Movement Disorders Vol. 8, No. 2, 1993, pp. 230-231 0 1993 Movement Disorder Society

Brief Report

Stereotyped Hand Clasping: An Unusual Tardive Movement Disorder

Kiyotoshi Kaneko, Tatsuhiko Yuasa, "Tadashi Miyatake, and Shoji Tsuji

Department of Neurology, Brain Research Institute, Niigata University, Niigata; and *Department of Neurology, Tokyo Medical and Dental University, Tokyo, Japan

Summary: We report an 83-year-old woman with vascular parkinsonism who presented with stereotyped rhythmical hand-clasping movements after 18 months exposure to neu- roleptics. Although stereotypical rhythmical orolinguomasticatory and limb movements are the most common tardive dyskinesia in the elderly, we feel this woman's hand clasping represents another unusual expression of a tardive movement disorder. Key Words: Stereotypy-Hand clasping-Tardive movement disorder-Tardive dyskine- sia-Parkinsonism-Rett's syndrome.

The most common tardive movement disorder associ- ated with chronic neuroleptic therapy affects the orofacial region (1). Dystonia, akathisia, and rarely tics are other, less frequent, tardive dyskinesias that may occur alone or in combination with the classical form affecting the oro- facial region. We report an 83-year-old woman who pre- sented with persistent involuntary hand clasping that be- gan 18 months after initiation of neuroleptic treatment.

CASE REPORT This 83-year-old woman was well until age 60, when

she insidiously developed a tremor in her hands and head. She had a history of hypertension and diabetes, but no family history of movement or psychiatric disorders. There were no rituals in her life that involved hand clasp- ing. During the subsequent 20 years, her tremor gradually increased.

Neurological examination showed coarse resting tremor of the hands bilaterally and head, mild rigidity predominantly of her left arm, and gait abnormality mim- icking parkinsonism. Bilateral tendon reflexes were slightly hyperactive, but pathological reflexes were ab- sent. Her cognitive function was normal for age according to the Mini Mental Scale Test (26 of 30 points) and the Diagnostic and Statistical Manual of Mental Disorders (3rd ed.) criteria (2). No other abnormal findings, includ-

Videotape segments accompany this article. Address correspondence and reprint requests to Dr. Kiyotoshi

Kaneko, Department of Neurology, Tokyo Medical and Dental University, 1-5-45, Bunkyo-ku, Tokyo 113 , Japan.

ing disorientation, aphasia, agnosia, apraxia, cranial nerve deficits, sensory disturbance, incoordination, aki- nesia, or autonomic failure, were revealed.

Her laboratory evaluation, including thyroid function tests, showed only a mildly elevated fasting blood glucose level. A computed tomography (CT) scan showed small bilaterally distributed ischemic lesions in the globus pal- lidus and putamen with penventricular lucency. An elec- troencephalogram showed diffuse slowing and no focal abnormalities.

Antiparkinsonian therapy, including levodopa/ carbidopa (200/20 mg/day), amantadine (200 mgday), tn- hexyphenidyl (6 mdday), and bromocriptine (5 mg/day), failed to provide any significant benefit. Based on these data, a diagnosis of vascular parkinsonism was made, al- though Parkinson's disease or parkinsonism in combina- tion with essential tremor were also considered. Because she had a coarse tremor, treatment with propranolol (30 mg/day) or pimozide (2 mg/day) was tried. However, the treatment also failed to provide any significant benefit for 6 months, and mild oral dyskinesia was observed during this therapy.

The addition of haloperidol (0.75 mg/day) to a regimen of trihexyphenidyl, amantadine, and pimozide was also tried to alleviate her tremor, and led to a significant re- duction of the tremor and oral dyskinesia. Other symp- toms, including gait abnormality, rigidity, and hyperre- flexia, continued without change. Five months later, she had onset of involuntary hand clasping. These stereo- typed movements were unassociated with restlessness or any subjective perception. She could voluntarily suppress these movements for up to half a minute, and experienced

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STEREOTYPED HAND CLASPING 231

no feeling of inner tension during these periods. The movements disappeared in sleep. There was no evidence of new ischemic lesions on a repeated CT scan after these movements developed. These movements continued after the solo withdrawal of haloperidol, and continued for over a year without change.

DISCUSSION This woman with parkinsonism or in combination with

essential tremor developed stereotyped hand clasping af- ter an 18-month exposure to neuroleptics. Early in her treatment, she had developed mild orolingual dyskinesia, but these abated during our observation period.

Stereotypical rhythmical movements in the orolinguo- masticatory region are the most common form of tardive dyskinesia, and we have never seen rhythmical hand clasping as observed in this woman. One consideration in this woman could be tardive akathisia (3). Although the movements in akathisia may appear purposeful, the ab- sence of any feeling of restlessness in this woman or need to move make this diagnosis unlikely. Pseudoakathisia, in which the typical motor signs of akathisia occurred with- out restlessness, is a controversial variant of akathisia. However, many of these cases have associated orofacial dyskinesia, which is moderate to severe, and limb move- ment (4). The lack of any build-up of psychic tension when the woman attempted to voluntarily suppress the movement may suggest a diagnosis against tardive tics. Mannerisms or stereotypes associated with schizophre- nia, Huntington’s disease, or senile chorea are obviously excluded by history or absence of particular neurological findings, although we cannot absolutely exclude dementia as a possible cause for her stereotyped movement.

Stereotyped hand movements are a cardinal feature of Rett’s syndrome. Rett’s syndrome is a disorder that be- gins in infancy and is characterized by verbal, motor, and social regression, in addition to hand-wringing move- ments (5). Although the age of onset and cognitive fea- tures would obviously exclude consideration of this diag- nosis, it is interesting that extrapyramidal features (6) and abnormalities of the dopaminergic system (7) also have been described in Rett’s syndrome.

In conclusion, the stereotyped rhythmical hand clasp-

ing observed in this woman may be an unusual presenta- tion of a tardive movement disorder. When stereotypic movements occur in elderly individuals, neuroleptic ex- posure should be considered, even if denied by the pa- tients.

Acknowledgment: We thank Drs. Toshiharu Anezaki and Isao Hozumi for their assistance; Ms. Misako Sat0 for her help in preparation of this manuscript; and Dr. Torbjoern Nygaard for constructive suggestions in preparation of this report.

LEGENDS TO VIDEOTAPE

Segment 1. Stereotypical hand clasping observed 42 months after first neuroleptic exposure. The patient was taking amantadine (200 mg/day), tnhexyphenidyl (6 mpl day), and pimozide (2 mg/day) at the time of this video- tape.

Segment 2. The patient was asked to suppress her hand- clasping during this segment. She can only do this for half a minute before the movements reappear.

REFERENCES 1. Tanner CM. Drug-induced movement disorders (tardive dys-

kinesia and dopa-induced dyskinesia). In: Vinken PJ, Bruyn GW, Klawans HL, eds. Handbook of clinical neurology. New York: Elsevier, 1986;5: 185-204.

2. American Psychiatric Association (APA). Diagnostic and statistical manual of mental disorders. 3rd ed. Washington, DC: APA, 1980.

3. Burke RE, Kang UJ, Jankovic J, Miller LG, Fahn S. Tardive akathisia: an analysis of clinical features and response to open therapeutic trials. Mov Disord 1989;4: 157-175.

4. Barnes TRE, Brande WM. Akathisia variants and tardive dyskinesia. Arch Gen Psychiatry 1985;42:874-878.

5 . Hagberg B, Aicardi J, Dias K, Ramos 0. A progressive syn- drome of autism, dementia, ataxia, and loss of purposeful hand use in girls: Rett’s syndrome: report of 35 cases. Ann Neurol 1983 ; 14:471479.

6. FitzGerald PM, Jankovic J, Glaze DG, Schultz RN, Percy AK. Extrapyramidal involvement in Rett’s syndrome. Neu- rology 1990;40:293-295.

7. Wenk GL, Naidu S, Casanova MF, Kitt CA, Moser H. Al- tered neurochemical markers in Rett’s syndrome. Neurol- ogy 199 1 ;41: 1753-1 756.

Movement Disorders, VoI. 8 , No. 2, 1993