NCT04113083

Brief Summary

The prevalence of CTX in our country is estimated to be 1 / 50.000. The aim of this study is to screen more volunteers by conducting a larger screening from neurology and pediatric metabolism clinics in Turkey. This observational study was designed retrospectively and prospectively in two stages. In the retrospective section, the patient database and / or patient files will be screened in the neurology and pediatric metabolism clinics and the patients aged 40 and below in the neurology clinics with at least two of the following will be enrolled to the study:

  • Ataxia and / or spasticity
  • Bilateral cataract (except senile cataract)
  • Intellectual limitation
  • Non-enhancing hyperintensity on T2 sections in MR imaging of dentate nuclei
  • Autosomal recessive transition pattern. (Ex: Relative Marriage) In the pediatric metabolism centers, cases suspected of CTX and planned to apply the Mignarri Index according to the investigator's opinion will be identified.

Trial Health

57
Monitor

Trial Health Score

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

Enrollment
22

participants targeted

Target at below P25 for all trials

Timeline
Completed

Started Oct 2019

Typical duration for all trials

Geographic Reach
1 country

22 active sites

Status
terminated

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

September 25, 2019

Completed
7 days until next milestone

First Posted

Study publicly available on registry

October 2, 2019

Completed
17 days until next milestone

Study Start

First participant enrolled

October 19, 2019

Completed
2.4 years until next milestone

Primary Completion

Last participant's last visit for primary outcome

March 16, 2022

Completed
3 months until next milestone

Study Completion

Last participant's last visit for all outcomes

June 10, 2022

Completed
Last Updated

June 15, 2022

Status Verified

June 1, 2022

Enrollment Period

2.4 years

First QC Date

September 25, 2019

Last Update Submit

June 10, 2022

Conditions

Outcome Measures

Primary Outcomes (2)

  • Proportion of patients with CTX possibility in Neurology Clinics

    \- Proportion of patients aged 40 years or younger with at least two of the following (2) in patients with a cholestanol test threshold (3.75 mg / mL) in neurology clinics: * Ataxia and / or spasticity * Bilateral cataract (except senile cataract) * Intellectual limitation * Nonintensitive hyperintensity in T2 sections on MRI of the dentate nucleus * Forming an autosomal recessive transition pattern. (Ex: Relative Marriage)

    3 years

  • Proportion of patients with CTX possibility in Pediatric Metabolism Clinics

    \- Proportion of cases above the cholestanol test threshold (3.75 mg / mL) in pediatric metabolism centers

    3 years

Secondary Outcomes (7)

  • Total of Mignarri Suspicion Index (SI)

    3 Years

  • Cholestanol Levels

    3 Years

  • Patient demographics

    3 Years

  • CTX Family History

    3 Years

  • Presence of consangunious marriage

    3 Years

  • +2 more secondary outcomes

Interventions

Blood sampling will be performed by Düzen Laboratories group for cholestanol

Eligibility Criteria

AgeUp to 40 Years
Sexall
Healthy VolunteersNo
Age GroupsChild (0-17), Adult (18-64)
Sampling MethodProbability Sample
Study Population

200 patients

You may qualify if:

  • I-1. Giving written informed consent
  • I-2. Patients in neurology clinics should have been identified with at least two of the following:
  • Ataxia and / or spasticity
  • Bilateral cataract (except senile cataract)
  • Intellectual limitation
  • Nonintensitive hyperintensity in T2 sections on MRI of the dentate nucleus
  • Forming an autosomal recessive transition pattern. (Ex: Relative Marriage)
  • I-3. In the pediatric metabolism centers, cases suspected of CTX and planned to apply the Mignarri Index according to the investigator's opinion.
  • I-4. On the day the patient signed the Informed Consent Form, the patient did not get older than 41 years of age (subjects aged 40 and under will be included in the study)

You may not qualify if:

  • E-1. The patient's ataxia and / or spasticity, cataract, intellectual limitation, and non-contrasted hyperintensity of T2 sections in MR imaging of dentate nuclei with typical MRI findings are due to a known cause other than CTX or other underlying disease.
  • E-2. The patient has participated in an interventional clinical study in the last 30 days,
  • E-3. The patient and / or his / her legal representative does not give consent to participate in the study,
  • E-4. In the opinion of the investigator, the patient is not able to fulfill the working requirements appropriately,
  • E-5. Pregnancy and / or lactation
  • E-6. If the patient was 41 years old when included in the study.

Contact the study team to confirm eligibility.

Sponsors & Collaborators

Study Sites (22)

Çukurova University Medical Faculty Deparment of Metabolism

Adana, Turkey (Türkiye)

Location

Çukurova University Medical Faculty Department of Neurology

Adana, Turkey (Türkiye)

Location

Ankara Child and Heamatology Hospital Deparment of Metabolism

Ankara, Turkey (Türkiye)

Location

Ankara City Hospital

Ankara, Turkey (Türkiye)

Location

Ankara Dışkapı Yıldırım Beyazıt Research and Training Hospital Clinic of Neurology

Ankara, Turkey (Türkiye)

Location

Gazi University Medical Faculty Department of Pediatric Metabolism

Ankara, Turkey (Türkiye)

Location

Hacettepe University Medical Faculty Deparment of Metabolism

Ankara, Turkey (Türkiye)

Location

Hacettepe University Medical Faculty Department of Neurology

Ankara, Turkey (Türkiye)

Location

Osmangazi University Medical Faculty Department of Neurology

Eskişehir, Turkey (Türkiye)

Location

Osmangazi University Medical Faculty Department of Pediatric Metabolism

Eskişehir, Turkey (Türkiye)

Location

Bezmi Alem Vakıf University Medical Faculty Department of Neurology

Istanbul, Turkey (Türkiye)

Location

Hamidiye Şişli Etfal Research and Training Hospital Clinic of Neurology

Istanbul, Turkey (Türkiye)

Location

Hamidiye Şişli Etfal Research and Training Hospital Clinic of Pediatric Metabolism

Istanbul, Turkey (Türkiye)

Location

İstanbul University Cerrahpasa Medical Faculty Department of Pediatric Metabolism

Istanbul, Turkey (Türkiye)

Location

İstanbul University Cerrahpaşa Medical Faculty Department of Neurology

Istanbul, Turkey (Türkiye)

Location

İstanbul University İstanbul Medical Faculty Department of Neurology

Istanbul, Turkey (Türkiye)

Location

İstanbul University İstanbul Medical Faculty Department of Pediatric Metabolism

Istanbul, Turkey (Türkiye)

Location

Kanuni Sultan Suleyman Research and Training Hospital Clinic of Pediatric Metabolism

Istanbul, Turkey (Türkiye)

Location

Medeniyet University Göztepe Research and Training Hospital Clinic of Neurology

Istanbul, Turkey (Türkiye)

Location

Mersin City Hospital Department of Metabolism

Mersin, Turkey (Türkiye)

Location

Mersin University Medical Faculty Department of Neurology

Mersin, Turkey (Türkiye)

Location

Cumhuriyet University Medical Faculty Department of Neurology

Sivas, Turkey (Türkiye)

Location

Biospecimen

Retention: SAMPLES WITHOUT DNA

Blood sampling for cholestanol analysis

MeSH Terms

Conditions

Xanthomatosis, Cerebrotendinous

Interventions

Blood Specimen Collection

Condition Hierarchy (Ancestors)

Lipid Metabolism, Inborn ErrorsMetabolism, Inborn ErrorsGenetic Diseases, InbornCongenital, Hereditary, and Neonatal Diseases and AbnormalitiesLipid Metabolism DisordersMetabolic DiseasesNutritional and Metabolic DiseasesXanthomatosis

Intervention Hierarchy (Ancestors)

Specimen HandlingClinical Laboratory TechniquesDiagnostic Techniques and ProceduresDiagnosisPuncturesSurgical Procedures, OperativeInvestigative Techniques

Study Design

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

Study Record Dates

First Submitted

September 25, 2019

First Posted

October 2, 2019

Study Start

October 19, 2019

Primary Completion

March 16, 2022

Study Completion

June 10, 2022

Last Updated

June 15, 2022

Record last verified: 2022-06

Data Sharing

IPD Sharing
Will not share

Locations