The impact of highly effective modulator therapy on sinusitis and dysosmia in young children with cystic fibrosis: a prospective study protocol

Background Chronic rhinosinusitis (CRS) and olfactory dysfunction (OD) are prevalent disease complications in people with cystic fibrosis. These understudied comorbidities significantly impact quality of life. The impact of highly effective modulator therapy (HEMT) in young children with cystic fibr...

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Main Authors: Christine M. Liu, Jakob L. Fischer, Edith T. Zemanick, Jason C. Woods, Karolin K. Markarian, Sean B. Fain, Deborah Froh, Sonya L. Heltshe, Lucas R. Hoffman, Stephen M. Humphries, Elizabeth L. Kramer, Katie Larson Ode, Michael Lewis, Douglas A. Li, Jaime Mata, Sarah S. Milla, Peter J. Niedbalski, Benjamin D. Sawatzky, Myung-Shin Sim, Jillian S. Sullivan, Andrew T. Trout, Christopher H. Goss, Jennifer L. Taylor-Cousar, Daniel M. Beswick
Format: Article
Language:English
Published: European Respiratory Society 2025-01-01
Series:ERJ Open Research
Online Access:http://openres.ersjournals.com/content/11/1/00137-2024.full
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author Christine M. Liu
Jakob L. Fischer
Edith T. Zemanick
Jason C. Woods
Karolin K. Markarian
Sean B. Fain
Deborah Froh
Sonya L. Heltshe
Lucas R. Hoffman
Stephen M. Humphries
Elizabeth L. Kramer
Katie Larson Ode
Michael Lewis
Douglas A. Li
Jaime Mata
Sarah S. Milla
Peter J. Niedbalski
Benjamin D. Sawatzky
Myung-Shin Sim
Jillian S. Sullivan
Andrew T. Trout
Christopher H. Goss
Jennifer L. Taylor-Cousar
Daniel M. Beswick
author_facet Christine M. Liu
Jakob L. Fischer
Edith T. Zemanick
Jason C. Woods
Karolin K. Markarian
Sean B. Fain
Deborah Froh
Sonya L. Heltshe
Lucas R. Hoffman
Stephen M. Humphries
Elizabeth L. Kramer
Katie Larson Ode
Michael Lewis
Douglas A. Li
Jaime Mata
Sarah S. Milla
Peter J. Niedbalski
Benjamin D. Sawatzky
Myung-Shin Sim
Jillian S. Sullivan
Andrew T. Trout
Christopher H. Goss
Jennifer L. Taylor-Cousar
Daniel M. Beswick
author_sort Christine M. Liu
collection DOAJ
description Background Chronic rhinosinusitis (CRS) and olfactory dysfunction (OD) are prevalent disease complications in people with cystic fibrosis. These understudied comorbidities significantly impact quality of life. The impact of highly effective modulator therapy (HEMT) in young children with cystic fibrosis (YCwCF) on these disease complications is unknown. This proposed study aims to characterise CRS and OD in YCwCF and assess the efficacy of HEMT in improving sinus and olfactory health in this young age group. Methods This six-centre, prospective, observational study will enrol 80 YCwCF aged 2–8 years. Patients are divided into two groups: those receiving HEMT and those not on HEMT based on clinical indication. Both groups undergo sinus magnetic resonance imaging, psychophysical olfactory tests, and complete patient- or parent-reported quality of life surveys over 2 years. Outcomes will be compared before and after initiation of HEMT and between groups. Ethical approval has been obtained for all sites, and this study has been registered on ClinicalTrials.gov (NCT06191640). Results Enrolment began in April 2023. 21 participants have been enrolled as of October 2023 with ongoing enrolment at all sites. Conclusion This investigation is expected to provide critical insights into the potential benefits of early HEMT initiation in managing CRS and OD in YCwCF. It will assist in developing targeted interventions and contribute to the understanding of HEMT's role in altering the disease course in this demographic.
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spelling doaj-art-13ece56839174cbca6340602165b2d722025-08-20T03:02:16ZengEuropean Respiratory SocietyERJ Open Research2312-05412025-01-0111110.1183/23120541.00137-202400137-2024The impact of highly effective modulator therapy on sinusitis and dysosmia in young children with cystic fibrosis: a prospective study protocolChristine M. Liu0Jakob L. Fischer1Edith T. Zemanick2Jason C. Woods3Karolin K. Markarian4Sean B. Fain5Deborah Froh6Sonya L. Heltshe7Lucas R. Hoffman8Stephen M. Humphries9Elizabeth L. Kramer10Katie Larson Ode11Michael Lewis12Douglas A. Li13Jaime Mata14Sarah S. Milla15Peter J. Niedbalski16Benjamin D. Sawatzky17Myung-Shin Sim18Jillian S. Sullivan19Andrew T. Trout20Christopher H. Goss21Jennifer L. Taylor-Cousar22Daniel M. Beswick23 Department of Otolaryngology-Head and Neck Surgery, University of California, Los Angeles, Los Angeles, CA, USA Department of Otolaryngology-Head and Neck Surgery, University of California, Los Angeles, Los Angeles, CA, USA Department of Pediatrics, University of Colorado School of Medicine, Aurora, CO, USA Department of Pediatrics, Cincinnati Children's Hospital Medical Center, Cincinnati, OH, USA University of California, Los Angeles, CTSI, David Geffen School of Medicine, Los Angeles, CA, USA Department of Radiology, University of Iowa, Iowa City, IA, USA Department of Pediatrics, University of Virginia, Charlottesville, VA, USA Department of Pediatrics, University of Washington and Seattle Children's Research Institute, Seattle, WA, USA Department of Pediatrics, University of Washington and Seattle Children's Research Institute, Seattle, WA, USA Department of Radiology, National Jewish Health, Aurora, CO, USA Department of Pediatrics, Cincinnati Children's Hospital Medical Center, Cincinnati, OH, USA Department of Pediatrics, University of Iowa, Iowa City, IA, USA Department of Pediatrics, University of Kansas Medical Center, Kansas City, KS, USA Department of Pediatrics, University of California, Los Angeles, Los Angeles, CA, USA Department of Radiology and Medical Imaging, University of Virginia, Charlottesville, VA, USA Department of Radiology, Children's Hospital Colorado, Aurora, CO, USA Department of Pulmonary, Critical Care, and Sleep Medicine, University of Kansas Medical Center, Kansas City, KS, USA Department of Radiology, University of Vermont, Colchester, VT, USA Department of Pediatrics, University of California, Los Angeles, Los Angeles, CA, USA Department of Pediatrics, University of Vermont, Colchester, VT, USA Department of Pediatrics, Cincinnati Children's Hospital Medical Center, Cincinnati, OH, USA Department of Medicine and Pediatrics, University of Washington, Seattle, WA, USA Department of Medicine, National Jewish Health, Aurora, CO, USA Department of Otolaryngology-Head and Neck Surgery, University of California, Los Angeles, Los Angeles, CA, USA Background Chronic rhinosinusitis (CRS) and olfactory dysfunction (OD) are prevalent disease complications in people with cystic fibrosis. These understudied comorbidities significantly impact quality of life. The impact of highly effective modulator therapy (HEMT) in young children with cystic fibrosis (YCwCF) on these disease complications is unknown. This proposed study aims to characterise CRS and OD in YCwCF and assess the efficacy of HEMT in improving sinus and olfactory health in this young age group. Methods This six-centre, prospective, observational study will enrol 80 YCwCF aged 2–8 years. Patients are divided into two groups: those receiving HEMT and those not on HEMT based on clinical indication. Both groups undergo sinus magnetic resonance imaging, psychophysical olfactory tests, and complete patient- or parent-reported quality of life surveys over 2 years. Outcomes will be compared before and after initiation of HEMT and between groups. Ethical approval has been obtained for all sites, and this study has been registered on ClinicalTrials.gov (NCT06191640). Results Enrolment began in April 2023. 21 participants have been enrolled as of October 2023 with ongoing enrolment at all sites. Conclusion This investigation is expected to provide critical insights into the potential benefits of early HEMT initiation in managing CRS and OD in YCwCF. It will assist in developing targeted interventions and contribute to the understanding of HEMT's role in altering the disease course in this demographic.http://openres.ersjournals.com/content/11/1/00137-2024.full
spellingShingle Christine M. Liu
Jakob L. Fischer
Edith T. Zemanick
Jason C. Woods
Karolin K. Markarian
Sean B. Fain
Deborah Froh
Sonya L. Heltshe
Lucas R. Hoffman
Stephen M. Humphries
Elizabeth L. Kramer
Katie Larson Ode
Michael Lewis
Douglas A. Li
Jaime Mata
Sarah S. Milla
Peter J. Niedbalski
Benjamin D. Sawatzky
Myung-Shin Sim
Jillian S. Sullivan
Andrew T. Trout
Christopher H. Goss
Jennifer L. Taylor-Cousar
Daniel M. Beswick
The impact of highly effective modulator therapy on sinusitis and dysosmia in young children with cystic fibrosis: a prospective study protocol
ERJ Open Research
title The impact of highly effective modulator therapy on sinusitis and dysosmia in young children with cystic fibrosis: a prospective study protocol
title_full The impact of highly effective modulator therapy on sinusitis and dysosmia in young children with cystic fibrosis: a prospective study protocol
title_fullStr The impact of highly effective modulator therapy on sinusitis and dysosmia in young children with cystic fibrosis: a prospective study protocol
title_full_unstemmed The impact of highly effective modulator therapy on sinusitis and dysosmia in young children with cystic fibrosis: a prospective study protocol
title_short The impact of highly effective modulator therapy on sinusitis and dysosmia in young children with cystic fibrosis: a prospective study protocol
title_sort impact of highly effective modulator therapy on sinusitis and dysosmia in young children with cystic fibrosis a prospective study protocol
url http://openres.ersjournals.com/content/11/1/00137-2024.full
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