Long-Term Severe Mental Disorders Preceding Behavioral Variant Frontotemporal Dementia: Frequency and Clinical Correlates in an Outpatient Sample

Gambogi, Leandro Boson; Guimarães, Henrique Cerqueira; Souza, Leonardo Cruz de; Caramelli, Paulo

doi:10.3233/jad-180528

Cited by 12 publications

(21 citation statements)

References 48 publications

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“…Also, there are case reports of late-onset (>85) bvFTD ( 50 ). It is also interesting to mention a paper on long-term severe mental disorders preceding bvFTD in a Brazilian cohort ( 51 ).…”

Section: Resultsmentioning

confidence: 99%

Frontotemporal Dementias in Latin America: History, Epidemiology, Genetics, and Clinical Research

et al. 2021

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Introduction: The historical development, frequency, and impact of frontotemporal dementia (FTD) are less clear in Latin America than in high-income countries. Although there is a growing number of dementia studies in Latin America, little is known collectively about FTD prevalence studies by country, clinical heterogeneity, risk factors, and genetics in Latin American countries.Methods: A systematic review was completed, aimed at identifying the frequency, clinical heterogeneity, and genetics studies of FTD in Latin American populations. The search strategies used a combination of standardized terms for FTD and related disorders. In addition, at least one author per Latin American country summarized the available literature. Collaborative or regional studies were reviewed during consensus meetings.Results: The first FTD reports published in Latin America were mostly case reports. The last two decades marked a substantial increase in the number of FTD research in Latin American countries. Brazil (165), Argentina (84), Colombia (26), and Chile (23) are the countries with the larger numbers of FTD published studies. Most of the research has focused on clinical and neuropsychological features (n = 247), including the local adaptation of neuropsychological and behavioral assessment batteries. However, there are little to no large studies on prevalence (n = 4), biomarkers (n = 9), or neuropathology (n = 3) of FTD.Conclusions: Future FTD studies will be required in Latin America, albeit with a greater emphasis on clinical diagnosis, genetics, biomarkers, and neuropathological studies. Regional and country-level efforts should seek better estimations of the prevalence, incidence, and economic impact of FTD syndromes.

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“…Also, there are case reports of late-onset (>85) bvFTD ( 50 ). It is also interesting to mention a paper on long-term severe mental disorders preceding bvFTD in a Brazilian cohort ( 51 ).…”

Section: Resultsmentioning

confidence: 99%

Frontotemporal Dementias in Latin America: History, Epidemiology, Genetics, and Clinical Research

et al. 2021

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“…Finally, it is becoming clear that psychosis and other psychiatric disorders are likely part of the broader spectrum of ADCY5-dyskinesia. 1,3,5 Patients in the early stages of frontotemporal dementia, another tauopathy, 19 have been given a diagnosis of schizophrenia before development of substantial cognitive impairment. Additional brain autopsies will need to be evaluated to determine whether the tau abnormalities in our case comprise a pathological signature of ADCY5-dyskinesia and whether it is restricted to cases with psychosis.…”

Section: Discussionmentioning

confidence: 99%

Hyperphosphorylated Tau, Increased Adenylate Cyclase 5 (ADCY5) Immunoreactivity, but No Neuronal Loss in ADCY5‐Dyskinesia

Chen

Latimer

Spencer

et al. 2019

Movement Disord Clin Pract

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Background Background: Adenylate cyclase 5 (ADCY5)-related dyskinesia is a childhood-onset movement disorder. Manifestations vary in frequency and severity and may include chorea, tremor, dystonia, facial twitches, myoclonus, axial hypotonia, and limb hypertonia. Psychosis is likely part of the broader spectrum. ADCY5 is widely expressed in the brain, especially in the striatum. Previous reports of brain autopsies of 2 subjects with likely ADCY5-dyskinesia were limited by the absence of a molecular diagnosis. In 1 case, normal gross pathology was reported. In the other case, ADCY5 expression was not examined and neuropathological findings were confounded by age and comorbidities. Objectives Objectives: To examine ADCY5 expression and neuropathological changes in ADCY5-dyskinesia. Methods Methods: An extensive brain autopsy, including immunohistochemical analyses with antibodies to paired helical filament tau, α-synuclein, amyloid-β, microtubule-associated protein 2, and ADCY5, was performed. ResultsResults: The patient, with a p.M1029K ADCY5 variant, had severe dyskinesias from early childhood, later recurrent episodes of psychosis, and died at age 46. Gross pathology was unremarkable, but we detected increased immunoreactivity for ADCY5 in neurons in multiple brain regions. Despite no history of brain trauma to suggest chronic traumatic encephalopathy, we found tau deposits in the deep cortical sulci, midbrain, and hippocampus with minimal amyloid pathology and no Lewy bodies. Conclusions Conclusions: We present the first brain autopsy findings in a molecularly proven case of ADCY5-dyskinesia, showing increased ADCY5 immunoreactivity in neurons and evidence of tau deposition. Additional patients will need to be studied to determine whether increased immunoreactivity for ADCY5 is a signature for ADCY5-dyskinesia and whether this disease has a tauopathy component.

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“…Semantic fluency in LA bvFTD cohorts may also be influenced by cerebrovascular disease, as patients without such comorbidity had lower scores on specific categories (animals) ( 66 ). Finally, there seems to be no difference in semantic fluency between bvFTD patients with and without psychiatric history ( 65 ).…”

Section: Linguistic Research In La Bvftdmentioning

confidence: 99%

Language in Behavioral Variant Frontotemporal Dementia: Another Stone to Be Turned in Latin America

et al. 2021

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Beyond canonical deficits in social cognition and interpersonal conduct, behavioral variant frontotemporal dementia (bvFTD) involves language difficulties in a substantial proportion of cases. However, since most evidence comes from high-income countries, the scope and relevance of language deficits in Latin American bvFTD samples remain poorly understood. As a first step toward reversing this scenario, we review studies reporting language measures in Latin American bvFTD cohorts relative to other groups. We identified 24 papers meeting systematic criteria, mainly targeting phonemic and semantic fluency, naming, semantic processing, and comprehension skills. The evidence shows widespread impairments in these domains, often related to overall cognitive disturbances. Some of these deficits may be as severe as in other diseases where they are more widely acknowledged, such as Alzheimer's disease. Considering the prevalence and informativeness of language deficits in bvFTD patients from other world regions, the need arises for more systematic research in Latin America, ideally spanning multiple domains, in diverse languages and dialects, with validated batteries. We outline key challenges and pathways of progress in this direction, laying the ground for a new regional research agenda on the disorder.

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Long-Term Severe Mental Disorders Preceding Behavioral Variant Frontotemporal Dementia: Frequency and Clinical Correlates in an Outpatient Sample

Cited by 12 publications

References 48 publications

Frontotemporal Dementias in Latin America: History, Epidemiology, Genetics, and Clinical Research

Frontotemporal Dementias in Latin America: History, Epidemiology, Genetics, and Clinical Research

Hyperphosphorylated Tau, Increased Adenylate Cyclase 5 (ADCY5) Immunoreactivity, but No Neuronal Loss in ADCY5‐Dyskinesia

Language in Behavioral Variant Frontotemporal Dementia: Another Stone to Be Turned in Latin America

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