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Quetiapine for Mania With Wilson's Disease

Isin Baral Kulaksizoglu, M.D., and Aslhan Polat, M.D., Istanbul, Turkey

TO THE EDITOR: Wilson's disease is an autosomal recessive disorder of copper accumulation in the liver, brain, cornea, and kidneys. Tremor, extrapyramidal symptoms—particularly dystonia—and gait abnormalities are the most frequent neurological features of Wilson's disease. A 20% incidence of psychiatric symptoms has been reported in Wilson's disease.¹ Anxiety, psychosis, affective disorders, personality changes, cognitive impairment, and schizophrenia-like states commonly occur as the disease progresses. Affective disorders, such as mania, are one of the main psychiatric symptoms of Wilson's disease.² The selection of antipsychotic medication for treatment must be carefully made because of the incapacitating side effects of these drugs.

Case Report

Andrew was a 17-year-old male adolescent with Wilson's disease who presented with first-episode bipolar I disorder (mania). He had been diagnosed with Wilson's disease 6 months before, and it gradually increased in severity. A neurological examination revealed extrapyramidal signs, rigidity, and cogwheel sign positivity. A physical examination revealed splenohepatomegaly and grade I esophageal varicose enlargement. A biochemical evaluation showed a low plasma copper level (0.66 µg/ml, normal range=0.9–11 µg/ml) and a normal SMA. His liver enzyme levels—SGOT: 36 (normal range=5–42 U/liter) and SGPT: 33 (normal range=5–45 U/liter)—were normal and stayed within normal ranges during antipsychotic therapy. Andrew also had low ceruloplasmin levels (0.07 g/liter, normal range= 0.15–0.60 g/liter) and a high urine copper level (196 µg/ml/24 hours). His EEG was normal. An MRI of his brain showed a bilateral hyperintense signal in both heads of the caudate nucleus and putamen. Andrew was diagnosed with bilateral Kayser-Fleischer rings. He was given penicillamine, 600 mg/day.

Andrew's psychiatric symptoms were mood elevation, psychomotor agitation, grandiose delusions, such as being extra-talented, and erotomania. He had no previous psychiatric illness history nor did his family. After initial treatment with quetiapine (maximum dose=400 mg/day), lithium (1200 mg/day) was added to his treatment regimen 8 months later since his manic symptoms, such as irritability and excessive energy, recurred whenever quetiapine tapering was attempted. Quetiapine treatment continued for 10 months without extrapyramidal symptoms that would have exacerbated the neurological symptoms of Wilson's disease. Lithium was discontinued at the end of 1 year. Andrew is still being followed 24 months later and is doing well.

Discussion

There is a lack of consensus over the pharmacological treatment of Wilson's disease in patients with psychiatric disorders. The possible side effects of classical neuroleptics are a limiting factor in the choice of psychopharmacology in neurological disease. There are reports that the symptoms of Wilson's disease may worsen with the use of typical neuroleptics.^3,4 Psychiatric symptoms often respond poorly to conventional psychiatric medication, and it may be difficult to differentiate adverse effects of psychiatric medications from the developing neurological symptoms of Wilson's disease itself. Atypical neuroleptics may be used in the treatment of the psychotic and affective symptoms of Wilson's disease because of their fewer extrapyramidal side effects. There is at least one case report regarding the use of clozapine in a psychotic patient with Wilson's disease.⁵ Quetiapine is an atypical neuroleptic with reportedly safe use in terms of extrapyramidal side effects, even in patients with Parkinson's disease. Our case is an example of the successful use of quetiapine in the treatment of psychotic symptoms in a patient with Wilson's disease.

REFERENCES

1. Medalia A, Scheinberg IH: Psychopathology in patients with Wilson's disease. Am J Psychiatry 1989; 146:662–664[Abstract/Free Full Text]
2. Akil M, Brewer G: Psychiatric and behavioral abnormalities in Wilson's disease, in Advances in Neurology, vol 65: Behavioral Neurology of Movement Disorders. Edited by Weiner WJ, Lang AE. New York, Raven Press, 1995, pp 171–178
3. Chroni E, Lekka NP, Tsiribri E, Economou A, Paschalis C: Acute progressive akinetic-rigid syndrome induced by neuroleptics in a case of Wilson's disease (letter). J Neuropsychiatry Clin Neurosci 2001; 13:531–532[Free Full Text]
4. Tu J: The inadvisability of neuroleptic medication in Wilson's disease. Biol Psychiatry 1981; 16:963–968[Medline]
5. Krim E, Barroso B: Psychiatric disorders treated with clozapine in a patient with Wilson's disease. J Neuropsychiatry Clin Neurosci 2001; 13:531–532

Full Text (PDF) Alert me when this article is cited Alert me if a correction is posted Email this article to a Colleague Similar articles in this journal Similar articles in PubMed Alert me to new issues of the journal Add to My Articles & Searches Download to citation manager Citing Articles via Google Scholar Articles by Kulaksizoglu, I. B. Articles by Polat, A. Search for Related Content PubMed Citation Articles by Kulaksizoglu, I. B. Articles by Polat, A. Syndromes Secondary to General Medical Disorders

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