Erethism describes severe cases of unprovoked aggressive behavior, usually associated with some degree of mental impairment and gross brain damage. The etiology can be epileptic, postencephalitic, or posttraumatic, or the condition can be caused by brain malformations or perinatal insults. Erethism is often refractory to medication, and patients must often be interned in institutions, where they are managed with major restraining measures. The hypothalamus is a crucial group of nuclei that coordinate behavioral and autonomic responses and play a central role in the control of aggressive behavior. Deep brain stimulation (DBS) of the posteromedial hypothalamus (PMH) has been proposed as a treatment for resistant erethism, although experience with this treatment around the world is scarce. The objective of this study was to examine the long-term outcome of PMH DBS in 6 patients with severe erethism treated at the authors' institution.
Medical records of 6 patients treated with PMH DBS for intractable aggressiveness were reviewed. The therapeutic effect on behavior was assessed by the Inventory for Client and Agency Planning preoperatively and at the last follow-up visit.
Two patients died during the follow-up period due to causes unrelated to the neurosurgical treatment. Five of 6 patients experienced a significant reduction in aggressiveness (the mean Inventory for Client and Agency Planning general aggressiveness score was −47 at baseline and −25 at the last follow-up; mean follow-up 3.5 years). Similar responses were obtained with low- and high-frequency stimulation. In 4 cases, the patients' sleep patterns became more regular, and in 1 case, binge eating and polydipsia ceased. One of the 3 patients who had epilepsy noticed a 30% reduction in seizure frequency. Another patient experienced a marked sympathetic response with high-frequency stimulation during the first stimulation trial, but this subsided when stimulation was set at low frequency. A worsening of a previous headache was noted by 1 patient. There were no other side effects.
In this case series, 5 of 6 patients with pathological aggressiveness had a reduction of their outbursts of violence after PMH DBS, without significant adverse effects. Prospective controlled studies with a larger number of patients are needed to confirm these results.
Abbreviations used in this paper:AVM = arteriovenous malformation; DBS = deep brain stimulation; EEG = electroencephalography; ICAP = Inventory for Client and Agency Planning; OCD = obsessive-compulsive disorder; PMH = posteromedial hypothalamus.
Address correspondence to: Cristina V. Torres Díaz, M.D., Ph.D., Department of Neurosurgery, University Hospital La Princesa, C/Diego de León 32, 28006 Madrid, Spain. email: firstname.lastname@example.org.
Please include this information when citing this paper: published online June 7, 2013; DOI: 10.3171/2013.4.JNS121639.
BallangerBLozanoAMMoroEvan EimerenTHamaniCChenR: Cerebral blood flow changes induced by pedunculopontine nucleus stimulation in patients with advanced Parkinson's disease: a [(15)O] H2O PET study. Hum Brain Mapp30:3901–39092009
BruininksRHGilmanCJLa observación y el currículum de las destrezas de conducta adaptativa: dos instrumentos de evaluación y ayuda en la intervención. BruininksRH: Conducta Adaptativa y DiscapacidadBilbaoICE de la Universidad de Deusto-Mensajero1993
CostaACarlesimoGACaltagironeCMazzonePPierantozziMStefaniA: Effects of deep brain stimulation of the peduncolopontine area on working memory tasks in patients with Parkinson's disease. Parkinsonism Relat Disord16:64–672010
DuprezTPSeriehBARaftopoulosC: Absence of memory dysfunction after bilateral mammillary body and mammillothalamic tract electrode implantation: preliminary experience in three patients. AJNR Am J Neuroradiol26:195–1982005. (Letter)
HernandoVPastorJPedrosaMPeñaESolaRG: Low-frequency bilateral hypothalamic stimulation for treatment of drug-resistant aggressiveness in a young man with mental retardation. Stereotact Funct Neurosurg86:219–2232008
KuhnJLenartzDMaiJKHuffWKlosterkoetterJSturmV: Disappearance of self-aggressive behavior in a brain-injured patient after deep brain stimulation of the hypothalamus: technical case report. Neurosurgery62:E11822008
SeijoFSaizALozanoBSantamartaEAlvarez-VegaMSeijoE: Neuromodulation of the posterolateral hypothalamus for the treatment of chronic refractory cluster headache: experience in five patients with a modified anatomical target. Cephalalgia31:1634–16412011