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Posterior Cortical Dementia with Alexia: Neurobehavioural, MRI, and Petfindings 445

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Posterior Cortical Dementia with Alexia: Neurobehavioural, MRI, and Petfindings 445 Journal ofNeurology, Neurosurgery, and Psychiatry 1991;54:443-448 4L43 Posterior cortical dementia with alexia: J Neurol Neurosurg Psychiatry: first published as 10.1136/jnnp.54.5.443 on 1 May 1991. Downloaded from neurobehavioural, MRI, and PET findings L Freedman, D H Selchen, S E Black, R Kaplan, E S Garnett, C Nahmias Abstract The syndrome of posterior cortical demen- A progressive disorder of relatively focal tia (PCD) described by Benson et al'4 and De but asymmetric biposterior dysfunction Renzi' is clinically dominated by disturbances is described in a 54 year old right handed in visual function including object agnosia, male. Initial clinical features included prosopagnosia, alexia, and visuospatial dis- letter-by-letter alexia, visual anomia, orientation. The development of transcortical acalculia, mild agraphia, constructional sensory aphasia and a complete Gerstmann's apraxia, and visuospatial compromise. and Balint's syndrome was also present in Serial testing demonstrated relentless several patients. Computed tomography34 and deterioration with additional develop- MRI4 in PCD revealed prominent posterior ment of transcortical sensory aphasia, atrophy characterised by enlargement of the Gerstmann's tetrad, and severe occipital horns. However, the topography of visuoperceptual impairment. Amnesia cerebral metabolic dysfunction using positron was not an early clinical feature. Judg- emission tomography (PET) has yet to be ment, personality, insight, and aware- described for PCD. ness remained preserved throughout We examined a patient with serial neurobe- most of the clinical course. Extinction in havioural testing complimented by MRI and the right visual field to bilateral stimula- PET who exhibited PCD. The dementia was tion was the sole neurological abnor- notable for the presence of a severe letter-by- mality. Early CT was normal and late letter alexia and profound bioccipital (left > MRI showed asymmetrical bioc- right) hypometabolism verified by PET. cipitoparietal atrophy with greater involvement of the left hemisphere. Case report Results from positron emission A 54 year old, right handed man with two tomography (PET) showed bilaterally doctoral degrees was referred for neurological The Mississauga asymmetric (left > right) occipitotem- in 1986 after Hospital, Mississauga, examination February complain- Ontario poroparietal hypometabolism. The ing to his family physician of progressive Department of metabolic decrement was strikingly difficulty in reading, mathematical ability, and Psychology asymmetric with a 50% reduction in memory function over the previous three L Freedman glucose consumption confined to the left years. These problems were at first mild, but http://jnnp.bmj.com/ Divisions of Neurosurgery and occipital cortex. The- picture of became disturbing- given his aptitude in Neurology occipitotemporoparietal compromise mechanical engineering and economics. He D H Selchen verified by MRI, PET, and neurobe- was aware of the impairments and was able to Department of havioural testing would be unusual for manage a private business despite his reduced Neurology, such degenerative dementias as Alz- cognitive capabilities. Sunnybrook Medical Centre, University of heimer's (AD) and Pick's disease, alth- A neurological examination performed in Toronto, Toronto, ough atypical AD with predominant February 1986 revealed an alert and oriented Ontario occipital lobe involvement cannot be man with intact cranial nerves. There were no on October 2, 2021 by guest. Protected copyright. S E Black excluded. This case supports the con- orbital, cranial, or carotid bruits and visual Department of cepts of posterior cortical dementia full to Psychology, Chedoke- fields were confrontation. Sensoximotor McMaster Hospitals (PCD) as a clinicaily distinct entity and function, gait, and coordination were intact and Department of for the first time documents its corre- and primitive reflexes were absent. Examina- Psychiatry, McMaster sponding metabolic deficit using PET. tion of the skull, chest, and heart was University, Hamilton, Ontario negative. Haematological studies revealed a R Kaplan Focal and diffuse cortical syndromes distinct normal CBC, electrolytes, enzymes, thyroid, Department of from Alzheimer's and Pick's disease are B12, folate levels, VDRL, ESR, and ANF Nuclear Medicine, becoming increasingly recognised." Included counts. Analysis of the CSF- was normal. An Chedoke-McMaster are of in the tem- Hospitals, Hamilton, among these the syndromes progressive EEG showed non-specific slowing Ontario, Canada aphasia without dementia,' 2 5 progressive poral lobes, bilaterally, and CT revealed no E S Garnett apraxia,' frontal lobe dementia,67 argyrophilic focal lesions or significant atrophy. Medical C Nahmias grain disease,8 and a posterior dementia.'4 history was negative for cerebrovascular and Correspondence to: Dr While some scepticism still remains that some cardiovascular disease, subarachnoid Freedman, Department of Psychology, The Mississauga of these syndromes represent atypical variants haemorrhage, intracranial infection, cerebral Hospital, 100 Queensway of Alzheimer's (AD) or Pick's disease,9 11 tumour, hypertension, diabetes, and psy- West, Mississauga, Ontario, Canada L5B 1B8 evidence derived from metabolic,12 regional chiatric illness. Family history was negative Received 23 April 1990 and blood flow," and neuropathological'6-5 inves- for dementia. in revised form 1 October have their status as Serial neurobehavioural examinations were 1990. tigations clearly supported Accepted 15 October 1990 distinct neurological entities. conducted over a three year period beginning 444 Freedman, Selchen, Black, Kaplan, Garnett, Nahmias Table I Language and reading data: serialfindings plete Gerstmann's syndrome (acalculia, agra- phia, finger agnosia, right-left disorientation) J Neurol Neurosurg Psychiatry: first published as 10.1136/jnnp.54.5.443 on 1 May 1991. Downloaded from February December April October and ocular dysmetria, which is one component Measure 86 86 88 88 of Balint's syndrome. Despite evolving WRAT reading 39th% 37th% - - visuoperceptual compromise, he was able to PIAT reading recognition - 3rd% < lst% PIAT reading comprehension - 1st% < lst% - recognise inanimate objects but had difficulty Boston Naming Test (60-items) - 33 22 19 identifying animals. He was, however able to Tactile Naming Test* - - - 5/16 Token Test (36-items) - 22 9 - point to photographs of inanimate and Word discrimination (BDAE) - 60/72 52/72 - animate objects named aloud by the examiner Written naming (BDAE) 8/10 6/10 4/10 - Word-picture matching (BDAE) - 8/10 0/10 - without error using a multiple-choice format. Writing-to-Dictation 15/19 13/19 3/10 - The matching of black and white photographs Word repetition (BDAE) - 90% - 100% High-phrase repetition (BDAE) - 100% - 40% of unfamiliar faces29 was relatively intact Low-phrase repetition (BDAE) - 75% - 25% (80%) and he was able to recognise 14/19 (73%) photographs of famous faces using a WRAT = Wide range achievement test. PIAT = Peabody individual achievement test. BDAE = Boston diagnostic aphasia examination. WRAT and PIAT scores in percentiles. multiple choice format. He never failed to *Naming of 16 common objects using the right hand. recognise the examiner (LF) over the serial examinations. He was totally unable to locate any continents, oceans, or countries on a large in February 1986. Initial testing of language world map and did not exhibit any environ- function (table 1) showed a mild anomia and mental agnosia' until late in the course. He agraphia. He also exhibited a severe alexic did not exhibit any colour agnosia, disturbance. Latency and accuracy in oral achromatopsia, or ideomotor apraxia. Person- reading of single words'4 as a function of ality, insight, judgment, comportment, and word-length revealed a positive linear awareness remained intact throughout most of relationship consistent with a letter-by-letter the clinical course until the development of or pure alexia (fig la-b). Further analysis of paranoid ideation three years after the initial his oral reading ability revealed a reading rate neurological evaluation. of approximately one second per letter which Repeat neurological testing performed five, is in keeping with the word-length effect 27, and 39 months after the initial examina- of previously described letter-by-letter tion revealed normal cranial nerves, sen- readers who had focal left occipitotemporal sorimotor function, reflexes, and gait. lesions.'""8 Visual paralexias were common17 However, visual confrontation testing and he committed right-sided derivational revealed persistent right visual field extinc- reading errors'9 as well. Both oral reading and tion. MRI scanning performed in September comprehension20 of letters and words even- 1988 showed bilateral occipitoparietal atrophy tually became impossible. The early anomia progressed into a severe transcortical sensory 90- aphasia with considerably compromised nam- 0 ing2' and auditory comprehension.22 Single 80- 0 word repetition remained intact. Tactile nam- 70- ing was also severely defective although he o 60- was able to correctly pantomime the function 0 50- http://jnnp.bmj.com/ of objects he failed to name. Ca 40- 0 Results of the general neurobehavioural . assessment (table 2) revealed moderate com- ae30 36 20 r=0 promise in overall IQ23 characterised by a p< 0Q05 large discrepancy between average verbal and 10 his severely impaired visuospatial abilities. He 0 4- and 5 6 7 8
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