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| CASE REPORT |
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| Year : 2022 | Volume
: 5
| Issue : 1 | Page : 65-67 |
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Levosulpiride-induced arm-raising stereotypy in a 65-year-old man: an unusual tardive phenomenon
Pankaj A Agarwal1, Jimmy A Lalkaka2
1 Movement Disorders Clinic, Department of Neurology, Global Hospitals and Department of Neurology, KEM Hospital and Seth GS Medical College, Mumbai 400012, Maharashtra, India
2 Department of Neurology, Bombay Hospital Institute of Medical Sciences, Mumbai 400020, Maharashtra, India
| Date of Submission | 21-Jan-2021 |
| Date of Decision | 19-Feb-2021 |
| Date of Acceptance | 10-May-2021 |
| Date of Web Publication | 14-Nov-2021 |
Correspondence Address:
Dr. Pankaj A Agarwal
Room 213, Movement Disorders Clinic, Department of Neurology, Global Hospitals, Dr Ernest Borges Road, Parel, Mumbai 400012
India
 Source of Support: None, Conflict of Interest: None
DOI: 10.4103/AOMD.AOMD_2_21
A 65-year-old man presented to us with a 6-month history of recurrent, bilateral, purposeless, arm-raising and lowering movements that fulfilled all criteria for stereotypy. He was on a dopamine blocking agent levosulpiride before the onset of the movements. After the diagnosis of tardive, levosulpiride-induced, armraising stereotypy was made, the offending drug was stopped. Tetrabenazine at 100 mg/day produced near-complete improvement at 3 months. Proximal limb involvement—such as arm-raising—is not reported as a phenotype in tardive stereotypy to date, with all cases having distal hand involvement. Furthermore, any limb stereotypy is not reported with levosupride to date. Keywords: Arm-raising, levosulpiride, stereotypy, tardive
How to cite this article:
Agarwal PA, Lalkaka JA. Levosulpiride-induced arm-raising stereotypy in a 65-year-old man: an unusual tardive phenomenon. Ann Mov Disord 2022;5:65-7 |
How to cite this URL:
Agarwal PA, Lalkaka JA. Levosulpiride-induced arm-raising stereotypy in a 65-year-old man: an unusual tardive phenomenon. Ann Mov Disord [serial online] 2022 [cited 2023 May 29];5:65-7. Available from: https://www.aomd.in/text.asp?2022/5/1/65/330446 |
A 65-year-old man presented to our hospital with a six-month history of involuntary, recurrent raising and lowering of both arms above his head. These movements began gradually over one to two weeks and involved both arms simultaneously. Over the next two to three weeks, they became more frequent and persistent throughout the day. His wife observed that the arm-raising movement would start when he sat idle for a few minutes, and then it would continue unabated for long periods, sometimes for up to 3–4 h. As the movement would stop during voluntary activity with his upper limbs, he was able to carry out all activities with his hands; thus, his daily routine was largely unhindered. He had no other symptoms, no behavioral or cognitive complaints, and no other medical comorbidities. Family history was negative. A review of his drug records revealed ongoing daily use of the dopamine receptor-antagonist levosulpiride (50 mg/day) for hyperacidity, for the previous eight months.
On examination, the stereotypic arm-raising movement persisted practically throughout the interview [Video 1, segment 1]. It stopped briefly when the patient was distracted, for instance, when he was asked to count numbers [Video 1, segment 2]. It also ceased when he performed volitional movements with his hands. In addition, tardive orofacial, neck, and trunk dyskinesia was noted [Video 1, segment 3]. There was no bradykinesia or rigidity suggestive of Parkinsonism More Details. The rest of the neurological examination was normal. Routine laboratory testing and an MRI brain were normal. The diagnosis of levosulpiride-induced tardive stereotypy was made, the offending drug was discontinued, and tetrabenazine (TBZ) was commenced. At three months’ follow-up (TBZ dose 100 mg/day), the arm-raising stereotypy had almost completely resolved [Video 2]. Cranial and trunk dyskinesia had also improved, but to a lesser degree.
[Additional file 1]
[Additional file 2]
[Additional file 3]
In 2011, Edwards, et al. defined stereotypy as “a repetitive, purposeless, non–goal-directed movement pattern that is repeated continuously for a period of time in the same form, and on multiple occasions, and which is typically distractible.”[1] The movement pattern in our patient had all the features just cited. It was also the dominant motor pattern, occurring at the expense of all other motor output, and had an invariant “looping” character, thus satisfying all criteria for stereotypy.[1] Unlike a complex motor tic, there was lack of a premonitory urge. It was a single predictable movement that did not have the rapid, random, and variable nature of tics. Unlike a compulsion, it was not driven by obsessive thought. Improvement with distraction is a feature that is common to both functional (psychogenic) movements and organic stereotypy. However, the lack of variability and inconsistency in the movement seen in our patient, its congruence with all features of organic stereotypy, and its co-occurrence with other typical tardive movements all argued against a functional origin.
[Additional file 4]
The presence of stereotypy in adults, especially in those who are otherwise neurologically normal, should prompt a careful search for previous exposure to dopamine receptor-blocking drugs.[2],[3] In a series of 100 patients with drug-induced tardive dyskinesia (TD) reported by Jankovic,[2] stereotypy (68%) was the most common phenomenology observed, occurring mostly in the orofacial region (61%), that is, “classic TD.” However, orofacial TD has more recently been considered distinct from stereotypy by other authorities,[1] given its worsening with distraction, and the random, unpredictable order of occurrence of its constituent movements. Tardive stereotypy most commonly involves the lower limbs (toe or foot tapping, marching in place) and the trunk (rocking or swaying movements). When it occurs in the upper limbs, it is typically distal,[3] involving fingers or hands, with reported patterns including picking or rubbing clothes, thumb twiddling, finger rubbing, hand waving, hand clasping, hair-twisting and rubbing of face, scalp, and other body parts. Proximal upper limb involvement by tardive stereotypy is distinctly uncommon in adults.[2],[3]
In children, distal hand stereotypies are seen in autism, Rett syndrome, intellectual disability, and in up to 20% of normal children, and these commonly include hand-wringing, shaking, flapping, hand-waving, or wrist rotation.[4] Proximal limb involvement with arm-waving or arm-flapping stereotypies is also described in autism,[4] and in adolescents with Tourette syndrome.[5] Interestingly, arm-raising per se has also been reported in a few cases: as a primary stereotypy pattern in normal children,[6] and more recently in pediatric NMDAR encephalitis.[7] Thus, nearly all cases of arm stereotypy in the literature are reported in children. Arm-raising stereotypy has not been reported as a tardive phenomenon in adults.
Levosulpiride is commonly used as a prokinetic drug, especially in Asia, and is known to cause various tardive syndromes.[8] However, limb stereotypy due to levosulpiride has not been reported to date. Interestingly, the relatively more rapid improvement in the arm stereotypy compared with that of other dyskinesia (orofacial, neck, and trunk), seen in our patient, is in keeping with the observation by Jankovic and Orman, that tardive stereotypy improves with TBZ therapy to a greater extent than other forms of TD.[9]
Through this case, we aim at bringing to attention an unusual and novel tardive stereotypy in adults. Prompt recognition of this hyperkinetic movement as a stereotypy would help not only in diagnosis of the clinical syndrome, but also in providing clues to its causation and, thus, would curtail unnecessary diagnostic testing.
Declaration of patient consent
The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.
Acknowledgments
The authors are thankful to the patient and his family for their cooperation.
Author contribution
Pankaj A. Agarwal: Conception, clinical examination, literature review, video recording, and video editing. Writing the first draft of the article, review and critique, and final approval of the version to be published.
Jimmy A. Lalkaka: Conception, clinical examination, literature review, writing the first draft of the article, review and critique, and final approval of the version to be published.
Ethical compliance statement
The authors confirm that the approval of an institutional review board was not required for this work. They also confirm that they have read the Journal’s position on issues involved in ethical publication and affirm that this work is consistent with those guidelines
Financial support and sponsorship
Nil.
Conflicts of interest
There are no conflicts of interest.
Informed consent for video
Written valid and informed consent was obtained from the patient for video recording, and both print and online publication of the videos.
| References | |  |
| 1. | Edwards MJ, Lang AE, Bhatia KP Stereotypies: A critical appraisal and suggestion of a clinically useful definition. Mov Disord 2012;27:179-85.  |
| 2. | Jankovic J Tardive syndromes and other drug-induced movement disorders. Clin Neuropharmacol 1995;18:197-214. |
| 3. | Savitt D, Jankovic J Tardive syndromes. J Neurol Sci 2018;389:35-42. |
| 4. | Goldman S, Greene PE Stereotypies in autism: A video demonstration of their clinical variability. Front Integr Neurosci 2012;6:121. |
| 5. | Ubhi M, Achinivu K, Seri S, Cavanna AE Motor stereotypies in adult patients with Tourette syndrome. Future Neurol 2020;15:FNL42. |
| 6. | Mahone EM, Bridges D, Prahme C, Singer HS Repetitive arm and hand movements (complex motor stereotypies) in children. J Pediatr 2004;145:391-5. |
| 7. | Granata T, Matricardi S, Ragona F, Freri E, Zibordi F, Andreetta F, et al. Pediatric NMDAR encephalitis: A single center observation study with a closer look at movement disorders. Eur J Paediatr Neurol 2018;22:301-7. |
| 8. | Joe J Levosulpiride-induced neurological adverse effects: A prospective study from a tertiary care center. Ann Indian Acad Neurol 2020;23:174-6. |
| 9. | Jankovic J, Orman J Tetrabenazine therapy of dystonia, chorea, tics, and other dyskinesias. Neurology 1988;38:391-4. |
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