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Pulmonary arterial hypertension with features of venous involvement: a detective's task

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Resumo:Pulmonary veno-occlusive disease (PVOD) and pulmonary capillary hemangiomatosis are rare types of histopathological substrates within the spectrum of pulmonary arterial hypertension (PAH) with a very poor prognosis. They are characterized by a widespread fibroproliferative process of the small caliber veins and/or capillaries with sparing of the larger veins, resulting in a pre-capillary pulmonary hypertension phenotype. Clinical presentation is unspecific and similar to other PAH etiologies. Definitive diagnosis is obtained through histological analysis, although lung biopsy is not advised due to a higher risk of complications. However, some additional findings may allow a presumptive clinical diagnosis of PVOD, particularly a history of smoking, chemotherapy drug use, exposure to organic solvents (particularly trichloroethylene), low diffusing capacity for carbon monoxide (DLCO), exercise induced desaturation, and evidence of venous congestion without left heart disease on imaging, manifested by a classical triad of ground glass opacities, septal lines, and lymphadenopathies. Lung transplant is the only effective treatment, and patients should be referred at the time of diagnosis due to the rapid progression of the disease and associated poor prognosis. We present a case of a 58-year-old man with PAH with features of venous/capillary involvement in which clinical suspicion, prompt diagnosis, and early referral for lung transplantation were determinant factors for the successful outcome.
Autores principais:Cazeiro, Daniel
Outros Autores:Plácido, Rui; Azaredo Raposo, Miguel; Brito, Joana; Borba, Alexandra; Guimarães, Tatiana; Pinto, Eugénia; Freitas, Pedro; Pinto, Fausto J.
Assunto:Pulmonary arterial hypertension Pulmonary veno-oclusive disease Lung transplantation Hemangioma capillary Tomography-X-Ray Computed Surgery
Ano:2024
País:Portugal
Tipo de documento:artigo
Tipo de acesso:acesso aberto
Instituição associada:Universidade de Lisboa
Idioma:inglês
Origem:Repositório da Universidade de Lisboa
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author Cazeiro, Daniel
author2 Plácido, Rui
Azaredo Raposo, Miguel
Brito, Joana
Borba, Alexandra
Guimarães, Tatiana
Pinto, Eugénia
Freitas, Pedro
Pinto, Fausto J.
author2_role author
author
author
author
author
author
author
author
author_facet Cazeiro, Daniel
Plácido, Rui
Azaredo Raposo, Miguel
Brito, Joana
Borba, Alexandra
Guimarães, Tatiana
Pinto, Eugénia
Freitas, Pedro
Pinto, Fausto J.
author_role author
contributor_name_str_mv Repositório Científico de Acesso Aberto da ULisboa
country_str PT
creators_json_txt [{\"Person.name\":\"Cazeiro, Daniel\",\"Person.identifier.orcid\":\"0009-0004-0277-7398\"},{\"Person.name\":\"Plácido, Rui\",\"Person.identifier.orcid\":\"0000-0003-4164-5481\"},{\"Person.name\":\"Azaredo Raposo, Miguel\",\"Person.identifier.orcid\":\"0000-0002-1744-8843\"},{\"Person.name\":\"Brito, Joana\",\"Person.identifier.orcid\":\"0000-0001-6672-7542\"},{\"Person.name\":\"Borba, Alexandra\"},{\"Person.name\":\"Guimarães, Tatiana\",\"Person.identifier.orcid\":\"0000-0001-7054-0465\"},{\"Person.name\":\"Pinto, Eugénia\"},{\"Person.name\":\"Freitas, Pedro\"},{\"Person.name\":\"Pinto, Fausto J.\",\"Person.identifier.orcid\":\"0000-0002-8034-4529\"}]
datacite.contributors.contributor.contributorName.fl_str_mv Repositório Científico de Acesso Aberto da ULisboa
datacite.creators.creator.creatorName.fl_str_mv Cazeiro, Daniel
Plácido, Rui
Azaredo Raposo, Miguel
Brito, Joana
Borba, Alexandra
Guimarães, Tatiana
Pinto, Eugénia
Freitas, Pedro
Pinto, Fausto J.
datacite.date.Accepted.fl_str_mv 2024-01-01T00:00:00Z
datacite.date.available.fl_str_mv 2024-06-12T14:48:24Z
datacite.date.embargoed.fl_str_mv 2024-06-12T14:48:24Z
datacite.rights.fl_str_mv http://purl.org/coar/access_right/c_abf2
datacite.subjects.subject.fl_str_mv Pulmonary arterial hypertension
Pulmonary veno-oclusive disease
Lung transplantation
Hemangioma capillary
Tomography-X-Ray Computed
Surgery
datacite.titles.title.fl_str_mv Pulmonary arterial hypertension with features of venous involvement: a detective's task
Hipertensão arterial pulmonar com características de envolvimento venoso: um trabalho investigativo
dc.contributor.none.fl_str_mv Repositório Científico de Acesso Aberto da ULisboa
dc.creator.none.fl_str_mv Cazeiro, Daniel
Plácido, Rui
Azaredo Raposo, Miguel
Brito, Joana
Borba, Alexandra
Guimarães, Tatiana
Pinto, Eugénia
Freitas, Pedro
Pinto, Fausto J.
dc.date.Accepted.fl_str_mv 2024-01-01T00:00:00Z
dc.date.available.fl_str_mv 2024-06-12T14:48:24Z
dc.date.embargoed.fl_str_mv 2024-06-12T14:48:24Z
dc.format.none.fl_str_mv application/pdf
dc.identifier.none.fl_str_mv http://hdl.handle.net/10451/65034
dc.language.none.fl_str_mv eng
dc.publisher.none.fl_str_mv Sociedade Brasileira de Cardiologia - SBC
dc.rights.cclincense.fl_str_mv http://creativecommons.org/licenses/by/4.0/
dc.rights.none.fl_str_mv http://purl.org/coar/access_right/c_abf2
dc.subject.none.fl_str_mv Pulmonary arterial hypertension
Pulmonary veno-oclusive disease
Lung transplantation
Hemangioma capillary
Tomography-X-Ray Computed
Surgery
dc.title.fl_str_mv Pulmonary arterial hypertension with features of venous involvement: a detective's task
Hipertensão arterial pulmonar com características de envolvimento venoso: um trabalho investigativo
dc.type.none.fl_str_mv http://purl.org/coar/resource_type/c_6501
description Pulmonary veno-occlusive disease (PVOD) and pulmonary capillary hemangiomatosis are rare types of histopathological substrates within the spectrum of pulmonary arterial hypertension (PAH) with a very poor prognosis. They are characterized by a widespread fibroproliferative process of the small caliber veins and/or capillaries with sparing of the larger veins, resulting in a pre-capillary pulmonary hypertension phenotype. Clinical presentation is unspecific and similar to other PAH etiologies. Definitive diagnosis is obtained through histological analysis, although lung biopsy is not advised due to a higher risk of complications. However, some additional findings may allow a presumptive clinical diagnosis of PVOD, particularly a history of smoking, chemotherapy drug use, exposure to organic solvents (particularly trichloroethylene), low diffusing capacity for carbon monoxide (DLCO), exercise induced desaturation, and evidence of venous congestion without left heart disease on imaging, manifested by a classical triad of ground glass opacities, septal lines, and lymphadenopathies. Lung transplant is the only effective treatment, and patients should be referred at the time of diagnosis due to the rapid progression of the disease and associated poor prognosis. We present a case of a 58-year-old man with PAH with features of venous/capillary involvement in which clinical suspicion, prompt diagnosis, and early referral for lung transplantation were determinant factors for the successful outcome.
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eu_rights_str_mv openAccess
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id ul_e4c1dc90bc452e5cbe4dfca37cf71d3b
identifier.url.fl_str_mv http://hdl.handle.net/10451/65034
instacron_str ul
institution Universidade de Lisboa
instname_str Universidade de Lisboa
language eng
network_acronym_str ul
network_name_str Repositório da Universidade de Lisboa
oai_identifier_str oai:repositorio.ulisboa.pt:10451/65034
organization_str_mv urn:organizationAcronym:ul
person_str_mv Cazeiro, Daniel
Cazeiro, Daniel
http://orcid.org/0009-0004-0277-7398
0009-0004-0277-7398
Plácido, Rui
Plácido, Rui
https://www.ciencia-id.pt/1A11-7BF5-0D37
1A11-7BF5-0D37
http://orcid.org/0000-0003-4164-5481
0000-0003-4164-5481
Azaredo Raposo, Miguel
Azaredo Raposo, Miguel
https://www.ciencia-id.pt/201D-88DC-5C86
201D-88DC-5C86
http://orcid.org/0000-0002-1744-8843
0000-0002-1744-8843
Brito, Joana
Brito, Joana
https://www.ciencia-id.pt/281C-A194-3753
281C-A194-3753
http://orcid.org/0000-0001-6672-7542
0000-0001-6672-7542
Borba, Alexandra
Guimarães, Tatiana
Guimarães, Tatiana
http://orcid.org/0000-0001-7054-0465
0000-0001-7054-0465
Pinto, Eugénia
Freitas, Pedro
Pinto, Fausto J.
Pinto, Fausto J.
https://www.ciencia-id.pt/C311-AEDD-6DBB
C311-AEDD-6DBB
http://orcid.org/0000-0002-8034-4529
0000-0002-8034-4529
publishDate 2024
publisher.none.fl_str_mv Sociedade Brasileira de Cardiologia - SBC
reponame_str Repositório da Universidade de Lisboa
repository_id_str urn:repositoryAcronym:ul
service_str_mv urn:repositoryAcronym:ul
spelling engSociedade Brasileira de Cardiologia - SBCpt_PTPulmonary veno-occlusive disease (PVOD) and pulmonary capillary hemangiomatosis are rare types of histopathological substrates within the spectrum of pulmonary arterial hypertension (PAH) with a very poor prognosis. They are characterized by a widespread fibroproliferative process of the small caliber veins and/or capillaries with sparing of the larger veins, resulting in a pre-capillary pulmonary hypertension phenotype. Clinical presentation is unspecific and similar to other PAH etiologies. Definitive diagnosis is obtained through histological analysis, although lung biopsy is not advised due to a higher risk of complications. However, some additional findings may allow a presumptive clinical diagnosis of PVOD, particularly a history of smoking, chemotherapy drug use, exposure to organic solvents (particularly trichloroethylene), low diffusing capacity for carbon monoxide (DLCO), exercise induced desaturation, and evidence of venous congestion without left heart disease on imaging, manifested by a classical triad of ground glass opacities, septal lines, and lymphadenopathies. Lung transplant is the only effective treatment, and patients should be referred at the time of diagnosis due to the rapid progression of the disease and associated poor prognosis. We present a case of a 58-year-old man with PAH with features of venous/capillary involvement in which clinical suspicion, prompt diagnosis, and early referral for lung transplantation were determinant factors for the successful outcome.application/pdfpt_PTPulmonary arterial hypertension with features of venous involvement: a detective's taskAlternativeTitlept_PTHipertensão arterial pulmonar com características de envolvimento venoso: um trabalho investigativoPersonalCazeiro, DanielDSpacehttp://dspace.org/items/1429ae42-ed17-4df5-8b40-b950cbbd8562DSpacehttp://dspace.org/items/1429ae42-ed17-4df5-8b40-b950cbbd8562CazeiroDanielORCIDhttp://orcid.org0009-0004-0277-7398PersonalPlácido, RuiDSpacehttp://dspace.org/items/f94a2173-a4ff-4460-b47d-194f917cb98bDSpacehttp://dspace.org/items/f94a2173-a4ff-4460-b47d-194f917cb98bPlácidoRuiCiência IDhttps://www.ciencia-id.pt1A11-7BF5-0D37ORCIDhttp://orcid.org0000-0003-4164-5481Scopus Author IDhttps://www.scopus.com18438045300PersonalAzaredo Raposo, MiguelDSpacehttp://dspace.org/items/0acbacea-532e-410d-894b-924fbabe662aDSpacehttp://dspace.org/items/0acbacea-532e-410d-894b-924fbabe662aAzaredo RaposoMiguelCiência IDhttps://www.ciencia-id.pt201D-88DC-5C86ORCIDhttp://orcid.org0000-0002-1744-8843PersonalBrito, JoanaDSpacehttp://dspace.org/items/3298f703-5e47-4fdd-9d3e-2f643355df53DSpacehttp://dspace.org/items/3298f703-5e47-4fdd-9d3e-2f643355df53BritoJoanaCiência IDhttps://www.ciencia-id.pt281C-A194-3753ORCIDhttp://orcid.org0000-0001-6672-7542Researcher IDhttps://www.researcherid.comH-8224-2016Borba, AlexandraPersonalGuimarães, TatianaDSpacehttp://dspace.org/items/bee0ac18-190d-43ca-9d70-dc16f5655515DSpacehttp://dspace.org/items/bee0ac18-190d-43ca-9d70-dc16f5655515GuimarãesTatianaORCIDhttp://orcid.org0000-0001-7054-0465Pinto, EugéniaFreitas, PedroPersonalPinto, Fausto J.DSpacehttp://dspace.org/items/5f44176f-69f5-482c-83cd-ab94425a6ec3DSpacehttp://dspace.org/items/5f44176f-69f5-482c-83cd-ab94425a6ec3PintoFausto J.Ciência IDhttps://www.ciencia-id.ptC311-AEDD-6DBBORCIDhttp://orcid.org0000-0002-8034-4529Researcher IDhttps://www.researcherid.comG-9363-2015Researcher IDhttps://www.researcherid.comGQA-4584-2022Scopus Author IDhttps://www.scopus.com7102740158HostingInstitutionOrganizationalRepositório Científico de Acesso Aberto da ULisboae-mailmailto:repositorio@reitoria.ulisboa.ptrepositorio@reitoria.ulisboa.ptISSNIsPartOf0066-782XDOIIsPartOf10.36660/abc.202305652024-06-12T14:48:24Z20242024-01-01T00:00:00ZHandlehttp://hdl.handle.net/10451/65034http://purl.org/coar/access_right/c_abf2open accessPulmonary arterial hypertensionPulmonary veno-oclusive diseaseLung transplantationHemangioma capillaryTomography-X-Ray ComputedSurgery360249 bytesliteraturehttp://purl.org/coar/resource_type/c_6501journal article2024http://creativecommons.org/licenses/by/4.0/http://purl.org/coar/access_right/c_abf2application/pdffulltexthttps://repositorio.ulisboa.pt/bitstreams/a94d8bae-e133-454d-979b-3a0fc4ccfc9f/downloadArquivos Brasileiros de Cardiologia1214
spellingShingle Pulmonary arterial hypertension with features of venous involvement: a detective's task
Cazeiro, Daniel
Pulmonary arterial hypertension
Pulmonary veno-oclusive disease
Lung transplantation
Hemangioma capillary
Tomography-X-Ray Computed
Surgery
status SINGLETON
subject.fl_str_mv Pulmonary arterial hypertension
Pulmonary veno-oclusive disease
Lung transplantation
Hemangioma capillary
Tomography-X-Ray Computed
Surgery
title Pulmonary arterial hypertension with features of venous involvement: a detective's task
title_full Pulmonary arterial hypertension with features of venous involvement: a detective's task
title_fullStr Pulmonary arterial hypertension with features of venous involvement: a detective's task
title_full_unstemmed Pulmonary arterial hypertension with features of venous involvement: a detective's task
title_short Pulmonary arterial hypertension with features of venous involvement: a detective's task
title_sort Pulmonary arterial hypertension with features of venous involvement: a detective's task
topic Pulmonary arterial hypertension
Pulmonary veno-oclusive disease
Lung transplantation
Hemangioma capillary
Tomography-X-Ray Computed
Surgery
topic_facet Pulmonary arterial hypertension
Pulmonary veno-oclusive disease
Lung transplantation
Hemangioma capillary
Tomography-X-Ray Computed
Surgery
url http://hdl.handle.net/10451/65034
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