Association between ribs shape and pulmonary function in patients with Osteogenesis Imperfecta

Sanchís-Gimeno, Juan Alberto; Lois Zlolniski, Stephanie; González Ruiz, José María; Palancar, Carlos A.; Torres-Tamayo, Nicole; García-Martínez, Daniel; Aparicio, Luis; Perez-Bermejo, Marcelino; Blanco-Pérez, Esther; Mata-Escolano, Federico; Llidó, Susanna; Torres Sánchez, Isabel; García-Río, Francisco; Bastir, Markus

Association between ribs shape and pulmonary function in patients with Osteogenesis Imperfecta

The aim of the present study was to test the hypothesis that ribs shape changes in patients with OI are more relevant for respiratory function than thoracic spine shape. We used 3D geometric morphometrics to quantify rib cage morphology in OI patients and controls, and to investigate its relationshi...

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Autores:	Sanchís-Gimeno, Juan Alberto, Lois Zlolniski, Stephanie, González Ruiz, José María, Palancar, Carlos A., Torres-Tamayo, Nicole, García-Martínez, Daniel, Aparicio, Luis, Perez-Bermejo, Marcelino, Blanco-Pérez, Esther, Mata-Escolano, Federico, Llidó, Susanna, Torres Sánchez, Isabel, García-Río, Francisco, Bastir, Markus
Tipo de recurso:	artículo
Estado:	Versión publicada
Fecha de publicación:	2019
País:	España
Institución:	Consejo Superior de Investigaciones Científicas (CSIC)
Repositorio:	DIGITAL.CSIC. Repositorio Institucional del CSIC
OAI Identifier:	oai:digital.csic.es:10261/235399
Acceso en línea:	http://hdl.handle.net/10261/235399
Access Level:	acceso abierto
Palabra clave:	Geometric morphometrics Osteogenesis imperfecta Pulmonary function Rib cage Scoliosis Thoracic spine

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spelling	Association between ribs shape and pulmonary function in patients with Osteogenesis ImperfectaSanchís-Gimeno, Juan AlbertoLois Zlolniski, StephanieGonzález Ruiz, José MaríaPalancar, Carlos A.Torres-Tamayo, NicoleGarcía-Martínez, DanielAparicio, LuisPerez-Bermejo, MarcelinoBlanco-Pérez, EstherMata-Escolano, FedericoLlidó, SusannaTorres Sánchez, IsabelGarcía-Río, FranciscoBastir, MarkusGeometric morphometricsOsteogenesis imperfectaPulmonary functionRib cageScoliosisThoracic spineThe aim of the present study was to test the hypothesis that ribs shape changes in patients with OI are more relevant for respiratory function than thoracic spine shape. We used 3D geometric morphometrics to quantify rib cage morphology in OI patients and controls, and to investigate its relationship with forced vital capacity (FVC) and forced expiratory volume in 1 s (FEV1), expressed as absolute value and as percentage of predicted value (% pred). Regression analyses on the full sample showed a significant relation between rib shape and FEV1, FVC and FVC % pred whereas thoracic spine shape was not related to any parameter. Subsequent regression analyses on OI patients confirmed significant relations between dynamic lung volumes and rib shape changes. Lower FVC and FEV1 values are identified in OI patients that present more horizontally aligned ribs, a greater antero-posterior depth due to extreme transverse curve at rib angles and a strong spine invagination, greater asymmetry, and a vertically short, thoraco-lumbar spine, which is relatively straight in at levels 1–8 and shows a marked kyphosis in the thoraco-lumbar transition. Our research seems to support that ribs shape is more relevant for ventilator mechanics in OI patients than the spine shape.This project was funded by grants of the Fundacion Eresa (grant number: BF14_005), the Ministry of Economy, Industry and Competitiveness (grant number: CGL2015-63648-P), the Care4 Brittlebones Foundation (grant number: OTR2016-15543INVES), and the University of Valencia (grant number: UV-INV_AE18-773873).ElsevierMinisterio de Economía y Competitividad (España)Care4 Brittlebones FoundationUniversidad de ValenciaConsejo Superior de Investigaciones Científicas [https://ror.org/02gfc7t72]2021202120192021info:eu-repo/semantics/articlehttp://purl.org/coar/resource_type/c_6501Publisher's versioninfo:eu-repo/semantics/publishedVersionhttp://hdl.handle.net/10261/235399reponame:DIGITAL.CSIC. Repositorio Institucional del CSICinstname:Consejo Superior de Investigaciones Científicas (CSIC)Inglés#PLACEHOLDER_PARENT_METADATA_VALUE#info:eu-repo/grantAgreement/MINECO/Plan Estatal de Investigación Científica y Técnica y de Innovación 2013-2016/CGL2015-63648-Phttp://dx.doi.org/10.1016/j.jare.2019.10.007Síinfo:eu-repo/semantics/openAccessoai:digital.csic.es:10261/2353992026-05-22T06:33:51Z
dc.title.none.fl_str_mv	Association between ribs shape and pulmonary function in patients with Osteogenesis Imperfecta
title	Association between ribs shape and pulmonary function in patients with Osteogenesis Imperfecta
spellingShingle	Association between ribs shape and pulmonary function in patients with Osteogenesis Imperfecta Sanchís-Gimeno, Juan Alberto Geometric morphometrics Osteogenesis imperfecta Pulmonary function Rib cage Scoliosis Thoracic spine
title_short	Association between ribs shape and pulmonary function in patients with Osteogenesis Imperfecta
title_full	Association between ribs shape and pulmonary function in patients with Osteogenesis Imperfecta
title_fullStr	Association between ribs shape and pulmonary function in patients with Osteogenesis Imperfecta
title_full_unstemmed	Association between ribs shape and pulmonary function in patients with Osteogenesis Imperfecta
title_sort	Association between ribs shape and pulmonary function in patients with Osteogenesis Imperfecta
dc.creator.none.fl_str_mv	Sanchís-Gimeno, Juan Alberto Lois Zlolniski, Stephanie González Ruiz, José María Palancar, Carlos A. Torres-Tamayo, Nicole García-Martínez, Daniel Aparicio, Luis Perez-Bermejo, Marcelino Blanco-Pérez, Esther Mata-Escolano, Federico Llidó, Susanna Torres Sánchez, Isabel García-Río, Francisco Bastir, Markus
author	Sanchís-Gimeno, Juan Alberto
author_facet	Sanchís-Gimeno, Juan Alberto Lois Zlolniski, Stephanie González Ruiz, José María Palancar, Carlos A. Torres-Tamayo, Nicole García-Martínez, Daniel Aparicio, Luis Perez-Bermejo, Marcelino Blanco-Pérez, Esther Mata-Escolano, Federico Llidó, Susanna Torres Sánchez, Isabel García-Río, Francisco Bastir, Markus
author_role	author
author2	Lois Zlolniski, Stephanie González Ruiz, José María Palancar, Carlos A. Torres-Tamayo, Nicole García-Martínez, Daniel Aparicio, Luis Perez-Bermejo, Marcelino Blanco-Pérez, Esther Mata-Escolano, Federico Llidó, Susanna Torres Sánchez, Isabel García-Río, Francisco Bastir, Markus
author2_role	author author author author author author author author author author author author author
dc.contributor.none.fl_str_mv	Ministerio de Economía y Competitividad (España) Care4 Brittlebones Foundation Universidad de Valencia Consejo Superior de Investigaciones Científicas [https://ror.org/02gfc7t72]
dc.subject.none.fl_str_mv	Geometric morphometrics Osteogenesis imperfecta Pulmonary function Rib cage Scoliosis Thoracic spine
topic	Geometric morphometrics Osteogenesis imperfecta Pulmonary function Rib cage Scoliosis Thoracic spine
description	The aim of the present study was to test the hypothesis that ribs shape changes in patients with OI are more relevant for respiratory function than thoracic spine shape. We used 3D geometric morphometrics to quantify rib cage morphology in OI patients and controls, and to investigate its relationship with forced vital capacity (FVC) and forced expiratory volume in 1 s (FEV1), expressed as absolute value and as percentage of predicted value (% pred). Regression analyses on the full sample showed a significant relation between rib shape and FEV1, FVC and FVC % pred whereas thoracic spine shape was not related to any parameter. Subsequent regression analyses on OI patients confirmed significant relations between dynamic lung volumes and rib shape changes. Lower FVC and FEV1 values are identified in OI patients that present more horizontally aligned ribs, a greater antero-posterior depth due to extreme transverse curve at rib angles and a strong spine invagination, greater asymmetry, and a vertically short, thoraco-lumbar spine, which is relatively straight in at levels 1–8 and shows a marked kyphosis in the thoraco-lumbar transition. Our research seems to support that ribs shape is more relevant for ventilator mechanics in OI patients than the spine shape.
publishDate	2019
dc.date.none.fl_str_mv	2019 2021 2021 2021
dc.type.none.fl_str_mv	info:eu-repo/semantics/article http://purl.org/coar/resource_type/c_6501 Publisher's version info:eu-repo/semantics/publishedVersion
format	article
status_str	publishedVersion
dc.identifier.none.fl_str_mv	http://hdl.handle.net/10261/235399
url	http://hdl.handle.net/10261/235399
dc.language.none.fl_str_mv	Inglés
language_invalid_str_mv	Inglés
dc.relation.none.fl_str_mv	#PLACEHOLDER_PARENT_METADATA_VALUE# info:eu-repo/grantAgreement/MINECO/Plan Estatal de Investigación Científica y Técnica y de Innovación 2013-2016/CGL2015-63648-P http://dx.doi.org/10.1016/j.jare.2019.10.007 Sí
dc.rights.none.fl_str_mv	info:eu-repo/semantics/openAccess
eu_rights_str_mv	openAccess
dc.publisher.none.fl_str_mv	Elsevier
publisher.none.fl_str_mv	Elsevier
dc.source.none.fl_str_mv	reponame:DIGITAL.CSIC. Repositorio Institucional del CSIC instname:Consejo Superior de Investigaciones Científicas (CSIC)
instname_str	Consejo Superior de Investigaciones Científicas (CSIC)
reponame_str	DIGITAL.CSIC. Repositorio Institucional del CSIC
collection	DIGITAL.CSIC. Repositorio Institucional del CSIC
repository.name.fl_str_mv
repository.mail.fl_str_mv
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score	15.81155

Association between ribs shape and pulmonary function in patients with Osteogenesis Imperfecta

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