Deep brain stimulation as a palliative treatment for myorhythmia: A case of failure.
Guillain-Mollaret triangle
deep brain stimulation
myorhythmia
tremor
Journal
European journal of neurology
ISSN: 1468-1331
Titre abrégé: Eur J Neurol
Pays: England
ID NLM: 9506311
Informations de publication
Date de publication:
03 2022
03 2022
Historique:
received:
02
11
2021
accepted:
29
11
2021
entrez:
10
2
2022
pubmed:
11
2
2022
medline:
5
4
2022
Statut:
ppublish
Résumé
Myorhythmia is a hyperkinetic movement disorder that derives from a disruption of the Guillain-Mollaret triangle, due to an identifiable structural lesion. It is often disabling and with disappointing control under medical treatment. Herein, a case of myorhythmia secondary to a vascular insult in the brainstem is reported and an unsuccessful attempt to palliate it with functional neurosurgery. A 67-year-old man displayed a repetitive, rhythmic, slow 2-3 Hz movement, 6 months after suffering a pontomesencephalic hypertensive haematoma. The kinetic phenomenon affected the orbicular and low facial muscles, the neck, the thorax and the upper limbs. Furthermore, he exhibited tremor of the soft palate and pendular nystagmus. On T2-weighted magnetic resonance imaging, hypertrophic degeneration of the inferior olivary complex was seen. He was diagnosed with secondary myorhythmia and multiple pharmacological treatments were tested, but failed. Ultimately, deep brain stimulation with bilateral electrodes placed in the thalamic ventralis intermedius nucleus was offered. Unfortunately, no alleviation of the symptoms was achieved other than mild improvement in involuntary eye movements. This is the first case to report the use of deep brain stimulation for myorhythmia. Better understanding of the pathophysiology of this condition, and localization of the pacemaker, may allow identification of reliable neurosurgical therapeutic targets.
Sections du résumé
BACKGROUND AND PURPOSE
Myorhythmia is a hyperkinetic movement disorder that derives from a disruption of the Guillain-Mollaret triangle, due to an identifiable structural lesion. It is often disabling and with disappointing control under medical treatment.
METHODS
Herein, a case of myorhythmia secondary to a vascular insult in the brainstem is reported and an unsuccessful attempt to palliate it with functional neurosurgery.
RESULTS
A 67-year-old man displayed a repetitive, rhythmic, slow 2-3 Hz movement, 6 months after suffering a pontomesencephalic hypertensive haematoma. The kinetic phenomenon affected the orbicular and low facial muscles, the neck, the thorax and the upper limbs. Furthermore, he exhibited tremor of the soft palate and pendular nystagmus. On T2-weighted magnetic resonance imaging, hypertrophic degeneration of the inferior olivary complex was seen. He was diagnosed with secondary myorhythmia and multiple pharmacological treatments were tested, but failed. Ultimately, deep brain stimulation with bilateral electrodes placed in the thalamic ventralis intermedius nucleus was offered. Unfortunately, no alleviation of the symptoms was achieved other than mild improvement in involuntary eye movements.
CONCLUSIONS
This is the first case to report the use of deep brain stimulation for myorhythmia. Better understanding of the pathophysiology of this condition, and localization of the pacemaker, may allow identification of reliable neurosurgical therapeutic targets.
Types de publication
Case Reports
Langues
eng
Sous-ensembles de citation
IM
Pagination
937-941Informations de copyright
© 2021 European Academy of Neurology.
Références
Masucci EF, Kurtzke JF, Saini N. Myorhythmia: a widespread movement disorder: clinico pathological correlations. Brain. 1984;107:53-79.
Ure RJ, Dhanju S, Lang AE, Fasano A. Unusual tremor syndromes: know in order to recognise. J Neurol Neurosurg Psychiatry. 2016;87(11):1191-1203.
Baizabal-Carvallo JF, Cardoso F, Jankovic J. Myorhythmia: phenomenology, etiology, and treatment. Mov Disord. 2015;30(2):171-179.
Carvalho CH, Kimmig H, Lopez WO, Lange M, Oeckler R. Hypertrophic olivary degeneration: a neurosurgical point of view. J Neurol Surg A Cent Eur Neurosurg. 2016;77(1):59-62.
Wang D, Sanchez J, Foote KD, et al. Failed DBS for palliation of visual problems in a case of oculopalatal tremor. Parkinsonism Relat Disord. 2009;15(1):71-73.