Case reports

CASE 1

A 28-year-old kimono shop manager had a 20-year history of paroxysmal fearful feelings of being left alone. At age 11, complex partial seizures began to follow these moments of fear. As his age advanced, his seizures increased in intensity as well as frequency, despite maximum drug therapy. At age 26, the first manifest postictal mental derangement occurred, after several bouts of complex partial seizures. One day after this cluster of seizures, he struck his father, the owner of the kimono shop, who had asked whether he was all right, out of uncontrollable rage. This peculiar dysphoric state lasted for a week, during which he was continuously prone to violent behaviour over minor matters. He reported that alien ideas had invaded him and that opposing thoughts battled each other during this state. Afterwards, he could recall perfectly the details of his violent behaviour, but could not understand why he had acted in such a manner. Such postictal episodes and repeated violent behaviours tormented those around him, and recurred every 2 months before admission. An MRI revealed a marked asymmetry of the hippocampi (the left side was smaller than the right) with a lower signal intensity from the left hippocampus on a reversed T2 condition. Although he was right-handed, a 60 mg injection of Amytal into the right, but not the left, carotid artery caused the patient to become aphasic. Ictal EEG recordings, including those with depth, plus subdural electrodes, unanimously suggested that the left hippocampus was the origin of both the simple and complex partial seizures. Subsequently, a left inferior lobectomy with a hippo-campoamygdalotomy was performed. The resected tissue revealed Ammon's horn sclerosis. On awakening from anaesthesia, the patient showed a peculiar dysphoria, just as he had during his postictal psychoses, which spontaneously disappeared within 3 days. However, one month after the lobectomy, he became increasingly euphoric and elevated in mood. His speech was loud, rapid and difficult to interrupt, while full of jokes, puns and other amusing irreverence. He even exposed his genitalia in public during his elevated moods and courted several copatients. Since he sang throughout the night, we needed strong sedatives to put him to sleep. This manic state, lasting for 4 weeks, gradually turned into a depressive one. Two months after the operation, he felt quite upset and lost interest in all activities. He became so agitated that he could not stand still for a moment, and walked around restlessly. He complained of slowed thinking and difficulties in making decisions. After treatment with tricyclic antidepressants, his mood improved steadily, but only gradually, over half a year. Six months after the operation, he set out to do his previous work and his relations with colleagues improved dramatically. A year after the operation, his mood was stabilized without the help of antidepressants and he was accepted once again as the manager of the kimono shop. He has been completely seizure-free for 4 years postoperatively. Moreover, neither the previous dysphoric episodes nor the mood disorders have recurred.

CASE 2

At age 3, a 38-year-old housewife suffered from an episode of prolonged febrile convulsions, which lasted for more than 30 minutes and resulted in transient paralysis of the left upper extremities. At age 12, the first episode of complex partial seizures occurred, in which she unknowingly handed over an examination paper to a classmate who happened to be sitting next to her. After that, every time during a seizure, she would unconsciously reiterate the same phrase; 'He's coming to collect my examination paper. What should I do?'. At age 15, paroxysmal feelings of a peculiar familiarity began to precede the complex partial seizures, during which she felt that the atmosphere of her environment suddenly changed and it seemed as if she had dissolved into the immediate surroundings. Her seizures remained uncontrollable despite intensive medication. As the patient's age advanced, she joined a local religious sect as a devoted member and became increasingly eccentric. The first manifest postictal psychosis occurred after bouts of complex partial seizures at age 29. After an intervening 36-hour lucid interval, she would rapidly become more and more elevated in mood, with loud rapid speech that was difficult to interrupt.

She would change subjects kaleidoscopically from one to another. She screamed to her husband repeatedly, 'I love you, darling', and hugged and kissed him in public in a sensual manner. Three days after the cluster of seizures, the euphoric state culminated in agitated exaltation. She said, 'I am directly feeling all that is happening in every corner of the world through the palpitating movement of my teeth. The circular movements of my teeth are synchronized with the circular movements of the world. Through nerves in my teeth, I can sense the future 2000 years from now'. Because of extreme psychomotor agitation, a short stay in the psychiatric ward as well as potent sedatives were required. This state disappeared completely within 10 days. Throughout the episode, her orientation and memory remained intact. After several episodes of such postictal psychotic states, she agreed to surgery. An MRI revealed a marked asymmetry of the hippocampi (the left side was smaller than the right) with a lower signal intensity from the left hippocampus on a reversed T2 condition. Although she was right-handed, her dominant language side proved to be right. Ictal EEG recordings unanimously suggested that the left hippocampus was the origin of both the simple and complex partial seizures. During the course of intensive seizure monitoring, postictal psychosis recurred once after a cluster of complex partial seizures. Subsequently, a left inferior lobectomy with a hippocampoamygdalotomy was performed and the resected tissue revealed Ammon's horn sclerosis. She recovered steadily without complications. A year after the operation, the patient began to work as a manager of a Japanese restaurant. She completely lost interest in the religious activity as if exorcised. She has been completely seizure-free for 7 years postoperatively and no episodes of postictal psychosis have recurred.

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