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The Skin as a Window to the Central Nervous System in Frontotempolar Lombar Degeneration

Not Applicable
Recruiting
Conditions
Frontotemporal Lobar Degeneration
Interventions
Procedure: Skin biopsy
Registration Number
NCT06490822
Lead Sponsor
Nantes University Hospital
Brief Summary

Frontotemporal lobar degeneration (FTLD) is a clinically heterogeneous syndrome, characterized by progressive decline in behaviour and/or language. From a pathological standpoint, like the great majority of neurodegenerative disorders, FTLD are proteinopathies, which are characterized by the presence of specific protein deposits in the Central Nervous System (CNS). Accordingly, the two main deposits observed in FTLD are either made of Tau or transactive response DNA binding protein 43 (TDP-43).

In pathological conditions such as FTLD, both proteins are aggregated and hyperphosphorylated.

It is now well established that the pathological process in some proteinopathies such as synucleinopathies (of which Parkinson's disease is the main representative) is not limited to the brain but also widespread throughout the peripheral autonomic networks, including the autonomic innervation of the skin. In this context, many independent studies have shown that the pathological process in PD could be detected using routine punch skin biopsies opening the way for the development of original histopathological markers of the disease.

Our hypothesis is that such a scenario could also occur in FTLDs and that the detection of the pathological tau or TDP-43 protein in the skin could help in diagnosing FTLD. This is especially relevant as, despite the recent progress in genetics, neurobiology and neuroimaging, there are no available biomarkers for FTLD.

Detailed Description

Participant with Frontotemporal Lobar degeneration equally distributed into behavioral variant of frontotemporal dementia (bvFTD), language variant with primary progressive aphasia (PPA) and motor presentations with atypical parkinsonian disorders (corticobasal degeneration-CBD and progressive supranuclear palsy-PSP) and motoneuron disorder (amyotrophic lateral sclerosis -ALS) will be included at Nantes University Hospital during a period of 24 months.

Healthy volunteers will be included as comparative group. A skin biopsy and venous blood samples will be collected for all participants.

Recruitment & Eligibility

Status
RECRUITING
Sex
All
Target Recruitment
80
Inclusion Criteria

Not provided

Exclusion Criteria

Not provided

Study & Design

Study Type
INTERVENTIONAL
Study Design
SINGLE_GROUP
Arm && Interventions
GroupInterventionDescription
Single arm studySkin biopsyA single 3 mm-diameter punch skin biopsy will be obtained from FTLD patients and healthy volunteers at the C8 paravertebral under local anesthesia to analyze cutaneous innervation
Primary Outcome Measures
NameTimeMethod
Amount of Tau assessed by Western blot and qPCRDay of inclusion

assessed by Western blot and qPCR to determine level of expression of Tau in the skin

Amount of TDP 43Day of inclusion

assessed by Western blot and qPCR to determine level of expression of TDP-43 in the skin

Secondary Outcome Measures
NameTimeMethod
Amount of TDP 43 phosphorylationDay of inclusion

phospho-TDP-43 /TDP43 ratio in the skin will be measured by Western blot

FTLD mutationbefore inclusion

Mutation carrier patient will be identified among participant (not all the participant) who had a genetic screening had neen carried out during their follow up.

Neuropsychological characterizationday of inclusion for patients vFTD, PPA and corticobasal degeneration-CBD and progressive supranuclear palsy-PSP within 12 months of inclusion for patients ALS

During their follow-up FTLD patients underwent a complete neuropsycological assessment define as:

* mini-mental state examination (MMSE)

* the 16-item Free Recall/Indicated Recall test (RL/RI-16 items)

* Rey figure and recall

* Trail Making Test (TMT A \& B)

* Wechsler Adult Intelligence Scale

* Literal (letter P) and categorical (animals) fluency

* Rapid Frontal Efficiency Battery (BREF)

* Wisconsin Card Sorting Test (WCST)

* Zoo test of Behavioural Assessment of the Dysexecutive Syndrome (BADS)

* Stroop test - 4 boards

* Oral image naming test (DO 80)

* Theory of mind (ToM-15)

* ekman facial emotion recognition test

Amount of Tau phosphorylationDay of inclusion

phospho-tau/tau ratio in the skin will be measured by Western blot

Behaviour profileday of inclusion for patients vFTD, PPA and corticobasal degeneration-CBD and progressive supranuclear palsy-PSP within 12 months of inclusion for patients ALS

The 10-items scale Daphne will be used to explore disinhibition (4 items), apathy(1 item), perseveration (1 item), hyperorality (2items), personal neglect (1 item) and loss of empathy (1 item)

Trial Locations

Locations (1)

Nantes University Hospital

🇫🇷

Nantes, France

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