Q0619–CN0401 / Mar 4, 03 (Tue)/ [16 pages, 14 tables, 4 figures, 1 footnotes] – S endings EDITED FROM DISK COGNITIVE NEUROPSYCHOLOGY, 2003, 20 (2), 99–114 VOWELS IN THE BUFFER: A CASE STUDY OF ACQUIRED DYSGRAPHIA WITH SELECTIVE VOWEL SUBSTITUTIONS Maria Cotelli I.R.C.C.S. S. Giovanni di Dio, Fatabene Fratelli, Brescia, Italy Jubin Abutalebi Vita Salute San Raffaele University, Milano, Italy Marco Zorzi and Stefano F. Cappa Vita Salute San Raffaele University and San Raffaele Scientific Institute, Milano, Italy We report the case of a patient who recovered from a clinical picture of fluent aphasia to selective dysgraphia. The features of the writing disorder were compatible with a graphemic output buffer dysfunction (errors in all spelling tasks and for all type of material, affected by word length and consisting mostly of graphemic deviations), with the exception of the lack of transposition errors and position preference. Further, the spelling disorder was selective for vowels, replicating the original observation by Cubelli (1991). A similar, although milder, error pattern was also observed in reading tasks, in particular for nonwords, suggesting that the locus of dysfunction involves a processing stage shared by reading and writing. These findings support the notion that the consonant-vowel status is a property of graphemic representations, and is compatible with that a common buffer is involved in spelling and reading. We discuss the implications of selective vowel disorders for current models of the spelling system. INTRODUCTION The investigation of patients with acquired dysgraphia has played a crucial role in increasing our understanding of the spelling system. All models of the spelling system (Campbell, 1983; Caramazza, Miceli, Villa, & Romani, 1987; Ellis, 1982, 1988; Goodman & Caramazza, 1986; Houghton & Zorzi, 1998) converge in postulating a processing stage, called the “graphemic buffer”. This structure is thought to hold the graphemic representations computed temporarily, according to dual-route models, by the lexical or the sublexical phonology-to-orthography conversion components, in preparation for subsequent, more peripheral, spelling processes. The graphemic representations of both familiar and nonfamiliar words are both held within this graphemic buffer during the execution of the appropriate output processes, that is the computation of specific letter shape representations for written spelling, or the computation of letter name representations for oral spell- Requests for reprints should be addressed to Prof Stefano F. Cappa, DIBIT, via Olgettina 58, 20132 Milano, Italy. We wish to thank Dr Roberto Cubelli and Dr Lorella Algeri who referred the patient to us and contributed to the investigation in many different ways. 2003 Psychology Press Ltd http://www.tandf.co.uk/journals/pp/02643294.html 99 DOI:10.1080/02643290244000158 COTELLI ET AL. ing. In the case of written spelling, the abstract letter-form has been suggested to include both visually and motorically based representations (Rapp & Caramazza, 1997). Selective damage to the graphemic buffer Several recent studies have indicated that the graphemic memory store can be selectively involved in cases of acquired dysgraphia (Aliminosa, McCloskey, Goodman-Schulman, & Sokol, 1993; Caramazza et al., 1987; Hillis & Caramazza, 1989; Kay & Hanley, 1994; McCloskey, Badecker, Goodman-Schulman, & Aliminosa, 1994; Miceli, Silveri, & Caramazza, 1985; Piccirilli, Petrillo, & Poti, 1992; Posteraro, Zinelli, & Mazzucchi, 1988). In their landmark study, Caramazza and coworkers (1987) have attempted to delineate the specific features of the spelling disorders caused by a selective functional lesion to the graphemic buffer (henceforth GBD, graphemic buffer disorder). First, the errors should be quantitatively and qualitatively similar in all spelling tasks, irrespective of both the input modalities (written naming, writing to dictation, spontaneous writing, and delayed copying) and the output modalities (written spelling, oral spelling, or typing), because the graphemic buffer is involved in each of these spelling tasks. Second, the errors should be present for the spelling of both familiar words and nonwords, as the buffer holds both types of stimuli. Spelling performance should therefore not be affected by linguistic factors, such as lexical frequency, grammatical class, morphological structure, or concreteness. Third, as the graphemic buffer is a temporary repository, which can be conceived of as a working memory storage system, characterised by limited space capacity, word length may be expected to affect spelling performance. Fourth, the spelling errors expected from a buffer deficit should mostly consist of graphemic deviations from the target words; they should essentially take the form of substitutions, deletions, additions, or transpositions of letters and of various combinations of these error types. These errors should result in the production of nonwords (Miceli et al., 1985; Nolan & Caramazza, 1983). 100 COGNITIVE NEUROPSYCHOLOGY, 2003, 20 (2) Subsequently, Caramazza and Miceli (1990) have suggested that the nature of the graphemic representations within the graphemic buffer consists of more than a simple linear arrangement of letter identities. According to this simplistic view, the information contained in graphemic representations basically consists of the identity of individual letters and the specification of their correct linear order. By analysing the errors produced by their patient LB, the authors concluded that the graphemic representations included in the graphemic buffer have a multidimensional structure, which codes several different properties of written words separately. In particular, LB’s errors were constrained by the written syllabic structure: For instance, words with a regular alternating CV pattern (e.g., the CVCVCV structure of the Italian word “TAVOLO,” table) were correctly spelled more frequently than words with a more complex syllabic structure (e.g., the VCCVCV structure of the Italian word “ALBERO,” tree). Furthermore, a different distribution of error types in simple and more complex words was observed. Errors like deletion, insertion, and adjacent-letter transposition were found to be uncommon for simple CV words, when compared with words with more complex syllabic words. A further crucial characteristic of the errors produced by LB was the presence of letter substitutions: These errors virtually always involved the substitution of a vowel for a vowel or of a consonant for a consonant. The authors reported also that the performance with geminate consonants was radically different from other CC patterns: Geminate consonants behaved like a single functional unit, were more resistant to error, and were never involved by some types of errors commonly described for other CC patterns. Finally, a crucial observation was that it was the graphemic structure, and not the phonological structure, that determined the distribution of errors. In particular, consonant or vowel clusters that corresponded to a single phoneme behaved as two graphemic elements, and not as a single phonological unit. On the basis of these observations, the authors concluded that graphemic representations have a multidimensional structure that specifies, besides identity and position of the graphemes, gemination, A STUDY OF ACQUIRED DYSGRAPHIA graphosyllabic structure, and the consonant/vowel status. The model of the graphemic buffer put forth by Caramazza and Miceli (1990) is in essence a model of spelling representation (Shallice et al., 1995), and has little to say about serial output processes. In contrast, Houghton and colleagues have developed a connectionist model of spelling in which the “problem of serial order” in the production of letters plays a central role in explaining spelling disorders (Houghton, Glasspool, & Shallice, 1994; Shallice, Glasspool, & Houghton, 1995). Indeed, problems with the control of serial order are manifest in GBD patients, because many aspects of the patients’ errors are strikingly similar to the kinds of errors generally found in serial recall tasks (i.e., mis-orderings and substitutions). The model of Houghton and colleagues is based on a connectionist architecture known as “competitive queuing” (CQ) (Houghton, 1990). When a word is to be spelled, a group of letter nodes in the CQ model are activated in parallel, but with a gradient of activation over them such that letters are more active the sooner they are to be produced. Letters compete to be produced depending on their activation level. Under disruption (in the form of noise added to letter activations), the model has been shown to account for many basic features of the errors found in GBD, such as word length effect, serial position curve, and error types (Shallice et al., 1995). The CQ model, however, could not account for the preservation of the consonant/vowel status. This is because a word was represented simply as a series of letters, with no further structure other than a special marking of doubled letters (geminates). Glasspool and Houghton (2002) have recently developed a successor of the model, which includes a representation of the consonant/vowel status of letters. The revised CQ model produces very detailed simulations of GBD errors, including the preservation of CV status. Peripheral dysgraphia Some agraphic patients appear to be affected at a stage that follows the graphemic buffer. These include disorders at a stage, defined as allographic, which contains spatially coded representations of letter shapes. These patients usually display dissociation between upper-case and lower-case writing (Hanley & Peters, 1996, 2001), or letter case confusions (De Bastiani & Barry, 1989). This is considered to be followed by a stage characterised by the selection of graphic motor patterns (Baxter & Warrington, 1986; Zesiger, Pegna, & Rilliet, 1994). Patients with damage at this stage often write shapes that are not real letters. If the patient produces mostly letter substitution despite good oral spelling, the disorder is considered to affect the access to graphic motor codes from intact allographic representations (Black, Behrmann, Bass, & Hacker, 1989; Del Grosso Destreri et al., 2000). Dysgraphia with a selective deficit for vowels Additional, direct evidence in favour of the hypothesis that the consonant/vowel status of graphemes is represented in the spelling process was provided by Cubelli (1991). The author studied the spelling performance of two patients with a selective deficit in writing vowels. His first patient, CF, had a transient dysgraphia after an ischaemic infarction located in the left parietal lobe of the left hemisphere. When writing words, he omitted all vowels, leaving a blank space between consonants or consonant clusters (e.g., TAVOLINO, little table → T_V_L_N_). The second patient, CW, who had an infarction in the subcortical region of the left frontal lobe, produced errors that almost exclusively involved vowels. His spelling abilities were investigated in depth: His spelling performance was affected only by stimulus length, and not by input or output modalities, lexical status, word frequency, or word class, suggesting a deficit to the graphemic buffer. The most frequent errors were letter substitutions, which, however, respected the consonant/ vowel status of the involved graphemes. In the writing tasks, CW produced 409 letters incorrectly, with 340 of them (83%) consisting of letter substitutions. Vowels were significantly more affected than consonants (e.g., DIETRO, behind → DIATRO; AUTO, car → UUTO; VILONTA, nonword → VELENTO). These two patients provided the first clinical evidence that vowels can COGNITIVE NEUROPSYCHOLOGY, 2003, 20 (2) 101 COTELLI ET AL. be selectively affected in a spelling disorder due to brain damage. We present here a new case of acquired dysgraphia, characterised by selective vowel substitutions. Besides replicating the previous observations, this case study provided evidence for a similar, although much milder, pattern of impairment in reading performance, thus providing insight about the role of the graphemic buffer not only for spelling but also for reading (Caramazza, 1997; Caramazza, Capasso, & Miceli, 1996; Hanley & Kay, 1998; Hanley & McDonnell, 1997). We discuss how computational models of spelling could explain a selective deficit for vowels and we conclude that the pattern represents a challenge to these models. CASE REPORT Clinical background LiB is a 39-year-old, right-handed woman, a textile industry worker, with 8 years of schooling. In January 1996, she suffered an acute left hemispheric cerebral haemorrhage. The CT scan showed a left temporal haematoma, in the white matter adjacent to the posterior corn of the lateral ventricle. A surgical evacuation was performed. Post-operatively, the neurological examination disclosed slight weakness of the right limbs, right homonymous hemianopia, and a fluent aphasia with impaired auditory comprehension. The first neuropsychological examination was carried out 3 months after the cerebral haemorrhage (April 1996). At that time the patient had already made a good neurological recovery. The weakness of the right limbs had resolved, and the aphasia was substantially improved. A formal language evaluation with the Aachener Aphasie Test (Italian version, Luzzati, Wilmes, & De Bleser, 1992) showed a profile of mild Wernicke’s aphasia, associated with a severe disorder of writing. Her performance in reading tasks was qualitatively very similar to writing, but less severely affected. On Raven’s Colored Progressive Matrices, a test of nonverbal intelli- 102 COGNITIVE NEUROPSYCHOLOGY, 2003, 20 (2) gence, LiB had a normal score (26/36 correct responses). Three months after this first neuropsychological evaluation LiB was referred to our laboratory for an in-depth examination of her linguistic abilities. The experimental investigations described here were all conducted during the summer of 1996. Her language abilities were largely improved at the time of the evaluation, with the exception of writing and reading deficits, which were found to be stable. Spontaneous speech LiB’s spontaneous speech was fluent, with only a few anomic pauses as investigated with the BADA (Miceli, Laudanna, Burani, & Capasso, 1994). Phonemic and verbal substitutions were rare. A transcript of LiB’s production, in response to the request to tell the story of Little Red Riding Hood, is reported below: Cappuccetto Rosso (The story of Little Red Riding Hood) .. è andata per preparare il cestino...per preparare la pappa per portare uova per la loro nonna, Hanno incontrato....’sto olio.....lupo, e voleva andare a tirare dentro questa bambina... e lei è riuscita a staccarsi. Però è riuscito ad entrare dentro nella .. nella casa della nonna ... per mangiare, voleva mangiare e ha cercato di mangiare un po’ di nonna. L’ha messa nella loro, dentro la loro, allora è passato e diceva: “sono io la nonna”...... (...she was going to prepare the basket...to prepare the pap to take the eggs to their grandma. They have met.... this oil.....wolf, and he wanted to go to draw inside this girl....and she succeeded to pull off. But he managed to enter in the.. in grandma’s house...to eat, he wanted to eat, he tried to eat a little bit of grandma. He put her in their, inside their, and then he passed and said: “It’s me the grandma” .....) EXPERIMENTAL INVESTIGATIONS Table 1 reports LiB’s performance in single-word processing tasks, and clearly indicates that her basic impairment was in writing (only 62% correct). The features of the writing disorder were in general compatible with a graphemic output buffer dysfunction whereas allographic errors, letter sequence errors, and micrographia never occurred. Fewer difficulties were present in reading and repetition. A A STUDY OF ACQUIRED DYSGRAPHIA Table 1. LiB’s performance in the repetition, the reading, and the writing-to-dictation of single words and nonwords Task Repetition Reading Writing Correct responses 665/676 610/668 420/681 % 93 91 62 series of investigations was carried out to better characterise her written language disorder. Writing A single researcher (MC) performed the analysis of writing: In cases of doubt in the interpretation of a letter, the evaluation was checked independently by five native Italian speakers. In cases of disagreement the error was not considered for the analysis. Written spelling to dictation The written spelling-to-dictation evaluation was performed with a large corpus of single words (n = 681), which included sublists controlled for frequency, grammatical class, lexical status, concreteness, and stimulus length. LiB was invited to spell to dictation 616 words and 65 nonwords, presented in random order during the testing sessions. The stimulus length of the words ranged from 2 to 13 letters. LiB wrote with her (dominant) right hand. Her written production was motorically unimpaired, and was executed without any noticeable delay after word presentation. No reliable differences were observed between her ability to write words and nonwords. LiB spelled correctly a total of 420/681 stimuli (62%), consisting of 380/616 words (62%) and 40/65 nonwords (62%). Neither the writing modality (e.g., cursive or capital letters), nor lexical factors (word frequency, grammatical class) affected her spelling performance. The results for the controlled sublists are presented in Table 2. Only the stimulus length had a significant effect: LiB spelled correctly 359/539 (67%) stimuli (words and nonwords) ranging from 2 to 7 letters, whereas she spelled correctly only 61/142 (43%) stimuli ranging from 8 to 13 letters, χ2 = 26.59; p < .0001. It is, however, important to notice that the length effect was abol- Table 2. Results obtained by LiB on controlled sublists in the writing-to-dictation tasks Total stimuli Lexicality Words Nonwords Grammatical word class Nouns Adjectives Verbs Functors Frequency High Low Cursive Capital letters Correct responses % 420/681 62 380/616 40/65 62 62 21/40 24/40 28/39 26/40 53 60 72 65 68/108 67/108 369/602 51/79 63 62 61 64 ished if the effective number of correct letters within the total number of letters present in the whole corpus of stimuli was considered (see Table 3), indicating that the error unit was the single letter and not the whole word. LiB’s errors were predominantly letter substitutions, followed by letter insertions, deletions, and transpositions. These features were observed in the written spelling-to-dictation of both familiar words and nonwords. Although a more detailed error pattern analysis for writing to dictation will be reported later, we will mention here that LiB made significantly more errors with vowels than with consonants. Her writing deficit was thus characterised by the relatively selective substitutions of vowels (e.g., ENTRARE, to enter → INTRARE; DAVANTI, in front of → DAVONTE; SIRBOLO, nonword → SIRBELE). A sample of her writing production is reported in Table 4, and examples of her handwriting are reproduced in Figure 1 on page 105 (see also Figure 4 on page 111 for examples of handwritten capital letters). Table 3. Incidence of correct responses as a function of stimulus length for the writing-to-dictation of words/nonwords No. of letters Correct responses % Correct a letters % 2–7 8–13 359/539 61/142 67 43 2613/2814 1147/1241 93 92 a Amount of correctly spelled letters within the total number of letters provided by the stimuli. COGNITIVE NEUROPSYCHOLOGY, 2003, 20 (2) 103 COTELLI ET AL. Table 4. Examples of LiB’s errors in the writing tasks Item (In English) Written uomo ultimi inutile insomma purtroppo atteso poi successo cospetto sud tatto invidia volonta accordo entrare credi ferrovia nuova beato termina cattivo ogni davvero gruppo davanti entrare piuttosto giornali vota intanto rigazzo dottere pano bortuna mettina (man) (last) (unnecessary) (in short) (unfortunately) (attended) (then) (success) (presence) (south) (touch) (envy) (will) (agreement) (to enter) (you believe) (railway) (new) (happy) (to finish) (bad) (every) (really) (group) (in front of) (to enter) (rather) (newspapers) (to vote) (meanwhile) (nonword) (nonword) (nonword) (nonword) (nonword) iomo iltime enotile insemma purtrappo otteso pui siccesso caspitto sod tatti invedia volento eccarde intrare crodi forrovia niova biato tormina cottivo ogne dovvero groppa davonte intrare piottosta giarnali veta entante rigazzi dottero pani bertuna muttina Spontaneous writing LiB was invited to describe her daily life in a written narrative. Also in this task her written production was fast, without noticeable pauses. In the sample, the errors are underlined and followed by the correct word in parentheses: Prima salata (saluto) Gloria e totti (tutti); faccio la pipì cal (col ) cagnolino pui (poi) praparo (preparo) tè con biscotti. Pai (poi) andiamo a trovare Gloria, col cagnolino. Quando ritorno a casa chiuda (chiudo) gli occhi, poi comincia (comincio) a pulire un po’, pachissimo (pochissimo); puoi (poi) camincio (comincio) a mangiare. 104 COGNITIVE NEUROPSYCHOLOGY, 2003, 20 (2) ( I first say hello to Gloria and all; I pee with the little dog then I prepare tea with cookies. Then we go to see Gloria, with the little dog. When I come back home I close the eyes, then I begin to clean a bit, a little bit; then I begin to eat.) The sample consists of 35 words (not taking into consideration words formed only by one letter, such as “a,” to or “e,” and ). LiB spelled incorrectly 11 words (31%), making 12 errors, most of them consisting of substitutions (n = 11/12; 92%), with only one insertion. By dividing the words in two main groups—words from 2 to 5 letters (n = 17) and from 6 to 10 letters (n = 18)—five errors affected shorter words (42%), and seven (58%) longer words. All the errors involved vowels. Written naming LiB was asked to write the names of 44 pictures of objects and actions, controlled for frequency and length. She spelled correctly 10 items (23%), whereas 16 items (36%) were omitted. Within the group of incorrect responses (18/44; 41%), she produced 12 letter substitutions (67%), 5 of which could be classified as semantically related to the target (3 with letter substitutions) (28%), and 1 neologism (5%). All errors, except for two semantic errors, resulted in the production of nonwords. It is noteworthy that 11 of the 12 letter substitutions (91%) affected vowels (e.g., TORTA, cake → TARTA; BANDIERA, flag → BANDIARA; LAVARE, to wash → LAVARA), and only 1 was a consonant substitution (PIANGERE, to cry → MIANGERE). Similarly, in three of the five semantically related errors, vowel substitutions were found (e.g., CULLA, cradle → LETTINA, instead of the semantically related word LETTINO, little bed); VOLARE, to fly→ COLEMBA, instead of the semantically related word COLOMBA, dove; and BOCCA, mouth → LABBRI, instead of LABBRO/LABBRA, lip/ lips). Errors were not influenced by frequency or word length. Delayed copy The patient was presented with 16 typewritten stimuli, including 10 familiar words controlled for frequency, grammatical class and length, and 6 A STUDY OF ACQUIRED DYSGRAPHIA Figure 1. Samples of LiB’s handwritten words. nonwords. She was allowed to look at each stimulus, presented individually, for 10 s, then the stimulus was removed. She spelled incorrectly two nonword stimuli (33%), producing one substitution (SEGREMO, nonword → SEGRAMO) and one deletion (PRI, nonword → PR_). Within the familiar words, three stimuli (33%) were not spelled correctly, producing three substitutions (FERIE, vacation → FERIO; MENTO, chin → MANTE) and one insertion (NOVEMBRE, November → NOVEMBRIE). All six errors involved selectively vowels, most of them consisting of substitutions (67%). Oral spelling-to-dictation tasks were not carried out, because of the unfamiliar nature of oral spelling tasks for native Italian speakers. Italian, in contrast to English, has a largely transparent orthographic system: Oral spelling is thus unnecessary as a learning/teaching strategy, and most Italians have never practiced oral spelling at school. The difficulty of performing an oral spelling task has also been observed in other investigations of Italian dysgraphics, e.g., Caramazza et al. (1987) and Posteraro et al. (1988). Spelling errors analysis In the present section we provide a detailed characterisation of the errors produced by LiB in the written spelling-to-dictation tasks (e.g., the tasks based on the largest corpus of stimuli). In the first place, it must be underlined that two of the errors resulted in illegal graphemic sequences for Italian orthography (e.g., SOSTANZA, substance → SOSTANRA). In a few instances, one of the graphemes of double consonant clusters corresponding, in Italian orthography, to a single phoneme was also affected by substitution errors (e.g., FINISCE, it finish → FINIGRE). Both these features indicate that the errors are graphemic, rather than phonological in nature. The distribution of spelling errors as a function of letter position in words/nonwords was assessed using the Wing and Baddeley method (see Figure 2). These authors proposed (1980) that the letter position within words/nonwords might affect the liability to errors within the graphemic buffer. Errors were expected to occur more frequently for letters located in the middle positions of the stimuli, because of a so-called “read-out failure” from COGNITIVE NEUROPSYCHOLOGY, 2003, 20 (2) 105 COTELLI ET AL. the graphemic buffer. Our results, reported in Figure 2, indicate that error rates are distributed in a somewhat equal manner for initial, medial, and final letter positions. Thus, we did not observe “read-out” errors in LiB. Other patients with graphemic buffer deficits failed to show the middle position effect (Aliminosa et al., 1993; De Partz, 1995; Katz, 1991). Recently, Schiller et al. (2001) reported two dysgraphic patients, whose spelling errors most often occurred in the final part of words, suggesting that different types of impairment to the graphemic buffer may result in contrasting patterns of position effects. The most striking aspect of LiB’s spelling errors was represented by the significant occurrence of letter substitutions. In the entire corpus of responses, a total of 295 letters were not produced correctly, with 283 (96%) of the errors represented by letter substitutions. From Table 5, it appears that within the spelling-to-dictation of familiar words, the incorrect responses (e.g., BEATO, happy → BIATO) were overwhelmingly represented by letter substitutions (263/270; 97%), followed by only a few insertions (6/270; 2%; e.g., TIZIO, fellow → TIZZIO). A similar error pattern was observed for the writing to dictation of nonwords, in which letter substitutions (e.g., BORTUNA, nonword → BERTUNA) were the most common error type (20/25; 80%), followed by insertions (4/25; 16%; for example, ARVE, nonword → ARVEN). Transpositions of letters never occurred and only two deletions (BRO, nonword → BR_, and STRADA, road → ST_ADA) were present within the entire corpus. The substitution, insertion, and deletion errors always respected the consonant/vowel status of the involved graphemes. A predominant 85% of the Wing and Baddeley’s method (1980) was utilised to conform letter positions in words/nonwords of different length. This method assigns a position between A and E in a word of any length, according to the following pattern: Number of letters 3 4 5 6 7 8 9 10 11 12 A B C K K K K K+1 K+1 K+1 K+1 K+2 K+2 K K K K K K+1 K+1 K+1 K+1 K K K+1 K K+1 K K+1 K K+1 D E K K K K K K+1 K+1 K+1 K+1 K K K K K+1 K+1 K+1 K+1 K+2 K+2 Figure 2. Distribution of LiB’s errors in the writing-to-dictation task as a function of letter position in words/nonwords. errors (250/295) affected vowels (as indicated in Table 6a), whereas only 15% involved consonants, χ2 = 284.9; p < .0001. In Table 6b, the incidence of these errors is reported by considering the number of incorrect letters within the whole corpus of letters administered (divided by their consonant/ Table 5. The general distribution of the various error types (substitutions, insertions, deletions, and transpositions) made by LiB in the writing-to-dictation of words/nonwords Substitutions Insertions Deletions Transpositions Total 106 Words % Nonwords % Total % 263 6 1 0 270 97.0 2.0 0.4 0.0 20 4 1 0 25 80.0 16.0 4.0 0.0 283 10 2 0 295 96.0 3.0 0.7 0.0 COGNITIVE NEUROPSYCHOLOGY, 2003, 20 (2) A STUDY OF ACQUIRED DYSGRAPHIA Table 6. (a) Incidence of vowels and consonants within LiB’s entire corpus of incorrect letters (n = 295) obtained from her errors in the writing-to-dictation of words/nonwords; (b) Incidence of writing errors selectively affecting vowels and consonants within the whole corpus of letters administered in the spelling-to-dictation tasks, shared by their vowel/consonant status (1850 vowels and 2205 consonants, for a total of 4055 letters) (a) Incorrect letters Vowels Consonants % 250/295 45/295 (b) Writing errors % 250/1850 45/2205 13.5 2.0 85 15 vowel status), and a similar pattern emerges. Moreover, it is noteworthy that within the group of vowel errors, 99% (247/250) were selective vowel substitutions, that is, vowels were substituted for other vowels. Within the consonant errors, 80% (36) were selective consonant substitutions, as indicated in Table 7. Vowel-for-consonant substitutions and vice versa were never observed. The dissociation between vowels and consonants found in LiB’s spelling performance leads to the further characterisation of her “graphemic buffer” dysgraphia as being relatively selective for vowels. Table 8. Results obtained by LiB on controlled sublists in the reading tasks Stimuli Total stimuli Lexicality Words Nonwords Grammatical word class Nouns Adjectives Verbs Functors Frequency High Low Correct responses % 610/668 92 542/583 68/85 93 80 36/40 34/40 31/39 37/40 90 85 79 93 80/98 76/98 82 78 Reading The assessment of reading skills in a patient with an ascertained damage to the graphemic buffer offers the opportunity to gain more insight into an important issue: does the reading performance reflect the spelling difficulties, suggesting that a common buffer is involved in both reading and writing? LiB was asked to read aloud single words. She was administered a total corpus of 668 words, controlled for lexicality, grammatical class, frequency, abstractness, and stimulus length. The correct responses that LiB made while reading are shown in Table 8. She read correctly 92% of the words Table 7. Proportions of the various error types (substitutions, insertions, and deletions) distributed selectively as a function of the consonant/vowel status of letters Substitutions Insertions Deletions Total Vowels (%) 247 2 1 250 99 0.8 0.4 85 Consonants 36 8 1 45 (%) 80.0 18.0 1.0 15.0 from the entire corpus (610/668). Among them, 93% of correct responses were for real words (542/ 583). Non-word reading was more affected: LiB correctly read 80% of the stimuli (68/85). A lexicality effect was thus present, χ2 = 15.73; p < .0001. There was no reliable difference between high- and low-frequency words. By dividing the whole corpus of stimuli into two groups of different stimulus length (from 2 to 7 letters and from 8 to 13 letters), no significant length effect was found, χ2 = 0.07; p < 0.78 (see Table 9). Table 9. Incidence of correct responses as a function of stimulus length within the reading of words/nonwords Stimulus 2-7 8-13 Correct responses % 462/505 148/163 91 91 COGNITIVE NEUROPSYCHOLOGY, 2003, 20 (2) 107 COTELLI ET AL. Figure 3. A CV constraint can be implemented in a CQ model by adding a bias to the activation of all vowel letters in vowel positions (left) and all consonant letters in consonant positions (right). Activation is in arbitrary units (adapted from Glasspool & Houghton, 1998). Error pattern analysis in reading LiB’s error pattern in reading was very similar to that observed in spelling. Letter substitutions were the leading error type. She read a total of 133 letters incorrectly. Among these errors, 98.5% consisted of letter substitutions (131/133), with only a few letter insertions (2/133: 1.5%) (Table 10). In word reading, letter substitutions were 98% of the errors (110/112 (e.g., PUDORE, shame → PODORE; PENSIERO, thought → PENSIERE); only two letter insertions were observed (e.g., COSCIENZA, conscience → COSCHIENZA). All errors (21/21) in nonword reading were letter substitutions (e.g., SUMILE, nonword → SAMILE; STIGHE, nonword → STIGRE). Transpositions and deletions never occurred in reading. The analysis of incorrect responses in reading words and nonwords yield the following results: 59% of errors in word reading resulted in real words (24/41), while 41% resulted in nonwords (17/41), χ2 = 2.39; p = .122, n.s. Errors in nonword reading never resulted in real words. Also in reading, the substitutions and insertions respected the consonant/vowel status of the involved phonemes. Similarly to the spelling tasks, in which 85% of the errors affected vowels and only 15% affected consonants, in reading 84% (112/133) of the errors involved vowels whereas only 16% (21/133) involved consonants, χ2 = 124.53; p < .0001, as indicated in Table 11a. As in the case of writing, the selectivity of the errors is confirmed when considering the number of incorrect letters within the whole corpus (see Table 11b). Again, within the group of vowel errors, all (112/112; 100%) were formed by selective vowel substitutions, that is, vowels were substituted only with vowels (see Table 12). The nonwords were divided into two main groups according to stimulus length (from 2 to 5 letters and from 6 up to 9 letters: 35% (13/37) of the Table 10. The general distribution of the various error types (substitutions, insertions, deletions, and transpositions) made by LiB when reading words/nonwords Words Substitutions Insertions Deletions Transpositions Total 108 110 2 0 0 112 % 98.0 1.7 0.0 0.0 84.0 COGNITIVE NEUROPSYCHOLOGY, 2003, 20 (2) Nonwords 21 0 0 0 21 % 100.0 0.0 0.0 0.0 15.7 Total % 131 2 0 0 133 98.5 1.5 0.0 0.0 100.0 A STUDY OF ACQUIRED DYSGRAPHIA Table 11. (a) Incidence of vowels and consonants within the total amount of incorrect letters (n = 133) obtained from LiB’s errors in the reading tasks; (b) Incidence of reading errors selectively affecting vowels and consonants within the whole corpus of letters read in the reading tasks, shared by their vowel/consonant status (1917 vowels and 1985 consonants, for a total of 3902 letters) (a) Incorrect letters Vowels Consonants % 112/133 21/133 84 16 Table 12. Proportions of the various error types that occurred in the reading tasks (substitutions and insertions), distributed selectively as a function of the consonant/vowel status of letters Substitutions Insertions Total Vowels % Consonants % 112 0 112 100 0 19 2 21 90 10 100 longer nonwords were not correctly read. For the shorter nonwords the percentage of errors was 9% (see Table 13 for a comparison between spelling and reading performance). These findings lead to the conclusion that LiB’s reading impairment mirrors the writing disorder, although at a lesser level of severity, and might thus share the same underlying dysfunctional mechanism. Table 13. Distribution of LiB’s errors in writing and in reading as a function of stimulus length for words and nonwords Stimulus length (in letters) Words 3–7 8–13 Nonwords 2–5 6–9 Errors in writing ——————— No. % Errors in reading ——————— No. % 156/476 80/140 33 57 26/423 15/160 6.0 9.0 7/38 18/27 18 67 4/48 13/37 8.0 35 As indicated, a marked stimulus “length effect” occurred in 2 the writing of both words, χ = 27.19; p < .0001, and 2 nonwords, χ = 15.52; p < .0001, whereas in the reading 2 tasks this “length effect” was found only for, χ = 9.38; p < .01. No significant “length effect” was observed in the 2 reading of familiar words, χ = 1.85; p < .17, ns. (b) Writing errors % 112/1917 21/1985 5.8 1.1 DISCUSSION The first point to consider is the locus of functional impairment in LiB’s writing disorder. Most of the features of LiB’s spelling performance are consistent with the criteria for graphemic buffer (GB) dysfunction delineated by Caramazza and coworkers (1987). These are: the presence of spelling errors for both familiar words and nonwords, the lack of frequency effects, and the similar spelling performance in the various experimental tasks (written spelling-to-dictation, spontaneous writing, written naming, and delayed copying). There are, however, some atypical aspects that deserve careful consideration. In the first place, the significant word length effect (WLE), as assessed on the basis of the number of correct words, disappeared when single letters were considered. A WLE has been reported both in patients with GB and in those with post-graphemic (PG) impairments (Del Grosso Destreri et al., 2000; Hanley & Peters, 2001; Zesiger et al., 1994). Rapp and Caramazza (1997) reported an abolition of the effect if the number of letters was considered in their two PG cases, but not in two previously reported GB patients. The latter conclusion was based on the fact that the error rate increased much faster than stimulus length in the reported data. This however does not seem to apply to all patients (see, for example, case TH, Schiller, Greenhall, Shelton, & Caramazza, 2001), and thus cannot probably be considered as a distinctive feature. Second, the error distribution curve was flat, whereas most GB cases show a U-shaped function. It has already been mentioned that serial order effects appear to be heterogeneous in GB patients, probably reflecting different mechanisms of COGNITIVE NEUROPSYCHOLOGY, 2003, 20 (2) 109 COTELLI ET AL. dysfunction (interference, fast decay, defective activation; Schiller et al., 2001). A third atypical feature is that LiB’s incorrect responses were overwhelmingly represented by letter substitutions (97%). Letter insertions were few (2%) and transpositions of letters never occurred. Although patients vary in the relative proportion of error types, with substitutions as the most frequent error, most patients produce a relatively high number of transposition errors. For instance, the proportion of letter transpositions was 17% in patient LB (Caramazza et al., 1987; Caramazza & Miceli, 1990) and 22% in patient AS (Jonsdottir, Shallice, & Wise, 1996). However, in other cases (such as the patient reported by Posteraro et al., 1988) transpositions were quite rare, indicating that in this respect also there is a high degree of heterogeneity. Nevertheless, the prevalence of substitutions and the lack of transposition errors must lead to the careful consideration of a possible post-graphemic (PG) locus of impairment (Black et al., 1989; Del Grosso Destreri et al., 2000). This possibility appears to be unlikely given the specificity of the disorder for vowels, which cannot be accounted for by visual or motor similarity. An inspection of the direction of errors (Table 14) indicates that the most vulnerable vowel is O (substituted by A or E), followed by A (substituted by O): The errors are thus bidirectional in the case of A and O, and unidirectional from O to E. By comparing the performance on the same letters in cursive and capital form, visual and motor factors in error generation may be ruled out, since similar errors occurred also in capital-letter writing tasks, in which the letters are quite dissimilar (see Table 14b and Figure 4 for handwritten examples). It is also noteworthy, from this point of view, that in patients with PG disorders the errors may involve V to C substitution (Rapp & Caramazza, 1997)—and may result in orthotactically illegal sequences (Del Grosso Destreri et al., 2000). An attempt was made to apply the procedure described by Rapp and Caramazza to assess the relationship between target and errors. Unfortunately, the stroke similarity metrics could not be applied, given the unavailabil1 We wish to thank an anonymous reviewer for this suggestion. 110 COGNITIVE NEUROPSYCHOLOGY, 2003, 20 (2) a Table 14. (a) Confusion matrix of vowel errors in writing tasks (words and nonwords); (b) confusion matrix of vowel errors in capital letter writing tasks (words and nonwords); (c) confusion matrix of vowel errors in reading tasks (words and nonwords) (a) A E I O U A E I O U Total 497 9 0 60 1 567 5 419 7 6 2 439 0 19 399 1 0 419 66 35 2 451 6 560 0 6 9 16 84 115 (b) A E I O U A E I O U Total 49 1 0 7 0 57 0 46 0 0 0 46 0 7 27 0 0 34 6 7 0 44 2 59 0 3 1 2 4 10 (c) A E I O U A E I O U Total 541 2 0 19 3 565 8 443 13 7 0 471 0 5 406 2 1 416 16 26 3 446 3 494 3 0 0 1 81 85 a The columns report the observed responses for each target vowel. ity of the patients for further testing. We have thus applied only the visuospatial similarity metrics onto the relatively small number of errors observed in capital writing: only 1 out of 30 errors (increasing to 6 with a less stringent criterion) could be considered as visuospatially similar to the target. Finally, phonological-articulatory similarity in term of position does not appear to affect the errors, since I and E are anterior, A central, and O and U posterior. Taken together, these affects appear to be fully compatible with a categorical disorder for vowels in the GB. In this case, the probability of producing an error is expected to be independent from the position or the length of a word1. The present observation replicates the observation of a selective deficit for vowels in writing, which has been reported only once in the literature (Cubelli, 1991). The A STUDY OF ACQUIRED DYSGRAPHIA Figure 4. Handwritten samples of capital letter writing. The item word is followed by the error and the handwritten sample. graphemic buffer has typically been conceived as a sort of working memory storage system, where graphemic representations are temporarily held in preparation for further, more peripheral spelling processing. The production of correct words/ nonwords by combining letter sequences essentially implicates that several properties of the orthographic representations have to be considered, in particular the identity of the letters and their serial position. However, if letter identity and letter position were the only specified properties, in the case of letter substitution errors all the following possibilities would be expected (given the target word COGNITIVE): COGNITIVE → ROGNITIVE COGNITIVE → AOGNITIVE COGNITIVE → CAGNITIVE COGNITIVE → CRGNITIVE In other words, each letter would have the same possibility of being substituted by an another one. Caramazza and co-workers (1987, 1990) were the first to remark, investigating the spelling performance of their dysgraphic patient LB, that only the first and the third alternatives were actually observed. Their observation resulted in the identification of another important property of graphemic representations, namely the vowel/consonant status. The preservation of vowel/consonant status in GBD implies that CV information is available in some form. Caramazza and Miceli (1990) proposed that CV status is represented in an abstract “CV template,” separately from letter identity (also see Miceli, 1993; Tainturier & Caramazza, 1996): C V C C V C V C V | | | | | | | | | áGñ áOñ áGñ áNñ áIñ áTñ áIñ áVñ áEñ consonant/ vowel level of representation graphemic level of representation Glasspool and Houghton (1997, 2002) discuss the nature of “categorical constraints” (such as the CV status) in the more general context of serial behaviour. They distinguish two possible sources of categorical response biasing in CQ-type models: external and internal. In the case of external constraint, the response bias comes from an explicit representation of the template to which the sequence must conform. This form of constraint has often been used in connectionist models of speech production (e.g., Dell, Burger, & Svec, 1997; Hartley & Houghton, 1996). Glasspool and Houghton (2002) used a global biasing of all C or V responses in the competitive queue to favour consonants or vowels at different points in the recall process (see Figure 3). In the case of internal constraint, the response bias is produced by the partial activation of items sharing the same status of the target due to the similarity of their internal representations. For instance, Glasspool, Shallice, and Cipolotti (1999) suggested that letters have a COGNITIVE NEUROPSYCHOLOGY, 2003, 20 (2) 111 COTELLI ET AL. distributed internal representation, which includes a CV dimension. Whatever the exact nature of such constraint, it will cause a systematic bias on activation levels in the queue of a CQ model, which, under noise disruption, will show typical GBD errors but with a preservation of CV status (Glasspool & Houghton, 2002). Current CQ models, however, cannot account for a selective deficit for vowels. The level of noise can change the relative proportion of error types, but it cannot differentially affect consonants and vowels. Thus, a revised CQ model should incorporate an explicit distinction between consonants and vowels, as proposed by Caramazza and Miceli (1990). For instance, CV information can be part of the orthographic representation itself, as in Houghton and Zorzi’s (1998, 2002) connectionist model of spelling. In addition to the explicit coding of CV status in the orthographic representation, one might also postulate that vowels and consonants are stored in different ways, or in different loci, within the spelling system. Cubelli (1991), reporting the first two cases of selective vowel deletion (case CF) or substitution (case CW) in writing concluded that, when the identity of a grapheme is unspecified because of a brain lesion, residual information about the consonant/vowel status may affect error production. When the “identity defect” is selective for vowels, this would result in their deletion, or substitution with other vowels. The observation of the converse dissociation would be crucial to support this hypothesis. To the best of our knowledge, a selective disorder of consonant writing has never been reported. In contrast to the single dissociation in spelling, however, a double dissociation between consonant and vowels has been reported in oral production. It has been repeatedly observed that phonological errors affect consonants more often than vowels (Béland, Caplan, & Nespoulous, 1990). Romani and co-workers (Romani, Granà, & Semenza, 1996) have reported the case of a patient with conduction aphasia who made more errors on vowels (56%) than on consonants (44%). Recently, Caramazza and co-workers (Caramazza, Chialant, 112 COGNITIVE NEUROPSYCHOLOGY, 2003, 20 (2) Capasso, & Micelli, 2000) have reported two patients, with a comparable clinical picture, who showed a clear-cut double dissociation between consonant and vowel errors in repetition. These observations support the hypothesis that consonants and vowels are processed by two distinct neural mechanisms, which can be affected selectively by brain damage. LiB’s error pattern in reading indicates that the locus of dysfunction involves a processing stage, which is shared by spelling and reading, and is compatible with the hypothesis that a common graphemic buffer is involved in both reading and spelling. It must, however, be underlined that, according to several case studies of patients whose spelling disorder was attributed to a damage to the graphemic buffer (Caramazza et al., 1996; Friedman & Kohn, 1990; Jonsdottir et al., 1996; Katz, 1991; Tainturier & Caramazza, 1994), a mild deficit in reading words and much greater difficulties in reading nonwords should be expected. An exception is case JH (Hanley & Kay, 1998), who had normal nonword reading. The significant length effect on nonword reading, described here in our case, fits with the hypothesis of a common buffer, in that a “buffer effect” should become more evident in reading “long” nonwords (see Table 13), which presumably should increase the memory load of the graphemic buffer. 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