51051ournal ofNeurology, Neurosurgery, and Psychiatry 1996;61:510-514 J Neurol Neurosurg Psychiatry: first published as 10.1136/jnnp.61.5.510 on 1 November 1996. Downloaded from

SHORT REPORT

Increased writing activity in neurological conditions: a review and clinical study

P van Vugt, P Paquier, L Kees, P Cras

Abstract of behaviour and that similar increased writing Increased writing activity in a 70 year old, activities have been labelled differently. We right handed man presenting with a his- would like to clear up the terminological con- tory of alcohol misuse and maturity onset fusion in the literature on increased writing diabetes is reported. Brain CT disclosed activity by reporting another patient and by corticosubcortical atrophy and 99mTc- differentiating automatic writing behaviour HMPAO SPECT disclosed severe bilat- from hypergraphia. eral frontal hypoperfusion more prominent on the right. The patient's neuropsychological symptomatology con- Case report sisted of severe (verbal) aspontaneity, A 70 year old right handed man with a long intermittent utilisation behaviour, and history of alcohol and tobacco misuse and a pronounced increased writing activity, previous medical history of well controlled which mainly consisted of a persevera- non-insulin dependent diabetes mellitus was tive, micrographic written reproduction admitted after a fall causing a left orbitomaxil- of visually or verbally perceived language lar fracture. He had been drinking several fragments. Several neurological causes of beers a day for many years and had shown increased writing activity and the equivo- progressive memory disturbances during the cal terminology met in the medical litera- year before admission. Relatives reported dys- ture are reviewed. A distinction between praxic behaviour, decreased verbal output, hypergraphia and automatic writing and two fugue episodes. Clinical examination behaviour is proposed. It is concluded on admission showed a frail, apathetic old that our patient's increased writing activ- man who was disoriented in time and space. ity may be characterised as automatic His liver was slightly enlarged. There was no writing behaviour. tremor and no asterixis. He displayed bilateral grasping, and there was a clear bilateral pal- http://jnnp.bmj.com/ (3 Neurol Neurosurg Psychiatry 1996;61:510-514) momental reflex. During his stay in hospital the patient remained aspontaneous. Only rarely did he respond, with one or two words, Keywords: ; hypergraphia; compulsive adequately answering the question. He walked writing Unit of with a wide base and there was bilateral Neurolinguistics, reduced diadochokinesia. However, there was University of Antwerp Increased writing activity in mental illness is a no ataxia nor dysmetria. on September 25, 2021 by guest. Protected copyright. (UIA), Belgium P van Vugt well accepted topic of interest.'2 On the other Laboratory tests showed slightly raised L Kees hand, abnormal writing behaviour resulting ammonia (up to 66 mg/100 ml). Hepatic Department of ENT from neurological disorder is seldom reported. enzymes were normal, except for gGT which Surgery, University of Probably the best known example is the ictal3 was slightly raised. Chest radiography, urine, Antwerp (UIA) and Department of or interictal increased writing activity seen in thyroid function, serology (for Treponema pal- Neurology, University some patients with epilepsy.' 4 5 6 lidum, HIV), uric acid, lipids, vitamin B 12, Hospital Erasme However, its frequency and specificity in tem- and CSF tests were negative or normal. His (ULB), Belgium poral lobe epilepsy4 have been questioned: EEG showed theta waves in the occipital P Paquier increased writing activity has also been lobes. Brain CT disclosed pronounced, gener- Department of Neurology (Born- described in mentally retarded patients alised corticosubcortical atrophy and ventricu- Bunge Foundation), (epileptic and non-epileptic)7 and in patients lomegaly more prominent on the right. As University of Antwerp with brain tumours,"9 cerebral strokes,'0 multi- normal pressure hydrocephalus was suspected (UIA), Belgium P Cras ple sclerosis,"I or frontal lobe .'2 Omnipaqueg was injected intrathecally. In the past, increased has The of CSF was normal. 99mTc- Correspondence to: writing activity resorption Dr P van Vugt, Unit of been referred to as hypergraphia,' echo- HMPAO SPECT showed severe frontal Neurolinguistics, CBL-UIA, on Universiteitsplein 1, B-2610 graphia, graphomania,8 anosognosic graph- hypoperfusion, more pronounced the right Wilrijk, Belgium. omimia, 4 graphorrh6e,'5 and phonographie.'6 (fig 1). Received 22 November 1995 Although diagnostic criteria have been pro- The mini mental state examination22 was and in final revised form 3 July 1996 posed,'4 '7 a comparative study of case reports difficult to evaluate due to the lack of commu- Accepted 4 July 1996 shows that one term may cover different types nication, but the patient scored 11/30 at his Increased wniting activity in neurological conditions: a review and clinical study 511 J Neurol Neurosurg Psychiatry: first published as 10.1136/jnnp.61.5.510 on 1 November 1996. Downloaded from

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best. Psychiatric examination showed some care was taken to replace the ballpoint pen loss of decorum. The patient did not confabu- used the day before by a differently coloured late. There was no obsessive compulsive per- one. To observe the patient's communicative =~~~~~~~~sonality disorder and during clinical bedside reaction when confronted with unexpected examination no obsessional traits were found. written stimuli, we surreptitiously inserted On the ward he displayed a mild form of utili- questions about his youth, his family, the hos- http://jnnp.bmj.com/ sation behaviour and a pronounced increased pital food, etc in the otherwise blank note- writing activity. book. Instructions related to writing as well as During an observation period of five weeks, geometric figures alternated with these ques- the patient produced 22 925 written words. tions, as we wanted to assess the patient's abil- The writing was micrographic but legible (fig ity to adapt his behavioural pattern and the 2). Generally, the patient copied whatever he degree to which the imitation was servile

set his eyes on: a newspaper headline, the (table). on September 25, 2021 by guest. Protected copyright. instructions on the television set, the trade The differences in ink disclosed that during mark on his pencil. He went on copying until the observation period the patient wrote three the end of the page or until another visual to four pages a day. He tended to fill all the stimulus caught his eye (table). The copying space available on the pages, and would often was rather careless: punctuation, numbers, return to previous pages to fill up the margins and abbreviations were not always respected, horizontally and vertically. Except for one conjunctions were often added (and ... and ... instance of shadow copying, small as well as and), prepositions were substituted. The large geometric patterns were disregarded and intrusion of neologisms suggested lexical wan- simply covered with text. Horizontal lines did dering (for example, de eeuwige achtervelden, not influence the writing activity, whereas ver- the happy backing grounds). As he became tical lines seem to have been perceived and more distracted during the act of writing, he respected as margins, or at least as an element would insert questions asked to him or a sen- organising writing space. Usually, questions tence overheard in a conversation. As a result, and instructions were copied several times. the text sometimes became very awkward and Occasionally a semantically related one word difficult to understand. or one syntagma response was formulated and The patient's writing activity was closely repeated over and over again. More than once, monitored during weeks 2 and 3. The nursing the copied instruction was personalised-for staff made sure that the patient only had a example, "You must not write here" became notebook at his disposal and every morning "I must not write here". 512 van Vugt, Paquier, Kees, Cras J Neurol Neurosurg Psychiatry: first published as 10.1136/jnnp.61.5.510 on 1 November 1996. Downloaded from

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Some examples ofinstructions and stimuli randomly inserted in a blank DINAS notebook, and description of the patient's graphic response Instruction or stimulus Spatial display Patient's response Write the word steam engine See fig 2A The word is initially copied four times. Later on, the three times in the box patient wrote the word another five times in the box. Finally, the page is covered with the word. Copy below the following Printed at 130 mm The sentence is copied once. The next day we found sentence once from the top out that the same sentence had been used to cover the entire page as well as the next two pages. Do not write on this side of Printed in a 60 mm wide The ban is copied on the right side of the line; afterwards, the line margin, see fig 2B the patient fills up the restricted area. Do you like the food? Printed at the page centre Unrelated page covering writing, until the question is reached. Below the question is answered 64 times: "Yes, very much". Diamond 140 x 90 mm Page centred The patient keeps on writing straight across the geometric figure. Were you on good terms with Printed at the page centre The answering starts at the top of the page. "I was on your mother? Explain why good terms with my mother (2 x ) and I will think about it and I will not mince matters everything is OK OK love you (illegible) and I will ask Jimmy Frey (a popular singer) (4 x ) and I was on good terms with my mother (5 x ) and it depended on what father wanted in my small world mother is still not lost and (...)". Describe your youth Printed at 30 mm Unrelated text above the instruction, below is written from the top in a page covering fashion: "my youth was I always the youth what a world the youth and the youth and I was always and what a youth (...)". Serpentine curve Printed at the right At first the curve is copied, 4 mm at the left. Afterwards, of the page text is displayed at the left of both curves. Finally, both curves are covered with text.

When the pen was removed while the regarded. The sharpening of a pencil resem- patient was writing, he kept his meek, gentle, bled a psychomotor automatism and a consid- and slightly absent minded physiognomy. erable effort was needed to separate the There were no signs of anxiety or frustration. patient's hands. However, a very strong grasp reflex made it After a month, the patient's verbal output difficult to take the pen away. When the paper had slightly improved: he answered questions was slightly pulled aside by the examiner during with one word responses and occasionally writing, the patient would firmly hold it with uttered short sentences. Written questions and his left hand. instructions yielded a significantly faster The examination of the utilisation behav- response than their spoken counterparts (eight iour took place in a quiet and almost distracter instructions in an ABBA design: t = 4-41, free observation room. When a nail hammered P = 0003). The increased writing activity into a piece of wood was presented together was still very prominent and annoyed his room with a nail puller, the patient would extract the mates, who insisted very strongly that nurses nail, despite the examiner's warning not to do or speech therapists took away the patient's so. Similarly, although he had repeatedly been pencil and notebooks. instructed not to touch the objects, the patient could not resist using the pair of scissors in front of him to cut the sheet of paper which Discussion http://jnnp.bmj.com/ had been laid on the table. When a plastic cup Our patient's neuropsychological symptoma- and a bottle of motor oil were put in front of tology consisted of severe (verbal) aspontane- the patient, he seized the bottle and started ity, intermittent utilisation behaviour, and reading the directions for use. There was, abnormally increased writing activity, which however, no attempt to open the bottle or to mainly consisted of a perseverative written pour the oil. At other times, however, there reproduction of visually or verbally perceived

was no utilisation reaction. For instance, when language fragments. On this basis, as well as in on September 25, 2021 by guest. Protected copyright. given an open envelope containing a visible the rather loose semantic coherence of his letter, the patient would not start reading it; he written production, he closely resembled the held the envelope until he was asked to give it patient of Cambier et al.8 As in previous back. Similarly, the presentation of a nail, a reports on increased writing activity in patients hammer, and a wooden block did not result in with left brain damage," patients with right hammering the nail into the block. On the brain damage,9 10 and patients with temporal contrary, the objects were left untouched. It lobe epilepsy with ictal increased writing activ- must be said that, in the few instances of utili- ity3 our patient's writing behaviour was also sation behaviour, the initiation of the response avolitional, automatic, and mechanical. took a certain time. This was by contrast with A relation between features of increased the activities related to writing, which had writing activity and underlying causes or lesion much shorter response latencies. Moreover, site and side seems to exist in some, but cer- the presence of a pen, a pencil, a sharpener, tainly not in all patients. It has been sug- and a ruler always resulted in the prompt initi- gested'7 that conscious and attentive writing is ation of writing, pencil sharpening, line draw- associated with epilepsy, and automatic com- ing, etc. Whereas the use or manipulation of pulsive writing with stroke, degenerative, or other objects was seldom repetitive, writing space occupying disease. However, this related activities had a continuous aspect and assumption is thwarted by Joseph's' report of a were difficult to interrupt. Verbal or printed patient with who pre- orders not to write or to stop writing were dis- sented with frequent, sudden onset, non-voli- 514 van Vugt, Paquier, Kees, Cras J Neurol Neurosurg Psychiatry: first published as 10.1136/jnnp.61.5.510 on 1 November 1996. Downloaded from tional writing automatisms. Moreover, some to hypergraphia this is less conclusive. We patients with right brain damage reported by endorse the view of Cambier et a18 that in tem- Yamadori et al'° displayed in their notes the poral lobe epilepsy the desire to record facts overinclusive style described in patients with and events might underlie the hypergraphia, as temporal lobe epilepsy. To avoid confusion recent investigations have considered hyper- when causal mechanisms are discussed, we graphic behaviour an emotional response in would like to redefine some terms in a merely trying to cope with the sometimes frightening behavioural perspective. memory problems caused by cerebral lesions.22 Although it is not always easy to measure In view of these findings, the following the degree of content productivity in repetitive guidelines may be clinically useful. In severe language,3 19 the communicative intention of cases of automatic writing behaviour, taking the patient's writing may help to distinguish away all writing material might contribute to several forms of increased writing activity. the psychological comfort of the patient's rela- Moreover, in patients with temporal lobe tives and room mates. In cases of hypergraphia epilepsy or right brain damage'0 a rather the- it seems best to allow the writing activity, as matic repetitiveness is found, whereas in other discomfort or an increase of fear is likely to types of increased writing activity, a formal result when the patient is deprived of the repetition on syntactic, lexical, or graphemic means to write. level is seen. An aspect of continuity parallels this distinction: by contrast with the patients of Pick,'3 Cambier et al,8 and Gil et al'4 and 1 Trimble M. Hypergraphia. In: Trimble M, Bolwig T, eds. Aspects ofepilepsy and psychiatry. London: John Wiley and our patient, the increased writing activity is Sons, 1986,75-87. not constantly present or elicitable either in 2 Ludwig A. Mental illness and creative activity in female writers. Am_J Psychiatry 1994;151:1650-6. temporal lobe epilepsy or in stroke patients 3 Joseph A. A hypergraphic syndrome of automatic writing, with right brain damage. Therefore, we pro- affective disorder, and temporal lobe epilepsy in two patients. Jf Clin Psychiatry 1986;47:255-7. pose to call hypergraphia all transient increased 4 Davey D, Thompson P. Interictal language functioning writing activity with a non-iterative appear- in chronic epilepsy. Journal of Neurolinguistics 1991 ;6: 381-99. ance on syntactic or lexicographemic level. We 5 Hermann B, Whitman S, Wyler A, Richey E, Dells J. The suggest reserving the term automatic writing neurological, psychosocial and demographic correlates of hypergraphia in patients with epilepsy. _J Neurol Neurosurg behaviour to indicate a permanently present or Psychiatry 1988;51:203-8. elicitable, compulsive, iterative, and not neces- 6 Roberts J, Robertson M, Trimble M. The lateralizing sig- nificance of hypergraphia in temporal lobe epilepsy. 3 sarily complete written reproduction of visu- Neurol Neurosurg Psychiatry 1982;45: 131-8. ally or orally perceived messages, as is the case 7 Jancar J, Kettle L. Hypergraphia and mental handicap. Journal ofMental Deficiency Research 1984;28: 151-8. in our patient. With respect to the nature of 8 Cambier J, Masson C, Benammou S, Robine B. La the pressure of writing it is important to pay graphomanie, activite graphique compulsive manifesta- tion d'un gliome fronto-calleux. Rev Neurol (Paris) 1988; attention to the patient's behaviour when 144:158-64. engaged in writing activity. If a patient shows 9 Imamura T, Yamadori A, Tsuburaya K. Hypergraphia associated with a brain tumour of the right cerebral hemi- frustration or anxiety when pen and paper are sphere. J Neurol Neurosurg Psychiatry 1992;55:25-7. removed from him while writing, it may be 10 Yamadori A, Mori E, Tabuchi M, Kudo Y, Mitani Y. Hypergraphia: a right hemisphere syndrome. .7 Neurol assumed that the increased writing activity is Neurosurg Psychiatry 1986;49: 1160-4. related to an obsessive compulsive disorder.20 11 Kanemoto K, Akamatsu T, Itagaki Y, Nishitani H. Early On the other hand, if deprivation of writing onset multiple sclerosis with hypergraphia and Klu.ver- Bucy syndrome: a case report. Neurological Medicine http://jnnp.bmj.com/ material does not induce such a reaction, a (Tokyo) 1990;32:301-3. 12 Frisoni G, Scuratti A, Bianchetti A, Trabucchi M. truly compulsive syndrome is less likely to Hypergraphia and brain damage. J Neurol Neurosurg explain the increased writing activity. As far as Psychiatry 1993;56:576-7. 13 Pick A. On the pathology of echographia. Brain 1924;47: our aspontaneous patient is concerned, the 417-29. fact that he tried to immobilise the sheet that 14 Gil R, Neau JP, Aubert I, Fabre C, Agbo C, Tantot AM. was Graphomimie anosognosique: variete particuliere was pulled aside by the examiner while he d'hypergraphie au cours d'un infarctus sylvien droit. Rev writing, points to his desire to continue his Neurol (Paris) 1995;151:198-201. H. Dictionnaire whether such 15 Manuila A, Manuila L, Nicole M, Lambert on September 25, 2021 by guest. Protected copyright. activity. However, it is doubtful franfais de medecine et de biologie. Paris: Masson, 1970. a mild reaction can reasonably be considered 16 Bernard C. De l'aphasie. Paris, 1889?. 17 Okamura T, Motomura N, Asaba H, et al. Hypergraphia in an instance of obsessive compulsive behaviour. temporal lobe epilepsy, compared with stroke of the right Several case reports of automatic writing cerebral hemisphere. Japanese Journal of Psychiatry and Neurology 1989;43:524-5. behaviour suggest or document an involve- 18 Folstein MF, Folstein SE, McHugh PR. Mini-Mental ment of left or right frontal lobe areas using State. JPsychiatr Res 1975;12:189-98. 19 Perkins M. Repetitiveness in language disorders: a new imaging techniques and neurobehavioural analytical procedure. Clinical Linguistics and Phonetics descriptions. It seems reasonable to accept 1994;8:321-36. 20 Cooper J. Obsessional illness and personality. In: Russell automatic writing behaviour as a particular, G, Hersov L, eds. Handbook ofpsychiatry. Vol 4: the neu- sometimes isolated, form of utilisation behav- roses and personality disorders. Cambridge: Cambridge University Press, 1983,258-74. iour, in the sense that the inhibitory function 21 Lhermitte F. Utilization behaviour and its relation to of the frontal lobes is suppressed, thus leaving lesions of the frontal lobes. Brain 1983;106:237-55. 22 Okamura T, Fukai M, Yamadori A, Hidari M, Asaba H, the subject dependent on the slightest stimu- Sakai T. A clinical study of hypergraphia in epilepsy. lus.2' With respect to the mechanisms leading Neurol Neurosurg Psychiatry 1993;56:556-9.