Case Study Propranolol Treatment of Akathesia in Tourettes Syndrome JAMES D. CHANDLER, M.D., F.R.C.P.(C)

Abstract. Akathesia is a common side effect of neuroleptic medication and has been reported to occur in patients with Tourettcs Syndrome (1'5). In 1'5, the differentiation between untreated hypcractivity and akathcsia can bc difficult. A case ofneurolcptic treated 1'5 with hyperactivity versus akathesia is presented in which propranolol was successfully used to treat the akathcsia. The akathesia reappeared when the propranolol was withdrawn. This case illustrates an alternative to neuroleptic dose reduction in the management of akathcsia in TS . .I. Am. /vcad. Child Adolesc, Psychiatry, 1990,29,3:475-477. Key Words: Tourcttcs, akathcsia, hypcractivity, propranolol. Case Report

One of the more difficult clinical decisions an adult psychiatrist must make is whether restlessness and agitation are secondary to psychosis or akathesia. If akathesia is mistaken for psychosis, neuroleptics arc usually increased, leading to greater restlessness rather than less. The importance of the proper diagnosis and treatment in this situation is further accentuated by reports of suicide attempts secondary to akathesia (Drake and Ehrlich, 1985). The usual recommendation is that psychiatrists should err on the side of overcalling akathesia and consider adding one of the medications successfully used in the treatment of this condition such as beta-blockers, anticholinergics, or benzodiazepines (Talbott et al., 1988). There is an analogous situation in child psychiatry. One of the few indications for neuroleptics in children is Gilles de la Tourctte Syndrome (TS) (Talbott and Yudofsky, 1987). This disorder is diagnosed based upon the presence of vocal tics with or without motor tics in thc absence of any other neurologic or medical cause. It is also frequently accompanied by hyperactivity in addition to signs and symptoms of obsessive-compulsive disorder. The neuroleptics most frequently used in this condition, pimozide and haldoperidol , can often be associated with akathesia in adults. While these drugs can be quite effective in reducing tics, they are usually less useful in reducing hyperactivity and obsessive-compulsive behavior (Cohen et al., 1988). In children with TS treated with neuroleptics, the differentiation between the restlessness of akathesia and accompanying hyperactivity which has not responded to treatment can prove to be extremely difficult (Bruun, 1988). The case below describes a child with TS in which the clinical strategy of erring on the side of overcalling akathesia and prcscribing appropriate treatmen t was applied with great success.

Ann was a 7-year-old , 20 kilogram Caucasian girl with a 3- to 4-year history of facial tics, grunting, sighing, and clicking. She met DSM-III-R criteria forTS. Besides these core symptoms, she also showed impulsiveness, severe restlessness, inattentiveness, and met DSM-JIJ-R criteria for hyperactivity syndrome. She also had some compulsive rituals (touching people exactly twiec) and occasional echolalia. Her compulsive touching had ostracizcd her from her peers and she had few friends. The touching and vocal tics had also alienated her from her parents, especially her father. Her mother described her as a "different" but affectionate child. Educational testing had shown a delay in language development. However, the testing was in French rather than English. Although she lived in a French area, she had learned English from a baby sitter and spoke primarily English. No formal IQ scores were available. Her hyperactivity had been noticed by her teacher, and this, in combination with her lack of fluency in French, had resulted in her placement in "special education" classes on a weekly basis. The family history was positive for a mother with panic disorder, in remission, and a father with aleohol abuse, in remission. Her hyperactivity was of greater concern to her family than the tics and had resulted in difficulties in school, both behavioral and academic. She was referred to her [amily doetor who prescribed methylphenidate, 10 mg per day (mg/d). Two days later she was seen by the author on an emergency basis after her tics had increased in both frequency and severity to such a degree as to alarm the family. On examination, she was unable to sit still and was almost constantly clicking, sighing, and grunting. She was notcd to be frequently touching objects in the examination room with her open palms. Ann was hospitalized. A workup for an organic cause revealed the following normal studies: complete blood count and differential, electrocardiogram, electrolytes, ceruloplasmin, serum copper, and brain scan. She was started on pimozide I mg/d. She showed an immediate improvement in her tics and hyperactivity. After I week, the dosage was increased to 2 mg/d and was kept at that dose for the next 2 months. Her hyperactivity then seemed to worsen again and her tics continued at a low, but not disabl ing, frequency.

Accepted June 26, 19S9. Dr. Chandler is StaffPsychiatrist in the Department of Psychiatry, Western Regional Health Centre, Yarmouth. Nova Scotia. Request reprints [rom Dr. Chandler, Department of Psychiatry, Western Regional Health Centre, 60 Vancouver St., Yarmouth, Nova Scotia, B5A 21'5 Canada. 0890-8567/90/2903-0475$02.00/0@ 1990 by the American Academy of Child and Adolescent Psychiatry.

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In order to better manage her hyperactivity, a trial of clonidine at 0.025 mg/d was begun. After 3 days, the patient became extremely sedated, irritable, and appeared depressed. The clonidine was discontinued and her clinical picture returned to baseline. She was restarted on pimozidc and the dose was increased to 3 mg/d. After 2 weeks, the mother reported that the child was more' 'hyper" than ever and was running in circles. There had been no recent psychosocial strcssors. In the office, the child was found to be more restless and in fact did bcgin skipping in circles. The tics, however, were under very good control. The child did not appear anxious or irritated. When asked why she was engaging in these stcrcotypic movements, she stated, "I can't help it." In addition to this stereotypic movement, she showed her baseline level of random hyperactive behavior. The mother had also noticed mild slurring of speech. There was no sign of cogwhceling, rigidity, or tremor. Based on the author's personal experience in successfully treating akathesia in adults with beta blockers, propranolol, 10 mg twice daily, was begun. After 2 days, the mother reported that Ann was no longer running in circles in the evenings. She did continue to be unattentivc and slightly overactive. After 2 weeks of propranolol at this dosage, her blood pressure and pulse remained unchanged. On examination, her tics remained in good control, her speech continued to be slightly slurred, and her restlessness had decreased markedly. She showed no signs of depression. However, the child did not like the taste of the propranolol and the mother was unsure whether the improvement had resulted from this treatment. After discussing this with the mother, I discontinued the propranolol. After 2 days, Ann began running in circles. After a week, she was restarted on propranolol 10 mg twice daily with a resultant decrease in this behavior. At the next office visit, the mother informed the author that the propranolol prescription had run out 4 days earlier. The child was again running in circles and lying on her back and spinning herself in a stereotypic fashion. The mother was quite adamant at this time about the necessity of having su Ificicnt propranolol to last until the next appointment. Throughout these trials of propranolol, there were no new psychosocial strcssors. At follow up 12 week later, Ann continues to do well on this combination of pimozide and propranolol.

Discussion This case illustrates the difficulty in distinguishing akathcsia in children from hyperactivity. The author assumed when the patient presented with increased hyperactivity on 2 mg/d of pimozide that this was due to TS rather than akathcsia. It was only when this condition worsened and the patient presented with stereotypic restless movements that the diagnosis of akathesia was considered. Previous reports have found akathesia to be associated with an increase and not a decrease in tic frequency (Weiden and Bruun, 1987). The prevalence of akathesia in this population is relatively unknown. An open study of 208 TS patients treated with neuroleptics found akathesia to be clearly 476

present in nine and possibly preset in four others for an estimated prevalence of 5.1 % (Bruun, 1988). One of the possible criticisms of this report is that it was assumed that since the stereotypic restlessness decreased when the propranolol was added and returned when it was discontinued that this condition therefore must be akathesia. Patients with chronic motor tic disorder have been noted to show an "inner tension" which has some similarities to akathesia (Walsh et al., 1986). It can be argued that propranolol has not been found to be an effective treatment of hyperactivity or TS, and thus the likelihood of this beta blocker acting directly to decrease hyperactivity or TS symptomatology is very low. Another possibility is that the symptoms could represent panic attacks or aggressive outbursts, as these could have responded favorably to propranolol. However, there were no other signs of anxiety except restlessness, and there was no sign of aggression in the movements observed by the author. There are other significant limitations inherent in drawing conclusions about causation and treatment efficacy from one case report. No placebo control was used, only one patient was assessed, and the two conditions, akathesia and TS, arc notorious for being difficult to correctly diagnose. Nevertheless, this case does illustrate a possible explanation for the noted lack of efficacy of ncuroleptics in the treatment of secondary hyperactivity in TS. Studies using pimozide have not specifically evaluated patients for akathesia (Shapiro and Shapiro, 1984) and none to the present author's knowledge have considered giving drugs such as propranolol a therapeutic trial in questionable cases. Based on clinical experience, some investigators have noted patients with TS to be more susceptible than others to the side effects of neuroleptics (Bruun et aI., 1988). While a reduction in dose is always the optimal solution to the disabling side effects of neuroleptic use, this can often lead to an increase in tics and other manifestations of TS. If the results of this report are replicated by others in controlled settings, the use of drugs such as propranolol in the treatment of akathesia may result in a greater tolerance of neuroleptic treatment in TS. In summary, the successful treatment of this child's worsening movement disorder with propranolol points to the importance of considering akathesia as a possible explanation of worsening restlessness/hyperactivity in TS patients treated with neuroleptics. The observation that this condition was in this case quite easily treated with beta-blockers makes vigilance in regards to akathesia all the more vital.

References Bruun, R. D. (1988), Subtle and under recognized side effects of neuroleptic treatment in children with Tourette's disorder. Am. J,

Psychiatry, 145:621-624. Cohen, D. J., Brunn, R. D. & Leekman, J. F. (eds.) (1988), Tourette's Syndrome: Clinical Understanding and Treatment, New York: Wiley. Drake, R. E. & Ehrlich, J. (1985), Suicide attempts associated with akathcsia. Am. 1. Psychiatry, 142:499-501. Shapiro, A. K. & Shapiro, E. (1984), Controlled study of pimozide vs. placebo in Tourcttcs Syndrome. J, Am. Acad. Child Psychiatry,

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AK ATHES IA IN TO UR ETT E ' S S YND RO M E

Talbott, 1. A. , Hales, R. E. & Yudofsky, S . C . (cds .)( 1987) , The American Psychiatric Press Textbook of Neuropsychiatry , Washington, DC: American Psychiatric Press , pp . 366- 37 1. - - - _ . - - (cds .) (1988), The AmericanPsvchiatric Press Textbook or Psychiatry, Washington, DC: American Psychiatric Press , p. 780.

J. Am .Acad . Child Adolesc . Psychiatry, 29:3 , May 1990

Walsh , T . L. & Lavcnsticn, B. , Licamclc , W . L. , Bronh cim S . & O'Leary, J. (1986) , Calc ium antagoni sts in thc treatm e nt of To urcue's d isorder. Am . J . Psychiatry , 143: 1467- 1468. Wcidc n, P. & Bruun, R. D. ( J987) , Worsening of To urcttc' s d isorder due to neuroleptic-induced akath csia. Am. J. Psychiatr y: 14:504505 .

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Propranolol treatment of akathesia in Tourette's syndrome.

Akathesia is a common side effect of neuroleptic medication and has been reported to occur in patients with Tourette's Syndrome (TS). In TS, the diffe...
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