NCT03222947

Brief Summary

Taybi-Linder syndrome (TALS, OMIM 210710) is a rare autosomal recessive disorder belonging to the group of microcephalic osteodysplastic primordial dwarfisms (MOPD). This syndrome is characterized by short stature, skeletal anomalies, severe microcephaly with brain malformations and facial dysmorphism, and is caused by mutations in RNU4ATAC. Although RNU4ATAC-associated TALS is a recognizable phenotype, an atypical presentation is sometimes observed, thus expanding the clinical spectrum (TALS-like phenotype). This study aims to identify new variants involved in Taybi-Linder syndrome and associated phenotypes (i.e.TALS-like). This non interventional study will be performed on patients with no proven mutation of RNU4ATAC and their blood relatives (19 samples total) by high throughput sequencing and genetic analysis of already collected deoxyribonucleic acid samples. Altogether, such a study will allow a better understanding of the molecular mechanisms responsible for the Taybi-Linder syndrome and Taybi-Linder syndrome-like phenotypes as well as the pathophysiology of these devastating forms of microcephalic dwarfism.

Trial Health

43
At Risk

Trial Health Score

Automated assessment based on enrollment pace, timeline, and geographic reach

Trial has exceeded expected completion date
Enrollment
19

participants targeted

Target at below P25 for all trials

Timeline
Completed

Started Sep 2017

Shorter than P25 for all trials

Geographic Reach
1 country

1 active site

Status
unknown

Health score is calculated from publicly available data and should be used for screening purposes only.

Trial Relationships

Click on a node to explore related trials.

Study Timeline

Key milestones and dates

First Submitted

Initial submission to the registry

July 18, 2017

Completed
1 day until next milestone

First Posted

Study publicly available on registry

July 19, 2017

Completed
1 month until next milestone

Study Start

First participant enrolled

September 1, 2017

Completed
8 months until next milestone

Primary Completion

Last participant's last visit for primary outcome

May 1, 2018

Completed
1 month until next milestone

Study Completion

Last participant's last visit for all outcomes

June 1, 2018

Completed
Last Updated

July 19, 2017

Status Verified

July 1, 2017

Enrollment Period

8 months

First QC Date

July 18, 2017

Last Update Submit

July 18, 2017

Conditions

Outcome Measures

Primary Outcomes (1)

  • Identification of new variants involved in the Taybi-Linder syndrome

    A genetic high throughput exome capture sequencing of 19 deoxyribonucleic acid samples from patients diagnosed with a Taybi-Linder like syndrome and their blood relatives

    Collection at time of diagnosis = less than one day

Study Arms (2)

Taybi-Linder index cases

Taybi-Linder index cases who have already consented for the (re)use of their DNA samples for medical research.

Genetic: Deoxyribonucleic acid analysis

Blood relatives of Taybi-Linder index cases

Blood relatives of Taybi-Linder index cases who have already consented for the (re)use of their DNA samples for medical research.

Genetic: Deoxyribonucleic acid analysis

Interventions

This study consists in the high throughput exome sequencing and subsequent genetic bio-analysis of 19 deoxyribonucleic acid samples from 6 families, already collected and consented, including patients diagnosed with a Taybi-Linder syndrome and their relatives (parents and/or siblings).

Blood relatives of Taybi-Linder index casesTaybi-Linder index cases

Eligibility Criteria

Sexall
Healthy VolunteersNo
Age GroupsChild (0-17), Adult (18-64), Older Adult (65+)
Sampling MethodNon-Probability Sample
Study Population

Patients diagnosed with a Taybi-Linder or Taybi-Linder like syndrome and their blood relatives.

You may qualify if:

  • foetus or young children diagnosed with a Taybi-Linder or Taybi-Linder like syndrome, with no RNU4ATAC mutation (index case)
  • aged 20 weeks pregnant to 18 years old
  • parents or sibling of the index cases, with informed consent for the analysis of both their DNA sample and the one of the index case.

You may not qualify if:

  • no informed consent for the use of genetic samples for medical research

Contact the study team to confirm eligibility.

Sponsors & Collaborators

Study Sites (1)

Service de Génétique Clinique, Groupement Hospitalier Est, Hospices Civils de Lyon

Lyon, 69677, France

Location

Biospecimen

Retention: SAMPLES WITH DNA

Deoxyribonucleic acid of patients obtained either from blood samples (lymphocytes) or foetal tissue sampling

MeSH Terms

Conditions

Microcephalic osteodysplastic primordial dwarfism, type 1Genetic Diseases, Inborn

Condition Hierarchy (Ancestors)

Congenital, Hereditary, and Neonatal Diseases and Abnormalities

Central Study Contacts

Audrey PUTOUX, MCU-PH

CONTACT

Charles EDERY, PU-PH

CONTACT

Study Design

Study Type
observational
Observational Model
COHORT
Time Perspective
RETROSPECTIVE
Sponsor Type
OTHER
Responsible Party
SPONSOR

Study Record Dates

First Submitted

July 18, 2017

First Posted

July 19, 2017

Study Start

September 1, 2017

Primary Completion

May 1, 2018

Study Completion

June 1, 2018

Last Updated

July 19, 2017

Record last verified: 2017-07

Data Sharing

IPD Sharing
Will not share

Locations