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Patients with cerebral lesions out of motor and sensory strips are often overlooked, especially those with frontal lobe symptoms, which are subtle and elusive. We report on a patient with a left anterior cerebral artery infarct, who had an unusual manifestation—namely, repetitive sentence writing.
A 58 year old right handed woman had a transient lapse of consciousness when riding a bicycle on 3 June 1995. She had no vomiting, convulsion, or sphincter problems, and walked home after having taken a rest. Brain CT performed at a local hospital was normal. Next day, she visited our hospital because of right leg weakness, urinary urgency, persistent mutism, and slow mentality. On admission, the patient seemed alert and had normal vital signs, but was mute and responded slowly to oral commands. Neurological examination disclosed a grasp reflex on both hands, being stronger on the right, gegenhalten rigidity, right leg weakness, and clonic motor perseveration of the right hand. With the right hand, she would hit the bed, her own body, cutlery, and nearby objects before her. She knocked herself on the head if we asked her: “Do you have a headache?”, and on the leg if we asked her: “Do you have leg pain?”. The clonic motor perseveration stopped when she held objects, made a fist, changed posture, or restrained the right hand with the left one, but would recur several seconds later. It never ceased for long unless she fell asleep. On her second day in hospital, she was still abulic, mute, and indifferent. She used a toothbrush, chopsticks, towels, and fastened and undid buttons normally. On the fourth day, the frequency and the amplitude of the clonic motor perseveration decreased. She laughed, displayed more facial expressions, and pronounced an “e” sound. On the 11th day, she began to communicate with her family by writing. Next day, she spoke a complete sentence to complain about the noise made by the neighbouring patient, which was the only one sentence she had spoken since her admission. Brain MRI on the 13th day showed a left anterior cerebral artery infarct (figure A, B).
Neuropsychological tests were done from day 6 to 11. The patient had had a high school education. She had no spontaneous speech and could not repeat even simple words, but her verbal comprehension was intact and she could correctly name real objects and photographs of them. She performed line bisection and cancellation tasks very well. On day 11, the patient wrote about her illness on request (figure C), although she was still mute. The handwriting was legible and we easily transcribed it to a printed form (figure D). Initially, she wrote quite normally, “I am....My physical condition is not good and (I had) a syncope on the road, and was sent to this hospital by a friend. Now I am well” (figure D, the first and second lines). In figure D, the seventh character right of the comma on the second line (we defined as L2R7) and L3R6 should be L4R7, which is an orthographic paragraphia. Then, she began to rewrite sentences from L3, although not exactly identical. These repetitive sentences were divided into two clauses by a comma. The first clause said, “My physical condition is not very good”, which she repeated 17 times (L3 to L19). The second clause said, “( I ) was sent to this hospital by a doctor of this hospital”. There were several minor errors in the second clauses, including character omissions (between L3R4 and L3R5, L10R6 and L10R7, and L12R3 and L12R4) and redundant characters (at L7R6 and L23R10 to R12). She changed “friend” (L3R2 and L3R3) to “doctor” (L8R4 and L8R5) and unnecessarily repeated the modifier “this hospital” twice (L14R2 and L14R3 as well as L14R8 and L14R9). She wrote five identical copies of a long sentence (L14 to L18 of figure D). She stopped writing when the space on the paper was used up.
Our patient was discharged on the 14th day. Her husband reported that on the 17th day she watered flowers, but played with the water at the same time. She rapped kitchen utensils with a spatula when cooking, and rapped bowls with a spoon when eating. Three days later, she began to repeat phrases, and followed events in the stock market on television. However, she became angry with her husband without reason on the 21st day, which was unprecedented. On the 26th day, her verbal output seemed normal. The clonic motor perseveration subsided gradually and eventually stopped.
Most of this patient’s symptoms had been described elsewhere.1 The repetitive writing had been reported only for letters, characters, and phrases.2 To our knowledge, repetition of a long meaningful sentence has never been reported. Our patient seemed totally involved when writing the sentences, and would not be distracted. Furthermore, the clonic motor perseveration and the repetitive sentence writing showed similarities in that both were repetitive and elicited by a stimulus. The clonic motor perseveration was elicited by nearby objects attracting her, and the repetitive sentence writing by an idea to describe her illness.
Regarding our patient’s ability to write while she was mute, we suggest two points. Firstly, her inner language was intact. This resembles “dynamic aphasia” with which patients seem almost mute in conversational speech, but show a dramatic preservation of the ability to name objects, to read, and to repeat sentences.3 Kleist thought that patients with dynamic aphasia had intact verbal propositional thought and intact “sentence schema” but these were disconnected.4 Our patient wrote sentences with correct grammar and word selection to describe a meaningful idea, which supports this notion. Secondly, the mechanism for writing was intact. The neural substrates accounting for writing were unaffected in our patient, as shown by the proper sentence syntax and legible ideograms.
Dominant mesial frontal lesion may impair the processes of modification and monitoring of the motoric components of writing, resulting in disinhibition and loss of monitoring. Moreover, Shallice5proposed an idea of “supervisory attentional system” which controls and modulates the lower level processes. This system is activated when a person is dealing with a non-habitual condition and is thought to be a frontal lobe function. This explains why our patient rewrote the sentences so many times, yet did not detect the errors in the writing.
We thank Professor Atsushi Yamadori of Tohoku University, Japan for his helpful comments in the preparation of this manuscript.
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