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AusCADASIL: An Australian Cohort of CADASIL

Recruiting
Conditions
Cadasil
Registration Number
NCT06148051
Lead Sponsor
Perminder Sachdev
Brief Summary

The aim of this project is to establish an Australian cohort of patients diagnosed with Cerebral Autosomal Dominant Arteriopathy with Subcortical Infarcts and Leukoencephalopathy (CADASIL). This study will examine the clinical features and longitudinal course of CADASIL. Outcome measures include neuropsychological profile, neuroimaging, genetics, blood biomarkers, and retinal imaging.

Detailed Description

Using clinical examination, questionnaires, neuropsychological evaluation, brain MRI, blood sample evaluation and retinal imaging, we aim to characterise the clinical profile and progression of CADASIL in an Australian cohort.

This is multi-centre observational cohort study currently based at six sites (clinics, hospitals and universities) across three states in Australia (New South Wales, Victoria and Queensland). The multidisciplinary team aims to be the first to develop an Australian cohort of CADASIL which will contribute to global efforts and understanding of the disease.

Recruitment & Eligibility

Status
RECRUITING
Sex
All
Target Recruitment
300
Inclusion Criteria

Not provided

Exclusion Criteria

Not provided

Study & Design

Study Type
OBSERVATIONAL
Study Design
Not specified
Primary Outcome Measures
NameTimeMethod
Alphabet required for Trail Making A/BBaseline, Year 1, Year 2, Year 3, Year 4

As part of the neuropsychological battery, participants will need to complete the alphabet to inform ability to complete Trail Making tests A and B. These indicate executive function

Montreal Cognitive Assessment (MoCA)Baseline, Year 1, Year 2, Year 3, Year 4

As part of the neuropsychological battery, participants will complete the MoCA to provide a measure of global cognitive function

National Institute of Health Computerised Toolbox (NIHCTB)Baseline, Year 1, Year 2, Year 3, Year 4

As part of the neuropsychological battery, participants will complete the NIHCTB to provide measures of global function

Modified Rankin Scale (mRS)Baseline, Year 1, Year 2, Year 3, Year 4

Participants will undergo structured physical examination which will provide further details on their clinical profile. Scale from 0-5 (0 no symptoms, 5 severe disability)

National Institute of Health Stroke Scale (NIHSS)Baseline, Year 1, Year 2, Year 3, Year 4

Participants will undergo structured physical examination which will provide further details on their clinical profile. Individual items (11) relating to stroke symptoms and disability, for each item 0 indicates normal.

Brain MRI scan (total scan time 60 mins)- includes T1 weighted imagingBaseline, Year 3

To assess brain morphology

Brain MRI scan (total scan time 60 mins)- includes resting-state blood oxygenation level dependent (BOLD) imaging.Baseline, Year 3

Used to assess brain functional activity and amplitude of low-frequency fluctuations which embeds signal of cerebrovascular reactivity. For sites with available capnography monitors, participants' end-tidal carbon dioxide concentrations will be recorded during acquiring resting-state BOLD data for more accurate quantification of cerebrovascular reactivity

Weight and height will be combined to report BMI in kg/m^2Baseline, Year 1, Year 2, Year 3, Year 4

Participants will undergo structured physical examination which will provide further details on their clinical profile.

Blood PressureBaseline, Year 1, Year 2, Year 3, Year 4

Participants will undergo structured physical examination which will provide further details on their clinical profile. Systolic/diastolic blood pressure will be recorded 3 times during the physical examination

Category Fluency (animals)Baseline, Year 1, Year 2, Year 3, Year 4

As part of the neuropsychological battery, participants will complete Category Fluency to provide a measure of processing speed

Digit Span Backwards (WAIS-IV)Baseline, Year 1, Year 2, Year 3, Year 4

As part of the neuropsychological battery, participants will complete digit span backwards to provide a measure of executive function

Brain MRI scan (total scan time 60 mins)- includes pseudo-continuous arterial spin labelling (PCASL) perfusion imaging with multiple post-labelling delays.Baseline, Year 3

Used to assess quantitative cerebral blood flow and arterial transit time.

Online Medical QuestionnaireBaseline, Year 1, Year 2, Year 3, Year 4

Includes questions on participant's medical history (CADASIL and other), family history, and medication use. Outcome will be used to inform clinical profile

Letter Fluency (FAS)Baseline, Year 1, Year 2, Year 3, Year 4

As part of the neuropsychological battery, participants will complete the FAS to provide measures of executive function

Rey Auditory Verbal Learning Test (RAVLT)Baseline, Year 1, Year 2, Year 3, Year 4

As part of the neuropsychological battery, participants will complete the RAVLT to provide measures of learning and memory

Brain MRI scan (total scan time 60 mins)- includes T2-weighted fluid attenuated inversion recovery (FLAIR) imagingBaseline, Year 3

Combined with T1 outcome to assess brain morphology

Brain MRI scan (total scan time 60 mins)- includes susceptibility-weighted MRIBaseline, Year 3

Assesses cerebral microbleeds. Signal magnitude will be processed to estimate signal decay time (i.e., T2\* maps), which reflect compartmentalisation of magnetised tissue constituents (iron, calcium) \[22\]. Signal phase will be processed for quantitative susceptibility mapping.

Brain MRI scan (total scan time 60 mins)- includes multi-shell diffusion weighted MRIBaseline, Year 3

Measures of brain white matter microstructural integrity, such as fractional anisotropy and mean diffusivity, will be calculated using tensor models. Fixel-based analyses of fibre density and cross-section will also be performed. Markers of cerebrovascular diseases derived from diffusion data, such as Peak width of Skeletonised Mean Diffusivity (PSMD), will be calculated. Structural connectivity will also be examined.

Secondary Outcome Measures
NameTimeMethod
Study Partner Apathy Evaluation Scale (AES)Baseline

Completed online by nominated study partner (someone who knows the participant well e.g. spouse, carer, friend etc). Inform's participant's apathy by goal-directed behaviour, goal-related cognition, and goal-related emotional responses

Instrumental Activities of Daily Living Scale (IADL)Baseline

Completed online, 8 items to assess daily living skills in older adults. Outcome will be used to inform clinical profile

Multidimensional Fatigue Inventory (MFI)Baseline

Completed online, 20 items to assess physical and mental fatigue and motivation. Outcome will be used to inform clinical profile

Study Partner Instrumental Activities of Daily Living Scale (IADL)Baseline

Completed online by nominated study partner (someone who knows the participant well e.g. spouse, carer, friend etc). 8 items to assess daily living skills in older adults. Outcome will be used to inform participant's clinical profile

Ocular Assessment- StereopsisBaseline and year 3

The perception of depth will be measured using a stereo acuity instrument by viewing a series of stereograms.

Blood biochemistryBaseline, Year 3

Analysed by local pathology lab to indicate clinical profile. Measures will include fasting glucose, HbA1c, lipids, C-reactive protein, creatinine, vit D and liver function tests.

Ocular QuestionnaireBaseline, Year 3

Includes questions regarding vision ability and ocular symptoms, and history

Apathy Evaluation Scale (AES)Baseline

Completed online, 18 items to assess apathy by goal-directed behaviour, goal-related cognition, and goal-related emotional responses. Outcome will be used to inform clinical profile

Neuropsychiatric Inventory (NPI-Q)Baseline

Completed online by nominated study partner (someone who knows the participant well e.g. spouse, carer, friend etc). 12 domains, approx. 100 questions. Used to inform psychopathology in participants

Ocular Assessment-Visual AcuityBaseline and year 3

a measure of the vision function using high contrast letters will be performed with standardised vision charts at distance and at near.

Genetic ProfileBaseline

Genetic testing for NOTCH3 variants as well as potentially modifying genes

Ocular Assessment- Slit-lamp examinationBaseline and year 3

Involves viewing of the ocular structures using a slit-lamp illumination system and biomicroscope viewing system. The slit-lamp examination will be conducted by a trained observer and will allow identification of ocular media opacities, abnormalities of the eye lids, tear film and anterior structures.

Ocular Assessment- Standard automated perimetryBaseline and year 3

A measure of the sensitivity of the visual field.

Quality of Life Scale (EQ-5D-5L)Baseline

Completed online, 6 items to assess mobility, self-care, activities, pain/discomfort, anxiety/depression. Outcome will be used to inform clinical profile

Patient Health Questionnaire-9 (PHQ-9)Baseline

Completed online, 9 items to assess depression symptoms and severity. Outcome will be used to inform clinical profile

STOP-BANG Sleep QuestionnaireBaseline

Completed online, 8 items to assess Obstructive Sleep Apnoea risk. Outcome will be used to inform clinical profile

PROMIS Sleep DisturbanceBaseline

Completed online, 8 items to assess sleep quality. Outcome will be used to inform clinical profile

Hospital Anxiety and Depression Scale (HADS)Baseline

Completed online, includes 14 items to assess non-physical symptoms of anxiety and depression. Outcome will be used to inform clinical profile

Blood Omic investigationBaseline and year 3

Blood tubes will be transported to the University of New South Wales for genomics, proteomics, lipidomics, and epigenomics analysis

Ocular Assessment- Contrast SensitivityBaseline and year 3

A measure of vision function, relating to the ability to distinguish between an object and the background behind it.

Ocular Assessment- Intraocular pressureBaseline and year 3

A measure of the pressure within the eye.

Ocular Assessment- Fundus imaging (requires pupil dilation using tropicamide 0.5 or 1.0% eye dropsBaseline and year 3

Used to visualise the back of the eye

Ocular Assessment- Optical Coherence Tomography (OCT) and OCT-AngiographyBaseline and year 3

Used to visualise the back of the eye in retinal layers, and the retinal vessels

Trial Locations

Locations (5)

Prince of Wales Hospital

🇦🇺

Sydney, New South Wales, Australia

University of New South Wales

🇦🇺

Sydney, New South Wales, Australia

John Hunter Hospital

🇦🇺

Newcastle, New South Wales, Australia

Royal Brisbane and Women's Hospital

🇦🇺

Brisbane, Queensland, Australia

Royal Melbourne Hospital

🇦🇺

Melbourne, Victoria, Australia

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