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Slowly Progressive Aphasia: Three Cases with Language, Memory, CT and PET Data Journal ofNeurology, Neurosurgery, and Psychiatry 1990;53:987-993 987 J Neurol Neurosurg Psychiatry: first published as 10.1136/jnnp.53.11.987 on 1 November 1990. Downloaded from Slowly progressive aphasia: three cases with language, memory, CT and PET data D Kempler, E J Metter, W H Riege, C A Jackson, D F Benson, W R Hanson Abstract (yes/no questions, sequential commands), Three cases of slowly progressive speech repetition, naming, reading and writing. and language disturbance were studied Measures of praxis, drawing and calculation at various points post onset (three, five are also collected as part of the WAB. Scores and 15 years respectively). Language, are converted into a 10 point scale (10 = neuropsychological and brain imaging normal; 0 = grossly deficient). (computer tomography and positron To evaluate other aspects of mental func- emission tomography) evaluations were tioning, the patients were administered a completed on all three patients. The data neuropsychological battery which included suggest that the syndrome of "progres- standard administration of published tests,13-16 sive aphasia": 1) does not involve a and an additional set of verbal and non-verbal uniform symptom complex; 2) does not memory tests designed to discriminate left- necessarily develop into a full blown from right-hemisphere functions.'7 18 The ver- dementia syndrome; 3) varies greatly in bal tests included: rate of progression from case to case; 4) 1 Story recall. The subject listens to a short is associated with normal brain struc- story (an Aesop fable of 141 words) and is ture (on computer tomography); and 5) asked to retell it immediately. is associated with abnormal left tem- 2 Sentence memory. The subject is asked to poral lobe metabolism as measured by repeat well formed, semantically anomalous fluorodeoxyglucose (FDG) positron and grammatically anomalous sentences. emission tomography (PET). One 3 Picture recall. The subject is presented with patient had histological findings consis- nine line drawings on a single board and asked tent with Alzheimer's disease at to describe each one. After 20 minutes, he is necropsy. asked to describe the pictures from memory. 4 Immediate and delayed memory of visually presented (printed) words. The patient is Speech Pathology, Approximately 29 cases of slowly progressive presented with a series of 20 words on a School of Medicine dementia have been and School of aphasia without reported tachistoscope and then asked to recognise the Gerontology, in the literature,'2 but there remain unans- original words within a set of 40 items University of wered questions about this syndrome. It immediately after presentation and again Southern California, remains to be seen whether this syndrome twenty minutes (delayed) after presentation. http://jnnp.bmj.com/ Los Angeles, California develops into a generalised dementia,'17 and Only a simple verbal or gestural indication of D Kempler we have yet to determine its underlying "yes" or "no" is required. Non-verbal tasks Gerontology Research neuropathology."l Additionally, little is included both traditional tests of attention and Center, Baltimore, known about its development (rate of progres- reasoning (5, 6, 8 below) as well as an array of Maryland E J Metter sion) or the epidemiology. nonverbal memory tasks developed by one of The purpose of this report is to furnish the authors (WHR). These were: VA Medical Center, Sepulveda, California more information about such patients. We 5 Block design. The subject is shown red and on September 28, 2021 by guest. Protected copyright. W H Riege have identified three individuals with progres- white blocks and asked to reconstruct seven VA Medical Center, sive speech and language deficits of unknown printed designs.'4 15 Sepulveda and UCLA, aetiology. Our goals were: 1) to document via 6 Ravens Coloured Progressive Matrices. California serial and medical records the a with one C A Jackson testing through The subject is shown visual pattern presence of a progressive language deficit, 2) part removed and asked to point to one of six Reed Neurological Institute, UCLA to compare language problems with other pictured inserts which contain the correct pat- California areas of mental status, and 3) to investigate tern.13 D F Benson through neuroimaging the structural and 7 Face Matching. The patient is presented VA Medical Center, metabolic bases for such disorders. A recent with pictures of 20 famous people from each Sepulveda, California, United States neuropathological necropsy report is included of three decades (1950s, 1960s, 1970s), and W R Hanson for one case. The three patients are described asked to point to 10 of them when named by Correspondence to: below. the examiner. Dr Kempler, Speech 8 Rhythm discrimination. The subject is Pathology, OPD 2P52, Los Angeles County/USC asked to discriminate between like and unlike Medical Center, 1175 Method pairs of musical beats.16 Cummings Street, Los Angeles, CA 90033, United Language and Memory 9 Design recognition. The subject is shown a States. The patients were tested with the Western set of 20 slides of abstract paintings (by Received 20 November 1989 Aphasia Battery (WAB),'2 which includes Vasarely) and asked to recognize the original and in revised form 25 February 1989. measures of spontaneous speech (fluency and set when shown a larger set of 40 paintings. Accepted 14 March 1990 information content), auditory comprehension 10 Auditory non-verbal recognition. The 988 Kempler, Metter, Riege, Jackson, Benson, Hanson J Neurol Neurosurg Psychiatry: first published as 10.1136/jnnp.53.11.987 on 1 November 1990. Downloaded from subject listens to a set of eight distinct bird minutes, scanning was initiated. Values of calls through head phones and is asked to local cerebral metabolic rate for glucose recognise the original set (by responding (LCMRGlc) were calculated as previously des- "yes" or "no") when listening to a larger set cribed.'920 Fifteen cerebral regions from each of bird calls. Recognition is tested im- hemisphere (fig 1) were outlined on a video mediately after presentation and then again 20 monitor using an interactive program, and minutes later (delayed condition). regional LCMRGlc values were derived in 11 Pattern reconstruction. The patient is milligrams of glucose per lOOg of tissue per shown a checkerboard pattern card containing minute. Region of localisation was based on the a design of red dots for five seconds, asked to atlas of Matsui and Hirano.2" To reduce the wait five seconds, and then asked to recon- number of comparisons, cortical regions from struct the red-dot pattern on a blank checker- adjacent tomographic levels were averaged board card. to give nine measures per hemisphere (for Performance on these tests are compared example, regions 1 and 2 combined to form a with the performance of 200 normal controls, "high frontal" region, 3 and 4 to form a and converted into Z-scores. For our pur- "low frontal" region, and 5 and 6 to form poses, z-scores between - 1 and -2 indicate a Broca's area). The LCMRGlc values ofaphasic mild impairment, between -2 and -3 patients' right hemispheres differ little from indicate a moderate impairment, and greater those of healthy controls but may show inter- than -3 a severe impairment. individual variation; therefore, left-right ratios for homologous regions were calculated and Neuroimaging used in analyses. Ratios of less than 0 90 were To discover neurofunctional information, considered abnormal for each regional ratio patients were studied with (F-18) fluoro- when compared with 22 normal controls. deoxyglucose (FDG) positron emission tomo- Each subject also had non-contrast CT scan graphy* (PET) on a Neuro-ECAT'920 in a (on either a Picker 1200 SX or GE 8800) with resting state with eyes and ears unoccluded. scanning in the same plane as PET. The same Patients lay on the scanner bed in a darkened regions as measured for glucose metabolism room, listening to ambient room noise and were were rated. In addition, the anterior internal injected intravenously with 18 5 x 1010 to 37 x capsule, posterior internal capsule, insula, and 1010 becquerels (5 to 10 mCi) of 18-F- lenticular nuclei were rated. The regions of fluorodeoxyglucose, followed by the drawing of interest were rated using a five-point scale (0, arterialised venous blood samples. After 40 normal; 1, atrophy; 2, structural damage with no tissue loss; 3, structural damage with partial tissue loss; and 4, structural damage with complete tissue loss) by a neuroradiologist who Figure 1 Diagram of regions analysedfor was blind to the project and by one of the structural damage and authors (EJM). Regional scores showed a 90% glucose metabolic rates. A NF 7 11 9 agreement between raters, and the two ratings 2 --I1 1 2 Regions I and 2 are high -2 were averaged to obtain an estimate of the - frontal; 3 and 4, low =1 9::_..rl., -if= II 5 frontal; 5 and 6, Broca's; i 15 --< >- F10 8 degree of structural damage. The three cases 7 and 8, parietal; 9 and -1 are presented individually below. 10, 11 and 4 Wernicke's; 1 http://jnnp.bmj.com/ 12, temporal; 13, % -- i. II0 occipital; 14 caudate; and Case 1 15, thalamus. (Reprinted I This is a left-handed male with a history of from Kempler et al, Disconnection and difficulty in remembering words, names and Cerebral Metabolism, faces that has progressed very slowly for ap- Archives ofNeurology proximately 15 years before our first evalu- 1988;45:275-9. most recent he was 59 1988, American ation. At the testing Copyright years old. He has been a puzzle to his Medical Association.) on September 28, 2021 by guest. Protected copyright. physicians, and has accumulated an impressive list ofdiagnoses, including chronic endogenous or possibly psychotic depression, sarcoid brain lesion, slow growing glioma, toxin exposure, Alzheimer's Disease, hippocampal damage, and Pick's disease.
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