficacy of levetiracetam on myoc-lonus of different etiologies. Parkinsonism Relat Disord 2005; 11(2): 135−7.

Gelisse P, Crespel A, Genton P, Baldy-Moulinier M. Dramatic effect of levetiracetam on epileptic negative myoclonus. Acta Neurol Scand 2003; 107 (4): 302−3.

Yu HY, Kwan SY, Lirng JF, Liao KK, Chu YK, Liao SQ. Epilep-tic negative myoclonus: SPECT, PET, and video/EEG studies and the dramatic effects of levetiracetam. Epilepsy Behav 2009; 14(4): 687−90.

Krauss GL, Bergin A, Kramer RE, Cho YW, Reich SG. Suppres-sion of post-hypoxic and post-encephalitic myoclonus with le-vetiracetam. Neurology 2001; 56(3): 411−2.

Genton P, Gelisse P. Suppression of posthypoxic and post-encephalitic myoclonus with levetiracetam. Neurology 2001; 57(6): 1144−5.

Venot M, Weiss N, Espinoza S, Imbert A, Tadie JM, Fagon JY, et al. Improvement of early diagnosed post-anoxic myoclonus with levetiracetam. Intensive Care 2011; 37(1): 177−9.