Primary Ciliary DyskinesiaNCT05287022
Use of Nasal Nitric Oxide Testing in Improving Primary Ciliary Dyskinesia Clinical Care
An observational study of Primary Ciliary Dyskinesia, run by Arkansas Children's Hospital Research Institute at 1 site in United States.
Trial summary
this study is aiming at learning more about primary ciliary dyskinesia (PCD) and tests that are used to diagnose this condition. One purpose of this study is to measure the level of nitric oxide in the nasal passages and examine how often the results correlate with other tests currently done to make the diagnosis. Read more about this study
- Who is it for?
- Ages 2 to 99 years
- Conditions the study is for
- Primary Ciliary Dyskinesia
- How many people
- 150 participants
- Where
- 1 siteUnited States
- Study dates
- 14 Jun 2021to Jun 2030
- Phase
- Observational
- Are there results?
- Not yet: the study is under way
Key requirements
Who can take part, as listed by the sponsor. Only the study team can confirm whether you are eligible, so take this list to your doctor.
- Ages 2 to 99 years
- All sexes
- No healthy volunteers
- Child, Adult, Older adult
Study population: Participants will include individuals who are diagnosed with PCD or undergoing work-up for primary ciliary dyskinesia based on their clinical symptoms as determined by the primary pulmonologist or other specialist
Who may be able to join
- Individuals who are diagnosed with Primary Ciliary Dyskinesia OR
- Individuals with abnormal PCD diagnostics (abnormal ciliary biopsy or PCD genetics) OR
- Individuals with unexplained bronchiectasis OR
- Individuals undergoing PCD diagnostic testing (ciliary biopsy, PCD genetic testing) or concern based on clinical symptoms (at least two of the following):
- Neonatal respiratory distress
- Organ laterality defects
Show all 13Show fewer
- Year-round cough starting in first year of life or bronchiectasis on chest CT
- Year-round nasal congestion starting in first year of life or pansinusitis
- Multiple ear infections in the first two years of life with sequelae (e.g. ear tubes, chronic effusion, abnormal audiological exam)
- History of recurrent pneumonias (at least 2 in one year or more than 3 at any time)
- Ability to provide consent for participation in study by the participants or guardian
- Ability to perform the test
- Age >= 2 years of age
Who cannot join
- Individuals who are unable to understand the requirements of the study.
- Individuals (or guardians) who are unwilling to provide consent.
- Individuals who are unable to complete the testing
- Recent history of sinus surgery (within four weeks) or bloody nose (within one week) of testing (they can be included at a later date)
- Patients who are currently being treated (within one week) with antibiotics for sinusitis or respiratory symptoms (they can be included at a later date)
- Age <2 years of age
Trial locations and contacts
1 site in 1 country. Contact the study team or a site near you to ask about taking part.
United States1 site, 1 recruiting
- Recruiting
Arkansas Children's Hospital
Little Rock, Arkansas, 72202
Zena Ghazala
About this study
this study is aiming at learning more about primary ciliary dyskinesia (PCD) and tests that are used to diagnose this condition. One purpose of this study is to measure the level of nitric oxide in the nasal passages and examine how often the results correlate with other tests currently done to make the diagnosis.
Read the detailed description
This will be an observational study. Participants who are referred by their clinician for nasal NO testing and meet the inclusion and exclusion criteria will be allowed to undergo testing. Informed consent will be completed prior to testing. The participant and/or guardian will be informed that this testing is not a clinically approved test at this time and thus it cannot confirm diagnosis of PCD. This test is to correlate with other diagnostic tests so that in the future it can be an approved diagnostic test. Data will be collected from the electronic medical records regarding participants.
The data collected will be stored on a REDCap (Research Electronic Data Capture) registry. The registry will contain participants' direct identifiers (like medical record). De-identified data will be used from the registry for monitoring, further analysis and presentations. The results of the testing may be included in the medical chart of each participant as part of the diagnosis discussion with family.
Consent forms will be obtained from participants >18 years of age or who are emancipated minors, or guardian for patients <18 years of age. Guardian refers to parent, guardian, or legally authorized personnel throughout this document.
The data collected will be used to refine and improve the diagnostic testing for PCD at Arkansas Children's Hospital. This will also involve collaboration with other sub-specialties like otolaryngology and allergy/immunology. With continued research regarding nasal nitric oxide testing, this may involve the use of this test as a screening tool for PCD in the right clinical setting. This will involve analyzing the association of nasal nitric oxide levels with approved diagnostic tests (ciliary biopsy and genetic testing).
Testing will be completed using (Eco-Physics Analyzer CLD 88 sp with DENOX 88). this testing will be done in the Arkansas Children's hospital pulmonary function laboratory, once all training is completed.
This equipment is not currently FDA approved for PCD diagnostic testing and therefore, referring clinicians will be informed of the results of this test and its limitations as a research tool will be discussed. However, there is growing evidence that this testing has utility as a non-invasive screening tool for PCD and continued data from the general pediatric pulmonary population is needed.
In addition to being non-invasive testing, it has very little risk as it simply monitors nasal nitric oxide levels in the sinus cavities during regular breathing without sedation. There is ongoing research to better determine normative values and thresholds for further PCD confirmatory testing, especially in the younger population. In the general population this testing has a greater negative predictive value than a positive predictive value. Consequently, the testing will be done on individuals who have at least two of the following clinical criteria for PCD: history of neonatal respiratory distress, chronic rhinitis, chronic cough, or situs defects.
- Conditions
- Primary Ciliary Dyskinesia
- Keywords
- nNO, Diagnosis, PCD
- Medical subject headings
- Ciliary Motility Disorders, Disease
What is being measured
The primary outcome is the main question the study is designed to answer.
Measuring nasal nitric oxide in comparison to ciliary biopsy and genetic testing for the diagnosis of PCD
Individuals with PCD have characteristically low values of nNO; values below an established cut-off of 77nL/min are strongly associated with PCD in compatible clinical settings. These values are not well established for individuals who are in 2-5 years of age. nNO measurements (in nL/min) in 2-5 years of age will be compared to the diagnosis of PCD and assessed to establish diagnostic cut-offs.
Measured: throughout the study, an average of 3 years
Secondary outcomes (1)
Comparing tidal breathing to resistor method
comparing nNO values in nL/min obtained via tidal breathing technique to those obtained via resistor method.
Measured: throughout the study, an average of 3 years
Timeline
From the start of the study to its completion; the mark is today. Estimated dates are the sponsor’s plan.
- First posted18 Mar 2022
- Study start14 Jun 2021
- Primary completionJun 2030 (estimated)
- Study completionJun 2030 (estimated)
Resources
- Official record on ClinicalTrials.govThe registry entry this page is built from, with its full change history.
- Publications (4)Papers the sponsor has linked to this study.
- Other trials for Primary Ciliary DyskinesiaStudies of the same condition, open and under way.
- What the trial phases meanHow a treatment moves from first tests in people to approval.
More details
For doctors and researchers: how the study is designed, who runs it, and its registry IDs.
Study design
- Study type
- Observational
- Observational model
- Other
- Time perspective
- Prospective
- Enrolment
- 150 participants (estimated)
Sponsor and investigators
- Lead sponsor
- Arkansas Children's Hospital Research Institute
- Sponsor type
- University, hospital or other
- Responsible party
- Sponsor
IDs, dates and publications
- ClinicalTrials.gov ID
- NCT05287022
- Sponsor's protocol ID
- 260432
- Status last verified
- Jan 2025
- First submitted
- 9 Apr 2021
- Last updated
- 30 Jan 2025
- Results posted
- No
- FDA-regulated drug
- No
- FDA-regulated device
- No
Publications
Shapiro AJ, Davis SD, Polineni D, Manion M, Rosenfeld M, Dell SD, Chilvers MA, Ferkol TW, Zariwala MA, Sagel SD, Josephson M, Morgan L, Yilmaz O, Olivier KN, Milla C, Pittman JE, Daniels MLA, Jones MH, Janahi IA, Ware SM, Daniel SJ, Cooper ML, Nogee LM, Anton B, Eastvold T, Ehrne L, Guadagno E, Knowles MR, Leigh MW, Lavergne V; American Thoracic Society Assembly on Pediatrics. Diagnosis of Primary Ciliary Dyskinesia. An Official American Thoracic Society Clinical Practice Guideline. Am J Respir Crit Care Med. 2018 Jun 15;197(12):e24-e39. doi: 10.1164/rccm.201805-0819ST.
PubMed 29905515Shapiro AJ, Zariwala MA, Ferkol T, Davis SD, Sagel SD, Dell SD, Rosenfeld M, Olivier KN, Milla C, Daniel SJ, Kimple AJ, Manion M, Knowles MR, Leigh MW; Genetic Disorders of Mucociliary Clearance Consortium. Diagnosis, monitoring, and treatment of primary ciliary dyskinesia: PCD foundation consensus recommendations based on state of the art review. Pediatr Pulmonol. 2016 Feb;51(2):115-32. doi: 10.1002/ppul.23304. Epub 2015 Sep 29.
PubMed 26418604Shapiro AJ, Dell SD, Gaston B, O'Connor M, Marozkina N, Manion M, Hazucha MJ, Leigh MW. Nasal Nitric Oxide Measurement in Primary Ciliary Dyskinesia. A Technical Paper on Standardized Testing Protocols. Ann Am Thorac Soc. 2020 Feb;17(2):e1-e12. doi: 10.1513/AnnalsATS.201904-347OT.
PubMed 31770003Collins SA, Behan L, Harris A, Gove K, Lucas JS. The dangers of widespread nitric oxide screening for primary ciliary dyskinesia. Thorax. 2016 Jun;71(6):560-1. doi: 10.1136/thoraxjnl-2015-208056. Epub 2016 Feb 19.
PubMed 26896442
Study at a glance
From the registry record, last updated by the sponsor on 30 Jan 2025. Recruitment status is as the sponsor reported it: confirm with the study team.
- Official title
- Use of Nasal Nitric Oxide Testing in Improving Primary Ciliary Dyskinesia Clinical Care
- ClinicalTrials.gov ID
- NCT05287022
- Acronym / sponsor ID
- 260432
- Conditions
- Primary Ciliary Dyskinesia
- Keywords
- nNO, Diagnosis, PCD
- Study type
- Observational
- Recruitment status
- Recruiting
- Enrolment
- 150 participants (estimated)
- Lead sponsor
- Arkansas Children's Hospital Research InstituteUniversity, hospital or other
- Study start
- 14 Jun 2021 (actual)
- Primary completion
- Jun 2030 (estimated)
- Study completion
- Jun 2030 (estimated)
- First posted
- 18 Mar 2022 (actual)
- Results
- Not posted in the source record
Clinical summary
The research question as the sponsor registered it. This describes what the study plans to test, not what it found.
this study is aiming at learning more about primary ciliary dyskinesia (PCD) and tests that are used to diagnose this condition. One purpose of this study is to measure the level of nitric oxide in the nasal passages and examine how often the results correlate with other tests currently done to make the diagnosis.
Detailed description
This will be an observational study. Participants who are referred by their clinician for nasal NO testing and meet the inclusion and exclusion criteria will be allowed to undergo testing. Informed consent will be completed prior to testing. The participant and/or guardian will be informed that this testing is not a clinically approved test at this time and thus it cannot confirm diagnosis of PCD. This test is to correlate with other diagnostic tests so that in the future it can be an approved diagnostic test. Data will be collected from the electronic medical records regarding participants.
The data collected will be stored on a REDCap (Research Electronic Data Capture) registry. The registry will contain participants' direct identifiers (like medical record). De-identified data will be used from the registry for monitoring, further analysis and presentations. The results of the testing may be included in the medical chart of each participant as part of the diagnosis discussion with family.
Consent forms will be obtained from participants >18 years of age or who are emancipated minors, or guardian for patients <18 years of age. Guardian refers to parent, guardian, or legally authorized personnel throughout this document.
The data collected will be used to refine and improve the diagnostic testing for PCD at Arkansas Children's Hospital. This will also involve collaboration with other sub-specialties like otolaryngology and allergy/immunology. With continued research regarding nasal nitric oxide testing, this may involve the use of this test as a screening tool for PCD in the right clinical setting. This will involve analyzing the association of nasal nitric oxide levels with approved diagnostic tests (ciliary biopsy and genetic testing).
Testing will be completed using (Eco-Physics Analyzer CLD 88 sp with DENOX 88). this testing will be done in the Arkansas Children's hospital pulmonary function laboratory, once all training is completed.
This equipment is not currently FDA approved for PCD diagnostic testing and therefore, referring clinicians will be informed of the results of this test and its limitations as a research tool will be discussed. However, there is growing evidence that this testing has utility as a non-invasive screening tool for PCD and continued data from the general pediatric pulmonary population is needed.
In addition to being non-invasive testing, it has very little risk as it simply monitors nasal nitric oxide levels in the sinus cavities during regular breathing without sedation. There is ongoing research to better determine normative values and thresholds for further PCD confirmatory testing, especially in the younger population. In the general population this testing has a greater negative predictive value than a positive predictive value. Consequently, the testing will be done on individuals who have at least two of the following clinical criteria for PCD: history of neonatal respiratory distress, chronic rhinitis, chronic cough, or situs defects.
Study design
- Observational model
- Other
- Time perspective
- Prospective
Who may qualify
- Ages
- 2 to 99 years
- Sex
- All
- Healthy volunteers
- Not accepted
- Age groups
- Child, Adult, Older adult
- Study population
- Participants will include individuals who are diagnosed with PCD or undergoing work-up for primary ciliary dyskinesia based on their clinical symptoms as determined by the primary pulmonologist or other specialist
Key inclusion criteria (13 listed)
- Individuals who are diagnosed with Primary Ciliary Dyskinesia OR
- Individuals with abnormal PCD diagnostics (abnormal ciliary biopsy or PCD genetics) OR
- Individuals with unexplained bronchiectasis OR
- Individuals undergoing PCD diagnostic testing (ciliary biopsy, PCD genetic testing) or concern based on clinical symptoms (at least two of the following):
- Ability to provide consent for participation in study by the participants or guardian
Key exclusion criteria (6 listed)
- Individuals who are unable to understand the requirements of the study.
- Individuals (or guardians) who are unwilling to provide consent.
- Individuals who are unable to complete the testing
- Recent history of sinus surgery (within four weeks) or bloody nose (within one week) of testing (they can be included at a later date)
- Patients who are currently being treated (within one week) with antibiotics for sinusitis or respiratory symptoms (they can be included at a later date)
Eligibility is decided by the study team after screening; this overview does not establish that a patient qualifies. Full inclusion and exclusion criteria.
Primary outcome
Measuring nasal nitric oxide in comparison to ciliary biopsy and genetic testing for the diagnosis of PCD
Time frame: throughout the study, an average of 3 years
Individuals with PCD have characteristically low values of nNO; values below an established cut-off of 77nL/min are strongly associated with PCD in compatible clinical settings. These values are not well established for individuals who are in 2-5 years of age. nNO measurements (in nL/min) in 2-5 years of age will be compared to the diagnosis of PCD and assessed to establish diagnostic cut-offs.
1 secondary outcome
Comparing tidal breathing to resistor method
Time frame: throughout the study, an average of 3 years
Results and safety
This study is still under way. Results are not available in the source record yet.
Research links
- Official record on ClinicalTrials.govLast updated by the sponsor 30 Jan 2025.
- Background: PubMed 29905515Shapiro AJ, Davis SD, Polineni D, Manion M, Rosenfeld M, Dell SD, Chilvers MA, Ferkol TW, Zariwala MA, Sagel SD, Josephson M, Morgan L, Yilmaz O, Olivier KN, Milla C, Pittman JE, Daniels MLA, Jones MH, Janahi IA, Ware SM, Daniel SJ, Cooper ML, Nogee LM, Anton B, Eastvold T, Ehrne L, Guadagno E, Knowles MR, Leigh MW, Lavergne V; American Thoracic Society Assembly on Pediatrics. Diagnosis of Primary Ciliary Dyskinesia. An Official American Thoracic Society Clinical Practice Guideline. Am J Respir Crit Care Med. 2018 Jun 15;197(12):e24-e39. doi: 10.1164/rccm.201805-0819ST.
- Background: PubMed 26418604Shapiro AJ, Zariwala MA, Ferkol T, Davis SD, Sagel SD, Dell SD, Rosenfeld M, Olivier KN, Milla C, Daniel SJ, Kimple AJ, Manion M, Knowles MR, Leigh MW; Genetic Disorders of Mucociliary Clearance Consortium. Diagnosis, monitoring, and treatment of primary ciliary dyskinesia: PCD foundation consensus recommendations based on state of the art review. Pediatr Pulmonol. 2016 Feb;51(2):115-32. doi: 10.1002/ppul.23304. Epub 2015 Sep 29.
- Background: PubMed 31770003Shapiro AJ, Dell SD, Gaston B, O'Connor M, Marozkina N, Manion M, Hazucha MJ, Leigh MW. Nasal Nitric Oxide Measurement in Primary Ciliary Dyskinesia. A Technical Paper on Standardized Testing Protocols. Ann Am Thorac Soc. 2020 Feb;17(2):e1-e12. doi: 10.1513/AnnalsATS.201904-347OT.
- Background: PubMed 26896442Collins SA, Behan L, Harris A, Gove K, Lucas JS. The dangers of widespread nitric oxide screening for primary ciliary dyskinesia. Thorax. 2016 Jun;71(6):560-1. doi: 10.1136/thoraxjnl-2015-208056. Epub 2016 Feb 19.
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Referral and contact
AzTrials does not enrol participants or take referrals. Contact the study team or a site listed below; they decide who can take part.
1 site in 1 country: see every site and its status.
Questions about this trial
What is this clinical trial studying?
An observational study of Primary Ciliary Dyskinesia, run by Arkansas Children's Hospital Research Institute at 1 site in United States.
The full registered description is in the About this study section and the official record.
Who may be eligible to take part?
The sponsor lists 13 inclusion and 6 exclusion criteria. Eligibility is decided by the study team after screening, so read the full criteria and contact a listed site.
Is this clinical trial currently recruiting?
The record says: looking for participants now, as of the sponsor's last update on 30 Jan 2025. Status can change between updates, so confirm with the study contact before making plans.
Where is the study taking place?
The record lists 1 site in United States. Whether a site is taking part right now should be confirmed with that site. See every site.
What are the possible benefits and risks?
They depend on the study and the treatment, and no study can promise a benefit. The study team explains the known risks during informed consent, before anyone joins; the record itself does not list them.
Are results available for this study?
This study is still under way. Results are not available in the source record yet.
How can a doctor refer a patient or contact the research team?
Contact Zena Ghazala, MD (5013641006, zbghazala@uams.edu), or a site near the patient. AzTrials does not enrol participants or take referrals.
What does the primary outcome measure?
Measuring nasal nitric oxide in comparison to ciliary biopsy and genetic testing for the diagnosis of PCD (time frame: throughout the study, an average of 3 years). It is what the study was designed to measure, not a finding.
Where can I read the original study information?
On the ClinicalTrials.gov record for NCT05287022, which also lists 4 linked publications.
Does a listing mean the treatment is approved or proven to work?
No. A listing means the study is registered. It does not show that the treatment is safe, works, or is approved by a regulator.
How often is this information updated?
AzTrials copies changed records from ClinicalTrials.gov every night and reloads the whole registry every week. The sponsor last updated this record on 30 Jan 2025.