BRAIN AND LANGUAGE 47, 197~213 (1994) Jargonagraphia in Kanji and Kana in a Japanese Crossed Wernicke’s Aphasic Nami [Hort Kyoritsu Rehabilitation Hospital, Kawanishi, Japan TosHiniro KasHtwaci Kyowakai Hospital, Japan ASAKO KaSHIWAGI Kyoritsu Rehabilitation Hospital, Kawanishi, Japan AND Hirotaka TANABE Osaka University Medical School, Japan A right-handed Japanese crossed Wernicke’s aphasic showed complete neolo- gistic jargonagraphia in kanji and kana with anosognosia of his writing deficits. Prominent jargonagraphia in kanji is quite rare and has not been previously de- scribed in the literature. Marked dissociation between speaking and writing during the course suggested that his jargonagraphia might be unique to crossed aphasia. His condition was interpreted for the main part by Yokoyama, Okubo, Doseki, and Yamadori’s hypothesis (1981) that free-running on of motor engrams of char- acters stored in the left hemisphere caused jargonagraphia in crossed aphasia. This case of jargonagraphia in kanji suggests that this hypothesis should be sup- plemented by the following points: (1) in kanji, not necessarily a character as a We thank Dr. Yoshitaka Ohigashi (Kyoto University) for many valuable comments, Dr. Atsushi Yamadori (Hyogo Institute for Aging Brain and Cognitive Disorders) for helpful suggestions on the explanation of the patient's symptoms, Dr. Kazuhiko Kinugawa (Kyowa- kai Hospital) for help in interpreting the patient’s medical history, and Drs. Eiichi Murakami and Kenzo Kiso (Kyoritsu Rehabilitation Hospital) for cooperation in this study. We also thank Dr. Tsuneo Takai and Nobuko Ohta (Yodogawa Christian Hospital) for providing medical and neuropsychological information on the patient at the onset. Portions of this case were presented at the 15th annual meeting at the Neuropsychology Association of Japan, Ohita, September, 199]. Address correspondence and reprint requests to Nami hori, Kyoritsu Rehabilitation Hospital, 1-39-1 Hirano, Kawanishi, Hyogo, 666-01, Japan. 197 0093-934X/94 $6.00 Copyright © 1994 by Academic Press, Inc, All rights of reproduction in any form reserved, 198 IHORI ET AL. whole, but radicals or parts of radicals, function as motor units, which may be released to produce jargonagraphia in kanji; (2) free-running on of visual images as well as of kinesthetic images should be considered especially in written jargon in kanji. © 1994 Academic Press, Inc. INTRODUCTION Crossed aphasia (CA) was first described by Bramwell (1899) as apha- sia due to hemispheric lesions ipsilateral to the preferred hand. This term was recently used for aphasic syndromes caused by right hemisphere lesions in dextrals (Hécaen & Albert, 1978). Whether language functions in the right hemisphere of crossed aphasics (CAs) exactly mirror those in the left hemisphere of standard (right-handed, Jefi-hemisphere damaged) aphasics remains unclear. Brown and Hécaen (1976) described similari- ties between CA and childhood aphasia, suggesting incomplete lateraliza- tion of language in each hemisphere in CAs. Carr, Jacobson, and Boller (1981), Henderson (1983), and Basso, Capitani, Laiacona, and Zanobio (1985) argued that the correlation between types of aphasia and loci of lesions in CAs might not be very different from that observed in standard aphasics. Alexander, Fischette, and Fischer (1989) reviewed the litera- ture and concluded that 65% of CAs could be considered to mirror stan- dard aphasia, although 35% of CAs have anomalous representation of language. In addition, several specific features in CA such as written jargon have been noted (Basso et al., 1985; Alexander et al., 1989). Since Pillon, Desi, and Lhermitte (1979) described patients with CA who showed jargonagraphia, several case reports of CA with jargona- graphia have been published (Yokoyama, Okubo, Doseki, & Yamadori, 1981; Assal, 1982; Habib, Joanette, Ali-Cherif, & Poncet, 1983; Basso et al., 1985; Sapir, Kokmen, & Rogers, 1986; Demeurisse, Hublet, Coe- kaerts, Derouck, & Capon, 1986; Washimi, Makishita, Miyasaka, Fujita, & Yanagisawa, 1987; Alexander et al., 1989; Ochipa & Rothi, 1989). According to Lecours and Rouillon (1976), jargonagraphia in general is characterized by (1) normal or greater than normal written output, (2) preservation of graphism and written automatisms, (3) production of para- graphias and of written neologisms, and (4) production of dyssyntactic transformations. Although some of the above reports do not provide suf- ficient information concerning the extent to which the writing deficits fit the definition of jargonagraphia, the literature suggested that jargona- graphia can appear in any aphasic type of CA, and that the written jargon tends to be dissociated from spoken output. Alexander et al. (1989) noted jargonagraphia as a feature in an anomalous type of CA, and a relatively frequent occurrence of jargonagraphia in CA is suggested. However, in standard aphasia, jargonagraphia appears in Wernicke’s or jargon aphasia (Kinsbourne & Warrington, 1963; Hécaen et al., 1963; Kertesz & Benson, 1970; Kertesz, 1979; Lecours & Lhermitte, 1983; Cappa et al., 1987), JARGONAGRAPHIA IN CROSSED APHASIA 199 and written jargon tends to show the same propensity as spoken jargon. Furthermore, jargonagraphia in standard aphasia is considered quite rare (Kertesz & Benson, 1970; Benson & Geschwind, 1985). Cappa et al. (1987) reported that 6 of 1076 aphasics (.6%) showed jargonagraphia, although one was left-handed and another was bilaterally damaged. These differences between jargonagraphia in CA and in standard aphasia sug- gest that specific mechanisms cause jargonagraphia in CA. Although such mechanisms remain unknown, several interpretations were suggested (Assal, 1982; Basso et al., 1985; Alexander et al., 1989). Assuming that motor engrams of characters in dextrals are stored in the left hemisphere, Yokoyama et al. (1981) proposed the hypothesis in which those engrams run on freely without mediation of the language area in the right hemisphere.' This hypothesis seems to be the most persuasive, since it might explain phenomena such as the appearance of jargona- graphia in any aphasic type in CA and the dissociation between speaking and writing. In the Japanese literature, there are four reported cases of CA with jargonagraphia (Imura, Asakawa, Hotta, & Nihonmatsu, 1959; Yokoy- ama et al., 1981; Washimi et al., 1987). The Japanese orthographic system has two kinds of characters, kanji and kana, Kanji are morphograms representing phonetic and semantic values simultaneously. Kana, simpli- fied forms of kanji, are syllabograms (see Appendix). Neuropsychological studies have suggested that kanji and kana are processed differently (Sa- sanuma, 1974; for reviews, see Paradis, Hagiwara, & Hildebrandt, 1985). The above cases demonstrated jargonagraphia predominantly in kana. Since kana are graphically simple, thought to be closely related to kines- thetic image (Iwata, 1986) and, therefore, expected to freely run on easily, it seems possible to interpret jargonagraphia in kana in terms of the hy- pothesis of Yokoyama et al. (1981). A similar condition might occur in alphabetic languages. However, the fiterature suggests that jargona- graphia in kanji rarely occurs. Most kanji are graphically much more complex than kana and are considered strongly related to visual images (Iwata, 1986). Semantic and visual factors may restrain jargon-like usage of kanji. However, if graphically simpler components of kanji such as radicals or parts of radicals (see Appendix) function as motor units as suggested by Kashiwagi and Kashiwagi (1989), it may be possible for those units in kanji to run on freely. We present a Japanese case of crossed Wernicke's aphasia with severe jargonagraphia in kanji as well as in kana. Prominent, neologistic jargon persisted in writing but not in speaking. Such a dissociation between ' Geschwind (1969) considered jargon speech to be due to ‘*. . . the running on of the relatively isolated Broca’s area.”’ Yokoyama et al. (1981) applied this hypothesis to jargona- graphia in CA. 200 THORI ET AL. speaking and writing suggested that his jargonagraphia was typical of CA rather than of standard Wernicke’s aphasia. Since his production of jar- gon in kanji was interesting, we investigated his kanji processing and the characteristics of his written jargon in kanji. We concluded that Yoko- yama et al.’s hypothesis (1981) could be applied to jargonagraphia in kanji as well as in kana in this case. We also discuss the free-running on of visual as well as kinesthetic images of characters. CASE REPORT Case History ES, a 68-year-old male bus driver with 8 years of formal education, had always been right-handed in daily activity with no history of left- handedness. He had no brothers or sisters, but his mother and two chil- dren were all right-handed. He had no history of developmental problems or neurologic illness in childhood. He sustained a head injury at the age of 23 during a battle in the second World War. As recounted by both the patient and his wife, he hit his head against metal, but he had no neurolog- ical or neuropsychological problems after the injury. His past medical history included hypotension and dyspepsia. He began having headaches without vomiting since the beginning of February, 1990 and subsequently experienced articulatory problems and anomia on 13 February. On February 16 and 18, he suffered mild hemipa- resis in the left limbs. He also had transient amaurosis in the left eye due to transient insufficiency of the left ophthalmic artery during this period. He was diagnosed with cerebral infarction at another hospital and contin- ued there as an outpatient once a week. On the carotid Doppler test performed February 22, severe stenosis before the bifurcation of the right carotid artery was suspected. A CT scan performed on February 28 showed hypodense areas in the right temporal lobe. Speech and language were evaluated by a speech therapist on March 6. His spontaneous speech was normal without a paraphasia. His performances on auditory comprehension, repetition, oral reading, and calculation were within nor- mal limits, although slight deficits in written commands were found. Mild anomia (3 of 20 items could not be named on confrontation naming) and some phonological paragraphias in kana were also observed. On March 25, he experienced left extremity weakness again, and on April 6, fluent aphasia appeared. He was hospitalized at another hospital for rehabilitation on April 13 and transferred to Kyowakai Hospital for further rehabilitation on May 28. Neurological Findings On admission, he was alert and cooperative. There was very mild hemi- paresis on the left side of the body including the face. Barré’s sign was JARGONAGRAPHIA IN CROSSED APHASIA 201 positive in the left upper limb. No abnormality was found on the right side of the body. Visual fields were normal on confrontation testing. A CT scan (Fig. 1) performed on May 21, 1990 revealed hypodense areas in the lateral portion of the right putamen which extended laterally to the insula and upwardly to the corona radiata. Another diffuse hypodense area was found in the right temporal lobe. No abnormal density was found in the left hemisphere. SPECT (single photon emission CT) performed on April 16, 1991 (Fig. 2) disclosed a decrease in regional cerebral blood flow in the right temporal and parietal lobe, a part of the right occipital lobe, the right basal ganglia, and the left cerebellum. There were no abnormalities in the left cerebral hemisphere. Neuropsychological Findings Neuropsychological investigations were initiated 7 weeks after the on- set of aphasia and continued for 6 weeks. A questionnaire consisting of 14 questions concerning hand use (e.g, using a pencil, combing hair, holding chopsticks, etc.), 4 for leg use (e.g., kicking a ball, etc.), and 1 for eye (peeping through a hole) was answered by the patient and his wife. Although two questions concerning leg use were answered ‘‘unclear,”’ all other responses indicated a pronounced preference for the right side. The profile of the Standard Language Test of Aphasia (SLTA) (Hase- gawa et al. 1977), one of the aphasia batteries popularly used in Japan, is shown in Fig. 3. Since the lesions were localized in the right hemisphere Fic. 1. CT scan (May 21, 1990). 202 IHORI ET AL. Fic. 2. SPECT (April 16, 1991). and he was strongly right-handed, crossed aphasia Wernicke’s type was diagnosed, based upon the following features of his language deficits. Spontaneous speech was fluent and well articulated. although some com- binations of sounds could not be differentiated. He had marked word- finding difficulties. Sentences included verbal paraphasias and neolo- gisms, and his speech sometimes became jargon-like. There was no abnormal press of speech and the quantity of speech was almost normal. On confrontation naming, repetition, and reading aloud both of kanji and kana, almost all of his responses were neologistic. He seemed to be con- scious of his speech deficits to some degree: he often tried to use gestures to compensate for word-finding difficulties. Auditory comprehension was relatively preserved compared to speech; in conversation, he could understand simple questions about matters of immediate personal relevance; on SLTA, he had difficulty responding to multi-stage oral commands. Disturbances in comprehending written materials were similar to those in auditory comprehension. However, dissociation was observed between kanji and kana, comprehension of the former being much better than that of the latter. JARGONAGRAPHIA IN CROSSED APHASIA 203 PERCENT CORRECT . Word Recogn. . Sentence Recogn. » Oral Commands . Kana Recogn. . Confront. Naming . Word Repetition . Description Activities . Narrative . Sentence Repetition . Animal Naming . Oral Rdg. (Kanji wds.) (Kanas} (Kana wds.) (Sentences) . Kanji Word Recogn. . Mana Word Recogn. . Sentence Recogn. _ Written Commands . Written Naming(Kanji) (Kana) . Narrative Writing . Dictation(Kanas) (Kanji wds.} Fyc. 3. The profile of the Standard Language Test of Aphasia (SLTA). (@) 90.6.5, (©) 91.2.19, (—) the mean score for 150 non-aphasics. The patient demonstrated severe writing disturbances, forming a num- ber of meaningless clusters of well-formed characters. Most of the char- acters were hiragana, although there were kanji, katakana, and numerals. Perseveration was observed. Figure 4 shows a sample of his spontaneous writing. His writing in kana was completely neologistic, and he used well-formed kanji in a meaningless or perseverative manner; syntactic structure was not recognizable. Such disturbances were observed in writ- ten tasks with either hand. Most kana used were seion, and a smali num- ber of dakuon was also used; handakuon and youon were not used at all (see Appendix). In contrast, his graphic movements such as the order of strokes and the speed of writing were normal. He also wrote abundantly. The characteristics of his writing fit the definition of jargonagraphia by Lecours and Rouillon (1976) quite well. He copied sentences well except for a few mistakes such as omission, substitution, or addition of charac- ters. He seemed unaware of his writing deficits. For example, he kept a diary and showed it to a ward nurse without hesitation. He sometimes tried to write down what he wanted to say to compensate for his speech disturbances. Awareness of deficits was dissociated between speech and writing. Calculations were severely disturbed. Matching one-digit numbers and Arabic numerals and reading and writing numerals were defective. He 204 IHORI ET AL. ie mid SX be a4 fircaite &, TP idofs Ha = AN (Rat he a K pita ia ost wy peqie HASAS Bite 42175 ~ 7A Ht MP boats 1 ott em st SD IR a VU B21 3. tbs WR array sal Dora grry (Piles A$ Up as ot ER at t= Fic. 4, An extract from the patient's diary (2 months after the onset of aphasia). Kanji are underlined. The patient wrote meaningless combinations of real kana. He also used well-formed kanji in a meaningless manner. also had difficulty in counting and writing numerals sequentially; he counted in a random order in two-digit numbers, and his sequential writ- ing of more than one-digit numerals was iterative. He had great difficulty in reading a watch and using money. His performance in Raven’s Colored Progressive Matrices was 22/36, and that in Kohs’ Block Design Test was 60 (IQ). His performance IQ in WAIS was 74. His copies of Rey’s diagram and a cube with each hand were good. He had no problems in copying line drawings. He demon- strated good ability in visuo-spatial functions and praxis. There was no sign of hemispatial neglect. He did not show ideomotor apraxia or ide- ational apraxia in either hand. Slight bucco-facial apraxia was observed. Melodies of familiar songs were preserved, but he sang them with neolo- gisms. He appeared normal in speech prosody and in emotion. Investigation of Kanji Processing The following tasks were used to investigate the patient’s kanji pro- cessing. These investigations began 2'2 months after the onset of aphasia and continued for 3 weeks. Task 1. Reading comprehension in kanji. Eighty concrete nouns writ- ten in kanji were presented one by one. The patient was required to choose one of six pictures to match the stimulus. His correct response was 86%. The same task was also performed in kana; the percentage correct was 50%. Task 2. Copying kanji. Eleven single kanji containing 3~10 strokes and 6 compound kanji consisting of 2 characters containing 7-21 strokes were JARGONAGRAPHIA IN CROSSED APHASIA 205 presented. With the left hand, there were three minor mistakes such as omission of a stroke or the substitution of another visually similar radical. However, overall the copying kanji was well preserved in both hands. Task 3. Written naming. Nineteen pictorial stimuli of concrete nouns selected from Task 1 were used. Target words were composed of 1 to 4 kanji for each and 36 in total. The patient was required to write the name of a picture in kanji. Examples of his responses are shown in Fig. 5. There were no correct responses. Most of the responses were meaning- less combination of characters (17 of 19 responses with both hands). Most of the kanji he wrote did exist in Japanese, but some combined existing radicals or parts of them into meaningless characters (6 of 50 kanji written with the right hand and 10 of 38 with the left hand). More than half of the kanji were perseverated. Task 4. Written description of activities. Five action pictures were pre- sented one by one and the patient was required to describe each picture in writing. Figure 6 shows an example of his response. Neologistic jargon, predominantly in hiragana, was prominent, and no syntactic markers Right Left Hand Hand * cherry 70 WY ? blossoms Lo 31 = sVe iit # 1 ree clinical > a thermometer < a [ee] thunder * * iv he * td oa p> fe y thread a] Fic. 5. Examples of written naming in kanji with the right and left hands. All of the Tesponses are neologisms which consist of irrelevant kanji. For example, for the stimulus #& (‘cherry blossoms’’), he wrote #* (#% means “‘to fall’’ and * means ‘‘a book’’). The kanji marked with asterisks are nonexistent ones combining existing radicals or parts of radicals. 206 THORI ET AL. BED co FSP RA op BO FDR CHEMO TWD ‘A boy is having a haircut at a _barbarshop.* Right Hand Bd Beg yv Goto -7 Ub bo Rt Caos te Fic. 6. An example of written description of activities. An example of a correct Japanese expression is shown on the top. Kana transcription for each kanji are presented above kanji in small hiragana. The patient’s responses are neologistic jargon and the kanji # (‘answer’) and 44 (‘ta word’’) are not relevant at all in this situation. The kanji marked with asterisks are nonexistent combinations of existing radicals. The kanji + means *‘a child,’’ but it is combined in a meaningless manner with the nonexistent kanji. #& . were discernible. Some kanji appeared, but most were irrelevant to the contexts. There were some non-existent kanji composed of existing radi- cals. No obvious difference was observed between performances with the right and left hands. Touten or a Japanese period (a small circle) was appropriately used at the end of each sentence. Task 5. Visual construction of kanji corresponding to meaning. To investigate the ability to construct kanji corresponding to meaning through a visual modality without the help of motor-kinesthetic factors normally used in writing, the kanji construction test developed by Asano, Takizawa, Hadano, Morimune, and Hamanaka (1985) was used. The pa- tient was first required to orally name a line-drawn picture; when he failed, the name was given verbally, and then he was asked to choose radicals printed on cards placed in front of him to construct the kanji. Correct responses were made in only 2 of 36 trials. Summary. The patient’s graphic movements were normal in each hand. The comprehension of kanji was relatively good. He had difficulty in recalling the visual image of kanji corresponding to meaning. Since most of the kanji he wrote, including the complicated ones, did exist, his mem- ory for the visual images of graphic forms seemed to function relatively well, although the small number of nonexistent kanji might reveal gaps in his memory. The Evolution of the Patient’s Aphasia and Jargonagraphia The results of SLTA performed 10 months after the onset of aphasia are profiled by the dotted lines in Fig. 3. In conversation, neologisms JARGONAGRAPHIA IN CROSSED APHASIA 207 decreased and appropriate words as well as verbal paraphasias increased. However, his writing was still severely disturbed. Discrepancies between speaking and writing were observed during the course. Table 1 shows the results of confrontation naming and written naming in SLTA performed 7 months after the onset of aphasia. In con- frontation naming, four of six responses were correct; the errors were verbal paraphasia and neologism. In written naming, most responses in kanji and all responses in kana were neologistic. These results suggest that his spoken and written disturbances may be caused by different mechanisms. Jargonagraphia remained, but a discrepancy between kanji and kana writing appeared. In spontaneous writing such as the diary, kanji usage decreased remarkably. In addition, a follow-up study of kanji processing performed 5 through 7 months after the onset of aphasia revealed some improvements. In Task 3, he often indicated ‘‘don’t know”’ or tried to write in kana instead. He became self-conscious of writing deficits in kanji. In Task 5, the correct responses increased from two to five. The results of SLTA in Fig. 3 also show some improvements in writing kanji. However, the quantity of kana usage did not change. He was still anosog- nostic toward jargonagraphia in kana. His diary entries became less frequent. In Fig. 7, a writing sample 13 months after the onset of aphasia shows completely neologistic jargon written only in hiragana. Follow-up study | year and 9 months after the onset of aphasia showed that dissociation between speaking and writing was more salient and jargonagraphia remained prominent, although he did not write as prolifically as before. TABLE 1 Discrepancy between Oral and Written Language (7 Months after the Onset of Aphasia) Target words Written naming Kanji Kana Oral naming Kanji Kana Mountain fy Bord /jama/ correct “Ea By BRTOH Dog R We finu/ correct correct ct Book x A ‘hon/ (ka, kami] . * ied < Clock Wat kit /tokei/ correct ek EAE < Newspaper BM LABA /finbun/ correct wp ® SARC OT Pencil %® AAUVD /enpitsu/ [denwa, jaiku] AF Lao (hiragana) or 7 (katakana) (the phonological representation for both is /tsu/) which is a geminate consonant marker. In the above exam- ples, underlined kana, a, b, c, d, and e are seion, dakuon, handakuon, youon, and sokuon, respectively. 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