Dystonia as a Presenting Feature of Alcohol Withdrawal
Case Report 1
“MR. R,” A 36-year-old man with a 15-year history of alcohol dependence (DSM-IV-TR) presented in July 2010 to the emergency room, after a period of abstinence for more than a year, in a state of withdrawal, with symptoms characterized by restlessness; tremors, after which, he had dystonia in upper limbs, trunk, and neck. His abnormal involuntary movement scale (AIMS) severity score was 4, with awareness and distress. He was immediately started on a trial of promethazine 50 mg in addition to detoxification with lorazepam. The response of dystonia to promethazine was dramatic, and the patient was completely symptom-free within few minutes. The detoxification regimen continued with lorazepam, and withdrawal symptoms subsided completely within 3 days. Mr. R is known to have retroviral infection, with CD4 count of 557, and his liver enzymes were deranged, suggestive of liver disease. Serum phosphorous and calcium levels were normal, and the CPK levels were elevated at the time of dystonia.
Case Report 2
“MR. M,” A 58-year-old man, presented in July 2010 with a 25-year history of alcohol dependence (DSM-IV-TR) in a withdrawal state after abrupt discontinuation of alcohol. Also, he developed symptoms characterized by restlessness, tremors, dragging sensation in his tongue, and deviation of mouth to one side, with stiffening of the tongue, which responded immediately to promethazine and lorazepam injections. He had a significant past history of lingual and nuchal dystonia associated with alcohol withdrawal and had shown a similar response to phenergan and lorazapam.
Case Report 3
“MR. U,” A 43-year-old man, presented in July 2010 with an 18-year history of alcohol dependence (DSM-IV-TR). He had relapsed after a period of abstinence for more than a year. After about 2 days of abrupt cessation of alcohol use, he developed symptoms characterized by restlessness and tremors, after which he had rigidity in both upper limbs. The AIMS severity score was 3, with awareness and distress. He was started on a lorazapam detoxification and trihexyphenydyl. His withdrawal symptoms and extrapyramidal symptoms subsided completely within 15 days. All the above patients had no previous history of neurological diseases, and their clinical neurological examinations were otherwise normal. Biochemical blood investigations, including electrolytes, renal and liver function tests, and blood glucose, were within normal limits. CPK was obtained for the first patient reported and it was high. In the first patient, EEG, computed tomography imaging of the brain, and serum phosphorous and calcium levels revealed no abnormal findings. None of the patients had received any injections containing dopamine-receptor blockers (antipsychotics or antiemetics), and, hence, drug-induced dystonia was ruled out.
Discussion
GABA-ergic neurotransmission is important in shaping plastic responses of the CNS to somatosensory and other stimuli, including the maintenance and plasticity of cortical receptive fields. GABA, along with dopamine, is, therefore,a neurotransmitter most plausibly linked to the electrophysiological mechanisms identified in dystonia.1 Most of the work related to the role of GABA in this condition is reported with primary dystonia. A decreased GABA level selectively in cortical and subcortical regions contralateral to the dystonic limb in task-induced dystonia has been reported. Levy and Hallett2 speculated that deficient GABA levels could be consistent with reduced dopaminergic neurotransmission in the putamen, since D2 dopamine receptors mostly inhibit the GABA-ergic striatal medium spiny neurons. Drugs that potentiate GABA neurotransmission, such as baclofen, are effective against dystonic movements.3 Moreover, application of the GABA antagonist bicuculline to the motor cortex of monkeys causes abnormal co-contraction of agonist and antagonist muscle groups during activities requiring fine motor skills, and produces abnormal movements reminiscent of task-specific dystonia.4 Alcohol withdrawal is a state characterized by an imbalance of GABA-ergic and glutaminergic systems in the brain. Long-term alcohol consumption affects brain receptors, which undergo adaptive changes in an attempt to maintain normal function. Withdrawal state involves reduced brain GABA levels and GABA-receptor sensitivity and activation of glutamate systems, which leads to nervous system hyperactivity in the absence of alcohol.5 Benzodiazepines are an established treatment modality for alcohol withdrawal,6 and, similarly, baclofen has been effectively used both in the treatment of alcohol-withdrawal and dystonia.7 Dystonia can be conceptualized as an epiphenomenon of alcohol withdrawal, as given in this report, due to this shared neurochemical abnormality. To our knowledge, this is the first report of dystonia as a presenting feature of the alcohol-withdrawal state.
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