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CompletedNCT02099552ECP-015Updated Sep 5, 2017

Natural History and Outcomes in X-Linked Hypohidrotic Ectodermal Dysplasia

An observational study in X-Linked Hypohidrotic Ectodermal Dysplasia, sponsored by Edimer Pharmaceuticals. Completed at 7 sites in 5 countries. Open to participants aged Up to 36 Months. Per ClinicalTrials.gov, last updated 2017-09-05.

Sponsored by Edimer Pharmaceuticals · Observational

Study type
Observational
Model
Cohort
Time perspective
Other
Enrollment
150
Ages
Up to 36 Months
Sex
All
01

Study summary

The proposed natural history study will enroll male and female patients, ages 36 months and younger, who have a diagnosis of XLHED based on genetic testing and who have not received an investigational study drug. The study protocol will include collection of all relevant medical history and documentation of clinical outcomes using age-appropriate, minimally invasive technologies. Data will be collected both retrospectively, back to pregnancy assessments that may be available, and prospectively through age 5 yrs.

Read the detailed description

Important to the development and regulatory approval of therapies for XLHED will be the collection of data on the clinical history and prospective health of those affected by XLHED. The proposed natural history study will enroll male and female patients, ages 36 months and younger, who have a diagnosis of XLHED based on genetic testing and who have not received an investigational study drug. The study protocol will include collection of all relevant medical history and documentation of clinical outcomes using age-appropriate, minimally invasive technologies. Data will be collected both retrospectively, back to pregnancy assessments that may be available, and prospectively through age 5 yrs. Genotype-phenotype correlations in XLHED, based on well-documented health records and prospective assessments on genetically-confirmed individuals, may now provide new and clinically-predictive information for the benefit of patients, families, health care providers and clinical investigators designing trials for therapeutic interventions.

02

Conditions studied

  • X-Linked Hypohidrotic Ectodermal Dysplasia

Keywords

  • X-Linked hypohidrotic ectodermal dysplasia
  • XLHED
  • Hypohidrotic ectodermal dysplasia
  • HED
  • Christ-Siemens-Touraine syndrome
03

Who can participate

Ages eligible
Up to 36 Months
Sexes eligible
All
Accepts healthy volunteers
No
Sampling method
Non-probability sample

Study population

Those with the condition of XLHED up to age 36 months

Inclusion criteria

Subjects must meet all of the following criteria to be enrolled in this study:

  1. Confirmed genetic diagnosis of XLHED
  2. Written informed consent of both parents (if reasonably available)

Exclusion criteria

Exclusion Criteria:

Subjects who meet any of the following criteria cannot be enrolled in this study:

  1. Medically-significant complications or congenital anomalies outside of those considered to be associated with the diagnosis or status of XLHED
  2. Having received an investigational study drug prior to enrollment. For subjects less than 6 months of age, the mother cannot have taken an investigational drug during her pregnancy.
  3. Known hypersensitivity to pilocarpine or pilocarpine-like muscarinic agonists
  4. Presence of pacemakers
04

Study design

Observational model
Cohort
Time perspective
Other
Enrollment
150 participants (estimated)
Patient registry
No

Groups and cohorts

  • XLHED

    Those with the condition of XLHED

05

What researchers measure

Primary outcomes

  1. To assess clinical course of untreated XLHED individuals

    To characterize the clinical course of untreated XLHED male and female subjects in early childhood, capturing data from physician and hospital records, medical history including growth and development, and family interviews.

    Time frame: Up to 5 years of life

  2. To assess the phenotype of untreated XLHED individuals

    To characterize the phenotype of untreated XLHED male subjects and female in early childhood with endpoint assessments including sweat (males only), dentition, craniofacial development, pulmonary and ocular health.

    Time frame: Up to 5 years of life

Secondary outcomes

  1. To assess changes in endpoint assessments over time (growth and development)

    Time frame: Baseline and yearly up through 5 years of age

  2. To assess changes in endpoint assessments over time (Mortality/Hospitalizations/Infections/Fevers/Heat Intolerance)

    Time frame: Baseline and yearly up through 5 years of age

  3. To assess changes in endpoint assessments over time (sweat rate)

    Time frame: Baseline and yearly through 5 years of age

  4. To assess changes in endpoint assessments over time (Dentition)

    Time frame: Baseline and yearly through 5 years of age

  5. To assess changes in endpoint assessments over time (dry eye)

    Time frame: Baseline and yearly through 5 years of age

  6. To assess changes in endpoint assessments over time (skin, hair and nail health)

    Time frame: Baseline through 5 years of age

  7. To assess changes in endpoint assessments over time (respiratory health)

    Time frame: Baseline and yearly through 5 years of age

  8. To assess changes in endpoint assessments over time (craniofacial development)

    Time frame: Baseline and yearly through 5 years of age

  9. To assess genotype-phenotype correlation in XLHED affected individuals

    To correlate clinical course and endpoint outcomes with EDA genotype in untreated XLHED-affected male and female subjects.

    Time frame: Baseline through 5 years of age

06

Study locations

7 sites
  • University of California, San Francisco
    San Francisco, California 94143, United States
  • Children's National Medical Center
    Washington, D.C., District of Columbia 20010, United States
  • Washington University School of Medicine
    Saint Louis, Missouri 63110, United States
  • Hôpital Necker-Enfants Malades
    Paris, 75015, France
  • University Hospital Erlangen
    Erlangen, Bavaria 91054, Germany
  • Azienda Ospedaliera-Polo Universitario "Luigi Sacco"
    Milan, 20157, Italy
  • University Hospital of Wales
    Cardiff, CF14 4XW, United Kingdom
07

Registry details

Key details

Study ID
NCT02099552
Lead sponsor
Edimer Pharmaceuticals
Responsible party
Sponsor
First posted
Mar 31, 2014
Start date
Apr 2014
Primary completion
Dec 2016
Completion
Dec 2016
Last update
Sep 5, 2017

Study contacts

Ramsey Johnson, MSM
study director · Edimer Pharmaceuticals

Oversight

Data monitoring committee
No
View the source record on ClinicalTrials.gov ↗

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This study is completed, as verified in Aug 2017. You cannot join it, but the record below documents what was studied.

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