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Identification of genetic variants in CFAP221 as a cause of primary ciliary dyskinesia.

Bustamante-Marin XM, Shapiro A, Sears PR, Charng WL, Conrad DF, Leigh MW, Knowles MR, Ostrowski LE, Zariwala MA.

J Hum Genet. 2019 Oct 21. doi: 10.1038/s10038-019-0686-1. [Epub ahead of print]


Mice with a Deletion of Rsph1 Exhibit a Low Level of Mucociliary Clearance and Develop a Primary Ciliary Dyskinesia Phenotype.

Yin W, Livraghi-Butrico A, Sears PR, Rogers TD, Burns KA, Grubb BR, Ostrowski LE.

Am J Respir Cell Mol Biol. 2019 Sep;61(3):312-321. doi: 10.1165/rcmb.2017-0387OC.


Lack of GAS2L2 Causes PCD by Impairing Cilia Orientation and Mucociliary Clearance.

Bustamante-Marin XM, Yin WN, Sears PR, Werner ME, Brotslaw EJ, Mitchell BJ, Jania CM, Zeman KL, Rogers TD, Herring LE, Refabért L, Thomas L, Amselem S, Escudier E, Legendre M, Grubb BR, Knowles MR, Zariwala MA, Ostrowski LE.

Am J Hum Genet. 2019 Feb 7;104(2):229-245. doi: 10.1016/j.ajhg.2018.12.009. Epub 2019 Jan 18.


Mucociliary Clearance in Mice Measured by Tracking Trans-tracheal Fluorescence of Nasally Aerosolized Beads.

Rogers TD, Ostrowski LE, Livraghi-Butrico A, Button B, Grubb BR.

Sci Rep. 2018 Oct 3;8(1):14744. doi: 10.1038/s41598-018-33053-2.


Primary ciliary dyskinesia: keep it on your radar.

Rosenfeld M, Ostrowski LE, Zariwala MA.

Thorax. 2018 Feb;73(2):101-102. doi: 10.1136/thoraxjnl-2017-210776. Epub 2017 Nov 13. No abstract available.


Direct monitoring of pulmonary disease treatment biomarkers using plasmonic gold nanorods with diffusion-sensitive OCT.

Blackmon RL, Kreda SM, Sears PR, Chapman BS, Hill DB, Tracy JB, Ostrowski LE, Oldenburg AL.

Nanoscale. 2017 Apr 13;9(15):4907-4917. doi: 10.1039/c7nr00376e.


Quantitative Proteomic Analysis of Human Airway Cilia Identifies Previously Uncharacterized Proteins of High Abundance.

Blackburn K, Bustamante-Marin X, Yin W, Goshe MB, Ostrowski LE.

J Proteome Res. 2017 Apr 7;16(4):1579-1592. doi: 10.1021/acs.jproteome.6b00972. Epub 2017 Mar 27.


High-speed and high-sensitivity parallel spectral-domain optical coherence tomography using a supercontinuum light source.

Barrick J, Doblas A, Gardner MR, Sears PR, Ostrowski LE, Oldenburg AL.

Opt Lett. 2016 Dec 15;41(24):5620-5623. doi: 10.1364/OL.41.005620.


Cilia and Mucociliary Clearance.

Bustamante-Marin XM, Ostrowski LE.

Cold Spring Harb Perspect Biol. 2017 Apr 3;9(4). pii: a028241. doi: 10.1101/cshperspect.a028241. Review.


Diffusion-sensitive optical coherence tomography for real-time monitoring of mucus thinning treatments.

Blackmon RL, Kreda SM, Sears PR, Ostrowski LE, Hill DB, Chapman BS, Tracy JB, Oldenburg AL.

Proc SPIE Int Soc Opt Eng. 2016;9697. pii: 969724.


Reduced mucociliary clearance in old mice is associated with a decrease in Muc5b mucin.

Grubb BR, Livraghi-Butrico A, Rogers TD, Yin W, Button B, Ostrowski LE.

Am J Physiol Lung Cell Mol Physiol. 2016 May 1;310(9):L860-7. doi: 10.1152/ajplung.00015.2016. Epub 2016 Mar 11.


The dual phosphodiesterase 3 and 4 inhibitor RPL554 stimulates CFTR and ciliary beating in primary cultures of bronchial epithelia.

Turner MJ, Matthes E, Billet A, Ferguson AJ, Thomas DY, Randell SH, Ostrowski LE, Abbott-Banner K, Hanrahan JW.

Am J Physiol Lung Cell Mol Physiol. 2016 Jan 1;310(1):L59-70. doi: 10.1152/ajplung.00324.2015. Epub 2015 Nov 6.


Continuous mucociliary transport by primary human airway epithelial cells in vitro.

Sears PR, Yin WN, Ostrowski LE.

Am J Physiol Lung Cell Mol Physiol. 2015 Jul 15;309(2):L99-108. doi: 10.1152/ajplung.00024.2015. Epub 2015 May 15.


Cryo-electron tomography reveals ciliary defects underlying human RSPH1 primary ciliary dyskinesia.

Lin J, Yin W, Smith MC, Song K, Leigh MW, Zariwala MA, Knowles MR, Ostrowski LE, Nicastro D.

Nat Commun. 2014 Dec 4;5:5727. doi: 10.1038/ncomms6727.


Genetic regulation of Zfp30, CXCL1, and neutrophilic inflammation in murine lung.

Rutledge H, Aylor DL, Carpenter DE, Peck BC, Chines P, Ostrowski LE, Chesler EJ, Churchill GA, de Villena FP, Kelada SN.

Genetics. 2014 Oct;198(2):735-45. doi: 10.1534/genetics.114.168138. Epub 2014 Aug 11.


Mutations in RSPH1 cause primary ciliary dyskinesia with a unique clinical and ciliary phenotype.

Knowles MR, Ostrowski LE, Leigh MW, Sears PR, Davis SD, Wolf WE, Hazucha MJ, Carson JL, Olivier KN, Sagel SD, Rosenfeld M, Ferkol TW, Dell SD, Milla CE, Randell SH, Yin W, Sannuti A, Metjian HM, Noone PG, Noone PJ, Olson CA, Patrone MV, Dang H, Lee HS, Hurd TW, Gee HY, Otto EA, Halbritter J, Kohl S, Kircher M, Krischer J, Bamshad MJ, Nickerson DA, Hildebrandt F, Shendure J, Zariwala MA.

Am J Respir Crit Care Med. 2014 Mar 15;189(6):707-17. doi: 10.1164/rccm.201311-2047OC.


Restoring ciliary function to differentiated primary ciliary dyskinesia cells with a lentiviral vector.

Ostrowski LE, Yin W, Patel M, Sechelski J, Rogers T, Burns K, Grubb BR, Olsen JC.

Gene Ther. 2014 Mar;21(3):253-61. doi: 10.1038/gt.2013.79. Epub 2014 Jan 23.


Mutations in SPAG1 cause primary ciliary dyskinesia associated with defective outer and inner dynein arms.

Knowles MR, Ostrowski LE, Loges NT, Hurd T, Leigh MW, Huang L, Wolf WE, Carson JL, Hazucha MJ, Yin W, Davis SD, Dell SD, Ferkol TW, Sagel SD, Olivier KN, Jahnke C, Olbrich H, Werner C, Raidt J, Wallmeier J, Pennekamp P, Dougherty GW, Hjeij R, Gee HY, Otto EA, Halbritter J, Chaki M, Diaz KA, Braun DA, Porath JD, Schueler M, Baktai G, Griese M, Turner EH, Lewis AP, Bamshad MJ, Nickerson DA, Hildebrandt F, Shendure J, Omran H, Zariwala MA.

Am J Hum Genet. 2013 Oct 3;93(4):711-20. doi: 10.1016/j.ajhg.2013.07.025. Epub 2013 Sep 19.


Exome sequencing identifies mutations in CCDC114 as a cause of primary ciliary dyskinesia.

Knowles MR, Leigh MW, Ostrowski LE, Huang L, Carson JL, Hazucha MJ, Yin W, Berg JS, Davis SD, Dell SD, Ferkol TW, Rosenfeld M, Sagel SD, Milla CE, Olivier KN, Turner EH, Lewis AP, Bamshad MJ, Nickerson DA, Shendure J, Zariwala MA; Genetic Disorders of Mucociliary Clearance Consortium.

Am J Hum Genet. 2013 Jan 10;92(1):99-106. doi: 10.1016/j.ajhg.2012.11.003. Epub 2012 Dec 20.


Computer-assisted image analysis of human cilia and Chlamydomonas flagella reveals both similarities and differences in axoneme structure.

O'Toole ET, Giddings TH Jr, Porter ME, Ostrowski LE.

Cytoskeleton (Hoboken). 2012 Aug;69(8):577-90. doi: 10.1002/cm.21035. Epub 2012 May 22.


Transgenic hCFTR expression fails to correct β-ENaC mouse lung disease.

Grubb BR, O'Neal WK, Ostrowski LE, Kreda SM, Button B, Boucher RC.

Am J Physiol Lung Cell Mol Physiol. 2012 Jan 15;302(2):L238-47. doi: 10.1152/ajplung.00083.2011. Epub 2011 Oct 14.


Cilia and models for studying structure and function.

Ostrowski LE, Dutcher SK, Lo CW.

Proc Am Thorac Soc. 2011 Sep;8(5):423-9. doi: 10.1513/pats.201103-027SD. Review.


Tubulin polyglutamylation is essential for airway ciliary function through the regulation of beating asymmetry.

Ikegami K, Sato S, Nakamura K, Ostrowski LE, Setou M.

Proc Natl Acad Sci U S A. 2010 Jun 8;107(23):10490-5. doi: 10.1073/pnas.1002128107. Epub 2010 May 24.


FoxJ1-dependent gene expression is required for differentiation of radial glia into ependymal cells and a subset of astrocytes in the postnatal brain.

Jacquet BV, Salinas-Mondragon R, Liang H, Therit B, Buie JD, Dykstra M, Campbell K, Ostrowski LE, Brody SL, Ghashghaei HT.

Development. 2009 Dec;136(23):4021-31. doi: 10.1242/dev.041129.


Conditional deletion of dnaic1 in a murine model of primary ciliary dyskinesia causes chronic rhinosinusitis.

Ostrowski LE, Yin W, Rogers TD, Busalacchi KB, Chua M, O'Neal WK, Grubb BR.

Am J Respir Cell Mol Biol. 2010 Jul;43(1):55-63. doi: 10.1165/rcmb.2009-0118OC. Epub 2009 Aug 12.


Ion transport across CF and normal murine olfactory and ciliated epithelium.

Grubb BR, Rogers TD, Boucher RC, Ostrowski LE.

Am J Physiol Cell Physiol. 2009 Jun;296(6):C1301-9. doi: 10.1152/ajpcell.00578.2008. Epub 2009 Mar 25.


Expression of CFTR from a ciliated cell-specific promoter is ineffective at correcting nasal potential difference in CF mice.

Ostrowski LE, Yin W, Diggs PS, Rogers TD, O'Neal WK, Grubb BR.

Gene Ther. 2007 Oct;14(20):1492-501. Epub 2007 Jul 19.


Olfactory epithelia exhibit progressive functional and morphological defects in CF mice.

Grubb BR, Rogers TD, Kulaga HM, Burns KA, Wonsetler RL, Reed RR, Ostrowski LE.

Am J Physiol Cell Physiol. 2007 Aug;293(2):C574-83. Epub 2007 Apr 11.


Lung development and repair: contribution of the ciliated lineage.

Rawlins EL, Ostrowski LE, Randell SH, Hogan BL.

Proc Natl Acad Sci U S A. 2007 Jan 9;104(2):410-7. Epub 2006 Dec 28.


Primary ciliary dyskinesia and upper airway diseases.

Kennedy MP, Ostrowski LE.

Curr Allergy Asthma Rep. 2006 Nov;6(6):513-7. Review.


Real-time analysis of cAMP-mediated regulation of ciliary motility in single primary human airway epithelial cells.

Schmid A, Bai G, Schmid N, Zaccolo M, Ostrowski LE, Conner GE, Fregien N, Salathe M.

J Cell Sci. 2006 Oct 15;119(Pt 20):4176-86. Epub 2006 Sep 19.


Culture of murine nasal epithelia: model for cystic fibrosis.

Grubb BR, Rogers TD, Diggs PC, Boucher RC, Ostrowski LE.

Am J Physiol Lung Cell Mol Physiol. 2006 Feb;290(2):L270-7. Epub 2005 Sep 9.


Voltage-dependent anion channel-1 (VDAC-1) contributes to ATP release and cell volume regulation in murine cells.

Okada SF, O'Neal WK, Huang P, Nicholas RA, Ostrowski LE, Craigen WJ, Lazarowski ER, Boucher RC.

J Gen Physiol. 2004 Nov;124(5):513-26. Epub 2004 Oct 11.


Investigation of the possible role of a novel gene, DPCD, in primary ciliary dyskinesia.

Zariwala M, O'Neal WK, Noone PG, Leigh MW, Knowles MR, Ostrowski LE.

Am J Respir Cell Mol Biol. 2004 Apr;30(4):428-34. Epub 2003 Nov 20.


Targeting expression of a transgene to the airway surface epithelium using a ciliated cell-specific promoter.

Ostrowski LE, Hutchins JR, Zakel K, O'Neal WK.

Mol Ther. 2003 Oct;8(4):637-45.


Characterization of an A-kinase anchoring protein in human ciliary axonemes.

Kultgen PL, Byrd SK, Ostrowski LE, Milgram SL.

Mol Biol Cell. 2002 Dec;13(12):4156-66.


A proteomic analysis of human cilia: identification of novel components.

Ostrowski LE, Blackburn K, Radde KM, Moyer MB, Schlatzer DM, Moseley A, Boucher RC.

Mol Cell Proteomics. 2002 Jun;1(6):451-65.


Identification of dynein heavy chain 7 as an inner arm component of human cilia that is synthesized but not assembled in a case of primary ciliary dyskinesia.

Zhang YJ, O'Neal WK, Randell SH, Blackburn K, Moyer MB, Boucher RC, Ostrowski LE.

J Biol Chem. 2002 May 17;277(20):17906-15. Epub 2002 Mar 4.


Cloning and characterization of KPL2, a novel gene induced during ciliogenesis of tracheal epithelial cells.

Ostrowski LE, Andrews K, Potdar P, Matsuura H, Jetten A, Nettesheim P.

Am J Respir Cell Mol Biol. 1999 Apr;20(4):675-83.


Discordant organ laterality in monozygotic twins with primary ciliary dyskinesia.

Noone PG, Bali D, Carson JL, Sannuti A, Gipson CL, Ostrowski LE, Bromberg PA, Boucher RC, Knowles MR.

Am J Med Genet. 1999 Jan 15;82(2):155-60.


Expression and regulation of gamma-glutamyl transpeptidase-related enzyme in tracheal cells.

Potdar PD, Andrews KL, Nettesheim P, Ostrowski LE.

Am J Physiol. 1997 Nov;273(5):L1082-9. doi: 10.1152/ajplung.1997.273.5.L1082.


A novel cytoplasmic dynein heavy chain: expression of DHC1b in mammalian ciliated epithelial cells.

Criswell PS, Ostrowski LE, Asai DJ.

J Cell Sci. 1996 Jul;109 ( Pt 7):1891-8.


Identification of seven rat axonemal dynein heavy chain genes: expression during ciliated cell differentiation.

Andrews KL, Nettesheim P, Asai DJ, Ostrowski LE.

Mol Biol Cell. 1996 Jan;7(1):71-9.


Inhibition of ciliated cell differentiation by fluid submersion.

Ostrowski LE, Nettesheim P.

Exp Lung Res. 1995 Nov-Dec;21(6):957-70.


Regulation of ciliated cell differentiation in cultures of rat tracheal epithelial cells.

Clark AB, Randell SH, Nettesheim P, Gray TE, Bagnell B, Ostrowski LE.

Am J Respir Cell Mol Biol. 1995 Mar;12(3):329-38.


Chrysotile asbestos stimulates platelet-derived growth factor-AA production by rat lung fibroblasts in vitro: evidence for an autocrine loop.

Lasky JA, Coin PG, Lindroos PM, Ostrowski LE, Brody AR, Bonner JC.

Am J Respir Cell Mol Biol. 1995 Feb;12(2):162-70.


Ciliogenesis of rat tracheal epithelial cells in vitro.

Ostrowski LE, Randell SH, Clark AB, Gray TE, Nettesheim P.

Methods Cell Biol. 1995;47:57-63. No abstract available.


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