LAMA2-MD (Merosin Deficient Congenital Muscular Dystrophy, MDC1A)NCT07125040
Characterization of the Natural History of LAMA2-RD and Identification of Novel Disease Biomarkers
An observational study of LAMA2-MD (Merosin Deficient Congenital Muscular Dystrophy, MDC1A) and LAMA2-MD \(Merosin Deficient Congenital Muscular Dystrophy, MDC1A\), run by Università Vita-Salute San Raffaele at 1 site in Italy.
Trial summary
The goal of this observational study is to learn about the natural history and multi-organ involvement of Laminin-Alpha-2-Related Dystrophy (LAMA2-RD) in pediatric and adult patients. Read more about this study
- Who is it for?
- Any age
- Conditions the study is for
- LAMA2-MD (Merosin Deficient Congenital Muscular Dystrophy, MDC1A), LAMA2-MD \(Merosin Deficient Congenital Muscular Dystrophy, MDC1A\), Merosin Deficient CMD (Full or Partial)
- What is being tested?
- Cardiac MRI
- How many people
- 45 participants
- Where
- 1 siteItaly
- Study dates
- 31 Jul 2025to May 2028
- Phase
- Observational
- Are there results?
- Not yet: the study is under way
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 Any age
- All sexes
- No healthy volunteers
- Child, Adult, Older adult
Study population: Adult and pediatric patients affected by LAMA2-RD
Who may be able to join
- Diagnosis of LAMA2-related dystrophy confirmed via:
- Two causative mutations in the LAMA2 gene or Muscle biopsy with absence of
- merosin (laminin-211) and at least one causative mutation in the LAMA2 gene or
- Consistent phenotype and affected siblings with criteria a) or b) and
- Ability to participate in study visits at least every 12 months during a 24 months period.
- Ability to sign informed consent for adults or parents/ legal tutors for children
Who cannot join
- Lack of a confirmed diagnosis of LAMA2-relate dystrophy
- Inability to participate in study visits at least every 12 months
- Medical fragility which precludes the ability to safely travel to the study site and/or participate in the study assessments
Trial locations and contacts
1 site in 1 country. Contact the study team or a site near you to ask about taking part.
Italy1 site, 1 recruiting
- Recruiting
Irccs Ospedale San Raffaele
Milan, 20132
Alberto A Zambon, MD, PhD, +390226435080, neuromuscolare@hsr.it
About this study
Official title: Characterization of the Natural History of Laminin-Alpha-2-Related Dystrophy (LAMA2-RD) Patients and Identification of Novel Disease Biomarkers
The goal of this observational study is to learn about the natural history and multi-organ involvement of Laminin-Alpha-2-Related Dystrophy (LAMA2-RD) in pediatric and adult patients. The main questions it aims to answer are:
- What is the prevalence and nature of cardiac involvement, and how do this relate to age and muscular phenotype?
- What is the prevalence of peripheral neuropathy, and how do this relate to age and muscular phenotype?
- What is the extent of respiratory, nutritional, skeletal, and cognitive/brain involvement, particularly in adults with more severe vs less severe phenotypes?
- How does quality of life and transition to adulthood occur in individuals with LAMA2-RD?
- Which nomenclature best reflects differences in disease severity and may support future clinical trial design?
Read more
Study participants will:
- Undergo retrospective and prospective clinical assessments every 12 months for 2 years across multiple centers.
- A subset of adult participants (n=20) will receive cardiac MRI with contrast enhancement.
- Provide biological samples during routine blood testing for future research.
Read the detailed description
Background: LAMA2-RD is an autosomal recessive disorder due mutations in the LAMA2 gene. The clinical manifestations of LAMA2-RD range from severe, early-onset congenital muscular dystrophy (CMD) to a milder limb-girdle type muscular dystrophy (LGMDR23). A few promising therapies are getting closer to clinical application, but clinical trial readiness is limited by the paucity of natural history studies. Although some groups have recently shed light on different aspects of the disease, these are usually focused on pediatric populations. A detailed description of the disease in adult patients as well as the importance of specific organs involvement (e.g., heart and peripheral nervous system) are lacking.
Objectives: To describe the natural history of a large cohort of patients affected by LAMA2-RD (n=40-45).
Specifically, the investigators aim 1) to clarify the prevalence and characteristics of cardiac involvement, and to correlate the latter with age and muscular phenotype 2) to clarify the degree of neuropathic involvement 3) to clarify respiratory, nutritional, skeletal, and brain/cognitive involvement, with a focus on adult population 4) to clarify which nomenclature better captures differences in terms of disease severity, to help refine inclusion criteria for trials 5) to understand how quality of life is impacted and transition to adulthood performed 6) to collect biological material for future research Design and methods: This will be a multicenter, retrospective and prospective longitudinal observational study with additional procedures for a subset of patients: cardiac MRI with contrast enhancement and an additional sample handling during routine blood test. Patients will be assessed every 12 months over a period of 2 years. In addition to routine clinical assessments, cardiac MRI will be performed in a selected 20 adult population. The differential involvement of specific organs between LAMA2-RD subpopulations will be analyzed.
- Conditions
- LAMA2-MD (Merosin Deficient Congenital Muscular Dystrophy, MDC1A), LAMA2-MD \(Merosin Deficient Congenital Muscular Dystrophy, MDC1A\), Merosin Deficient CMD (Full or Partial), Merosin Deficient Congenital Muscular Dystrophy
- Being tested
- Cardiac MRI
- Keywords
- LAMA2-RD, Natural history
- Medical subject headings
- Muscular dystrophy congenital, merosin negative
What participants receive
What participants receive. In a randomised study each person is assigned to one group by chance.
Cardiac MRIOther
On a subset of adult patients
What is being measured
The primary outcome is the main question the study is designed to answer.
Rhythm abnormalities
Prsence or absence of rhythm abnormalities (Brady arrhythmia, supraventricular and ventricular tachyarrhythmia) as detected by ECG and 24-hour Holter ECG
Measured: 0, 12, 24 months
Cardiac function
Left ventricular end-diastolic volume (LVEDV) and left ventricular ejection fraction (LVEF) measured by transthoracic echocardiogram
Measured: 0, 12, 24 months
Cardiac inflammation and fibrosis
Cardiac inflammation and fibrosis by cardiac Magnetic Resonance Imaging (MRI) (only in adult patients)
Measured: T0
Secondary outcomes (12)
Motor outcome 1
The Children's Hospital of Philadelphia Infant Test of Neuromuscular Disorders (CHOP INTEND) (0-2 years); maximum score 64
Measured: 0, 12, 24 months
Motor outcome 2
Motor Function Measure (MFM20 2-5 years; maximum score 60; MFM32 ≥ 5 years; maximum score 96)
Measured: 0,12, 24 months
Motor outcome 3 (Upper limbs)
Upper limbs function: Performance upper limb module 2.0. (PUL 2.0); maximum score 42
Measured: 0, 12, 24 months
Motor outcome 4 (Timed tests)
6 minutes walk test (6MWT) - metres
Measured: 0, 12, 24 months
Motor outcome 4 (Timed tests)
Timed rise from the floor (TRF) - seconds
Measured: 0, 12, 24 months
Motor outcome 5
North Star Assessment for Limb-Girdle Type Muscular Dystrophies (NSAD); maximum score 54
Measured: 0, 12, 24 months
Respiratory function
Lung function measurement (Forced vital capacity -FVC L/%predicted
Measured: 0, 12, 24 months
Respiratory function
Forced Expiratory Volume in 1 second -- FEV1 L/%predicted
Measured: 0, 12, 24 months
Respiratory function
Maximal Inspiratory Pressure- MIP
Measured: 0, 12, 24 months
Respiratory function
Maximal Expiratory Pressure - MEP
Measured: 0, 12, 24 months
Respiratory function
Peak exploratory flow - PEF
Measured: 0, 12, 24 months
Respiratory function
Peak cough flow - PCF
Measured: 0, 12, 24 months
Timeline
From the start of the study to its completion; the mark is today. Estimated dates are the sponsor’s plan.
- First posted15 Aug 2025
- Study start31 Jul 2025
- Primary completionMay 2027 (estimated)
- Study completionMay 2028 (estimated)
Resources
- Official record on ClinicalTrials.govThe registry entry this page is built from, with its full change history.
- Other trials for LAMA2-MD (Merosin Deficient Congenital Muscular Dystrophy, MDC1A)Studies 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
- Cohort
- Time perspective
- Other
- Enrolment
- 45 participants (estimated)
Sponsor and investigators
- Lead sponsor
- Università Vita-Salute San Raffaele
- Sponsor type
- University, hospital or other
- Responsible party
- Principal investigator, Alberto Andrea Zambon, Università Vita-Salute San Raffaele
IDs, dates and publications
- ClinicalTrials.gov ID
- NCT07125040
- Sponsor's protocol ID
- GUP24002_LAMA2
- Status last verified
- Aug 2025
- First submitted
- 17 Jul 2025
- Last updated
- 15 Aug 2025
- Results posted
- No
- Data monitoring committee
- No
- FDA-regulated drug
- No
- FDA-regulated device
- No
Study at a glance
From the registry record, last updated by the sponsor on 15 Aug 2025. Recruitment status is as the sponsor reported it: confirm with the study team.
- Official title
- Characterization of the Natural History of Laminin-Alpha-2-Related Dystrophy (LAMA2-RD) Patients and Identification of Novel Disease Biomarkers
- ClinicalTrials.gov ID
- NCT07125040
- Acronym / sponsor ID
- GUP24002_LAMA2
- Conditions
- LAMA2-MD (Merosin Deficient Congenital Muscular Dystrophy, MDC1A), LAMA2-MD \(Merosin Deficient Congenital Muscular Dystrophy, MDC1A\), Merosin Deficient CMD (Full or Partial), Merosin Deficient Congenital Muscular Dystrophy
- Keywords
- LAMA2-RD, Natural history
- Study type
- Observational
- Recruitment status
- Recruiting
- Enrolment
- 45 participants (estimated)
- Lead sponsor
- Università Vita-Salute San RaffaeleUniversity, hospital or other
- Study start
- 31 Jul 2025 (actual)
- Primary completion
- May 2027 (estimated)
- Study completion
- May 2028 (estimated)
- First posted
- 15 Aug 2025 (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.
The goal of this observational study is to learn about the natural history and multi-organ involvement of Laminin-Alpha-2-Related Dystrophy (LAMA2-RD) in pediatric and adult patients. The main questions it aims to answer are:
- What is the prevalence and nature of cardiac involvement, and how do this relate to age and muscular phenotype?
- What is the prevalence of peripheral neuropathy, and how do this relate to age and muscular phenotype?
- What is the extent of respiratory, nutritional, skeletal, and cognitive/brain involvement, particularly in adults with more severe vs less severe phenotypes?
- How does quality of life and transition to adulthood occur in individuals with LAMA2-RD?
- Which nomenclature best reflects differences in disease severity and may support future clinical trial design?
Study participants will:
- Undergo retrospective and prospective clinical assessments every 12 months for 2 years across multiple centers.
- A subset of adult participants (n=20) will receive cardiac MRI with contrast enhancement.
- Provide biological samples during routine blood testing for future research.
Detailed description
Background: LAMA2-RD is an autosomal recessive disorder due mutations in the LAMA2 gene. The clinical manifestations of LAMA2-RD range from severe, early-onset congenital muscular dystrophy (CMD) to a milder limb-girdle type muscular dystrophy (LGMDR23). A few promising therapies are getting closer to clinical application, but clinical trial readiness is limited by the paucity of natural history studies. Although some groups have recently shed light on different aspects of the disease, these are usually focused on pediatric populations. A detailed description of the disease in adult patients as well as the importance of specific organs involvement (e.g., heart and peripheral nervous system) are lacking.
Objectives: To describe the natural history of a large cohort of patients affected by LAMA2-RD (n=40-45).
Specifically, the investigators aim 1) to clarify the prevalence and characteristics of cardiac involvement, and to correlate the latter with age and muscular phenotype 2) to clarify the degree of neuropathic involvement 3) to clarify respiratory, nutritional, skeletal, and brain/cognitive involvement, with a focus on adult population 4) to clarify which nomenclature better captures differences in terms of disease severity, to help refine inclusion criteria for trials 5) to understand how quality of life is impacted and transition to adulthood performed 6) to collect biological material for future research Design and methods: This will be a multicenter, retrospective and prospective longitudinal observational study with additional procedures for a subset of patients: cardiac MRI with contrast enhancement and an additional sample handling during routine blood test. Patients will be assessed every 12 months over a period of 2 years. In addition to routine clinical assessments, cardiac MRI will be performed in a selected 20 adult population. The differential involvement of specific organs between LAMA2-RD subpopulations will be analyzed.
Study design
- Observational model
- Cohort
- Time perspective
- Other
Who may qualify
- Sex
- All
- Healthy volunteers
- Not accepted
- Age groups
- Child, Adult, Older adult
- Study population
- Adult and pediatric patients affected by LAMA2-RD
Key inclusion criteria (6 listed)
- Two causative mutations in the LAMA2 gene or Muscle biopsy with absence of
- merosin (laminin-211) and at least one causative mutation in the LAMA2 gene or
Key exclusion criteria (3 listed)
- Lack of a confirmed diagnosis of LAMA2-relate dystrophy
- Inability to participate in study visits at least every 12 months
- Medical fragility which precludes the ability to safely travel to the study site and/or participate in the study assessments
Eligibility is decided by the study team after screening; this overview does not establish that a patient qualifies. Full inclusion and exclusion criteria.
Interventions
| Intervention | Type | Arms |
|---|---|---|
| Cardiac MRIOn a subset of adult patients | Other | – |
Primary outcome
Rhythm abnormalities
Time frame: 0, 12, 24 months
Prsence or absence of rhythm abnormalities (Brady arrhythmia, supraventricular and ventricular tachyarrhythmia) as detected by ECG and 24-hour Holter ECG
Cardiac function
Time frame: 0, 12, 24 months
Left ventricular end-diastolic volume (LVEDV) and left ventricular ejection fraction (LVEF) measured by transthoracic echocardiogram
Cardiac inflammation and fibrosis
Time frame: T0
Cardiac inflammation and fibrosis by cardiac Magnetic Resonance Imaging (MRI) (only in adult patients)
12 secondary outcomes
Motor outcome 1
Time frame: 0, 12, 24 months
Motor outcome 2
Time frame: 0,12, 24 months
Motor outcome 3 (Upper limbs)
Time frame: 0, 12, 24 months
Motor outcome 4 (Timed tests)
Time frame: 0, 12, 24 months
Motor outcome 4 (Timed tests)
Time frame: 0, 12, 24 months
Motor outcome 5
Time frame: 0, 12, 24 months
Respiratory function
Time frame: 0, 12, 24 months
Respiratory function
Time frame: 0, 12, 24 months
Respiratory function
Time frame: 0, 12, 24 months
Respiratory function
Time frame: 0, 12, 24 months
Respiratory function
Time frame: 0, 12, 24 months
Respiratory function
Time frame: 0, 12, 24 months
Results and safety
This study is still under way. Results are not available in the source record yet.
Research links
- Official record on ClinicalTrials.govLast updated by the sponsor 15 Aug 2025.
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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.
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 LAMA2-MD (Merosin Deficient Congenital Muscular Dystrophy, MDC1A) and LAMA2-MD \(Merosin Deficient Congenital Muscular Dystrophy, MDC1A\), run by Università Vita-Salute San Raffaele at 1 site in Italy.
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 6 inclusion and 3 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: looking for participants now, as of the sponsor's last update on 15 Aug 2025. 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 Italy. 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?
This study is still under way. Results are not available in the source record yet.
How can a doctor refer a patient or contact the research team?
Contact Alberto A Zambon, MD, PhD (+390226435080, neuromuscolare@hsr.it), or a site near the patient. AzTrials does not enrol participants or take referrals.
What does the primary outcome measure?
Rhythm abnormalities (time frame: 0, 12, 24 months). The study lists 3 primary outcomes in all. It is what the study was designed to measure, not a finding.
Where can I read the original study information?
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 15 Aug 2025.