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LETTER TO EDITOR
Year : 2022  |  Volume : 70  |  Issue : 4  |  Page : 1720--1721

Delayed Cervical Dystonia with Tremors in a Patient with Wernicke Encephalopathy: An Expansion of Complication

Rohan R Mahale, Ameya Patwardan, Pooja Mailankody, Hansashree Padmanabha, PS Mathuranath 
 Department of Neurology, National Institute of Mental Health and Neurosciences (NIMHANS), Bangalore, Karnataka, India

Correspondence Address:
Rohan R Mahale
Department of Neurology, National Institute of Mental Health and Neurosciences (NIMHANS), Bangalore - 560 029, Karnataka
India




How to cite this article:
Mahale RR, Patwardan A, Mailankody P, Padmanabha H, Mathuranath P S. Delayed Cervical Dystonia with Tremors in a Patient with Wernicke Encephalopathy: An Expansion of Complication.Neurol India 2022;70:1720-1721


How to cite this URL:
Mahale RR, Patwardan A, Mailankody P, Padmanabha H, Mathuranath P S. Delayed Cervical Dystonia with Tremors in a Patient with Wernicke Encephalopathy: An Expansion of Complication. Neurol India [serial online] 2022 [cited 2022 Nov 30 ];70:1720-1721
Available from: https://www.neurologyindia.com/text.asp?2022/70/4/1720/355168


Full Text



Sir,

Wernicke's encephalopathy (WE) is a neuropsychiatric emergency having high morbidity (84%) and mortality (up to 20%).[1] It was first reported in 1881 by the German neuropsychiatrist Carl Wernicke as a syndrome of acute mental status change with associated ophthalmoplegia and ataxia.[2] Thiamine (vitamin B1) deficiency was discovered to be responsible for WE, decades after Wernicke's initial report. Movement disorders as a complication of WE are rarely reported. There are two reports of dystonia in patients with WE. Here, we report a patient who underwent gastrojejunostomy for gastroduodenal ulcer and developed WE in the postoperative period. After three months he developed cervical dystonia with dystonic tremors.

A 30-year-old male had a history of pain abdomen and tenderness in epigastrium. He was evaluated with upper gastrointestinal endoscopy in other hospital which showed gastroduodenal ulcer. He underwent gastrojejunostomy procedure for the same. On postoperative day 3, he developed progressive decrease in sensorium. Serum electrolytes were normal. Brain magnetic resonance imaging (MRI) showed T2 and fluid-attenuated inversion recovery hyperintensities in bilateral medial thalamus, periaqueductal area of the midbrain tectum, upper medulla suggestive of WE [Figure 1]. He was treated with intravenous thiamine supplementation and he had gradual improvement in sensorium but had gaze-evoked horizontal nystagmus, ophthalmoplegia, and ataxia. After 3months, he developed head tremors with involuntary head turn to left which was insidious on the onset and progressive causing pain and disability. He presented to us after 6 months of surgery. He was not prescribed levosulpride for his gastric complaints. On examination, he had normal cognition and speech. Fundus was normal. There was mild horizontal bilateral gaze-evoked nystagmus. Extraocular movements were normal. Motor and sensory examination was normal except mild impairment in tandem gait. No limb incoordination. Plantar responses were flexor. There was left rotational torticollis with dystonic “no-no” head tremors [Video 1]. A possibility of secondary dystonia due to the thalamic lesion was considered. There was no Kayser-Fleischer ring on slit-lamp examination. Serum copper, ceruloplasmin, ferritin, and 24-h urine copper were normal. There were no acanthocytes on peripheral smear examination. Ultrasonography of the abdomen was normal. He was treated with trihexyphenidyl and clonazepam. He made significant improvement over 4 months [Video 2].{Figure 1}

WE is caused by thiamine deficiency. Alcohol abuse remains the most common (up to 90%).[3] Other causes include gastrointestinal disorders and surgeries, hemodialysis, malignancies, infections, shock, prolonged nutritional deficiencies, or the administration of intravenous glucose prior to thiamine in malnourished or alcohol-dependent individuals.[4] The brain structures that are more vulnerable to damage from thiamine deficiency include the thalamus, mammillary bodies, periaqueductal and paraventricular areas, locus ceruleus, cranial nerves' nuclei, and reticular formation.[5] Lesions of the cortico-striato-pallido-thalamo-cortical and cerebello-thalamo-cortical pathways cause dystonia.[6] Thalamic lesions can cause focal, segmental dystonia, and hemidystonia. Moodley et al. (1989) reported an Indian man with WE who had marked dystonia and choreoathetosis during presentation which was thiamine responsive.[7] Yoon et al. (2008) presented a 23-year-old male patient who developed a progressive dystonic hand tremor following intravenous thiamine treatment for WE, thus suggesting movement disorders as a complication of WE.[8] These two cases had dystonia as a presenting manifestation of WE and appeared during treatment.

Our patient underwent gastrojejunostomy for gastroduodenal ulcer and developed WE in the postoperative period. He was promptly replenished with intravenous thiamine with improvement in consciousness. Brain MRI showed lesions in bilateral medial thalamus, periaqueductal region of midbrain, and upper medulla. However, he developed cervical dystonia with dystonic head tremors as delayed complication of WE.

The occurrence of movement disorders in WE is very rare. The occurrence of cervical dystonia in our patient is likely due to the involvement of the thalamus. This case adds to our knowledge regarding the occurrence of movement disorders in WE as presenting symptoms, during treatment initiation with thiamine or as a delayed complication.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

References

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