|Year : 2013 | Volume
| Issue : 1 | Page : 32-34
Choreoathetosis as a manifestation of thyrotoxicosis in a Nigerian woman: A case report and review of the literature
Lukman F Owolabi1, Aliyu Ibrahim1, Ibrahim D Gezawa2, Andrew E Uloko2
1 Department of Medicine, Neurology Unit, Aminu Kano Teaching Hospital, Bayero University, Kano, Nigeria
2 Endocrine Unit, Aminu Kano Teaching Hospital, Bayero University, Kano, Nigeria
|Date of Web Publication||18-Oct-2013|
Lukman F Owolabi
Department of Medicine, Aminu Kano Teaching Hospital, Bayero University, PMB 3452, Kano
Source of Support: None, Conflict of Interest: None
| Abstract|| |
We report the case of a 65-year-old Nigerian woman with thyrotoxicosis manifesting as choreoathetosis, which misdiagnosed as a seizure disorder. The movement abnormality was characterized by bilateral jerky, purposeless, intermittent, irregular, and sometimes writhing movements, predominantly in the distal joints. Thyroid function test showed elevated total serum triiodothyronine (T3) and thyroxine (T4), and reduced serum thyroid stimulating hormone (TSH). There was complete resolution of choreoathetoid movement after 1 week of propanolol use. The relationship between choreoathetosis and thyrotoxicosis is discussed.
Keywords: Choreoathetosis, seizure, thyrotoxicosis
|How to cite this article:|
Owolabi LF, Ibrahim A, Gezawa ID, Uloko AE. Choreoathetosis as a manifestation of thyrotoxicosis in a Nigerian woman: A case report and review of the literature. Ann Nigerian Med 2013;7:32-4
|How to cite this URL:|
Owolabi LF, Ibrahim A, Gezawa ID, Uloko AE. Choreoathetosis as a manifestation of thyrotoxicosis in a Nigerian woman: A case report and review of the literature. Ann Nigerian Med [serial online] 2013 [cited 2020 Feb 27];7:32-4. Available from: http://www.anmjournal.com/text.asp?2013/7/1/32/119985
| Introduction|| |
Several neurological symptoms such as nervousness, jitteriness, muscle weakness, tremor or tremulousness, and cognitive impairment, may accompany thyrotoxicosis. However, choreoathetosis associated with thyrotoxicosis is exceedingly rare with only one case previously reported from Nigeria. , Nonetheless, it is well-known that, at times, thyroxicosis presents in an atypical manner resulting in misdiagnosis.  We report a case of generalized choreoathethoid movements associated with thyrotoxicosis in a Nigerian woman, with a view to drawing attention of clinicians to this rare entity and the need for careful evaluation of patients presenting with suspected movement or seizure disorder.
| Case Report|| |
A 65-year-old woman presented to the neurology clinic of Aminu Kano Teaching Hospital, Kano, with insidious onset involuntary movement of the limbs of 1-year duration. The involuntary movement started from the left upper limb, and subsequently over 3 months, involved the other parts of the body. She had increased appetite, excessive sweating, occasional palpitation, and generalized dull headache. She did not have any visual impairment, diplopia, vomiting, or hyperdefecation. She was neither hypertensive nor diabetic. She was diagnosed with a seizure disorder in a private health facility for which she had an antiepileptic drug (AED). The AED was discontinued because there was no improvement on the medication.
Physical examination showed an elderly woman with normal findings apart from mild thyromegaly, exophthalmos and jerky, purposeless, intermittent, irregular, and sometimes writhing movements; predominantly in the distal joints. She also had a tachycardia of 104 beats per minute.
The initial diagnosis was that of choreoathetosis of unknown cause to exclude thyrotoxicosis and encephalitis. Thyroid function test showed elevated total serum triiodothyronine (T 3 ) and thyroxine (T 4 ), and reduced serum thyroid stimulating hormone (TSH). Total serum T 3 was 4.07 mmol/L (normal range: 1.3-3.1 nmol/L), total serum T 4 was 235 nmol/L (normal range: 66-181 nmol/L), and serum TSH was <0.005 mIU/L (normal range: 0.3-4.2 mIU/L). Thyroid scan did not reveal thyroid nodules. The patient was placed on propranolol 40 mg twice daily before she was reviewed by the endocrinologists. By the first week on propranolol, the choreoathetoid movement ceased. Subsequently, she was placed on carbimazole tablets 10 mg thrice daily. By the 3 rd week on carbimazole and propanolol, there was significant improvement in her condition and all choreoathetoid movement had subsided.
| Discussion|| |
In the patient being reported, choreoathetosis was the only reason the patient presented to neurology clinic. This observation agreed with similar case reports elsewhere that showed choreoathetosis as the presenting and sometimes the first feature of thyrotoxicosis. , Relationship between thyrotoxicosis and choreoathetoid movement disorder has been documented as far back as early 20 th century.  The first report of an association between Grave's disease and chorea was by Gowers in 1893, when he reported that true chorea existed in a few goiter cases in childhood or youth.  Subsequently, there were few similar reports of chorea in association with thyrotoxicosis from Europe and America, and unlike in the current report, all the patients reported were relatively young.  The mechanism underlying choreoathetosis from thyrotoxicosis is not fully known, however, it is thought to be due to amplified sensitivity of the dopaminergic receptor sites in the corpus striatum. The sensitivity of adrenergic and dopaminergic receptors to noradrenaline and dopamine have been found to be influenced by alterations in thyroid function. , It has been postulated that thyrotoxicosis influenced the brain neurotransmitter system causing alteration in the metabolism of dopamine in the corpus striatum thereby causing chorea. 
Responsiveness of thyrotoxicosis induced chorea to haloperidol which is a dopamine receptor blocker further lend support to this dopamine hypothesis. 
Previous reports have also suggested that choreoathetosis might be the most severe form of a continuum of movement disorder associated with thyrotoxicosis, with tremor representing the mildest form of the spectrum. 
In the current case, choreoathetoid movement was bilateral. This observation, which was similar to reports elsewhere, ,, is understandable, as the effect of thyrotoxicosis is supposed to be systemic rather than localized. However, there have been a few reports of rather enigmatic unilateral manifestation. ,
Although thyrotoxicosis is not rare, only one case has been reported to be associated with choreoathetosis in the literature from Nigeria.  However, the index case differs from the previous case in that there was a dramatic response to propanolol; treatment with propranolol produced resolution of the choreoathetosis in a matter of 1 week. This observation, as previously reported, would indicate that the choreoathetosis is subserved by the sympathetic nervous system.  The association between autonomic nervous system and thyrotoxicosis was first established in 1918 by Goetsch who showed that many features of thyrotoxicosis could be produced by epinephrine.  Earlier reports , have also noted dramatic response of the choreoathetosis to beta blockade and antithyroid drugs, as well as reserpine and dopamine antagonists. In these reports, , there was no relapse of choreoathetoid movements following stoppage of the medications after the patients had been made euthyroid.
The benefits of beta adrenergic blocking agents have been ascribed to their modulation of the effect of thyroxine on cathecholamine. Nonetheless, the effects of beta blockers are not very consistent, as there were some other reports that did not show any effect of beta blocking agents, , thereby casting some doubt on the sympathetic hypothesis of choreoathetosis in thyrotoxicosis.
The patient was wrongly diagnosed as having seizure disorder at the referring hospital. Seizure can imitate movement disorder. In fact, the word seizure often evokes images of involuntary movement, hence, it was not storming that, like in the present case, all episodic or paroxysmal movements were generally attributed to seizure in the early part of the 20 th century.  Thyrotoxicosis occasionally presents in an atypical fashion in which the diagnosis may not be clear cut.  However, to distinguish between movement and seizure disorders; both the history and the phenomenology of the abnormal movements will be useful. Direct observation of an episode is paramount, even though it is not always feasible. Thus, effort should be made to inquire specifically about which body parts move, and in what manner.
Because of the rarity of thyrotoxicosis manifesting as choreoathetosis, clinicians may, as exemplified by the case being reported, easily miss the diagnosis with resultant delay in making the appropriate diagnosis. That being the case, it behoves the clinician to become familiar with the fact that thyrotoxicosis can manifest in various atypical ways, choreoathetosis inclusive.
| References|| |
|1.||Baba M, Terada A, Hishida R, Matsunaga M, Kawabe Y, Takebe K. Persistent hemichorea associated with thyrotoxicosis. Intern Med 1992;31:1144-6. |
|2.||Adeleye JO, Akinyemi RO, Balogun WO, Onyegbutulem HO. Unusual presentation of thyrotoxicosis - A case report and review of literature. West Afr J Med 2005;24: 274-7. |
|3.||Greenspan F. The thyroid gland: Basic and clinical endocrinology, 5 th ed. Norwalk: Appleton and Lange; 1997. p. 192-262. |
|4.||Dhar SK, Nair CP. Choreoathetosis and thyrotoxicosis. Ann Intern Med 1974;80:426-7. |
|5.||Nagaoka T, Matsushita S, Nagai Y, Kobayashi K. A woman who trembled then chorea. Lancet 1998;351:1326. |
|6.||Sutherland GA. Chorea and Graves's disease. Brain 1903;26:210-4. |
|7.||Gowers W. A manual of diseases of the nervous system. vol. 2. London: Churchill; 1893. p. 814-18. |
|8.||Clements MR, Hamilton DV, Siklos P. Thyrotoxicosis presenting with choreoathetosis and severe myopathy. J R Soc Med 1981;74:459-60. |
|9.||Lipton MA, Prange AJ, Diarman W, Udeufriend S. Increased rate of norepinephine biosynthesis in hypothyroid rats. Fed Proc 1968;27:39. |
|10.||Javaid A, Hilton DD. Persistent chorea as a manifestation of thyrotoxicosis. Postgrad Med J 1988;64:789-90. |
|11.||Marks P, Anderson J, Vincent R. Choreo-athetosis with severe thyrotoxicosis. Postgrad Med J 1988;55:830-1. |
|12.||Shahar E, Shapiro MS, Shenkman L. Hyperthyroid-induced chorea. Case report and review of the literature. Isr J Med Sci 1988;24:264-6. |
|13.||Goetsch E. Newer methods in the diagnosis of thyroid disorders. New York J Med 1918;18:259. |
|14.||Fidler SM, O'Rourke RA, Bucksbaum HW. Chorioathetosis as a manifestation of thyrotoxicosis. Neurology 1971;21:55-7. |
|15.||Fahn S. Movement disorders. In: Engel J Jr, Pedley TA, editors. Epilepsy: A Comprehensive Textbook. Philadelphia: Lippincott-Raven Publisher; 1997. p. 2695-845. |