NOTE PURE DYSGRAPHIA WITH RELATIVE SPARING OF LOWER-CASE WRITING Luigi Trojano and Laura Chiacchio ("Clinica del Lavoro" Foundation, IRCCS, Medical Center of Rehabilitation, Campoli Monte Taburno, Italy; Second Department of Neurology, School of Medicine, "Federico II" University, Naples, Italy) INTRODUCTION Since the early 1980s writing disorders have been studied within information processing frameworks. Cognitive analyses of dysgraphia have often been based upon models of writing that propose some "central" linguistic processes and some "peripheral" processes related to the actual motor output and specific for written output (Ellis, 1982; Margolin, 1984). It has been proposed that at the end of the "central" spelling routes a short-term retention buffer is aimed at storing orthographic representations of words and nonwords alike, before the actual realization of output. This so-called Graphemic Output (orthographic) Buffer (GOB) would also be implied in oral spelling. Beyond the graphemic buffer, "peripheral" processes are devoted to transforming the orthographic string into graphic motor programs specifying the sequences of movements necessary for handwriting. A specific impairment of the GOB would imply quantitatively and qualitatively similar graphemic errors in all writing tasks (delayed copy, naming, writing to dictation) and in oral spelling. In fact, such a pattern has been reported in the literature (Caramazza, Miceli, Villa et aI., 1987; Posteraro, Zinelli and Mazzucchi, 1988; Hillis and Caramazza, 1989; Caramazza and Miceli, 1990). Diagnostic criteria for selective impairments of the GOB can be summarized as follows (Hillis and Caramazza, 1989): (1) similar patterns of spelling errors for words and nonwords; (2) number of errors increasing as a function of item length; (3) no effect of lexical factors; (4) no semantic or morphological errors; (5) spelling errors consisting of deletions, insertions, substitutions and transposition of letters which may lead to the production of graphemically implausible nonwords. Recently, some detailed observations have also been reported upon deficits of "peripheral" processes in handwriting (e.g., Baxter and Warrington, 1986; Papagno, 1992). Some of these patients, whose oral spelling was always correct, showed double dissociations between lowerand upper-case handwriting style (De Bastiani and Barry, 1986; Patterson and Wing, 1989; Kartsounis, 1992). In these cases, it was postulated that the selective impairment occurred in "peripheral" writing processes, where lower- and upper-cases style can be represented by independent graphic motor codes. In the present paper we describe a patient affected by acquired pure dysgraphia with a relative sparing of lower-case writing. In this case the dissociation between upper- and lowercase writing does not seem related to defects in "peripheral" writing processes. CASE REPORT D.V.M. was a 72-year-old right-handed housewife with 7 years of education. Her native language was Italian. In June 1991, she sustained a left ischemic cerebral lesion whose clinical signs were a right homonimous hemianopia and slight weakness of right arm. CT scan showed a left parietal-occipital ischemic lesion, with enlargement of left occipital ventricular horn (Figure 1). Doppler fluxirnetry showed marked stenosis of left common carotid artery. D.V.M. Cortex, (1994) 30, 499-507 500 L. Trojano and L. Chiacchio Fig. 1 - CT scan. complained of mild amnesic and moderate writing disturbances. She was evaluated from October to December 1991. She was well-oriented and cooperative. The patient was given some neuropsychological tests from a standardized Italian battery (Spinnler and Tognoni, 1987). Immediate verbal memory (Bysillabic word span=4), memory for complex verbal material (Story test = 10.3/16), constructional, oral and limb praxis were in the normal range for age and education. She obtained a poor score at Corsi' s visuospatial immediate memory task (3). Her Verbal I.Q. was in normal range (95). Speech-language Evaluation D.V.M. did not show overt aphasic disturbances. She performed flawlessly on tasks for spontaneous speech and naming, oral and written comprehension, repetition and reading aloud, comprised in an Italian standardized battery (Basso, Capitani and Vignolo, 1979). She could TABLE I Written Tasks of Standard Battery (scores expressed in percentages) Written naming Writing to dictation Letters (10) Syllables (10) Words (10) Nonwords (5) Sentences (5) Lower-case Upper-case 85 40 100 100 100 100 80 40 40 Word copy (10) 80 60 Letter transposition Upper- to Lower-case (21) Lower- to Upper-case* (21) 100 * This task is not comprised in the battery. 60 40 o 100 Lower-case sparing in dysgraphia 501 A) B) Fig. 2 - Examples of D. V.M.' s upper- and lower-case writing: A) delayed copy ("novembre" november - written as "noverbe" in upper-case and correctly in lower-case); B) writing to dictation ("fratello" - brother - written as "frtello" in upper-case and correctly in lower-case). sign her name but made many errors in writing tasks, and she reported more difficulties when writing upper-case letters. For example, when asked to write a letter to her husband, she managed to write two sentences in lower-case script, but refused to write in upper-case. For this reason, we asked her to perform standard writing tasks both in lower- and uppercase format. Results are shown in Table I. D.V.M. wrote single letters and CV syllables without errors, and correctly transposed single letters from upper-case to lower-case and viceversa. By contrast, she made many errors in copying and in writing words and nonwords spontaneously and to dictation, with a better performance with lower-case format (Figure 2). This picture suggested that D.V.M.'s impairment was neither in the selection nor in the actual motor output of single letters, but in the production of letter strings, especially in upper-case. 502 L. Trojano and L. Chiacchio TABLE II Correct Responses at Writing to Dictation and Spelling Lowercase Uppercase Spelling 27 17 10 Nouns (10) Verbs (10) Functors (10) 10 8 9 7 5 5 5 2 3 Bisyllabic (15) Trisyllabic (15) 14 13 9 8 9 1 21 18 14 . 2 2 19 Words (30) Nonwords (25) Monosyllabic (15) Bisyllabic (5) Trisyllabic (5) 15 3 3 15 3 1 Special Writing Evaluation In order to investigate D.V.M.'s difficulties in detail, we compared lower- and uppercase writing in several tasks (writing to dictation, copying and written naming) on different lists of words and nonwords. In particular, we tried to confirm discrepancies between lowerand upper-case writing and to identify the source of her spelling disorder. Part of the material used in this study was taken from an Italian battery for the cognitive analysis of language disorders (Miceli, Laudanna and Burani, 1990). Test 1: Word and Nonword Writing to Dictation and Oral Spelling The patient was presented with a list of 30 words, balanced for length and grammatical class, and with a list of 25 pronounceable nonwords, 2- to 8-letter long. During the same session, the patient first wrote the list in lower-case format, then in upper-case and finally she was asked to spell item orally. Results are given in Table II. D.V.M. spelled words in lower-case significantly better than in the other modalities tested (X 2 = 20, d.f. = 2, P = 0.(01). Upper-case writing and oral spelling did not differ from each other (X 2 =3.3, d.f.= 1, p=0.7), although the patient made more errors during spelling. Grammatical class did not affect either upper-case writing or spelling, while spelling showed a clear effect of stimulus-length (D.V.M. spelled correctly only one long word). The few lower-case errors were single graphemic errors (1 insertion and 1 deletion) and one double error (l deletion + 1 substitution), all resulting in nonwords. In upper-case writing and oral spelling, errors consisted of single or multiple letter deletions, substitutions, insertions and transpositions. Responses resulted in pronounceable and non pronounceable nonwords. One deletion in oral spelling resulted in a real word ("puniti" - punished - was spelled as "punti" - points). In nonwords writing, D.V.M. showed a similar accuracy in all three spelling tasks (X2= 1, dJ. = 2, P = 0.5). She made only 1 error with 2-letter items (in upper-case writing) but produced many errors with longer stimuli: length had a significant effect in all tasks (X 2 = 7.2, 8.4 and 14 for lower-, upper-case and spelling, dJ. = 2, p<0.02, 0.01, and 0.001 respectively). Also with nonwords, D.V.M.'s errors in all spelling tasks consisted of single and (rarely) multiple graphemic errors (mainly substitutions and deletions) resulting in pronounceable and nonpronounceable nonwords. A quantitative error analysis will be reported below. Test 2: Word and Nonword Writing to Dictation and Copying The patient was asked to write to dictation in lower- and upper-case, and to copy from the model in upper-case letters a list of 30 words, balanced for frequency and length, and 30 nonwords, balanced for length. D.V.M. read all stimuli flawlessly. Results are given in Lower-case sparing in dysgraphia 503 TABLE III Correct Responses at Word and Nonword Writing to Dictation and Copy Lowercase Uppercase Copy Words (30) High-Frequency (15) Low-Frequency (15) 4-1etter (6) 5-1etter (6) 6-1etter (6) 7-1etter (6) 8-1etter (6) 29 15 14 6 6 5 6 6 17 7 10 6 4 3 3 1 21 10 Nonwords (30) 4-1etter (6) 5-1etter (6) 6-1etter (6) 7-1etter (6) 8-letter (6) 22 5 5 5 5 2 16 5 4 4 2 1 23 6 6 5 3 3 11 6 6 4 4 1 Table III. The first relevant finding is that D.V.M.'s accuracy in upper-case copying was similar to that observed in upper-case writing to dictation. Moreover, D.V.M. made only one error in lower-case word writing to dictation. Performance on this task was significantly better than that on upper-case writing (X 2 = 13.4, dJ. = 1, P = 0.0002). Very good performance on lower-case spelling prevented the study of frequency and length. Upper-case writing (X2= IO.S, dJ. =4, P =0.03) and copy (X2= 14.S, dJ. =4, p