Manic illness associated with right temporo-thalamic arteriovenous malformation: A rare presentation

To the Editor

Krauthammer and Klerman (1978) introduced the concept of organic manic disorder, and Das and Khanna (1993) showed that it is present in 4.67% of patients with mania. The most common causes are neurological, especially focal brain lesions. Arteriovenous malformation is a rare neurological condition associated with organic manic episodes (Gross and Herridge, 1988). We describe a manic illness in a patient in whom a right medial temporal lobe and thalamic arteriovenous malformation was detected.

Ms A, a 15-year-old adolescent female, presented with complaints of irritability, over talkativeness, over familiarity and increased physical activity present for the last month. She gave a medical history of insidious progressive onset of left-sided weakness, starting over 4 years earlier. There was no history of fever, headache, head injury, incontinence, seizure episode or any other neurological abnormality. The weakness of the left side of body was permanent. No treatment was sought for it. Two months after onset of this neurological problem, she had experienced a manic episode, which resolved completely with 4 weeks of treatment from a psychiatrist (treatment details were not available). There is no family history of any neuro-psychiatric illness.

On neurological examination, there was atrophy and flaccidity of the left upper and lower limbs, power was 2/5, superficial reflexes were diminished, deep tendon reflexes were exaggerated, plantar reflex was extensor on left side. On mental status examination, affect was irritable and grandiose ideas and decreased need for sleep were elicited. All blood tests were normal. Computed tomography (CT) angiography revealed intracranial arteriovenous malformation embedded in right medial temporal lobe and thalamic regions. The patient was started on valproate 300 mg twice daily and lorazepam 2 mg per day. There was significant reduction in manic symptoms after 3 weeks of treatment and complete resolution of affective symptoms after 8 weeks. She was maintained on 600 mg of valproate per day and referred to the department of neurosurgery for evaluation and management of the intracranial arteriovenous malformation.

Organic manic disorder should be considered in any manic patient who presents outside the usual age of onset for idiopathic manic-depressive disease, has no family or personal history of affective disturbance or has concomitant neurologic deficit. Lack of acknowledgment of deficits, and the tendency for mania to overshadow other aspects of clinical presentation, may cause neurologic abnormalities to be overlooked and might account for the rarity of reported cases of organic manic disorder associated with cerebrovascular disease.