Frontal allographic agraphia in a patient with behavioral variant frontotemporal dementia.

We report a patient with behavioral variant frontotemporal dementia who developed agraphia, irritability, perseverative and stereotyped behavior, and dietary changes. MRI revealed bilateral frontal convexity atrophy. Neuropsychological examination showed fluent aphasia with perseverative allographic agraphia, mild semantic impairment, and dysexecutive syndrome. Allographic agraphia featured unidirectional conversion from hiragana (cursive form of Japanese phonograms) and kanji (Japanese morphograms) to katakana (square form of Japanese phonograms), as opposed to mutual (bidirectional) conversion between hiragana and katakana in parieto-occipital gyri lesions. Furthermore, all letters of the word were converted and this whole-word conversion may be characteristic of perseverative behavior in frontotemporal dementia.

Two types of developmental surface dysgraphia: to bee but not to bea.

We report on two types of developmental surface dysgraphia. One type, exhibited by 8 participants, is orthographic lexicon surface dysgraphia, which involves an impairment in the orthographic output lexicon, leading to nonword phonologically-plausible misspellings. The other type, shown by 3 participants, is disconnection surface dysgraphia. In this type, the orthographic output lexicon is disconnected from the semantic system and from the phonological input lexicon, but still contributes to spelling via support to the orthographic output buffer, resulting in mainly lexical phonologically-plausible misspellings (writing be as "bee" but not "bea").The specific localization of the impairment in spelling, in the lexicon or in its connections, allowed us to examine the question of one or two orthographic lexicons; four participants who had a deficit in the orthographic output lexicon itself in writing had intact orthographic-input-lexicon in reading. They made surface errors in writing but not in reading the same words, supporting separate input and output orthographic lexicons.

Capecitabine-related neurotoxicity presenting with agraphia.

INTRODUCTION: Capecitabine is a pre-metabolite of 5-fluorouracil and is used as a chemotherapeutic agent. Among the common side effects of capecitabine, there are gastrointestinal side effects including nausea, vomiting, and diarrhea, and dermatological side effects including hand-foot syndrome and skin pigmentation change. However, neurological side effects of capecitabine are very rare. We describe herein a patient who developed neurological side effects in the form of agraphia and dysarthria on the 7th day of capecitabine treatment. CASE REPORT: A 34-year-old male patient, who was being followed up with the diagnosis of colon cancer, presented with speech and writing disorder that developed while under capecitabine treatment. Dysarthria and agraphia were detected in his neurological examination. Diffusion-weighted magnetic resonance imaging (MRI) revealed acute diffusion restriction in the splenium of the corpus callosum and at the level of the bilateral centrum semiovale. Brain MRI revealed symmetrical T2-weighted fluid-attenuated inversion recovery (T2-FLAIR) signal increases at the right temporoparietal medial, corpus callosum level, and bilateral white matter level. MANAGEMENT & OUTCOME: The capecitabine treatment was terminated, and methylprednisolone treatment was administered and plasmapheresis procedure was carried out. Subsequently, significant improvement was observed in the clinical findings and neuroimaging. DISCUSSION: Capecitabine is used as an oral agent; thus, it provides ease of use. Neurological side effects associated with the use of capecitabine reportedly occur very rarely. The findings of this case demonstrated that leukoencephalopathy can be seen during the use of capecitabine, imaging results are very important in the diagnosis of leukoencephalopathy, and improvement can be achieved with the termination of the capecitabine treatment.

Two adult patients with moyamoya disease presenting with alexia with agraphia limited to kanji: can revascularization improve characteristic symptoms?

Patients with moyamoya disease (MMD) may exhibit higher brain dysfunction due to hypoperfusion, which may be ameliorated by revascularization. However, few studies have examined the relationship between cerebral perfusion and language function or the ameliorating effect of revascularization on language dysfunction. We present two cases with MMD who presented with alexia with agraphia, specifically for Japanese kanji. The patients had impaired perfusion in the left inferior temporal and lateral occipital lobes. Following superficial temporal artery-middle cerebral artery bypass, the symptoms improved dramatically. Thus, correction of hypoperfusion may be effective even in adult patients with MMD presenting with language dysfunction.

A case of aneurysmal subarchnoid haemorrhage and superficial siderosis complicated by prospagnosia, simultagnosia and alexia without agraphia.

A 42-year-old lady presented with acute aneurysmal subarachnoid haemorrhage and developed difficulty recognising faces (prosopagnosia), inability to process visual information in busy environments (simultagnosia) and difficulty to read (alexia). She was subsequently found to have superficial siderosis on MRI.

Handwriting Evaluation Using Deep Learning with SensoGrip.

Handwriting learning disabilities, such as dysgraphia, have a serious negative impact on children's academic results, daily life and overall well-being. Early detection of dysgraphia facilitates an early start of targeted intervention. Several studies have investigated dysgraphia detection using machine learning algorithms with a digital tablet. However, these studies deployed classical machine learning algorithms with manual feature extraction and selection as well as binary classification: either dysgraphia or no dysgraphia. In this work, we investigated the fine grading of handwriting capabilities by predicting the SEMS score (between 0 and 12) with deep learning. Our approach provided a root-mean-square error of less than 1 with automatic instead of manual feature extraction and selection. Furthermore, the SensoGrip smart pen SensoGrip was used, i.e., a pen equipped with sensors to capture handwriting dynamics, instead of a tablet, enabling writing evaluation in more realistic scenarios.

Investigating Visual Perception Impairments through Serious Games and Eye Tracking to Anticipate Handwriting Difficulties.

Dysgraphia is a learning disability that causes handwritten production below expectations. Its diagnosis is delayed until the completion of handwriting development. To allow a preventive training program, abilities not directly related to handwriting should be evaluated, and one of them is visual perception. To investigate the role of visual perception in handwriting skills, we gamified standard clinical visual perception tests to be played while wearing an eye tracker at three difficulty levels. Then, we identified children at risk of dysgraphia through the means of a handwriting speed test. Five machine learning models were constructed to predict if the child was at risk, using the CatBoost algorithm with Nested Cross-Validation, with combinations of game performance, eye-tracking, and drawing data as predictors. A total of 53 children participated in the study. The machine learning models obtained good results, particularly with game performances as predictors (F1 score: 0.77 train, 0.71 test). SHAP explainer was used to identify the most impactful features. The game reached an excellent usability score (89.4 ± 9.6). These results are promising to suggest a new tool for dysgraphia early screening based on visual perception skills.

Speech therapy and hyperbaric oxygen for aphasia after carbon monoxide intoxication.

Acute carbon monoxide (CO) intoxication may result in delayed neurological sequelae, which can include amnesia, ataxia, aphasia, emotional lability, disorientation, dysphagia, and other manifestations. A 27-year-old man reported symptoms of aphasia with agraphia and alexia in a review after CO intoxication. The patient received outpatient speech therapy, as well as repeated sessions of hyperbaric oxygen for 15 days, interspersing speech therapy with hyperbaric oxygen therapy for two months. After this period of combined treatment the aphasic symptomatology remitted, and oral and written language was normal. The complete disappearance of aphasia with agraphia and alexia confirms the efficacy of the combined intervention. More data from large clinical studies are needed to assess the outcomes of hyperbaric oxygen treatment in patients with delayed neurological sequelae after CO intoxication, but this case suggests it may be a good therapeutic option in combination with specific speech therapy.

Functional Assessment of Handwriting Among Children: A Systematic Review of the Psychometric Properties.

IMPORTANCE: Handwriting legibility and speed assessments have a critical role in identifying and evaluating handwriting problems, which are common among children. OBJECTIVE: The objective was to evaluate the psychometric properties and clinical utility of handwriting assessments for children ages 3 to 16 yr. DATA SOURCES: A systematic review was conducted in CINAHL, PubMed (MEDLINE), Scopus, and education databases, with no time limits. The search strategy included a combination of the following keywords: handwriting, write, children, assessment, and validity. The exclusion criteria were assessment tools that were electronic, that focused on cognitive components of handwriting, or that only evaluated alphabets other than Latin. STUDY SELECTION AND DATA COLLECTION: The systematic review was carried out on the basis of the COnsensus-based Standards for the selection of health Measurement INstruments (COSMIN) checklist and Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) recommendations. The protocol was registered in the Prospective Register of Systematic Reviews (PROSPERO). FINDINGS: The 14 included instruments had a total sample of 4,987 children. Internal consistency ranged from moderate (.73; Writing Readiness Inventory Tool in Context) to high (.98; Letter Writing). The interexaminer reliability values of the 11 direct assessment instruments ranged from .77 (Systematic Screening for Handwriting Difficulties) to 1.00 (Handwriting Speed Test). CONCLUSIONS AND RELEVANCE: In this systematic review, existing tools were evaluated by clinical utility and the quality of psychometric properties. Direct assessments showed good psychometric properties. Indirect and self-assessment tools demonstrated poor psychometric properties. Further research on screening tools and tools in other languages is needed. What This Article Adds: Specific learning disorders (e.g., dysgraphia) negatively affect academic learning and, when prolonged in time, self-concept. However, handwriting legibility and speed assessments could be used to identify and evaluate these learning disorders if an early referral to occupational therapy is carried out.

The Modularity of Dysgraphia.

Research regarding dysgraphia, an impairment in writing, is attaining more attention in recent times. The existing studies on dysgraphia draw insights from cognitive, behavioural, neurological, and genetic fields of knowledge. However, these multiple studies on dysgraphia fail to illustrate how these cognitive, behavioural, neurological, and genetic systems interact and intersect in dysgraphia. Therefore, the studies could not offer a comprehensive understanding of dysgraphia. In order to fill this gap, the review attempts to study dysgraphia using the notion of modularity by accommodating insights from cognitive, behavioural, neurological, and genetic aspects of dysgraphia. Such a profound understanding could facilitate an early diagnosis and holistic intervention towards dysgraphia.

Properties of graphic motor plans in the writing system.

The present study explores the extent to which properties of abstract graphemic representations are maintained at the post-graphemic level of graphic motor plans, where the sequences of writing strokes for producing the letters in a word are represented. On the basis of results from a stroke patient (NGN) who has a deficit affecting the activation of graphic motor plans, we explore the post-graphemic representation of 1) consonant/vowel status of letters; 2) geminate (double) letters, such as the BB in RABBIT; and 3) digraphs, such as the SH in SHIP. Through analyses of NGN's letter substitution errors, we conclude that 1) consonant-vowel status is not represented at the level of graphic motor plans; 2) geminates have special representations at the motor-plan level, as at the graphemic level; and 3) digraphs are represented by two separate single-letter graphic motor plans, and not by unitary digraph motor plans.

Tonal and orthographic analysis in a Cantonese-speaking individual with nonfluent/agrammatic variant primary progressive aphasia.

Clinical understanding of primary progressive aphasia (PPA) has been established based on English-speaking population. The lack of linguistic diversity in research hinders the diagnosis of PPA in non-English speaking patients. This case report describes the tonal and orthographic deficits of a multilingual native Cantonese-speaking woman with nonfluent/agrammatic variant PPA (nfvPPA) and progressive supranuclear palsy. Our findings suggest that Cantonese-speaking nfvPPA patients exhibit tone production impairments, tone perception deficits at the lexical selection processing, and linguistic dysgraphia errors unique to logographic script writer. These findings suggest that linguistic tailored approaches offer novel and effective tools in identifying non-English speaking PPA individuals.

Using a digital spelling aid to improve writing in persons with post-stroke aphasia: An intervention study.

BACKGROUND: Intervention studies aimed to improve the written production of single words by persons with aphasia have yielded promising results and there is growing interest in interventions targeting text writing. The development of technical writing aids offers opportunities for persons with aphasia, and studies have shown that using them can have a positive impact on written output. AIMS: The aim was to investigate what impact training to use a computerised spell checker had on text writing in persons with aphasia. METHODS & PROCEDURES: The study had a multiple-baseline single-case experimental design replicated across six male Swedish participants with mild-to-moderate post-stroke aphasia. The participants received training twice a week during 8 weeks, learning how to use the spell checker. At baseline and before every session, the participants wrote two texts which were logged in a keystroke-logging tool. Dependent variables were continuously measured in the texts, and the participants performed tests of language function and answered questionnaires on reading and writing habits and health-related quality of life before and after the intervention. The participants were also interviewed about how they had experienced the training. The results were evaluated on individual and group level. RESULTS: The study showed that systematic individual training involving a spell checker was experienced as positive by the participants and that they all described their writing ability in more positive terms after the intervention. Evaluation showed statistically significant improvements on group level for the dependent variables of spelling accuracy, rated syntax, writing speed and proportion of unedited text during text writing when using the spell checker. The intervention also had a generalising effect on writing speed and editing during text writing without the spell checker and on spelling accuracy in a dictation test. The participants who had the greatest spelling problems were the ones who showed the most progress, but participants with only minor writing difficulties at baseline also improved. CONCLUSIONS & IMPLICATIONS: The study shows that a digital spelling aid constitutes effective support for people with aphasia and may also affect levels other than spelling. The training had a generalising positive effect on text writing and spelling in a test. Although writing difficulties is a persisting symptom in aphasia, it can be supported and improved through use of digital spelling aids. Hence, treatment of writing ability should always be included in the rehabilitation of people with aphasia. WHAT THIS PAPER ADDS: What is already known on this subject Use of a technical writing aid can have a positive impact on the written output of persons with aphasia. Using a digital spell checker may improve spelling as well as other levels of writing, but it has not been investigated using a keystroke-logging tool in combination with language-test scores and results from questionnaires. What this paper adds to existing knowledge Through analyses on both individual and group level, this study shows that a digital spelling aid constitutes effective support for people with aphasia and also affects levels other than spelling. The training had a generalising positive effect on text writing and spelling in a test. What are the potential or actual clinical implications of this work? Digital spelling support, which is a relatively simple and inexpensive technology, can support and improve text writing in persons with post-stroke aphasia.

'I think writing is everything': An exploration of the writing experiences of people with aphasia.

BACKGROUND: Written communication has become an increasingly important part of everyday life in social, educational and professional spheres. The substantial increase in writing via the internet and mobile technologies provides both an opportunity for social engagement and distinct challenges for people with aphasia. Within the current literature there has been limited research into the lived experiences of people with aphasia of their writing difficulties and how these affect their ability to communicate. AIMS: This qualitative study aimed to explore the experiences of people with aphasia of living with language-related writing difficulties and the impact of these on their lives. METHODS & PROCEDURES: Eight people with post-stroke aphasia and writing difficulties took part in semi-structured interviews. The interviews were analysed using inductive reflexive thematic analysis. OUTCOMES & RESULTS: Two themes were found in the data. The first theme was a gradual and effortful improvement to writing: Participants described how writing had improved since their stroke due to strategies and support, but they still found writing to be difficult and frustrating and described many barriers to writing. The second theme was the importance of writing for fulfilling adult social roles: Participants found writing to be important for communicating with family, friends and organizations, but their participation in society and self-esteem and confidence were impacted by writing difficulties; reduced social roles meant reduced need for writing, but participants were still motivated to work towards writing goals. CONCLUSIONS & IMPLICATIONS: The findings demonstrate the emerging importance of writing skills for people with aphasia with respect to communication, well-being, participation and inclusion in society, and carrying out social roles. They provide an insight into the process of improvement, including the difficulties, facilitators and barriers. Implications for speech and language therapy assessment and management are discussed. WHAT THIS PAPER ADDS: What is already known on the subject People with aphasia have difficulties with writing that can affect their ability to communicate. A small body of qualitative research has provided insights into individuals' experiences of literacy difficulties. More research is needed to understand the writing experiences of people with aphasia to help design appropriate assessments and interventions. What this paper adds to existing knowledge Participants experienced gradual and effortful improvement since their stroke. They felt negative about aspects of their writing, including speed, accuracy and range of vocabulary. Writing was facilitated through assistive technologies, spelling practice and support from others; barriers included technology, lack of time, stroke-related symptoms and others' lack of awareness about aphasia. Participants considered writing skills to be important, particularly for communication, carrying out adult social roles and participating in society, and were therefore still working towards goals related to everyday writing activities. What are the potential or actual clinical implications of this work? This study suggests that speech and language therapy assessment should include interviewing participants about their activities, strengths, difficulties, facilitators and barriers in writing, and informal assessment of a range of functional writing tasks. Intervention should be tailored to the individual's needs. This should include meaningful activities that relate to functional everyday writing and, where appropriate, self-management, compensatory technologies and group approaches, while making use of existing strategies identified by the individual.

Pseudoword spelling ability predicts response to word spelling treatment in acquired dysgraphia.

Although rehabilitation of acquired dysgraphia can be quite effective, identifying predictors of responsiveness to treatment is useful for prognosis and individualization of treatment protocols. This study examined whether various features of treatment response were predicted by the integrity of one or more of the central cognitive components of spelling: orthographic long-term memory, orthographic working memory, and phoneme-grapheme conversion. Twenty dysgraphic individuals received 12 weeks of bi-weekly, individualized, lexically-based spelling rehabilitation using a spell-study-spell paradigm. Linear multiple regression modelling examined whether the type and severity of the dysgraphic deficit, assessed before rehabilitation, predicted the magnitude and rate of improvement, generalization to untrained items and maintenance of treatment gains. The results revealed that pseudoword spelling accuracy - indexing the integrity of the phoneme-grapheme conversion system - was the only factor examined that significantly predicted the rate of accuracy gains for trained words as well as the extent of generalization to untrained words. Pre-treatment pseudoword spelling accuracy also predicted retention of gains for trained and untrained words at 3-month follow-up. These findings reveal that the integrity of the phoneme-grapheme conversion system prior to dysgraphia rehabilitation may play a key role in rehabilitation-driven recovery, even when the treatment approach targets lexical rather than pseudoword spelling processes.

A Case Report on Dysgraphia in a Patient Receiving Blinatumomab: Complex Characters Are Easy to Find in a Handwriting Test.

Recent advances in chemotherapy have led to the emergence of new types of anticancer agents. With these advances, cases of side effects that have not been witnessed in the past have emerged. The systems of side effect evaluation and their grading have been based on the existing knowledge, such as the CTCAE (Common Terminology Standard for Adverse Events) for evaluating adverse drug reactions in cancer chemotherapy clinical trials. Therefore, new types of side effects may be overlooked or underestimated. Blinatumomab is a bispecific T-cell-engager (BiTE) antibody with specificity for CD19 on B cells and CD3 on T cells. Neurological events, such as neuropathy and encephalopathy, are serious side effects of BiTE antibodies. We encountered a case of a 62-year-old woman who experienced short-term memory impairment and dysgraphia after the first blinatumomab administration for Philadelphia chromosome negative (Ph-) B-cell acute lymphoblastic leukemia (ALL). The CTCAE does not include dysgraphia as a classifier for antibody therapies, such as blinatumomab, and immune effector cell-associated neurotoxicity syndrome, which is defined as a Chimeric antigen receptor T cell therapy-related toxicity; dysgraphia is included in the list of symptoms but is not graded. In this case, the severity of dysgraphia differed depending on the complexity of the letters examined. There is no report that the severity of dysgraphia depends on the letters' complexity, and therefore, it may be overlooked when using simple letters. We have reported the characteristics of dysgraphia in this case and the differences observed when judging different letters.

Serial position effects in graphemic buffer impairment: An insight into components of orthographic working memory.

This study investigated the nature of graphemic buffer functioning and impairment, through analysis of the spelling impairment shown by GEC, a man with acquired dysgraphia and clear characteristics of graphemic buffer impairment. We discuss GEC's error patterns in relation to different processes of orthographic working memory. This is the first study to show the contribution of these processes in one individual through performance on different spelling tasks. GEC's spelling errors in writing to dictation showed a linear serial position effect, including deletions of final letters. These "fragment errors" can be explained as the result of information rapidly decaying from the buffer (reduced temporal stability). However, in tasks that reduced working memory demands, GEC showed a different error distribution that may indicate impairment to a different buffer process (reduced representational distinctiveness). We argue that different error patterns can be a reflection of subcomponents of orthographic working memory that can be impaired separately.

Dysgraphia and dysprosody in a patient with arteriovenous malformation: a case report.

Arteriovenous malformation (AVM) results from development of abnormal connections between veins and arteries. This study reported anAVM case suffering from dysgraphia and dysprosody. According to the results after the trauma, the patient's handwriting was identified as macrographic and illegible, and written letters and verbs were neglected in free writing or dictation. Moreover, prosody of the patient's utterances was changed. Finally, an intervention was conducted to improve the writing impairments whereby they eventually enhanced. AVM can adversely affect communication opportunities and working life due to these impairments. Thus referring the patient to speech and language pathologists seems sensible and necessary.

Transcranial direct current stimulation with multiple oral re-reading therapy for pure alexia without agraphia: a case report.

Pure alexia without agraphia is characterized by impaired reading due to damage to the occipitotemporal cortex with preserved writing skills. In this case report, we investigate the effect of multiple oral re-reading (MOR) therapy adjunct with transcranial direct current stimulation (tDCS) in improving reading recovery of a 64-year-old patient with pure alexia without agraphia following a stroke. His MRI revealed an area of infarct with microhemorrhages at the left occipitotemporal region. The patient was blinded to each therapy and underwent seven consecutive sessions of sham tDCS followed by seven consecutive sessions of real tDCS, coupled with 1-hour MOR therapy during each session. Western Aphasia Battery (WAB) was performed at baseline, before sham and real-tDCS, and 6 weeks after completing tDCS therapy. The patient showed improvement using both sham and real-tDCS with better reading comprehension, average reading time, and word per minute after real-tDCS. This study suggests that MOR, coupled with tDCS therapy may accelerate the reading recovery in patients with pure alexia.

Could pure agraphia be the only sign of stroke? Lessons from two case reports.

Agraphia is defined as the disruption of the previously intact writing skills due to an acquired brain damage. Stroke remains the most common cause of language impairment; however, writing disorders, including agraphia, are underestimated in patients with stroke. In this regard, we report two patients presenting with pure agraphia as an early symptom of stroke. Both patients complained of at least two difficulties in visualizing letter formation beforehand, the frequent need for verbal cues, misuse of lines and margins, poorly legible signature, and writing and thinking at the same time (e.g., creative thinking and taking notes). They underwent brain magnetic resonance imaging which revealed a small lacunar infarction of the left insula and external capsule (patient 1) and a small hemorrhagic lesion in the posterior limb of the left internal capsule (patient 2). To our knowledge, this is the first report on pure agraphia as the presenting symptom of stroke. We suggest that all patients with acute agraphia, even when presenting as an isolated symptom, should be evaluated for stroke, in order to better facilitate its diagnosis and treatment.