Selective Lower-Case Letter Ideational Dysgraphia

Selective Lower-Case Letter Ideational Dysgraphia

NOTE SELECTIVE LOWER-CASE LETTER IDEATIONAL DYSGRAPHIA L.D. Kartsounis (The National Hospital for Neurology and Neurosurgery, London) The term agraph...

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NOTE SELECTIVE LOWER-CASE LETTER IDEATIONAL DYSGRAPHIA L.D. Kartsounis (The National Hospital for Neurology and Neurosurgery, London)

The term agraphia or dysgraphia refers to two main distinct types of impairment: (1) spelling disorders, and (2) writing disorders. Patients with spelling dysgraphia are able to form letters appropriately but make errors in the selection of letters. By contrast, patients with writing dysgraphia present with a motor disturbance in letter formation in the context of well preserved spelling skill (either in written or oral mode). Writing dysgraphia is considered to be independent of any motor or sensory disturbance and the term should be appropriately applied only in the absence of major apraxic or visuo-constructional impairments. There is a dearth of quantitative studies on writing dysgraphia which fulfil these criteria. Baxter and Warrington (1986) reported the first "pure" case of a patient who presented with a marked disturbance of writing in the absence of any spelling, praxic or visuoconstructional impairments. Furthermore this patient had no difficulty in copying letters or words. The authors argued that his selective deficit was due to his inability in accessing the correct motor programmes or sequences for writing and referred to this kind of impairment as "ideational agraphia" (i.e. an impairment analogous to the syndrome of ideational apraxia). More recently, Patterson and Wing (1989) reported the case of another patient whose reading and oral spelling were intact but he presented with a severe deficit in writing spontaneously or to dictation (as opposed to his copying which was satisfactory). Interestingly, he was significantly more impaired in the production of lower-case letters than production of upper-case letters. However, in addition to severe dyscaIculia, this patient was apraxic for relatively complex actions and, in particular, was rather poor at both copying and producing line drawings of objects. The present case report describes a patient with a marked disturbance of his ability to write lower-case letters in the context of relatively mild spelling dysgraphia and the absence of other significant apraxic difficulties. CASE REPORT

L.C.A. is a 67-year old, right-handed man (d.o.b. 9.3.23). He left school at the age of 15 and went on to an apprenticeship in tool making. In his late years prior to his retirement he worked as a pipe fitter. In September 1985 be slipped over while at work and fell a distance of ten feet, striking a concrete floor with the right side of his head. He remembered the moment of falling but his next recollection was of being in an ambulance which was transferring him to a hospital. He was treated only for somatic injuries to the right side of his body. However, in addition to pain in his back and upper limbs, during the following months he experienced intermittent headaches and dizziness. He was unable to return to work and was pensioned off. His wife reported that he was very forgetful and his personality had changed in that he showed apathy and lacked energy. Detailed neurological examination in October 1986 revealed no abnormality. Computerised tomography (C.T.), haematological and biochemical investigations were also normal but he was reported to present with "global intellectual impairment". Cortex, (1992) 28, 145-150

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Neuropsychological Examination In October 1989 the first (and only) comprehensive neuropsychological assessment was performed for medico-legal purposes. Both his verbal and performance I.Q.s on the WAISR were 91 and considered to represent only a mild degree of general intellectual deterioration. On the visual part of the Recognition Memory Test (Warrington, 1984) he scored at a high average level for his age group (46/50 correct) but on the verbal part at a low average level (41150 correct), suggesting a mild degree of verbal memory weakness. On the Graded Naming Test (McKenna and Warrington, 1978) he scored at an average level (14/30 correct) but made a few paraphasic errors. On a naming from description test (Coughlan and Warrington , 1978) he scored 11115 (correct) which also suggested mild word retrieval difficulties. By contrast, on tests of visual perception and visuospatial analysis (Warrington and James, 1967, 1988) his performance was errorless. There was no evidence of apraxic difficulties in that he copied hand movements and drawings of two- and three-dimensional geometric shapes satisfactorily (Figure la). He was also able to draw relatively complex objects without models satisfactorily (Figure Ib). However, on certain tests which are known to be sensitive to frontal lobe dysfunction (word fluency, interpretation of proverbs, Modified Card Sorting Test) his performance was uneven and suggestive of weakened abstract reasoning ability. His word reading on the Schone II and the National Adult Reading tests was equivalent to I.Q.s of 104 and 101 respectively (Nelson and O'Connell, 1978). There was a mild degree of hesitation in his reading, particularly with regard to irregular words, suggestive of "incipient" dyslexia (N.B. The reported I.Q. equivalents are unlikely to reflect accurately his optimallevel of general abilities). On the oral presentation of a spelling test of graded difficulty (Baxter, 1987) he scored at a low average level (8/30 correct) suggesting only a mild degree of impairment. His written spelling was weaker and appeared to be confounded by a motor disturbance in letter formation. Specifically, he was slow in writing even high frequency words such as "the" and "they", apparently due to his difficulty in "remembering" how to write individual letters in lower case (" how do you write 't' then?", he asked). However, he had no difficulty in writing to dictation the same words in upper-case letters or copying them either in lower- or upper-case letters. The variability in his writing skills prompted the investigations below. _ Before considering this matter further it was necessary to ensure that L.C.A. had not had any long-standing writing difficulties. Both the patient and independently his wife confirmed that, in the past , his writing skill was normal and he had never experienced it specific difficulty with lower-case letters. Indeed the detection of a writing difficulty during his assessment came as a surprise to both of them. It was not possible to obtain a sample of his writing from his earlier years. However, it is reasonable to assume that, having had nine years

Selective lower-case letter ideational dysgraphia

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schooling and his reading ability being at a good average level, he should certainly have acquired a proficient writing skill for both upper- and lower-case letters. It would be relevant to add that when he was first asked to write a sentence to dictation he wrote it spontaneously in lower-case letters, suggesting that writing in upper-case letters was not his preferred style. His neuropsychological profile above is consistent with an organic pattern affecting dominant hemisphere skills, apparently due to "contrecoup" effects. EXPERIMENTS

1. Writing the Alphabet and Numbers The first task was writing the alphabet from "memory", first in upper- and then in lower-case letters, and subsequently copying it in the reverse order, i.e. first in lower- and then in upper-case letters. L.C.A. 's writing of the alphabet from memory in lower-case letters was laborious and he made several errors, including incomplete graphemic representations of letters (f, g) and substitutions of lower-case letters by upper-case letters (q, I). However, his writing of the alphabet from memory in upper-case letters was fluent and well formed (Figure 2a). (N.B. He omitted the letter "N" both in lower- and upper-case letters versions of the alphabet). When required to copy the alphabet he was able to form the letters both in lower- and upper-case letters satisfactorily (Figure 2b). Temporal analysis of his performance indicated that it took him approximately twice as long to write the alphabet from memory in lower- than in upper-case letters (178 versus 93 sec.). There was only a small difference in time for copying the alphabet in lower- and upper-case letters (77 versus 67 sec.). In another test he was asked to write down (from memory) the numbers from 1 to 30 and he completed the task accurately in 65 seconds (Figure 2c).

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L.D. Kartsounis

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2. Writing Words to Dictation L.C.A. was asked to write to dictation the first 30 words from the Schonell three-column spelling test A', alternating every three words between lower- and upper-case letters. The words in the two tasks were matched for length. His writing was assessed by three criteria: legibility, spelling competence and speed of performance. All words in upper-case letters were written legibly. Two of the words in lower-case letters were not written legibly apparently due to wrong selection of letters and/or production of scrawls (Figure 2d). There was little difference in his ability to spell words correctly in upper- and lower-case letters. However, he needed substantially longer time to write each word in lower- than upper-case letters. His oral spelling of the same words was marginally more efficient than in the written mode (Table I).

L.C.A. 's performance on a different spelling test consisting of irregular words of graded difficulty (Baxter, 1987) was very similar (Table I). Although on this test the total number of letters comprising the words to be written in lower-case was inadvertently larger than the number of letters to be written in upper-case (65 vs 51), this discrepancy could not possibly account entirely for the observed substantial time effects in completing the two aspects of the task.

TABLE I

Summary of L.C.A.·s Performance on Two Spelling Tests Baxter test

Schonell test

Written mode

Number of words written legibly Mean time per word to be written (sees) Number of words spelt correctly

Oral mode

Number of words spelt correctly

Lower-case (N= 15)

Upper-case (N= 15)

Lower-case (N= 10)

Upper-case (N= 10)

13 20.3 (S.D. 11.4) 9

15 8.1 (S.D. 2.1) II

7 20.2 (S.D. 12.9)

10 8.7 (S.D. 6.0) 4

23/30

5

11120

3. Copying Words There was a little difference in the mean time L.C.A. required to copy each of the first ten words from the Baxter test (above) in lower- (6.4 sec.) and upper-case (5.0 sec.) letters.

4. Transcribing Words The test involved transcribing 15 words from lower- to upper-case letters and another 15 words from upper- to lower-case letters. The words were regular (Coltheart words), matched in terms of length in the two tasks and they were presented alternatively in blocks of five. L.C.A. transcribed legibly and correctly all 15 words from lower- to upper-case letters but only 8/15 from upper- to lower-case letters, making several substitutions of letters or omitting those which he was apparently unable to write (Figure 2e). The mean times in transcribing five words in the two rasks were 46.3 sec. (SD = 4.8) and 85 sec. (SD = 17.4) respectively. The reported data from different experiments suggest greater variability in the patient's performance on tasks involving writing in lower- than upper-case letters across different tests.

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Quantitative analysis of L.C.A.'s production of individual letters in terms of time was not attempted. However, inspection of the data did not suggest special vulnerability for any particular category of letters. DISCUSSION

The present case report describes an apraxic dysgraphia for lower-case letters in the context of only mild spelling dysgraphia and preserved praxic and constructional skills. There is clear evidence of a dissociation between the patient's competent performance in copying lower-case letters and his difficulty in producing them spontaneously. It may therefore be concluded that this patient presents with a selective ideational agraphia for lower-case letters. Previous reports by Kapur and Lawton (1983) and Baxter and Warrington (1986) indicated that neurological damage may give rise to selective ideational dysgraphia. Patterson and Wing (1989) described the case of a patient who, like L.C.A., presented with an ideational dysgraphia deficit specifically for lower-case letters. However, their patient had additional apraxic deficits. It appears that the present is the first case in which a selective ideational dysgraphia impairment for lower-case letters, in the absence of other apraxic deficits, is documented. Information-processing models of writing processes (Ellis, 1982; Margolin, 1984; Shallice, 1988) have distinguished between sets of graphemic motor programmes and those specifying neuromuscular instructions for the implementation of these programmes. Accordingly, patients may fail to select the appropriate graphemic motor programmes, or they may have difficulty in the executive stages of skilled writing actions. Clearly, L.C.A.'s writing deficit would be consistent with impaired retrieval or access to motor graphemic programmes. The fact that the converse dissociation of a selective impairment for writing upper- rather than lower-case letters has also been reported (De Bastiani and Barry, 1986) indicates a fairly independent representation of graphemic motor programmes for upper- and lower-case letters. ABSTRACT

A case study of a man with a selective ideational dysgraphia for lower-case letters is described. His spelling skills were only very mildly impaired and there was no evidence of other praxic or visuoconstructional impairments. The findings are discussed in the context of information processing models. It is suggested that the representation of upper- and lower-case letters must be fairly independent.

Acknowledgements. I wish to thank Dr. J.V. Jestico for his permission to investigate the patient and report the findings. I am also grateful to Professor E.K. Warrington for helpful comments on the manuscript. REFERENCES

BAXTER, D. Acquired spelling disorders. Ph. D. Thesis, University of London, 1987. BAXTER, D., and WARRINGTON, E.K. Ideational agraphia: A single case study. Journal oj Neurology, Neurosurgery and Psychiatry, 49: 369-374, 1986. COUGHLAN, A.K., and WARRINGTON, E.K. Word comprehension and word retrieval in patients with localised cerebral lesions. Brain, 101: 163-185, 1978. DE BASTIANI, P., and BARRY, C. After the graphemic buffer: Disorders of peripheral aspects of writing in Italian patients. Paper presented to the joint meeting of the Experimental Psychology Society and the Societa Italiana di Neuropsicologia, Padova, April 1986 (cited in Patterson and Wing, 1989). ELLIS, A.W. Spelling and writing (and reading and speaking). In A.W. Ellis (Ed.), Normality and Pathology in Cognitive Functions. London: Academic Press, 1982. KAPUR, N., and LAWTON, N.F. Dysgraphia for letters: A form of motor memory deficit? Journal oj Neurology, Neurosurgery and Psychiatry, 46: 573-575, 1983.

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MARGOLIN, D.1. The neuropsychology of writing and spelling. Semantic, phonological, motor, and perceptual processes. Quarterly Journal oj Experimental Psychology, 34A: 459-489, 1984. McKENNA, P., and WARRINGTON, E.K. Graded Naming Test. Windsor, Berks, England: NFER-Nelson Publishing Co. Ltd, 1983. NELSON, H.E., and O'CONNELL, A. Dementia: The estimation of premorbid intelligence levels using the New Adult Reading test. Cortex, 14: 234-244, 1978. PATTERSON, K., and WING, A.M. Processes in handwriting: A case for case. Cognitive Neuropsychology, 6: 1-23, 1989. SHALLICE, T. From Neuropsychology to Mental Structure. New York: Cambridge University Press, 1988. WARRINGTON, E.K. Recognition Memory Test. Windsor, Berks., England: NFER-Nelson Publishing Co. Ltd, 1984. WARRINGTON, E.K., and JAMES, M. Disorders of visual perception in patients with localised cerebral lesions. Neuropsychologia, 5: 253-266, 1967. WARRINGTON, E.K., and JAMES, M. Visual apperceptive agnosia: A c1inico-anatomical study of three cases. Cortex, 24: 13-32, 1988. L.D. Kartsounis, The National Hospital for Neurology and Neurosurgery, Queen Square, London WClN 38G, England, U.K.