RecruitmentNCT05631418
Chinese Regional Spinal Muscular Atrophy Patient Registration Study
An observational study of Recruitment, run by The Children's Hospital of Zhejiang University School of Medicine at 1 site in China.
Trial summary
The primary objectives of this study are to obtain clinically meaningful data on the survival, outcomes, prognosis and treatment effect of all the patients with spinal muscular atrophy (SMA) 5q types 1 to 3 (according to international classification), being followed in the Children's Hospital, Zhejiang University… Read more about this study
- Who is it for?
- Ages Up to 18 years
- Conditions the study is for
- Recruitment
- How many people
- 0 participants
- Where
- 1 siteChina
- Study dates
- Nov 2022to Dec 2026
- How long will I take part?
- 3 Years
- 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 Up to 18 years
- All sexes
- No healthy volunteers
- Child, Adult
Study population: Genetically confirmed 5qSMA patients through 1 to 3 types younger than 18 years old followed in our hospital since October 2019 with or without disease-modifying treatment.
Who may be able to join
- Genetically confirmed 5qSMA patients through 1 to 3 types younger than 18 years old followed in our hospital since October 2019;
- For prospective study: inform consent form signed by patient;
- Not combined with any other genetic and metabolic diseases;
Who cannot join
- Other type of SMA (not 5q).
- Under guardianship or curatorship.
- Unable to understand or cooperate with the study.
Trial locations and contacts
1 site in 1 country.
China1 site
The Children's Hospital, Zhejiang University School of Medicine
Hangzhou, 310052
About this study
Official title: The Children's Hospital, Zhejiang University School of Medicine, National Clinical Research Center for Child Health
The primary objectives of this study are to obtain clinically meaningful data on the survival, outcomes, prognosis and treatment effect of all the patients with spinal muscular atrophy (SMA) 5q types 1 to 3 (according to international classification), being followed in the Children's Hospital, Zhejiang University School of Medicine since October 2019. The registry will collect retrospectively and prospectively the longitudinal data of the long-term follow-up for children patients, under real life conditions of current medical practice, in order to document the clinical evolution of patients (survival, motor, respiratory, orthopedic and nutritional and so on), the conditions of use of the treatments, the mortality rates of treated and untreated patients, the tolerance of the treatments, adverse events.
Read the detailed description
The detailed objectives of this study included but not be limited to the following aspects:
- to collect and describe overall demographic, familial, clinical, biological, and genetic characteristics of patients with 5qSMA diagnosed and treated in regions of China, by the type of SMA (type 1,2, and 3);
- to study the impact of proactive and symptomatic medical interventions (such as bracing) and medications (disease-modifying treatment, anti-infectious, digestive, nutritional supplements, ect.) in the evolution of patients;
- to study the long-term evolution (survival, motor and respiratory functions, spinal shape, growth and nutritional function) of 5qSMA in treated and untreated populations, by new available therapies;
- to study the incidence and mortality rate of 5qSMA in treated and untreated populations;
- to identify and document the different therapeutic strategies by sub-populations and by discontinuation or follow-up of treatments;
- to evaluate prognostic factors of responses to different drug therapies;
- to study the tolerance of treatments by type of treatments, by type of SMA and overall tolerance (including adverse events);
- to help popularize newborn screening for SMA and pre-symptom treatment among Chinese population;
- to get knowledge of the probable costs of care for 5qSMA patients in different groups (disease types, ages);
- to provide needful elements to evaluate the health care costs for the disease;
- to study the autonomy and the quality of life of patients depending on different therapies and the impact on patients' caregivers;
- to help facilitate development of basic research on SMA in the conduct of trials on new treatment.
- Conditions
- Recruitment
- Keywords
- Spinal Muscular Atrophy, long-term follow-up, quality of life, disease-modifying treatment, clinical efficacy, disease prognosis, newborn screening
- Medical subject headings
- Muscular Atrophy, Spinal
What is being measured
The primary outcome is the main question the study is designed to answer.
World Health Organization (WHO) motor milestone change
Treated and untreated patients with SMA Type 1-3: change of WHO motor milestone from baseline to 3 years to track the patients' motor functional development/status. World Health Organization motor milestone score ranges from 0 to 18. The higher the score is, the better the motor function is.
Measured: baseline, 0.5 year, 1 year, 1.5 year, 2 year, 2.5 year, 3 year
Hammersmith Infant Neurological Examination-2 (HINE-2) scale score change
Treated and untreated patients with SMA Type 1-3: change of HINE-2 scale score from baseline to 3 years to help track the patients' motor functional development/status. The HINE-2 scale score ranges from 0 to 26. The higher the score is, the better the motor function is.
Measured: baseline, 0.5 year, 1 year, 1.5 year, 2 year, 2.5 year, 3 year
Self-reporting/Caregivers' reporting collection
Treated and untreated patients with SMA Type 1-3: motor function change acquired from patients themselves and their caregivers. (Subjective reports were collected through interviews and no evaluation scale was used here.)
Measured: baseline, 0.5 year, 1 year, 1.5 year, 2 year, 2.5 year, 3 year
Motor function evalution-Children's Hospital of Philadelphia Infant Test of Neuromuscular Disorders(CHOP INTEND) scale
For children ≤ 2 years: change of CHOP INTEND scale score. The CHOP INTEND scale score ranges from 0 to 64. The higher the score is, the better the motor function is.
Measured: treated/untreated: baseline, 0.5 year, 1 year, 1.5 year, 2 year, 2.5 year, 3 year
Motor function evalution-Hammersmith Functional Motor Scale-Expanded(HFMSE) scale
For children older than 2 years, ambulatory or not: Change of HFMSE scale score. The scale score ranges from 0 to 66. The higher the score is, the better the motor function is.
Measured: treated/untreated: baseline, 0.5 year, 1 year, 1.5 year, 2 year, 2.5 year, 3 year
Motor function evalution-Revised Upper Limb Module(RULM) scale
For children children older than 6 years, ambulatory or not: Change of RULM (Revised Upper Limb Module) scale score. The scale score ranges from 0 to 37. The higher the score is, the better the motor function is.
Measured: treated/untreated: baseline, 0.5 year, 1 year, 1.5 year, 2 year, 2.5 year, 3 year
Motor function evalution change-6 minutes walking test(6MWT)
For ambulatory individuals: added the change of 6MWT from baseline to 3 years to test exercise endurance. (The walking distance within 6 minutes is taken as the evaluation index, and there's no specific score range.)
Measured: treated/untreated: baseline, 0.5 year, 1 year, 1.5 year, 2 year, 2.5 year, 3 year
Yearly changes of incidence and morbi-mortality-vital events
Events of newly diagnosis, hospitalizations, recurrent infections, fractures, complications and death.
Measured: every year for 3 years
Change from baseline respiratory function
Onset of respiratory support or change in the mode and time (including intubations).
Measured: baseline(before treatment), 1 year, 2 year, 3 year
Change from baseline digestive-nutritional function
Digestive events, onset of nutritional support or change in the mode and time and special intervention.
Measured: baseline(before treatment), 0.5 year, 1 year, 1.5 year, 2 year, 2.5 year, 3 year
Change from baseline spinal scoliosis Cobb angle
Onset of spinal deformity, or increment of over 5° in the Cobb angle (examination in supine position or in the upright position, with or without brace, with or without implant (surgery), and the change of the bone mineral density in lumbar.
Measured: baseline(before treatment), 1 year, 2 year, 3 year
Secondary outcomes (6)
Pulmonary function
Pulmonary Function Test (PFT)will be evaluated at least once per year for children > 5 years, by specifying the posture of realization of the test, lying vs sitting.
Measured: baseline(before treatment), 1 year, 2 year, 3 year
Cardiological function monitoring
Conventional echocardiography will be evaluated to monitor the patients' cardiac function.
Measured: baseline(before treatment), 1 year, 2 year, 3 year
Body composition measurement-Lean body mass
Lean body mass will be measured in patients older than three years by DXA or Inbody device at least once a year.
Measured: baseline(before treatment), 1 year, 2 year, 3 year
Body composition measurement-Fat mass
Fat mass will be measured in patients older than three years by DXA or Inbody device at least once a year.
Measured: baseline(before treatment), 1 year, 2 year, 3 year
Biomarkers
Change of biomarkers: such as Neurofilaments.
Measured: baseline(before treatment), 0.5 year, 1 year, 1.5 year, 2 year, 2.5 year, 3 year
Patients and caregivers' quality of life
Questionary as the "PedsQL Child report/PedsQL parent report concerning child" will be used to evaluate patient's quality of life. The Pediatric Quality of Life Inventory Measurement Models (PedsQLTM) is used for this evalution. The minimum score is 0 and no specific maximum score of this scale. The higher the score is, the higher the quality of life is.
Measured: baseline(before treatment), 1 year, 2 year, 3 year
Timeline
From the start of the study to its completion; the mark is today. Estimated dates are the sponsor’s plan.
- First posted30 Nov 2022
- Study startNov 2022 (estimated)
- Primary completionDec 2025 (estimated)
- Study completionDec 2026 (estimated)
Why it stopped: Due to the lack of application materials, the normal research can not be carried out for the time being.
Resources
- Official record on ClinicalTrials.govThe registry entry this page is built from, with its full change history.
- Other trials for RecruitmentStudies of the same condition, open and under way.
- More trials run by The Children's Hospital of Zhejiang University School of MedicineThe sponsor's other registered studies.
- 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
- 0 participants (actual)
- Target duration
- 3 Years
Sponsor and investigators
- Sponsor type
- University, hospital or other
- Responsible party
- Principal investigator, Shanshan Mao, The Children's Hospital of Zhejiang University School of Medicine
- Investigators
- Shanshan Mao, MD, Principal investigator, The Children's Hospital of Zhejiang University School of Medicine
IDs, dates and publications
- ClinicalTrials.gov ID
- NCT05631418
- Sponsor's protocol ID
- CHZhejiang
- Status last verified
- Feb 2023
- First submitted
- 23 Oct 2022
- Last updated
- 23 Feb 2023
- 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 23 Feb 2023. Recruitment status is as the sponsor reported it: confirm with the study team.
- Official title
- The Children's Hospital, Zhejiang University School of Medicine, National Clinical Research Center for Child Health
- ClinicalTrials.gov ID
- NCT05631418
- Acronym / sponsor ID
- CHZhejiang
- Conditions
- Recruitment
- Keywords
- Spinal Muscular Atrophy, long-term follow-up, quality of life, disease-modifying treatment, clinical efficacy, disease prognosis, newborn screening
- Study type
- Observational
- Recruitment status
- WithdrawnReason stopped: Due to the lack of application materials, the normal research can not be carried out for the time being.
- Enrolment
- 0 participants (actual)
- Lead sponsor
- The Children's Hospital of Zhejiang University School of MedicineUniversity, hospital or other
- Study start
- Nov 2022 (estimated)
- Primary completion
- Dec 2025 (estimated)
- Study completion
- Dec 2026 (estimated)
- First posted
- 30 Nov 2022 (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 primary objectives of this study are to obtain clinically meaningful data on the survival, outcomes, prognosis and treatment effect of all the patients with spinal muscular atrophy (SMA) 5q types 1 to 3 (according to international classification), being followed in the Children's Hospital, Zhejiang University School of Medicine since October 2019. The registry will collect retrospectively and prospectively the longitudinal data of the long-term follow-up for children patients, under real life conditions of current medical practice, in order to document the clinical evolution of patients (survival, motor, respiratory, orthopedic and nutritional and so on), the conditions of use of the treatments, the mortality rates of treated and untreated patients, the tolerance of the treatments, adverse events.
Detailed description
The detailed objectives of this study included but not be limited to the following aspects:
- to collect and describe overall demographic, familial, clinical, biological, and genetic characteristics of patients with 5qSMA diagnosed and treated in regions of China, by the type of SMA (type 1,2, and 3);
- to study the impact of proactive and symptomatic medical interventions (such as bracing) and medications (disease-modifying treatment, anti-infectious, digestive, nutritional supplements, ect.) in the evolution of patients;
- to study the long-term evolution (survival, motor and respiratory functions, spinal shape, growth and nutritional function) of 5qSMA in treated and untreated populations, by new available therapies;
- to study the incidence and mortality rate of 5qSMA in treated and untreated populations;
- to identify and document the different therapeutic strategies by sub-populations and by discontinuation or follow-up of treatments;
- to evaluate prognostic factors of responses to different drug therapies;
- to study the tolerance of treatments by type of treatments, by type of SMA and overall tolerance (including adverse events);
- to help popularize newborn screening for SMA and pre-symptom treatment among Chinese population;
- to get knowledge of the probable costs of care for 5qSMA patients in different groups (disease types, ages);
- to provide needful elements to evaluate the health care costs for the disease;
- to study the autonomy and the quality of life of patients depending on different therapies and the impact on patients' caregivers;
- to help facilitate development of basic research on SMA in the conduct of trials on new treatment.
Study design
- Observational model
- Cohort
- Time perspective
- Other
- Target follow-up
- 3 Years
Who may qualify
- Ages
- Up to 18 years
- Sex
- All
- Healthy volunteers
- Not accepted
- Age groups
- Child, Adult
- Study population
- Genetically confirmed 5qSMA patients through 1 to 3 types younger than 18 years old followed in our hospital since October 2019 with or without disease-modifying treatment.
Key inclusion criteria (3 listed)
- Genetically confirmed 5qSMA patients through 1 to 3 types younger than 18 years old followed in our hospital since October 2019;
- For prospective study: inform consent form signed by patient;
- Not combined with any other genetic and metabolic diseases;
Key exclusion criteria (3 listed)
- Other type of SMA (not 5q).
- Under guardianship or curatorship.
- Unable to understand or cooperate with the study.
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
World Health Organization (WHO) motor milestone change
Time frame: baseline, 0.5 year, 1 year, 1.5 year, 2 year, 2.5 year, 3 year
Treated and untreated patients with SMA Type 1-3: change of WHO motor milestone from baseline to 3 years to track the patients' motor functional development/status. World Health Organization motor milestone score ranges from 0 to 18. The higher the score is, the better the motor function is.
Hammersmith Infant Neurological Examination-2 (HINE-2) scale score change
Time frame: baseline, 0.5 year, 1 year, 1.5 year, 2 year, 2.5 year, 3 year
Treated and untreated patients with SMA Type 1-3: change of HINE-2 scale score from baseline to 3 years to help track the patients' motor functional development/status. The HINE-2 scale score ranges from 0 to 26. The higher the score is, the better the motor function is.
Self-reporting/Caregivers' reporting collection
Time frame: baseline, 0.5 year, 1 year, 1.5 year, 2 year, 2.5 year, 3 year
Treated and untreated patients with SMA Type 1-3: motor function change acquired from patients themselves and their caregivers. (Subjective reports were collected through interviews and no evaluation scale was used here.)
Motor function evalution-Children's Hospital of Philadelphia Infant Test of Neuromuscular Disorders(CHOP INTEND) scale
Time frame: treated/untreated: baseline, 0.5 year, 1 year, 1.5 year, 2 year, 2.5 year, 3 year
For children ≤ 2 years: change of CHOP INTEND scale score. The CHOP INTEND scale score ranges from 0 to 64. The higher the score is, the better the motor function is.
Motor function evalution-Hammersmith Functional Motor Scale-Expanded(HFMSE) scale
Time frame: treated/untreated: baseline, 0.5 year, 1 year, 1.5 year, 2 year, 2.5 year, 3 year
For children older than 2 years, ambulatory or not: Change of HFMSE scale score. The scale score ranges from 0 to 66. The higher the score is, the better the motor function is.
Motor function evalution-Revised Upper Limb Module(RULM) scale
Time frame: treated/untreated: baseline, 0.5 year, 1 year, 1.5 year, 2 year, 2.5 year, 3 year
For children children older than 6 years, ambulatory or not: Change of RULM (Revised Upper Limb Module) scale score. The scale score ranges from 0 to 37. The higher the score is, the better the motor function is.
Motor function evalution change-6 minutes walking test(6MWT)
Time frame: treated/untreated: baseline, 0.5 year, 1 year, 1.5 year, 2 year, 2.5 year, 3 year
For ambulatory individuals: added the change of 6MWT from baseline to 3 years to test exercise endurance. (The walking distance within 6 minutes is taken as the evaluation index, and there's no specific score range.)
Yearly changes of incidence and morbi-mortality-vital events
Time frame: every year for 3 years
Events of newly diagnosis, hospitalizations, recurrent infections, fractures, complications and death.
Change from baseline respiratory function
Time frame: baseline(before treatment), 1 year, 2 year, 3 year
Onset of respiratory support or change in the mode and time (including intubations).
Change from baseline digestive-nutritional function
Time frame: baseline(before treatment), 0.5 year, 1 year, 1.5 year, 2 year, 2.5 year, 3 year
Digestive events, onset of nutritional support or change in the mode and time and special intervention.
Change from baseline spinal scoliosis Cobb angle
Time frame: baseline(before treatment), 1 year, 2 year, 3 year
Onset of spinal deformity, or increment of over 5° in the Cobb angle (examination in supine position or in the upright position, with or without brace, with or without implant (surgery), and the change of the bone mineral density in lumbar.
6 secondary outcomes
Pulmonary function
Time frame: baseline(before treatment), 1 year, 2 year, 3 year
Cardiological function monitoring
Time frame: baseline(before treatment), 1 year, 2 year, 3 year
Body composition measurement-Lean body mass
Time frame: baseline(before treatment), 1 year, 2 year, 3 year
Body composition measurement-Fat mass
Time frame: baseline(before treatment), 1 year, 2 year, 3 year
Biomarkers
Time frame: baseline(before treatment), 0.5 year, 1 year, 1.5 year, 2 year, 2.5 year, 3 year
Patients and caregivers' quality of life
Time frame: baseline(before treatment), 1 year, 2 year, 3 year
Results and safety
The study was withdrawn before enrolling participants, so there are no results.
Research links
- Official record on ClinicalTrials.govLast updated by the sponsor 23 Feb 2023.
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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.
Shanshan Mao, MD principal investigator
The Children's Hospital of Zhejiang University School of Medicine
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 Recruitment, run by The Children's Hospital of Zhejiang University School of Medicine at 1 site in China.
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 3 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: withdrawn, as of the sponsor's last update on 23 Feb 2023. 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 China. 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 was withdrawn before enrolling participants, so there are no results.
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?
World Health Organization (WHO) motor milestone change (time frame: baseline, 0.5 year, 1 year, 1.5 year, 2 year, 2.5 year, 3 year). The study lists 11 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 23 Feb 2023.