Charcot-Marie-Tooth DiseaseNCT03782883
The Impact of Charcot-Marie-Tooth Disease in the Real World
An observational study of Charcot-Marie-Tooth Disease, run by Vitaccess Ltd at 1 site in United Kingdom.
Trial summary
An observational, non-interventional registry study to collect real-world data from people living with Charcot-Marie-Tooth disease (CMT) and its treatment, which will be available to researchers to further the knowledge of Charcot-Marie-Tooth disease and improve patient care. Read more about this study
- Who is it for?
- Ages 18 years and older
- Conditions the study is for
- Charcot-Marie-Tooth Disease
- How many people
- 3,321 participants
- Where
- 1 siteUnited Kingdom
- Study dates
- 9 Oct 2018to 31 Dec 2023
- How long will I take part?
- 62 Months
- Phase
- Observational
- Are there results?
- None posted in the registry
Key requirements
Who can take part, as listed by the sponsor. Only the study team can confirm whether you are eligible, so take this list to your doctor.
- Ages 18 years and older
- All sexes
- No healthy volunteers
- Adult, Older adult
Study population: Adults (age >18 years) with any stage and any subtype of Charcot-Marie-Tooth disease, who are resident in France, Germany, Italy, Spain, the United Kingdom (UK) or the USA, so long as they have a National Health Service (NHS) or Community Health Index (CHI) number (or equivalent) and are willing to use their own smartphone / tablet.
Who may be able to join
- () Aged 18 years and over () Diagnosed with any stage and any subtype of Charcot-Marie-Tooth disease () Resident in France, Germany, Italy, Spain, the UK, or the USA () Have a National Health Service (NHS) or Community Health Index (CHI) number, or equivalent () Willing to use their own smartphone
Who cannot join
- () No specific exclusion criteria.
Trial locations and contacts
1 site in 1 country.
About this study
An observational, non-interventional registry study to collect real-world data from people living with Charcot-Marie-Tooth disease (CMT) and its treatment, which will be available to researchers to further the knowledge of Charcot-Marie-Tooth disease and improve patient care.
Read the detailed description
The registry uses Vitaccess' digital real-world evidence platform and has been developed in collaboration with CMT experts, Patient Advocacy Organizations (PAOs) and the biopharmaceutical company Pharnext.
Eligible participants install a study app on their smartphone. Researchers access aggregated, anonymised data via a cloud-based research portal.
The platform provides benefits to participants, which it is hoped will encourage persistence with data submission; these include options to download a copy of their symptom diary, a clinically-validated Knowledge feature, and access to high-level results from the study data.
The aggregated data are available in close to real time via "dashboards" and can be analysed according to a number of pre-set criteria (e.g. disease stage, age, geographic location).
State-of-the-art technologies and security policies are used in the platform to ensure industry-standard data storage and privacy for all users. Participants' personally identifiable information will remain confidential at all times, and researchers will not be able to identify individuals.
The study is ethics-approved in all scope countries and led by a Scientific Advisory Board comprising representatives from international PAOs, patients and key opinion leaders from each country, and study leads from both Pharnext Société Anonyme (SA) and Vitaccess Ltd.
- Conditions
- Charcot-Marie-Tooth Disease
- Medical subject headings
- Charcot-Marie-Tooth Disease
What is being measured
The primary outcome is the main question the study is designed to answer.
EuroQoL 5-dimension 5-level (EQ-5D-5L)
The EQ-5D-5L comprises two parts - the EQ-5D-5L descriptive system and the EQ Visual Analogue Scale (EQ VAS).
The descriptive system comprises 5 dimensions (mobility, self care, usual activities, pain/discomfort, and anxiety/depression), each with 5 levels (no problems, slight problems, moderate problems, severe problems, and extreme problems - i.e., higher scores represent worse health). The digits for 5 dimensions can be combined in a 5-digit number describing the respondent's health state.
The EQ VAS records the respondent's self-rated health on vertical, visual analogue scale with endpoints labelled 'the best health you can imagine' and 'the worst health you can imagine'. Higher scores represent better self-perceived health.
Measured: Throughout study completion, up to 62 months
Secondary outcomes (8)
Work Limitations Questionnaire (WLQ)
The WLQ measures the impact of CMT on participants' work ability and productivity. The measure comprises 25 items that ask respondents to rate their level of difficulty or ability to perform specific job demands, aggregated into four domains: time management, physical demands, mental-interpersonal demands, and output demands. Domain scores range from 0 to 100 and the recall period is the previous 2 weeks. WLQ domain scores can be converted into an estimate of productivity loss using an algorithm.
Measured: Throughout study completion, up to 62 months
Brief Fatigue Inventory (BFI)
The BFI assesses the fatigue severity in patients with CMT. The measure comprises 10 items, although the first item asks about usual fatigue over the past week (with the respondent answering "yes" or "no") and is not included in the overall score. The remaining nine items assess general fatigue within two subscales: fatigue severity (three items), and the impact of fatigue on daily functioning (six items). The measure uses a 10-point numeric rating scale, and a recall period of 24 hours. A global fatigue score can be calculated by averaging all nine items.
Measured: Throughout study completion, up to 62 months
Patient-Reported Outcomes Measurement Information System (PROMIS) Pain Intensity 3a and Interference 8a
The PROMIS pain intensity measure includes two items that assess pain intensity over the last 7 days (average and worst pain), and one for pain intensity "right now"; each scores using a 5-point scale. Scores range from 2-10. Higher scores represent worst pain. This measure is generic rather than disease-specific.
The PROMIS pain interference measure assesses the extent to which pain hinders engagement with social, cognitive, emotional, physical and recreational activities, sleep, and enjoyment in life over the last 7 days using a 5-point scale. Scores range from 8-40. Higher scores represent worse interference. This measure is generic rather than disease-specific.
Measured: Throughout study completion, up to 62 months
Bespoke questionnaire for this study
Two cramp-specific items were developed for inclusion in the survey, measuring cramp frequency and intensity. The cramp frequency item asks "In the past 7 days, how many days did you experience cramp?" and has five possible responses: had no cramp, 1-2 days, 3-4 days, 5-6 days, every day. The cramp intensity item asks "In the past 7 days, how intense was your cramp at its worst?" and has five possible responses: had no cramp, mild, moderate, severe, very severe. Higher scores on both items represent greater cramp frequency and intensity respectively.
Measured: Throughout study completion, up to 62 months
Patient-Reported Outcomes Measurement Information System (PROMIS) Sleep Disturbance 8a
The PROMIS sleep disturbance measure assesses self-reported perceptions of sleep quality, sleep depth, and restoration associated with sleep over the last 7 days using a 5-point scale. Scores range from 8-40. Higher scores represent worse sleep disturbance. This measure is generic rather than disease-specific.
Measured: Throughout study completion, up to 62 months
Falls Efficacy Scale - International (FES-I)
The FES-I measures the level of concern about falling during social and physical activities inside and outside hte home, whether or not the person actually does the activity. The "usual" level of concern is measured on a four-point Likert scale (1 = not at all concerned to 4 = very concerned), with a time frame of "usual". Scores range from 16-64. Higher scores represent greater concern about falling.
Measured: Throughout study completion, up to 62 months
Lower Extremity Function Scale (LEFS)
The LEFS evaluates difficulties because of lower limb problems in 20 activities, including work/school activities, hobbies, moving around the home, dressing, lifting, standing, sitting, walking, and running. The level of difficulty is assessed for "today" using a 5-point Likert scale (0 = extreme difficulty or unable to perform activity; 5 = No difficulty). Lower scores represent greater difficulties experience because of lower limb problems.
Measured: Throughout study completion, up to 62 months
QuickDash (Disabilities of the Arm, Shoulder and Hand)
The QuickDash measure uses 11 items to measure physical function and symptoms in people with any or multiple musculoskeletal disorders of the upper limb. Six questions relate to the ability to perform six activities over the last week, ranging from opening a jar, carrying, washing and recreational activities; two questions relate to impact on social and daily activities; two questions ask the person to rate the severity of pain and tingling in the arms, shoulder or hand, and one question relates to the effects of pain on sleep. All questions are rated 1-5 (no difficulty/none/not at all through to unable/extreme difficulty). Scores range from 11-55. Higher scores represent greater difficulties with physical function and symptoms.
Measured: Throughout study completion, up to 62 months
Timeline
From the start of the study to its completion; the mark is today. Estimated dates are the sponsor’s plan.
- First posted20 Dec 2018
- Study start9 Oct 2018
- Primary completion31 Dec 2023
- Study completion31 Dec 2023
Resources
- Official record on ClinicalTrials.govThe registry entry this page is built from, with its full change history.
- Publications (2)Papers the sponsor has linked to this study.
- Other trials for Charcot-Marie-Tooth DiseaseStudies of the same condition, open and under way.
- What the trial phases meanHow a treatment moves from first tests in people to approval.
More details
For doctors and researchers: how the study is designed, who runs it, and its registry IDs.
Study design
- Study type
- Observational
- Observational model
- Ecologic or community
- Time perspective
- Prospective
- Enrolment
- 3,321 participants (actual)
- Target duration
- 62 Months
Sponsor and investigators
- Lead sponsor
- Vitaccess Ltd
- Sponsor type
- Industry (pharma, biotech, devices)
- Collaborators
- ACMT-Rete per la malattia di Charcot-Marie-Tooth OdV, Charcot-Marie-Tooth Association, Charcot-Marie-Tooth UK, CMT France, Federación Española de Enfermedades Neuromusculares, Hereditary Neuropathy Foundation, Pharnext S.C.A.
- Responsible party
- Principal investigator, Mark Larkin, Vitaccess Ltd
- Investigators
- Mark JW Larkin, PhD, Principal investigator, Vitaccess Ltd
IDs, dates and publications
- ClinicalTrials.gov ID
- NCT03782883
- Sponsor's protocol ID
- 5101-01-2018
- Status last verified
- Apr 2024
- First submitted
- 17 Dec 2018
- Last updated
- 14 Nov 2024
- Results posted
- No
- Data monitoring committee
- Yes
- FDA-regulated drug
- No
- FDA-regulated device
- No
- Individual data sharing
- No: Aggregated data research dashboards will be available via industry subscriptions or free academic access.
Publications
Thomas FP, Saporta M, Attarian S, Sevilla T, Mascaro RS, Fabrizi GM, Genovese F, Gray A, Bull S, Tanesse D, Rego M, Moore A, Hollett C, Monteiro K, Paoli X, Llewellyn S, Larkin M, Boutalbi Y. Patient-reported impact of Charcot-Marie-Tooth disease: protocol for a real-world digital lifestyle study. Neurodegener Dis Manag. 2021 Feb;11(1):21-33. doi: 10.2217/nmt-2020-0044. Epub 2020 Oct 22.
PubMed 33086913Thomas FP, Saporta MA, Attarian S, Sevilla T, Sivera R, Fabrizi GM, Genovese F, Gray AJ, Bull S, Tanesse D, Rego M, Moore A, Hollett C, Paoli X, Senechal T, Day L, Ouyang C, Llewellyn S, Larkin M, Boutalbi Y. Patient-Reported Symptom Burden of Charcot-Marie-Tooth Disease Type 1A: Findings From an Observational Digital Lifestyle Study. J Clin Neuromuscul Dis. 2022 Sep 1;24(1):7-17. doi: 10.1097/CND.0000000000000426. Epub 2022 Aug 19.
PubMed 36005469
Study at a glance
From the registry record, last updated by the sponsor on 14 Nov 2024. Recruitment status is as the sponsor reported it: confirm with the study team.
- Official title
- The Impact of Charcot-Marie-Tooth Disease in the Real World
- ClinicalTrials.gov ID
- NCT03782883
- Acronym / sponsor ID
- 5101-01-2018
- Conditions
- Charcot-Marie-Tooth Disease
- Study type
- Observational
- Recruitment status
- Completed
- Enrolment
- 3,321 participants (actual)
- Lead sponsor
- Vitaccess LtdIndustry (pharma, biotech, devices)
- Collaborators
- ACMT-Rete per la malattia di Charcot-Marie-Tooth OdV, Charcot-Marie-Tooth Association, Charcot-Marie-Tooth UK, CMT France, Federación Española de Enfermedades Neuromusculares, Hereditary Neuropathy Foundation, Pharnext S.C.A.
- Study start
- 9 Oct 2018 (actual)
- Primary completion
- 31 Dec 2023 (actual)
- Study completion
- 31 Dec 2023 (actual)
- First posted
- 20 Dec 2018 (actual)
- Results
- Not posted in the source record
Clinical summary
The research question as the sponsor registered it. This describes what the study plans to test, not what it found.
An observational, non-interventional registry study to collect real-world data from people living with Charcot-Marie-Tooth disease (CMT) and its treatment, which will be available to researchers to further the knowledge of Charcot-Marie-Tooth disease and improve patient care.
Detailed description
The registry uses Vitaccess' digital real-world evidence platform and has been developed in collaboration with CMT experts, Patient Advocacy Organizations (PAOs) and the biopharmaceutical company Pharnext.
Eligible participants install a study app on their smartphone. Researchers access aggregated, anonymised data via a cloud-based research portal.
The platform provides benefits to participants, which it is hoped will encourage persistence with data submission; these include options to download a copy of their symptom diary, a clinically-validated Knowledge feature, and access to high-level results from the study data.
The aggregated data are available in close to real time via "dashboards" and can be analysed according to a number of pre-set criteria (e.g. disease stage, age, geographic location).
State-of-the-art technologies and security policies are used in the platform to ensure industry-standard data storage and privacy for all users. Participants' personally identifiable information will remain confidential at all times, and researchers will not be able to identify individuals.
The study is ethics-approved in all scope countries and led by a Scientific Advisory Board comprising representatives from international PAOs, patients and key opinion leaders from each country, and study leads from both Pharnext Société Anonyme (SA) and Vitaccess Ltd.
Study design
- Observational model
- Ecologic or community
- Time perspective
- Prospective
- Target follow-up
- 62 Months
Who may qualify
- Ages
- 18 years and older
- Sex
- All
- Healthy volunteers
- Not accepted
- Age groups
- Adult, Older adult
- Study population
- Adults (age >18 years) with any stage and any subtype of Charcot-Marie-Tooth disease, who are resident in France, Germany, Italy, Spain, the United Kingdom (UK) or the USA, so long as they have a National Health Service (NHS) or Community Health Index (CHI) number (or equivalent) and are willing to use their own smartphone / tablet.
Key inclusion criteria (1 listed)
- () Aged 18 years and over () Diagnosed with any stage and any subtype of Charcot-Marie-Tooth disease () Resident in France, Germany, Italy, Spain, the UK, or the USA () Have a National Health Service (NHS) or Community Health Index (CHI) number, or equivalent () Willing to use their own smartphone
Key exclusion criteria (1 listed)
- () No specific exclusion criteria.
Eligibility is decided by the study team after screening; this overview does not establish that a patient qualifies. Full inclusion and exclusion criteria.
Primary outcome
EuroQoL 5-dimension 5-level (EQ-5D-5L)
Time frame: Throughout study completion, up to 62 months
The EQ-5D-5L comprises two parts - the EQ-5D-5L descriptive system and the EQ Visual Analogue Scale (EQ VAS). The descriptive system comprises 5 dimensions (mobility, self care, usual activities, pain/discomfort, and anxiety/depression), each with 5 levels (no problems, slight problems, moderate problems, severe problems, and extreme problems - i.e., higher scores represent worse health). The digits for 5 dimensions can be combined in a 5-digit number describing the respondent's health state. The EQ VAS records the respondent's self-rated health on vertical, visual analogue scale with endpoints labelled 'the best health you can imagine' and 'the worst health you can imagine'. Higher scores represent better self-perceived health.
8 secondary outcomes
Work Limitations Questionnaire (WLQ)
Time frame: Throughout study completion, up to 62 months
Brief Fatigue Inventory (BFI)
Time frame: Throughout study completion, up to 62 months
Patient-Reported Outcomes Measurement Information System (PROMIS) Pain Intensity 3a and Interference 8a
Time frame: Throughout study completion, up to 62 months
Bespoke questionnaire for this study
Time frame: Throughout study completion, up to 62 months
Patient-Reported Outcomes Measurement Information System (PROMIS) Sleep Disturbance 8a
Time frame: Throughout study completion, up to 62 months
Falls Efficacy Scale - International (FES-I)
Time frame: Throughout study completion, up to 62 months
Lower Extremity Function Scale (LEFS)
Time frame: Throughout study completion, up to 62 months
QuickDash (Disabilities of the Arm, Shoulder and Hand)
Time frame: Throughout study completion, up to 62 months
Results and safety
The study is completed, but results are not available in the source record. That does not mean the treatment did or did not work.
Research links
- Official record on ClinicalTrials.govLast updated by the sponsor 14 Nov 2024.
- Background: PubMed 33086913Thomas FP, Saporta M, Attarian S, Sevilla T, Mascaro RS, Fabrizi GM, Genovese F, Gray A, Bull S, Tanesse D, Rego M, Moore A, Hollett C, Monteiro K, Paoli X, Llewellyn S, Larkin M, Boutalbi Y. Patient-reported impact of Charcot-Marie-Tooth disease: protocol for a real-world digital lifestyle study. Neurodegener Dis Manag. 2021 Feb;11(1):21-33. doi: 10.2217/nmt-2020-0044. Epub 2020 Oct 22.
- Results publication: PubMed 36005469Thomas FP, Saporta MA, Attarian S, Sevilla T, Sivera R, Fabrizi GM, Genovese F, Gray AJ, Bull S, Tanesse D, Rego M, Moore A, Hollett C, Paoli X, Senechal T, Day L, Ouyang C, Llewellyn S, Larkin M, Boutalbi Y. Patient-Reported Symptom Burden of Charcot-Marie-Tooth Disease Type 1A: Findings From an Observational Digital Lifestyle Study. J Clin Neuromuscul Dis. 2022 Sep 1;24(1):7-17. doi: 10.1097/CND.0000000000000426. Epub 2022 Aug 19.
- Registration page to enrol in the studyLink provided by the sponsor.
- Digital platform used to support the studyLink provided by the sponsor.
- Individual participant dataSharing planned: No. Aggregated data research dashboards will be available via industry subscriptions or free academic access.
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Referral and contact
AzTrials does not enrol participants or take referrals. Contact the study team or a site listed below; they decide who can take part. This study is not recruiting at the moment.
Mark JW Larkin, PhD principal investigator
Vitaccess Ltd
1 site in 1 country: see every site and its status.
Questions about this trial
What is this clinical trial studying?
An observational study of Charcot-Marie-Tooth Disease, run by Vitaccess Ltd at 1 site in United Kingdom.
The full registered description is in the About this study section and the official record.
Who may be eligible to take part?
The sponsor lists 1 inclusion and 1 exclusion criteria. Eligibility is decided by the study team after screening, so read the full criteria and contact a listed site.
Is this clinical trial currently recruiting?
The record says: finished, as of the sponsor's last update on 14 Nov 2024. Status can change between updates, so confirm with the study contact before making plans.
Where is the study taking place?
The record lists 1 site in United Kingdom. Whether a site is taking part right now should be confirmed with that site. See every site.
What are the possible benefits and risks?
They depend on the study and the treatment, and no study can promise a benefit. The study team explains the known risks during informed consent, before anyone joins; the record itself does not list them.
Are results available for this study?
The study is completed, but results are not available in the source record. That does not mean the treatment did or did not work.
How can a doctor refer a patient or contact the research team?
The record lists no contact. The official record names the sponsor, who can be asked directly. AzTrials does not enrol participants or take referrals.
What does the primary outcome measure?
EuroQoL 5-dimension 5-level (EQ-5D-5L) (time frame: Throughout study completion, up to 62 months). It is what the study was designed to measure, not a finding.
Where can I read the original study information?
On the ClinicalTrials.gov record for NCT03782883, which also lists 2 linked publications.
Does a listing mean the treatment is approved or proven to work?
No. A listing means the study is registered. It does not show that the treatment is safe, works, or is approved by a regulator.
How often is this information updated?
AzTrials copies changed records from ClinicalTrials.gov every night and reloads the whole registry every week. The sponsor last updated this record on 14 Nov 2024.