NCT05522374

Brief Summary

TIRCON-reg aims to

  • continue the provision of a global registry and natural history study for NBIA disorders
  • harmonize and cover existing national and single site registries
  • enable participation of countries and single sites that so far have no access to an NBIA registry
  • join forces in order to recruit sufficient numbers of patients
  • define the natural history of NBIA disorders
  • define the most appropriate outcome measures
  • inform the design and facilitate the conduction of clinical trials

Trial Health

83
On Track

Trial Health Score

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

Enrollment
2,000

participants targeted

Target at P75+ for all trials

Timeline
178mo left

Started Jun 2012

Longer than P75 for all trials

Geographic Reach
8 countries

9 active sites

Status
recruiting

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 Progress49%
Jun 2012Dec 2040

Study Start

First participant enrolled

June 14, 2012

Completed
10.2 years until next milestone

First Submitted

Initial submission to the registry

August 11, 2022

Completed
20 days until next milestone

First Posted

Study publicly available on registry

August 31, 2022

Completed
18.3 years until next milestone

Primary Completion

Last participant's last visit for primary outcome

December 1, 2040

Expected
Same day until next milestone

Study Completion

Last participant's last visit for all outcomes

December 1, 2040

Last Updated

June 5, 2025

Status Verified

June 1, 2025

Enrollment Period

28.5 years

First QC Date

August 11, 2022

Last Update Submit

June 2, 2025

Conditions

Keywords

NBIA, PKAN, BPAN, MPAN, FAHN, KRS, CoPAN, PLAN,

Outcome Measures

Primary Outcomes (4)

  • Change in Score on the Barry-Albright Dystonia (BAD) Scale

    The Barry-Albright Dystonia Scale is an instrument for rating the severity of dystonia in eight body regions. The individual scores are summed to provide a total score that ranges from 0 to 32; the higher the score, the more severe the dystonia. Patients with dystonia are assessed for the change in total BAD score over time since Baseline.

    The individual participants are followed with annual assessments over a long time period (up to 30 years) or until discontinuation or death.

  • Change in Score on Unified Parkinson's Disease Rating (UPDRS) Scale, Part I-III, VI

    The Unified Parkinson's Disease Rating Scale (UPDRS) is the major rating scale used to assess severity of symptoms of Parkinson's disease, some of which are similar to symptoms in NBIA. The UPDRS subscales used in this study are Part I: Mentation, Behavior and Mood, scored from 0 (best) to 16 (worst); Part II: Activities of Daily Living, scored from 0 (best) to 52 (worst); Part III: Motor Examination, scored from 0 (best) to 108 (worst); and Part VI: Schwab and England Activities of Daily Living Scale, scored from 0% (worst) to 100% (best).

    The individual participants are followed with annual assessments over a long time period (up to 30 years) or until discontinuation or death.

  • Change in Score on Pediatric Quality of Life (PedsQL)

    The Pediatric Quality of Life (PedsQL) questionnaire is used to measure functional health and well-being from the patient's point of view. Separate versions of the questionnaire are available for children, young adults aged 18-25 years, and adults older than 25 years. Patients are asked to indicate how they have felt over the past month, and the scores of the 23 questions are used to generate an overall score that ranges from 0 (worst) to 100 (best).

    The individual participants are followed with annual assessments over a long time period (up to 30 years) or until discontinuation or death.

  • Disease progression

    Disease progression as assessed by clinical examination and captured as HPO (Human Phenotype Ontology) Terms at each visit.

    The individual participants are followed with annual assessments over a long time period (up to 30 years) or until discontinuation or death.

Study Arms (1)

NBIA Patients

Patients with suspected or confirmed NBIA

Eligibility Criteria

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

children, adults, patients with limited decision-making capacity

You may qualify if:

  • suspected or confirmed NBIA
  • willingness to participate

You may not qualify if:

  • unwillingness to participate

Contact the study team to confirm eligibility.

Sponsors & Collaborators

Study Sites (9)

Children's Hospital of Eastern Ontario, Division of Neurology, Department of Pediatrics

Ottawa, K1H 8L1, Canada

RECRUITING

Charles University, Department of neurology

Prague, 120 00, Czechia

RECRUITING

LMU Klinikum, Friedrich-Baur-Institute

Munich, Bavaria, 80336, Germany

RECRUITING

The Foundation of the Carlo Besta Neurological Institute, IRCCS

Milan, 20133, Italy

RECRUITING

University medical Center Groningen (UMCG) Department of Neurology AB 51

Groningen, 9700 RB, Netherlands

RECRUITING

The Childrens Memorial Health Institute

Warsaw, 04-730, Poland

RECRUITING

University of Belgrade, Department of Movement Disorders and Degenerative Brain Diseases

Belgrade, Serbia

RECRUITING

Hospital Vall d'Hebron - Institut de Recerca (VHIR), Pediatric Neurology, Movement Disorders

Barcelona, 08035, Spain

ACTIVE NOT RECRUITING

Hospital Sant Joan de Déu, Universitat de Barcelona, Servei de Neurología

Barcelona, 08950, Spain

ACTIVE NOT RECRUITING

Related Publications (4)

  • Kalman B, Lautenschlaeger R, Kohlmayer F, Buchner B, Kmiec T, Klopstock T, Kuhn KA. An international registry for neurodegeneration with brain iron accumulation. Orphanet J Rare Dis. 2012 Sep 17;7:66. doi: 10.1186/1750-1172-7-66.

    PMID: 22985983BACKGROUND
  • Klopstock T, Tricta F, Neumayr L, Karin I, Zorzi G, Fradette C, Kmiec T, Buchner B, Steele HE, Horvath R, Chinnery PF, Basu A, Kupper C, Neuhofer C, Kalman B, Dusek P, Yapici Z, Wilson I, Zhao F, Zibordi F, Nardocci N, Aguilar C, Hayflick SJ, Spino M, Blamire AM, Hogarth P, Vichinsky E. Safety and efficacy of deferiprone for pantothenate kinase-associated neurodegeneration: a randomised, double-blind, controlled trial and an open-label extension study. Lancet Neurol. 2019 Jul;18(7):631-642. doi: 10.1016/S1474-4422(19)30142-5.

    PMID: 31202468BACKGROUND
  • Karin I, Buchner B, Gauzy F, Klucken A, Klopstock T. Treat Iron-Related Childhood-Onset Neurodegeneration (TIRCON)-An International Network on Care and Research for Patients With Neurodegeneration With Brain Iron Accumulation (NBIA). Front Neurol. 2021 Feb 22;12:642228. doi: 10.3389/fneur.2021.642228. eCollection 2021.

    PMID: 33692746BACKGROUND
  • Iankova V, Karin I, Klopstock T, Schneider SA. Emerging Disease-Modifying Therapies in Neurodegeneration With Brain Iron Accumulation (NBIA) Disorders. Front Neurol. 2021 Apr 15;12:629414. doi: 10.3389/fneur.2021.629414. eCollection 2021.

    PMID: 33935938BACKGROUND

Related Links

Biospecimen

Retention: SAMPLES WITH DNA

RNA, DNA, Plasma and urine

MeSH Terms

Conditions

Pantothenate Kinase-Associated NeurodegenerationFatty Acid Hydroxylase-Associated NeurodegenerationKufor-Rakeb syndromeNeuroferritinopathyFamilial apoceruloplasmin deficiencyWoodhouse Sakati syndromeNeuroaxonal Dystrophies

Condition Hierarchy (Ancestors)

Basal Ganglia DiseasesBrain DiseasesCentral Nervous System DiseasesNervous System DiseasesMovement DisordersHeredodegenerative Disorders, Nervous SystemNeurodegenerative DiseasesGenetic Diseases, InbornCongenital, Hereditary, and Neonatal Diseases and Abnormalities

Study Officials

  • Thomas Klopstock, Prof. Dr.

    LMU Klinikum, Friedrich-Baur-Institute

    PRINCIPAL INVESTIGATOR

Central Study Contacts

Study Design

Study Type
observational
Observational Model
COHORT
Time Perspective
PROSPECTIVE
Target Duration
30 Years
Sponsor Type
OTHER
Responsible Party
PRINCIPAL INVESTIGATOR
PI Title
Prof. Dr. med.

Study Record Dates

First Submitted

August 11, 2022

First Posted

August 31, 2022

Study Start

June 14, 2012

Primary Completion (Estimated)

December 1, 2040

Study Completion (Estimated)

December 1, 2040

Last Updated

June 5, 2025

Record last verified: 2025-06

Data Sharing

IPD Sharing
Will share

Any data or requests from the project can be shared with researchers upon written request. Data sharing requires i) the approval by the respective scientific committee and ii) an approval by the institutional review board of the researcher on the intended project.

Shared Documents
ICF
Time Frame
ime frame for data usage needs to be specified in the proposal for data sharing
Access Criteria
Approved written proposal including description of the research plan and data usage purpose.

Locations