NCT03357978

Brief Summary

Ataxia telangiectasia (A-T) is a rare devastating human recessive disorder characterized by progressive cerebellar ataxia, immunodeficiency, chromosomal instability and cancer susceptibility. The immunodeficiency is expressed by recurring infections. It's characterised by decreased lymphocytes data as well as lack of immunglobulin A, immunglobulin G subclasses and specific antibodies against pneumococcus. Aim of the present clinical trial is to investigate frequency-, intensity- and duration of the infections as well as changes oft immune status, dimension of liver disease and tumor risk in patients with A-T, with and without immunoglobulin G substitution therapy. Transient elastography (FibroScan) will be performed in order to measure liver stiffness as an indication of fatty liver and liver fibrosis. A bioelectrical impedance analysis (BIA) is conducted to investigate the exact body composition. Ataxia Score is determined to define neurological problems. Every subject receives a diary to compile symptoms of infection.

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
30

participants targeted

Target at below P25 for not_applicable

Timeline
Completed

Started Oct 2016

Typical duration for not_applicable

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

Study Start

First participant enrolled

October 1, 2016

Completed
7 months until next milestone

First Submitted

Initial submission to the registry

April 24, 2017

Completed
7 months until next milestone

First Posted

Study publicly available on registry

November 30, 2017

Completed
10 months until next milestone

Primary Completion

Last participant's last visit for primary outcome

September 30, 2018

Completed
1 year until next milestone

Study Completion

Last participant's last visit for all outcomes

September 30, 2019

Completed
Last Updated

November 30, 2017

Status Verified

November 1, 2017

Enrollment Period

2 years

First QC Date

April 24, 2017

Last Update Submit

November 29, 2017

Conditions

Keywords

immunodeficiencyliver diseasecancer risk

Outcome Measures

Primary Outcomes (1)

  • Infections in A-T

    Evaluation of frequency, severity and intensity of infections in A-T patients with and without immunoglobulin G substitution

    24 months

Secondary Outcomes (2)

  • Liver disease

    24 months

  • Cancer risk

    24 months

Study Arms (1)

A-T patients

OTHER

A-T patients aged 2 to 45 years with and without immunoglobulin G Substitution * bioelectrical impedance Analysis * blood draw * transient elastography (FibroScan) * ataxia score * Five-Times-Sit-to-Stand Test

Diagnostic Test: bioelectrical impedance analysisDiagnostic Test: blood drawDiagnostic Test: transient elastography (FibroScan)Diagnostic Test: ataxia scoreDiagnostic Test: Five-Times-Sit-to-Stand Test

Interventions

Electrophysical measurement that allows to determine the exact composition of single body compartments by producing a magnetic field and detecting the potential difference through the body

A-T patients
blood drawDIAGNOSTIC_TEST

Blood samples are taken from sober patients

A-T patients

FibroScan is a noninvasive tool to measure liver stiffness as an indication of fatty liver and liver fibrosis using ultrasound

A-T patients
ataxia scoreDIAGNOSTIC_TEST

Klockgether ataxia score ranges from 0 to 35 points in which 0 means no symptoms and 35 stands for final stage of disease. It includes seven ataxia associated symptoms: dysarthria, intention tremor, ataxia of gait, stance, dysdiadochokinesia, upper limb and lower limb

A-T patients

The test measures the complete time which is necessary for an individual to stand up and sit down on a chair five times in series

A-T patients

Eligibility Criteria

Age2 Years - 45 Years
Sexall
Healthy VolunteersNo
Age GroupsChild (0-17), Adult (18-64)

You may qualify if:

  • aim group: genetically and/or clinically diagnosed A-T
  • age 2-45 years
  • written informed consent

You may not qualify if:

  • age \< 2 or \> 45 years
  • other diseases with influence on the immune system (i.e. diabetes mellitus, malignoma, dialysis-dependent renal failure)

Contact the study team to confirm eligibility.

Sponsors & Collaborators

Study Sites (1)

Children's Hospital, Allergology, Pneumology and Cystic Fibrosis, Goethe University Frankfurt

Frankfurt am Main, Hesse, 60590, Germany

RECRUITING

Related Publications (1)

  • Zielen S, Duecker RP, Woelke S, Donath H, Bakhtiar S, Buecker A, Kreyenberg H, Huenecke S, Bader P, Mahlaoui N, Ehl S, El-Helou SM, Pietrucha B, Plebani A, van der Flier M, van Aerde K, Kilic SS, Reda SM, Kostyuchenko L, McDermott E, Galal N, Pignata C, Perez JLS, Laws HJ, Niehues T, Kutukculer N, Seidel MG, Marques L, Ciznar P, Edgar JDM, Soler-Palacin P, von Bernuth H, Krueger R, Meyts I, Baumann U, Kanariou M, Grimbacher B, Hauck F, Graf D, Granado LIG, Prader S, Reisli I, Slatter M, Rodriguez-Gallego C, Arkwright PD, Bethune C, Deripapa E, Sharapova SO, Lehmberg K, Davies EG, Schuetz C, Kindle G, Schubert R. Simple Measurement of IgA Predicts Immunity and Mortality in Ataxia-Telangiectasia. J Clin Immunol. 2021 Nov;41(8):1878-1892. doi: 10.1007/s10875-021-01090-8. Epub 2021 Sep 3.

MeSH Terms

Conditions

Ataxia TelangiectasiaImmunologic Deficiency SyndromesLiver Diseases

Interventions

Blood Specimen Collection

Condition Hierarchy (Ancestors)

Spinocerebellar AtaxiasCerebellar AtaxiaCerebellar DiseasesBrain DiseasesCentral Nervous System DiseasesNervous System DiseasesNeurocutaneous SyndromesAtaxiaDyskinesiasNeurologic ManifestationsTelangiectasisVascular DiseasesCardiovascular DiseasesGenetic Diseases, InbornCongenital, Hereditary, and Neonatal Diseases and AbnormalitiesPrimary Immunodeficiency DiseasesDNA Repair-Deficiency DisordersMetabolic DiseasesNutritional and Metabolic DiseasesImmune System DiseasesDigestive System Diseases

Intervention Hierarchy (Ancestors)

Specimen HandlingClinical Laboratory TechniquesDiagnostic Techniques and ProceduresDiagnosisPuncturesSurgical Procedures, OperativeInvestigative Techniques

Study Officials

  • Stefan Zielen, Prof. Dr.

    Children's Hospital, Allergology, Pneumology and Cystic Fibrosis, Goethe-University

    PRINCIPAL INVESTIGATOR

Central Study Contacts

Sandra Woelke, Dr.

CONTACT

Stefan Zielen, Prof. Dr.

CONTACT

Study Design

Study Type
interventional
Phase
not applicable
Allocation
NA
Masking
NONE
Purpose
DIAGNOSTIC
Intervention Model
SINGLE GROUP
Sponsor Type
OTHER
Responsible Party
PRINCIPAL INVESTIGATOR
PI Title
Prof. Dr. med. Stefan Zielen

Study Record Dates

First Submitted

April 24, 2017

First Posted

November 30, 2017

Study Start

October 1, 2016

Primary Completion

September 30, 2018

Study Completion

September 30, 2019

Last Updated

November 30, 2017

Record last verified: 2017-11

Locations