A Multicentric European Study to Promote Clinical Trial Readiness for STXBP1-related Disorders

Trial statusRecruiting
Trial phaseNot listed
Trial typeObservational
Biological sexAll
AgeNot listed
SponsorEuropean STXBP1 Consortium

About this trial

STXBP1-related disorders (STXBP1-RD) are rare genetic neurodevelopmental disorders, caused by pathogenic variants in the gene STXBP1. The core clinical features of the disorder are developmental delay often leading to (severe) intellectual disability and seizures in most patients, although the phenotypic spectrum is variable. Behavioral problems and movement disorders are frequent comorbidities. STXBP1-RD are severe disorders with significant impact on the quality of life of the patients and their caregivers. At the moment, there is no cure for STXBP1-RD and treatment is largely limited to symptom control. Recent advances in the field of precision medicine and gene therapy have led to the identification of potential novel disease modifying therapies for STXBP1-RD that hold promise to reach clinical trials in the coming years. However, accurate and successful evaluation of such novel precision therapies in STXBP1-RD patients is challenging, given the rarity of the condition and the variable clinical spectrum. Furthermore, relevant clinical endpoints, taking into account the patients' and caregivers' perspective have not been identified to date.

In this European collaborative study, the investigators will prospectively follow patients with STXBP1-RD during different phases of life (infantile period, childhood and adolescence/adulthood). The study aims to better understand the natural history and the phenotypic spectrum of the disease including the identification of disease modifiers. It further aims to identify relevant clinical endpoints (what to treat?) and robust outcome measures and biomarkers (how to measure?) for future clinical trials. The study is performed in close collaboration with different STXBP1 patient-caregiver communities across Europe.

Eligibility criteria

This trial does not accept healthy volunteers

Qualifiers

participant has a (likely) pathogenic, disease-causing STXBP1 variant, according to the American College of Medical Genetics and Genomics (ACMG) criteria; or participant has a larger structural variant including the STXBP1 gene where STXBP1 is thought to be (one of) the culprit gene(s) causing the phenotype •written informed consent from study participant and/or legal guardian.

Disqualifiers

Exclusion criteria for the study are: none if the inclusion criteria are met.

Trial population

Patients with STXBP1 living in one of the 8 ESCO member countries( Belgium, Italy, Israel, Denmark, France, Spain, Netherlands, Germany) and has a (likely) pathogenic, disease-causing STXBP1 variant, according to t.

Trial design

Design model

Case-only

Time perspective

Other

Treatments tested in this trial

Not listed

Trial groups

120 Participants
are grouped into 1 trial group
Group A: Natural history study participants

Trial outcomes

Primary outcomes

1

Changes in clinical assessment percentiles over time.

The primary analysis will include all participants who meet the inclusion and exclusion criteria and complete the first visit. For each participant, the percentage of correct responses on clinical assessments will be recorded. Variations in percentile scores over the study period will be examined using a linear mixed-effects model, allowing for the repeated observations collected from each participant.

Time frame
12 months

Secondary outcomes

Other outcomes

Sponsors and contacts

Click on the lead sponsor to view all of their trials.

European STXBP1 Consortium

Lead sponsor

Filadelfia Epilepsy Hospital

Collaborator

University Hospital Heidelberg

Collaborator

University Hospital, Antwerp

Collaborator

Istituto Giannina Gaslini, Genoa, Italy

Collaborator

Hospital Sant Joan de Deu

Collaborator

Amsterdam UMC, location VUmc

Collaborator

Sheba Medical Center

Collaborator

Aix Marseille Université

Collaborator

Hospital Ruber Internacional

Collaborator