Status:

COMPLETED

Functional Studies of Novel Genes Mutated in Primary Ciliary Dyskinesia II: Genotype to Phenotype

Lead Sponsor:

University of North Carolina, Chapel Hill

Collaborating Sponsors:

National Heart, Lung, and Blood Institute (NHLBI)

Conditions:

Primary Ciliary Dyskinesia

Eligibility:

All Genders

12-90 years

Phase:

EARLY_PHASE1

Brief Summary

The purpose of this study is to measure mucociliary clearance (MCC) in groups of subjects with the disease Primary Ciliary Dyskinesia (PCD) caused by mutations in different genes, and compare to healt...

Detailed Description

Participants will undergo screening with basic physical exam and lung function testing at the start of the study. Participants will then inhale a radiolabeled substance and undergo medical imaging to ...

Eligibility Criteria

Inclusion

  • Inclusion Criteria for PCD Patients
  • Confirmed PCD diagnosis with identified genetic mutations
  • Negative pregnancy test for females who are not s/p hysterectomy with oophorectomy
  • Forced Expiratory Volume (FEV1) of at least 30 percent of predicted
  • Inclusion Criteria for Healthy Controls:
  • Age ≥ 18 years old
  • Subjects must have an Forced vital capacity (FVC), Forced Expiratory Volume in one second (FEV1) and FVC/FEV1 of at least 80% of predicted.
  • Subjects who fall out of the normal range will be offered a copy of the test to share with their personal physician.
  • No pre-existing lung disease (asthma, cystic fibrosis, etc.).
  • Negative pregnancy test for females who are not s/p hysterectomy with oophorectomy.
  • Exclusion Criteria:
  • Any chronic medical condition considered by the PI as a contraindication to the exposure study including significant cardiovascular disease, diabetes, chronic renal disease, chronic thyroid disease, immunodeficiency, history of tuberculosis
  • Any acute infection requiring antibiotics within 4 weeks of study.
  • Mental illness or history of drug or alcohol abuse that, in the opinion of the investigator, would interfere with the participant's ability to comply with study requirements.
  • Medications which may impact the results of the study treatment, or may interfere with any other medications potentially used in the study (to include steroids, beta antagonists, non-steroidal anti-inflammatory agents)
  • Active smoking to include e-cigarettes within 1 year of the study, or lifetime of \> 10 pack years of smoking
  • History of vaping or current vaping.
  • Viral upper respiratory tract infection within 4 weeks of challenge.
  • Radiation exposure history in the past year which would be outside the safe levels
  • Pregnant or lactating women will also be excluded since the risks associated with radiation are unknown and cannot be justified
  • Use of the following medications:
  • Use of beta blocking medications
  • Receipt of Live Attenuated Influenza Vaccine (LAIV), also known as FluMist , within the prior 30 days, or any vaccine within the prior 5 days
  • Multivitamins, Vitamin C or E or herbal medications in the 4 days prior to the treatment visit
  • Non-steroidal anti-inflammatory drugs in the 4 days prior to the treatment visit.
  • Allergy/sensitivity to study drugs or their formulations:
  • Known Immunoglobulin E (IgE) mediated hypersensitivity to albuterol
  • Physical/laboratory indications:
  • Temperature \> 37.8 degrees Celsius (C)
  • Subjects \>15 years- Systolic BP \>150 mm hg or \< 90 mm Hg or diastolic BP\> 90 mm Hg or \< 50 and Subjects 12-15 years - Systolic BP \> 130 mmHg or \< 80 mmHg or diastolic BP \> 80 or \<40
  • Oxygen saturation of \< 93 percent
  • Inability or unwillingness of a participant to give written informed consent.

Exclusion

    Key Trial Info

    Start Date :

    June 10 2021

    Trial Type :

    INTERVENTIONAL

    Allocation :

    ACTUAL

    End Date :

    July 22 2024

    Estimated Enrollment :

    27 Patients enrolled

    Trial Details

    Trial ID

    NCT04901715

    Start Date

    June 10 2021

    End Date

    July 22 2024

    Last Update

    September 5 2025

    Active Locations (1)

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    1

    University of North Carolina Chapel Hill

    Chapel Hill, North Carolina, United States, 27514