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RecruitingNCT06262152Updated Feb 15, 2024

Sleep Profile of Patients With Septo-optic Dysplasia

An observational study in Septo-Optic Dysplasia, Agenesis of Corpus Callosum and Blindness, sponsored by IRCCS National Neurological Institute "C. Mondino" Foundation. Recruiting at 1 site in Italy. Open to participants aged 3 Years to 18 Years. Per ClinicalTrials.gov, last updated 2024-02-15.

Sponsored by IRCCS National Neurological Institute "C. Mondino" Foundation · Observational

Study type
Observational
Model
Cohort
Time perspective
Prospective
Enrollment
45
Ages
3 Years to 18 Years
Sex
All
01

Study summary

The aim of this study is to evaluate sleep of patients with septo-optic dysplasia compared to patients with an isolated disorder of peripheral visual system and patients with corpus callosum agenesis since both visus defict and agenesis of corpus callosum might be present SOD but associated to other features / structural and functional anomalies.

Included patients and their caregivers will be asked to compile standardize sleep questionnaires and a sleep screening through an interview will be scheduled. Patients will be asked to wear an actigraph on their non-dominant hand wrist for 7 days.

Read the detailed description

The primary objective of this study is to evaluate sleep of patients with septo-optic dysplasia compared to patients with an isolated disorder of peripheral visual system and patients with corpus callosum agenesis since both visus defict and agenesis of corpus callosum might be present SOD but associated to other features / structural and functional anomalies.

The secondary aims of this study are the following:

  1. identify which factors such as clinical and neuroanatomical features including sensory experience influence sleep outcome and how
  2. describe melatonin profile of patients with SOD
  3. describe sleep EEG of patients with SOD

Three groups of patients will be included:

  • Group A: patients with septo-optic dysplasia
  • Group B: patients with disorder of peripheral visual system
  • Group C: patients with Corpus Callosum agenesis

This is an observational monocentric study. The procedures in the study that are not included in current clinical practice in the follow-up pathway of the included patients are the following:

  • administration of standardized sleep questionnaires (Pittsburgh Sleep Quality Index: self-completed questionnaire assessing sleep quality over a 1-month time interval;Epworth Sleepiness Scale: self-completed questionnaire assessing daytime sleepiness; Children sleep habits questionnaire)
  • 7 days actigraph recording through wearing of Actiwatch spectrum plus at home
  • Performance of Sleep EEG
  • Performance of blood and salivary sampling for melatonin dosing (for each subject, one 4 mL blood and one 2 mL saliva samples will be collected at each assessment)
02

Conditions studied

  • Septo-Optic Dysplasia
  • Agenesis of Corpus Callosum
  • Blindness

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Keywords

  • sleep
  • actigraphy
  • brain malformation
  • visual deficit
  • SOD
  • genesis of corpus callous
  • pediatric patients
03

Who can participate

Ages eligible
3 Years to 18 Years
Sexes eligible
All
Accepts healthy volunteers
No
Sampling method
Non-probability sample

Study population

Three groups of patients will be included:

  • Group A: 15 patients with septo-optic dysplasia
  • Group B: 15 patients with disorder of peripheral visual system
  • Group C: 15 patients with Corpus Callosum agenesis

Eligibility criteria

Inclusion criteria group A:

  • diagnosis of SOD or SOD plus syndrome with or without a defined genetic diagnosis
  • age 3-18 years
  • availability of at least 2 serial sleep EEGs performed during clinical follow-up
  • stable drug therapy in the last three months

Inclusion criteria group B:

  • diagnosis of congenital or early acquired isolated peripheral visual deficit with or without a known genetic diagnosis (e.g., congenital cataract, inherited retinal dystrophies, isolated eye maldevelopment).
  • age 3-18 years
  • grating or visual acuity \< 3/10
  • availability of serial sleep EEGs performed during clinical follow-up
  • stable drug therapy in the last three months

Inclusion criteria group C

  • isolated corpus callosum agenesis at brain MRI
  • age 3-18 years
  • availability of at least 2 serial sleep EEGs performed during clinical follow-up
  • stable drug therapy in the last three months

Exclusion criteria group A:

  • absence of informed consent
  • severe Intellectual disability and/or severe motor impairment
  • melatonin assumption

Exclusion criteria group B:

  • absence of informed consent
  • CNS involvement ( malformations/ lesions)
  • severe Intellectual disability and/or severe motor impairment
  • melatonin assumption

Exclusion criteria group C

  • absence of informed consent
  • severe Intellectual disability and/or severe motor impairment
  • melatonin assumption
04

Study design

Observational model
Cohort
Time perspective
Prospective
Enrollment
45 participants (estimated)
Patient registry
No
Biospecimen retention
Samples without dna

Groups and cohorts

  • Patients with Septo-optic dysplasia, visual deficit and agenesis of corpus callosum

    * age 3-18 years * availability of at least 2 serial sleep EEGs performed during clinical follow-up * stable drug therapy in the last three months

    Diagnostic Test: Actigraphy, blood and salivary sample, and sleep evaluation

Interventions

  • Diagnostic testActigraphy, blood and salivary sample, and sleep evaluation

    * For each subject, one 4 mL blood and one 2 mL saliva samples will be collected at each assessment. * 7 days actigraph recording through wearing of Actiwatch spectrum plus at home * Standardized sleep questionnaire administration

05

What researchers measure

Primary outcomes

  1. Sleep profile ( Sleep quality, sleep efficiency) of patients with SOD

    SLEEP QUALITY will be assessed through standardized questionnaires :Pittsburgh Sleep Quality Index: self-completed questionnaire ;Epworth Sleepiness Scale: self-completed questionnaire assessing daytime sleepiness; Children sleep habits questionnaire . Sleep quality and sleep-wake cycle will be evaluated, namely Child's inability to go to bed, the delay in falling asleep, sleep duration, overnight awakenings, anxiety related to sleep, parasomnia, respiratory disorders, and daytime sleepiness. SLEEP EFFICIENCY will be measured by actigraphic assessment of sleep efficiency (the ratio of total sleep time to sleep period), total nighttime sleep duration, rest activity and number of awakenings.

    Time frame: 12 months

Secondary outcomes

  1. melatonin profile of patients with SOD

    For each subject, blood and and saliva samples will be collected and Serum and salivary concentrations of melatonin and of its main metabolite 6-hydroxymelatonin will be measured in samples collected from all patients by using a validated LC-MS method with minor modifications (Magliocco et al 2021).

    Time frame: 12 months

  2. Sleep EEG description of patients with SOD

    A Sleep EEG recording will also be scheduled at the time of study inclusion/evaluation. Previously performed EEG will be revised. EEG assessment will include the analysis of background activity, presence/absence of physiological sleep elements, interictal discharges, sleep macrostructure and microstructure ( spindle characteristics).

    Time frame: 12 months

06

Study locations

1 of 1 sites recruiting
  • IRCCS Casimiro Mondino Foundation
    Pavia, PV 27100, Italy
    Recruiting
07

References and documents

Individual participant data

Plan to share: Undecided

No publications or documents are linked to this record.

08

Registry details

Key details

Study ID
NCT06262152
Lead sponsor
IRCCS National Neurological Institute "C. Mondino" Foundation
Responsible party
Sponsor
First posted
Feb 15, 2024
Start date
Dec 20, 2023
Primary completion
Jan 20, 2024
Completion
May 20, 2025 (estimated)
Last update
Feb 15, 2024

Oversight

Data monitoring committee
Yes
FDA-regulated drug
No
FDA-regulated device
No
View the source record on ClinicalTrials.gov ↗

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