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

 Michael R. Knowles  ;  Lawrence E. Ostrowski  ;  Niki T. Loges  ;  Toby Hurd  ;  Margaret W. Leigh  ;  Lu Huang  ;  Whitney E. Wolf  ;  Johnny L. Carson  ;  Milan J. Hazucha  ;  Weining Yin  ;  Stephanie D. Davis  ;  Sharon D. Dell  ;  Thomas W. Ferkol  ;  Scott D. Sagel  ;  Kenneth N. Olivier  ;  Charlotte Jahnke  ;  Heike Olbrich  ;  Claudius Werner  ;  Johanna Raidt  ;  Julia Wallmeier  ;  Petra Pennekamp  ;  Gerard W. Dougherty  ;  Rim Hjeij  ;  Heon Yung Gee  ;  Edgar A. Otto  ;  Jan Halbritter  ;  Moumita Chaki  ;  Katrina A. Diaz  ;  Daniela A. Braun  ;  Jonathan D. Porath  ;  Markus Schueler  ;  Gyo¨rgy Baktai  ;  Matthias Griese  ;  Emily H. Turner  ;  Alexandra P. Lewis  ;  Michael J. Bamshad  ;  Deborah A. Nickerson  ;  Friedhelm Hildebrandt  ;  Jay Shendure  ;  Heymut Omran  ;  Maimoona A. Zariwala 
 AMERICAN JOURNAL OF HUMAN GENETICS, Vol.93(4) : 711-720, 2013 
Journal Title
Issue Date
Adolescent ; Adult ; Animals ; Antigens, Surface/genetics ; Axoneme/genetics ; Child ; Child, Preschool ; Cilia/genetics ; Ciliary Motility Disorders/genetics ; Cytoplasm/genetics ; Dyneins/genetics ; Epithelial Cells/metabolism ; Exome ; Female ; GTP-Binding Proteins/genetics ; Humans ; Infant ; Kartagener Syndrome/genetics ; Male ; Mutation/genetics ; Pedigree ; Phenotype ; Young Adult ; Zebrafish
OBJECTIVE: To fabricate the antibiotic-releasing coatings on TiO(2) nanotube surfaces for wide applications of implant and bone plate in medical and dental surgery, the optimal deposition time of amoxicillin/PLGA solution simultaneously performing non-toxicity and a high bactericidal effect for preventing early implant failures was found. MATERIALS AND METHODS: FE-SEM, ESD and FT-IR were used for confirming deposition of amoxicillin/PLGA on the TiO(2) surface. Also, the elution of amoxicillin/PLGA in a TiO(2) nanotube surface was measured by a UV-VIS spectrophotometer. The bactericidal effect of amoxicillin on the TiO(2) nanotube surface was evaluated by using Staphylococcus aureus (S. aureus). The cytotoxicity and cell proliferation were observed by WST assay using MC3T3-E1 osteoblast cells. RESULTS: The results indicated that the TiO(2) nanotube surface controlled by electro-spray deposition time with amoxicillin/PLGA solution could provide a high bactericidal effect against S. aureus by the bactericidal effect of amoxicillin, as well as good osteoblast cell proliferation at the TiO(2) nanotube surface without toxicity. CONCLUSIONS: This study used electro-spray deposition (ESD) methodology to obtain amoxicillin deposition in nanotube structures of TiO(2) and found the optimal deposition time of amoxicillin/PLGA solution simultaneously performing non-toxicity and a high bactericidal effect for preventing early implant failures.
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1. College of Medicine (의과대학) > Dept. of Pharmacology (약리학교실) > 1. Journal Papers
Yonsei Authors
Gee, Heon Yung(지헌영) ORCID logo https://orcid.org/0000-0002-8741-6177
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