Evidence map›Paper›PMID 42053522›Full record

ReviewAmerican journal of physiology. Lung cellular and molecular physiology2026

Considerations for early life genetic therapies in cystic fibrosis.

Ashley L Cooney, Ian M Thornell, Alejandro A Pezzulo, Anthony J Fischer, Daniel P Cook, Mahmoud H Abou Alaiwa, Katie M Larson Ode, Aliye Uc, Joseph Zabner, Amy L Ryan and 4 more

Abstract readReview
In one paragraph

Review in American journal of physiology. Lung cellular and molecular physiology, 2026. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 1 paper.

0numbers the graph read from it
0cells of the map it votes in
1citing papers in PubMed
–field-weighted citation impact
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

1 citing paper in PubMed.

  1. Review
4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

14 authors.

Ashley L CooneyDepartment of Pediatrics, Roy J and Lucille A. Carver College of Medicine, University of Iowa, Iowa City, Iowa, United States.ORCID 0000-0003-2717-9138
Ian M ThornellDepartment of Internal Medicine, Roy J and Lucille A. Carver College of Medicine, University of Iowa, Iowa City, Iowa, United States.ORCID 0000-0001-5535-2925
Alejandro A PezzuloDepartment of Internal Medicine, Roy J and Lucille A. Carver College of Medicine, University of Iowa, Iowa City, Iowa, United States.ORCID 0000-0001-7544-5109
Anthony J FischerDepartment of Pediatrics, Roy J and Lucille A. Carver College of Medicine, University of Iowa, Iowa City, Iowa, United States.
Daniel P CookDepartment of Internal Medicine, Roy J and Lucille A. Carver College of Medicine, University of Iowa, Iowa City, Iowa, United States.ORCID 0000-0002-1152-4454
Mahmoud H Abou AlaiwaDepartment of Internal Medicine, Roy J and Lucille A. Carver College of Medicine, University of Iowa, Iowa City, Iowa, United States.ORCID 0000-0003-4525-0217
Katie M Larson OdeDepartment of Pediatrics, Roy J and Lucille A. Carver College of Medicine, University of Iowa, Iowa City, Iowa, United States.
Aliye UcDepartment of Pediatrics, Roy J and Lucille A. Carver College of Medicine, University of Iowa, Iowa City, Iowa, United States.
Joseph ZabnerDepartment of Internal Medicine, Roy J and Lucille A. Carver College of Medicine, University of Iowa, Iowa City, Iowa, United States.ORCID 0000-0002-9606-1339
Amy L RyanDepartment of Internal Medicine, Roy J and Lucille A. Carver College of Medicine, University of Iowa, Iowa City, Iowa, United States.ORCID 0000-0003-1363-905X
David A StoltzDepartment of Internal Medicine, Roy J and Lucille A. Carver College of Medicine, University of Iowa, Iowa City, Iowa, United States.ORCID 0000-0003-0893-9015
Patrick L SinnDepartment of Pediatrics, Roy J and Lucille A. Carver College of Medicine, University of Iowa, Iowa City, Iowa, United States.ORCID 0000-0002-9746-966X
Paul B McCrayDepartment of Pediatrics, Roy J and Lucille A. Carver College of Medicine, University of Iowa, Iowa City, Iowa, United States.
David K MeyerholzDepartment of Pathology, Roy J and Lucille A. Carver College of Medicine, University of Iowa, Iowa City, Iowa, United States.ORCID 0000-0003-1552-3253

Funding

Respiratory virus infection in early cystic fibrosis lung diseaseP01HL091842 · NHLBI · UNIVERSITY OF IOWA · PI ZABNER, JOSEPH · 2008 to 2022
$34.2M
Vector Core-Core 2P30DK054759 · NIDDK · UNIVERSITY OF IOWA · PI Alejandro Antonio Pezzulo · 1998 to 2026
$30.5M
Pathology CoreP01HL152960 · NHLBI · UNIVERSITY OF IOWA · PI ENGELHARDT, JOHN F · 2020 to 2024
$11.6M
Life-long phenotypic correction of CF airwaysR01HL133089 · NHLBI · UNIVERSITY OF IOWA · PI PATRICK L SINN · 2017 to 2026
$5.3M
Adenine Base Edited Correction of Cystic Fibrosis AirwaysR01HL171035 · NHLBI · UNIVERSITY OF IOWA · PI PAUL B MCCRAY, PATRICK L SINN · 2024 to 2026
$1.9M
Receptor-mediated Control of Pulmonary IonocytesR01HL173260 · NHLBI · UNIVERSITY OF IOWA · PI Ian M Thornell · 2025 to 2026
$1.1M
Inhibiting CD4+ Th2 development and function by CFTR activity.K08AI181763 · NIAID · VANDERBILT UNIVERSITY MEDICAL CENTER · PI Daniel Paul Cook · 2024 to 2026
$553k
Cystic Fibrosis Foundation (CFF) COOK20L0Cystic Fibrosis Foundation (CFF) COOK24A0-KBCystic Fibrosis Foundation (CFF) COOK25R3Cystic Fibrosis Foundation (CFF) FISCHE24Y0Cystic Fibrosis Foundation (CFF) FISCHE25G0Cystic Fibrosis Foundation (CFF) SINN25G0Cystic Fibrosis Foundation (CFF) STOLTZ24R0Cystic Fibrosis Foundation (CFF) THORNE24G0HHS | NIH | National Heart, Lung, and Blood Institute (NHLBI) HL133089HHS | NIH | National Heart, Lung, and Blood Institute (NHLBI) HL171035HHS | NIH | National Heart, Lung, and Blood Institute (NHLBI) K08AI181763HHS | NIH | National Heart, Lung, and Blood Institute (NHLBI) P01 HL91842NHLBI NIH HHS P01 HL091842NHLBI NIH HHS P01 HL152960NHLBI NIH HHS R01 HL133089NHLBI NIH HHS R01 HL171035NHLBI NIH HHS R01 HL173260NIAID NIH HHS K08 AI181763NIDDK NIH HHS P30 DK054759
6 · The paper itself

Abstract

Cystic fibrosis (CF) is caused by mutations in the gene encoding for the CF transmembrane conductance regulator (CFTR) anion channel. Since the initial characterization of CF in the early 20th century, advances in clinical management have reduced disease burden and increased longevity. These advances are amplified by the recent development of highly effective CFTR modulator therapies (HEMTs) that have provided remarkable clinical results in people with CF (pwCF). However, some pwCF do not benefit from HEMT due to several limiting factors. Genetic therapies have increasingly emerged as prospective, complementary treatments to HEMTs for those unable to benefit from HEMTs. Genetic therapies have yet to be approved for clinical use in pwCF, but multiple clinical trials are in progress. In anticipation of future approval of one or more of these candidates, it is essential to identify and discuss how these genetic therapies might be used in early life, when many CF lesions originate, especially for those with severe mutations. In this multidisciplinary review, we discuss several pertinent factors such as CFTR localization and function, CF disease origins, emerging developments in genetic therapies, ethical considerations, and other perspectives needed to guide future genetic therapies in early life.

Indexed as

Cystic FibrosisCystic Fibrosis Transmembrane Conductance RegulatorGenetic TherapyAnimalsHumansMutationCFTR protein, humanCystic Fibrosis Transmembrane Conductance Regulatorcystic fibrosisearly lifefetalgenetic therapypediatric

Identifiers

PMID42053522
PMCPMC13262679

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Registered trials

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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.