Evaluation by a Vineland II Scale of Long-term Development of Children With Pyridoxine Dependent Epilepsy

NCT ID: NCT06054347

Last Updated: 2025-08-11

Study Results

Results pending

The study team has not published outcome measurements, participant flow, or safety data for this trial yet. Check back later for updates.

Basic Information

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Recruitment Status

COMPLETED

Total Enrollment

30 participants

Study Classification

OBSERVATIONAL

Study Start Date

2023-12-29

Study Completion Date

2025-05-21

Brief Summary

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This study aims to evaluate with the VINELAND II scale the long-term neurocognitive development of children above age 3 years with pyridoxine dependent epilepsy related to antiquitine deficiency.

Detailed Description

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Pyridoxine dependent epilepsy related to antiquitine deficiency is a rare and severe genetic epilepsy that usually starts within the first month of life. The treatment is based on high dose of Vitamin B6 (Pyridoxine) which is usually highly effective to control the seizures. However, most patients show impaired neurodevelopment.

Ten years ago, international recommendations have changed and a lysine restricted diet was added as an adjunct therapy with the hope to improve neurodevelopment.

Very few studies have evaluated the cognitive development of these patients in a standardized way, and these studies evaluate very few patients.

It seems therefore essential to study in a standardized way the neurocognitive development of these patients in order to evaluate the impact of the care.

The VINELAND adaptative behaviour scale II has been chosen because it can evaluate a patient whatever the age or the intellectual abilities through a semi-structured questionnaire completed with the parents, and gives a wide view of the neurocognitive development and everyday life autonomy of the patients.

Conditions

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Pyridoxine-Dependent Epilepsy

Study Design

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Observational Model Type

COHORT

Study Time Perspective

CROSS_SECTIONAL

Interventions

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VINELAND II

Standardised questionnaire filled by the investigator during an interview with the parents or with the patient himself if capable of it. Duration 1 hour to 1 hour and a half.

Intervention Type OTHER

Eligibility Criteria

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Inclusion Criteria

* Age above 3 years old
* Pyridoxine dependent epilepsy genetic diagnosis (mutation in ALDH7A1 gene)
* No objection of the patient or his legal representatives.

Exclusion Criteria

* Poor understanding of French language.
Minimum Eligible Age

3 Years

Eligible Sex

ALL

Accepts Healthy Volunteers

No

Sponsors

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University Hospital, Angers

OTHER_GOV

Sponsor Role lead

Responsible Party

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Responsibility Role SPONSOR

Principal Investigators

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Patrick VAN BOGAERT, PhD - MD

Role: PRINCIPAL_INVESTIGATOR

University Hospital, Angers

Locations

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CHU Angers

Angers, , France

Site Status

Centre Hospitalier Universitaire de Besançon

Besançon, , France

Site Status

CHRU Morvan

Brest, , France

Site Status

CHU d'Estaing

Clermont-Ferrand, , France

Site Status

Hôpital Bicêtre

Le Kremlin-Bicêtre, , France

Site Status

Hôpital Jeanne de Flandre

Lille, , France

Site Status

Hôpital de la Timone-Enfants

Marseille, , France

Site Status

CHU Gui de Chautiac

Montpeliier, , France

Site Status

Hôpital Necker-Enfants malades

Paris, , France

Site Status

Centre Hospitalier Saint Nazaire

Saint-Nazaire, , France

Site Status

CHU Toulouse

Toulouse, , France

Site Status

Hôpital Clocheville

Tours, , France

Site Status

Countries

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France

References

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Coughlin CR 2nd, Swanson MA, Spector E, Meeks NJL, Kronquist KE, Aslamy M, Wempe MF, van Karnebeek CDM, Gospe SM Jr, Aziz VG, Tsai BP, Gao H, Nagy PL, Hyland K, van Dooren SJM, Salomons GS, Van Hove JLK. The genotypic spectrum of ALDH7A1 mutations resulting in pyridoxine dependent epilepsy: A common epileptic encephalopathy. J Inherit Metab Dis. 2019 Mar;42(2):353-361. doi: 10.1002/jimd.12045. Epub 2019 Feb 22.

Reference Type BACKGROUND
PMID: 30043187 (View on PubMed)

Wilson MP, Plecko B, Mills PB, Clayton PT. Disorders affecting vitamin B6 metabolism. J Inherit Metab Dis. 2019 Jul;42(4):629-646. doi: 10.1002/jimd.12060. Epub 2019 Mar 20.

Reference Type BACKGROUND
PMID: 30671974 (View on PubMed)

Basura GJ, Hagland SP, Wiltse AM, Gospe SM Jr. Clinical features and the management of pyridoxine-dependent and pyridoxine-responsive seizures: review of 63 North American cases submitted to a patient registry. Eur J Pediatr. 2009 Jun;168(6):697-704. doi: 10.1007/s00431-008-0823-x. Epub 2008 Sep 2.

Reference Type BACKGROUND
PMID: 18762976 (View on PubMed)

Baxter P. Epidemiology of pyridoxine dependent and pyridoxine responsive seizures in the UK. Arch Dis Child. 1999 Nov;81(5):431-3. doi: 10.1136/adc.81.5.431.

Reference Type BACKGROUND
PMID: 10519720 (View on PubMed)

Coughlin CR 2nd, Tseng LA, Abdenur JE, Ashmore C, Boemer F, Bok LA, Boyer M, Buhas D, Clayton PT, Das A, Dekker H, Evangeliou A, Feillet F, Footitt EJ, Gospe SM Jr, Hartmann H, Kara M, Kristensen E, Lee J, Lilje R, Longo N, Lunsing RJ, Mills P, Papadopoulou MT, Pearl PL, Piazzon F, Plecko B, Saini AG, Santra S, Sjarif DR, Stockler-Ipsiroglu S, Striano P, Van Hove JLK, Verhoeven-Duif NM, Wijburg FA, Zuberi SM, van Karnebeek CDM. Consensus guidelines for the diagnosis and management of pyridoxine-dependent epilepsy due to alpha-aminoadipic semialdehyde dehydrogenase deficiency. J Inherit Metab Dis. 2021 Jan;44(1):178-192. doi: 10.1002/jimd.12332. Epub 2020 Dec 1.

Reference Type BACKGROUND
PMID: 33200442 (View on PubMed)

Tourrette C., Evaluer les enfants avec déficiences ou troubles du développement. PARIS : Dunod, 2006

Reference Type BACKGROUND

Fombonne E. ; Achard S. ; Tuffreau R. L'évaluation du comportement adaptatif : l'échelle de Vineland. In Handicap et inadaptations. Les Cahiers du CTNERHI, n° 67-68, juil. / déc. 1995. p. 79-90.3

Reference Type BACKGROUND

Gioia GA, Isquith PK, Guy SC, Kenworthy L. The Behaviour Rating Inventory of Executive Function. Lutz, FL: Psychological Assessment Resources; 2000.

Reference Type BACKGROUND

Gibaud M, Barth M, Lefranc J, Mention K, Villeneuve N, Schiff M, Maurey H, Barthez MA, Caubel I, Chouchane M, Doummar D, Kossorotoff M, Lamblin MD, Roubertie A, Nabbout R, Van Bogaert P. West Syndrome Is an Exceptional Presentation of Pyridoxine- and Pyridoxal Phosphate-Dependent Epilepsy: Data From a French Cohort and Review of the Literature. Front Pediatr. 2021 Mar 5;9:621200. doi: 10.3389/fped.2021.621200. eCollection 2021.

Reference Type BACKGROUND
PMID: 33748042 (View on PubMed)

Other Identifiers

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49RC23_0181

Identifier Type: -

Identifier Source: org_study_id

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