Rib Cage Deformities Alter Respiratory Muscle Action and Chest Wall Function in Patients with Severe Osteogenesis Imperfecta 英文参考文献.docVIP

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Rib Cage Deformities Alter Respiratory Muscle Action and Chest Wall Function in Patients with Severe Osteogenesis Imperfecta 英文参考文献.doc

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Rib Cage Deformities Alter Respiratory Muscle Action and Chest Wall Function in Patients with Severe Osteogenesis Imperfecta 英文参考文献

RibCageDeformitiesAlterRespiratoryMuscleAction andChestWallFunctioninPatientswithSevere OsteogenesisImperfecta AntonellaLoMauro1,SimonaPochintesta2,MariannaRomei2,MariaGraziaD’Angelo2,AntonioPedotti1, AnnaCarlaTurconi2,AndreaAliverti1* 1TBMLab,DipartimentodiBioingegneria,PolitecnicodiMilano,Milano,Italy,2IRCCSE.Medea,BosisioParini(Lc),Italy Abstract Background: Osteogenesis imperfecta (OI) is an inherited connective tissue disorder characterized by bone fragility, multiple fractures and significant chest wall deformities. Cardiopulmonary insufficiency is the leading cause of death in thesepatients. Methods: Seven patients with severe OI type III, 15 with moderate OI type IV and 26 healthy subjects were studied. In additiontostandardspirometry,ribcagegeometry,breathingpatternandregionalchestwallvolumechangesatrestin seatedandsupinepositionwereassessedbyopto-electronicplethysmographytoinvestigateifstructuralmodificationsof theribcageinOIhaveconsequencesonventilatorypattern.One-wayortwo-wayanalysisofvariancewasperformedto comparetheresultsbetweenthethreegroupsandthetwopostures. Results:BothOItypeIIIandIVpatientsshowedreducedFVCandFEV1comparedtopredictedvalues,onconditionthat updatedreferenceequationsareconsidered.Inbothpositions,ventilationwaslowerinOIpatientsthancontrolbecauseof lowertidalvolume(p,0.01).IncontrasttoOItypeIVpatients,whosechestwallgeometryandfunctionwasnormal,OItype IIIpatientswerecharacterizedbyreduced(p,0.01)angleatthesternum(pectuscarinatum),paradoxicalinspiratoryinward motionofthepulmonaryribcage,significantthoraco-abdominalasynchroniesandribcagedistortionsinsupineposition (p,0.001). Conclusions:Inconclusion,therestrictiverespiratorypatternofOsteogenesisImperfectaiscloselyrelatedtotheseverityof thediseaseandtothesternaldeformities.PectuscarinatumcharacterizesOItypeIIIpatientsandaltersrespiratorymuscles coordination,leadingtochestwallandribcagedistortionsandaninefficientventilatorpattern.OItypeIVischaracterized by lower alterations in the respirato