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The c.824 G>T mutation (p. Gly275Val) in the Australian patient was a de novo mutation. In both patients, TEM and IF did not reveal any changes in the respiratory cilia Fluvoxamine structure. However, following ciliogenesis in vitro, in contrast to the ciliary beat frequency, the ciliary beat coordination in the spheroids from the Polish proband and his relatives carrying the c.154G>A mutation was reduced. Analysis of the ciliary alignment indicated severely disturbed orientation of cilia. Therefore, we confirm that defects in the RPGR protein may contribute to syndromic PCD. Lack of ultrastructural defects in respiratory cilia of the probands, the reduced ciliary orientation and the decreased coordination of the ciliary bundles observed in the Polish patient suggested that the RPGR protein may play a role in the establishment of the proper respiratory cilia orientation. Pediatr Pulmonol. 2013; 48:352�C363. ? 2012 Wiley Periodicals, Inc. ""Perfluorochemical (PFC) is theoretically a good vehicle for delivering biological agents to the lungs. This study was designed to investigate the efficacy of intratracheal (IT) instillation of meropenem using PFC liquid as a vehicle in a piglet model of acute lung injury (ALI). Eighteen piglets were injured with lung lavages to induce ALI, and randomly assigned to intravenous (IV) infusion or IT instillation groups, the latter Pazopanib order using either PFC or normal saline (NS) as a I-BET-762 in vivo delivery vehicle for meropenem. Blood samples were obtained at 0, 15, 30, and 60?min, and then hourly for 6?hr. Sera and extracts of lung tissues were assayed for meropenem content using high-performance liquid chromatography. We found that the IV group had significantly higher serum concentrations of meropenem during the first hour after dosing (P?