Brain Injury ISSN: 0269-9052 (Print) 1362-301X (Online) Journal homepage: http://www.tandfonline.com/loi/ibij20 Agraphia with abnormal writing stroke sequences due to cerebral infarction Shinichiro Maeshima, Eri Sekiguchi, Koji Kakishita, Hideo Okada, Ryuji Okita, Fuminori Ozaki & Hiroshi Moriwaki To cite this article: Shinichiro Maeshima, Eri Sekiguchi, Koji Kakishita, Hideo Okada, Ryuji Okita, Fuminori Ozaki & Hiroshi Moriwaki (2003) Agraphia with abnormal writing stroke sequences due to cerebral infarction, Brain Injury, 17:4, 339-345, DOI: 10.1080/0269905031000070134 To link to this article: http://dx.doi.org/10.1080/0269905031000070134 Published online: 03 Jul 2009. Submit your article to this journal Article views: 20 View related articles Citing articles: 1 View citing articles Full Terms & Conditions of access and use can be found at http://www.tandfonline.com/action/journalInformation?journalCode=ibij20 Download by: [Tulane University] Date: 31 March 2016, At: 20:59 BRAIN INJURY, 2003, VOL. 17, NO. 4, 339–345 Case study Agraphia with abnormal writing stroke sequences due to cerebral infarction Downloaded by [Tulane University] at 20:59 31 March 2016 SHINICHIRO MAESHIMA{, ERI SEKIGUCHI{, KOJI KAKISHITA{, HIDEO OKADA{, RYUJI OKITA{, FUMINORI OZAKI{ and HIROSHI MORIWAKI{ { Department of Physical Medicine & Rehabilitation, Wakayama Medical University, Wakayama, Japan { Department of Neurological Surgery, Hidaka General Hospital, Wakayama, Japan (Received 1 May 2002; accepted 20 November 2002 ) A 65-year-old, right-handed man presented with speech and gait disturbances. He was alert and cooperative, showing mild right hemiparesis and sensory disturbance. Spontaneous speech was fluent; object naming, word fluency and reading were fully preserved. Sentence repetition and verbal comprehension were mildly impaired. Writing was slow, hesitant and difficult for both spontaneous writing and dictation. Copying was better, although he had some difficulty in copying letters and also complex figures. Sequences of strokes in forming written characters were abnormal; strokes were formed by piecing together several fragments. Computed tomography and magnetic resonance imaging showed a subcortical infarct in the left frontoparietal region. Characteristics of agraphia resembled ‘apractic agraphia’ and agraphia may have resulted largely from loss or unavailability of the memory of motor patterns necessary for writing letters. Introduction Dysgraphia unaccompanied by a speech disorder or disturbances in reading is called ‘agraphia’ [1], which may be classified into aphasic and apractic agraphia [2]. Aphasic agraphia is an abnormality in the process by which a semantically and phonemically appropriate letter or character is selected and arranged to express the content of writing; apractic agraphia is an abnormality in the process through which each letter or character selected is expressed in writing by movements [3, 4]. The Japanese language has two types of character system: Japanese syllabary (syllabograms) and Chinese characters (morphograms). The English alphabet consists of phonemic letters uniquely corresponding to English phonemes, while Japanese syllabic characters (preferably called letters) uniquely correspond to Japanese syllables. In contrast, a Chinese character often has its own complex letter form where several pronunciations are put together to indicate a meaning; this function is referred to as a semantic function. These differences in information processing Correspondence to: Shinichiro Maeshima, MD, PhD, Department of Physical Medicine & Rehabilitation, Wakayama Medical University, 811-1 Kimiidera, Wakayama 641-0012, Japan. e-mail: maeshima@wakayama-med.ac.jp Brain Injury ISSN 0269–9052 print/ISSN 1362–301X online # 2003 Taylor & Francis Ltd http://www.tandf.co.uk/journals DOI: 10.1080/0269905031000070134 S. Maeshima et al. 340 Japanese have been emphasized by Iwata [5], and few studies have considered details or order of writing for Chinese characters in Japanese patients with agraphia [6–9]. A patient is reported with a sub-cortical infarction in the dominant frontal and parietal lobes causing dysgraphia characterized by apractic agraphia. The patient showed differences in impairment between Japanese syllabary and Chinese characters. Case report Downloaded by [Tulane University] at 20:59 31 March 2016 A 65-year-old, right-handed businessman presented to the hospital 2 days after onset of difficulty in speaking and walking. He had been treated for diabetes and hypertension for 3 years, but had no history of neurological problems. He had a 10year history of cataract in the left eye. His educational level was 12 years. Neurologic examination Consciousness was clear, orientation was nearly normal. The cataract had essentially blinded the left eye; in the right eye visual fields were normal. Mild right hemiparesis and decreased superficial and deep sensation on the right side were noted. Deep tendon reflexes were increased on the right and the right plantar response was extensor. Figure 1. Results of Standard Language Test for Aphasia. Agraphia with abnormal stroke sequences 341 Neuropsychological examination The patient was polite and cooperative during the examination. Spontaneous speech was fluent and no abnormality was noted in naming objects or reading aloud. Sentence repetition was somewhat impaired as was comprehension of written characters. Writing was poor, writing names, dictation and copying were all impaired (figure 1). No unilateral spatial neglect was found. A standardized test of higher motor function did not indicate any ideomotor, ideational or buccofacial apraxia. Downloaded by [Tulane University] at 20:59 31 March 2016 Neuroradiologic examination Computed tomography (CT) of the brain performed on admission showed multiple small infarct in the deep white matter. Magnetic resonance imaging (MRI) 5 days after admission revealed a left frontal and parietal subcortical infarct (figure 2). The left carotid angiogram showed an occlusion in the middle cerebral artery as well as good collateral circulation from the anterior cerebral artery. Single-photon emission CT (SPECT) 2 weeks after admission revealed an area of low perfusion centred in the subcortical white matter and extending from the left frontal to the left parietal lobe (figure 3). Figure 2. Magnetic resonance imaging 5 days after admission revealed a left frontal and parietal subcortical infarct. S. Maeshima et al. Downloaded by [Tulane University] at 20:59 31 March 2016 342 Figure 3. Single-photon emission CT 2 weeks after admission revealed an area of low perfusion centered in the subcortical white matter and extending from the left frontal to the left parietal lobe. Clinical course The repetition and comprehension disorder disappeared in 3 weeks (figure 1), but dysgraphia persisted as described below (figure 4). Constructional apraxia was less severe than dysgraphia. Figure drawing was slow with many pauses, but eventually was accomplished well after much time. The revised Wechsler Adult Intelligence scale (WAIS-R) showed a verbal intelligence quotient (IQ) of 65, a performance (IQ) of 70 and a combined IQ of 65. Mini-Mental State and Raven’s Coloured Progressive Matrices scores were 26/30 and 21/36, respectively. Dysgraphia in the present case showed the following features: First, writing movements were slow with many pauses, so much time was required to write one character. Secondly, spontaneous writing and dictation showed agraphia in a similar manner when performed with either hand. Thirdly, agraphic symptoms were present with both Japanese syllabary and Chinese characters. Correct responses among the syllabograms numbered 16 characters out of 80 (20.0%); incorrect wrong responses resulted mainly from incomplete or collapsed forms. Complex Chinese characters having many vertical, transverse and oblique lines as their strokes consistently showed apractic collapses. Incorrect responses concerning Chinese characters consisted chiefly of difficulty with recall and partial responses to the correct forms of these characters. Of 76 Chinese characters in comon use that are learned in the first grade of elementary school, 51 characters (67.1%) were correctly produced during dictation. Consideration of errors involving the 25 other characters showed the following patterns; characters similar to the correct ones but collapsed in their form accounted for 22 characters with errors (88.0%). Characters recalled with Downloaded by [Tulane University] at 20:59 31 March 2016 Agraphia with abnormal stroke sequences 343 Figure 4. Examples of dictation and copy. Dictation and copy were impaired. difficulty or not at all numbered 3 (12.0%). One character was produced only in part, such as its left-hand or right-hand radical (4.0%). One character was given an entirely novel form (4.0%). Additionally, characters written by making strokes in the wrong order numbered 5 (20.0%). Of 145 Chinese characters in common use that are learned in the second grade, 34 characters (23.4%) were produced correctly during dictation. Among the other 111 characters, those recalled with difficulty or not at all included 21 (18.9%); characters similar to the correct ones but collapsed in form numbered 90 (81.1%); characters produced only in part were 7 (4.8%); characters with novel forms were 2 (1.8%). Characters written by making strokes in the wrong order included 15 of the 145 (10.3%), regularly resulting in incomplete or collapsed forms. Fourthly, the patient had difficulty in orally describing of components comprising the characters that posed problems. In other words, he could not relate the leftor right-hand radicals to the original Chinese character. At the same time, he could easily make a word by arranging several characters, select the correct Chinese character from among similar ones and point out what was wrong with miswritten characters. Fifthly, copying of characters showed improvement, as well as errors involving similar formed characters. Lastly, some strokes were made in an incorrect order in correctly produced Chinese characters and also some correctly copied characters. Discussion The present case included no disturbance of consciousness, and slight problems with comprehension and sentence repetition soon resolved. No other apraxis was present, nor was unilateral spatial neglect. On the other hand, writing was poor in writing names, writing to dictation and copying. Writing movements were slow and halting. Copying took much time and contained errors involving similarly Downloaded by [Tulane University] at 20:59 31 March 2016 344 S. Maeshima et al. formed characters, suggesting that this case had characteristics of apractic agraphia, as described by Alexander et al. [10]. The agraphia associated with a predominantly parietal lesion is sometimes classified into two categories, either one where a semantic and phonemic letter or character cannot be appropriately selected and arranged to express the content or one where the letter or character selected cannot be correctly produced by writing movements [3, 4]. The former type of disturbance affects writing to dictation and spontaneous writing, producing a variety of errors including a partial response (writing part of an existing character), an approximate response (writing an approximation of a correct character), a displacement response (writing another character in place of the correct character), a novel character (substituted for the correct character) and no response [11]. At the same time, copying is always preserved in this sub-type, and a capability of producing normal characters persists [12]. When writing movement patterns are affected, the disturbance resides in the movement programme containing the character strokes as well as the order of making strokes in writing the complete character [13], so almost all incorrect responses are expressed as characters similar to those intended to be copied. No displacement response occurs, but the ability to copy is affected. Since the present case showed mainly responses expressing the characters as similarly formed ones without any displacement response, the patient had an abnormality in the expression process involving writing movement patterns. Written Japanese has two types of characters; Japanese syllabary is rich in kinesthetic elements, while Chinese characters are rich in constructional elements. According to Iwata [5], in writing Japanese syllabary an auditory image leads directly to a kinesthetic image; however, in writing Chinese characters, auditory images lead first to visual images which lead to kinesthetic images. In other words, Japanese syllabary is presumably more automatic in writing than Chinese characters. In constructional agrapia, as defined as a dysgraphia associated with a marked constructional apraxia, impairment in writing of Chinese characters is severe, but writing of Japanese syllabary is usually intact. In contrast, the present patient had more correct responses with Chinese characters than with Japanese syllabary. For Japanese syllabary, his correct-response rates were remarkably low in writing letters demanding continuous movement patterns such as ‘ ¼ nu’ and ‘ ¼ ne’. For writing Chinese characters, errors were more frequent for Chinese characters learned in the second grade and showed more incorrect sequences of making strokes than for characters learned in the first grade. Correct-response rates should not simply be compared between Chinese characters and Japanese syllabary, because differences may reflect familiarity or experience with writing various characters rather than differences in information processing between character systems. At the same time, dysgraphia involving order of strokes, and also approximate responses, were more evident with the Chinese characters with low difficulty from the first grade than with those with high difficulty from the second grade; this may reflect a disturbance in writing movement patterns that had become more automatic by being mastered earlier in life. Furthermore, the present case was accompanied by a decrease in perfusion extending from the left frontal and parietal sub-cortex to the superior parietal lobule, but the decrease in perfusion in the angular gyrus was relatively slight. Case reports from Europe and the US have given great importance to the superior parietal lobule of the dominant hemisphere as the lesion site responsible for apractic Downloaded by [Tulane University] at 20:59 31 March 2016 Agraphia with abnormal stroke sequences 345 agraphia [10]. On the other hand, according to Kawamura [11], who had studied pure parietal-lobe agraphia in Japan, the displacement response results from a disturbance at the stage where a character is integrated as a visual memory. In contrast, a partial response involves disturbance of the process by which the associated visual, auditory and somatic sensory inputs that are involved in writing characters are integrated as a visual memory. So a writing movement can be carried out (i.e. a disturbance of the output process to the somatosensory association areas or premotor area). The present case included no displacement response and few partial responses, but still showed many approximate responses where the characters formed had similarities to and represented collapsed forms of the correct response. In other words, dysgraphia in the present case principally resulted from a disturbance of writing movement patterns, which may qualitatively differ from pure parietallobe agraphia. Agraphias resulting from a parietal lobe lesion are varied and may include extraneous cases, but the present case appeared to be apractic agraphia caused by a disturbance in the superior parietal lobule of the dominant hemisphere. References 1. Dubois, J., Hecaen, H. and Marcie, P.: L’agraphie ‘pure’. Neuropsychologia, 7: 271–286, 1969. 2. Roeltgen, D. P. and Heilmen, K. M.: Apractic agraphia in a patient with normal praxis. 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