Thoracic vasculitis presenting as surgical problems.

Hdl Handle:
http://hdl.handle.net/10147/200282
Title:
Thoracic vasculitis presenting as surgical problems.
Authors:
Jansen, Michael; Saleh, Sheikh; Bolster, Margot; O'Donnell, Aonghus; Ahern, Thomas; Spence, Liam; Sheppard, Mary N; Burke, Louise
Affiliation:
Department of Histopathology, Cork University Hospital, Wilton, Cork, Eire, Ireland. jansmichael@gmail.com
Citation:
Thoracic vasculitis presenting as surgical problems. 2010, 456 (1):91-6 Virchows Arch.
Journal:
Virchows Archiv : an international journal of pathology
Issue Date:
Jan-2010
URI:
http://hdl.handle.net/10147/200282
DOI:
10.1007/s00428-009-0865-0
PubMed ID:
20012090
Abstract:
We present four patients with vasculitis manifesting with unusual clinical or pathological features, generating surgical problems. Two cases presented with pulmonary hypertension, with investigations and radiological evidence prompting clinical suspicion of pulmonary thrombo-embolic disease. First case, with an antecedant history of Wegener's granulomatosis (WG), demonstrated following "embolectomy", WG involving the large pulmonary elastic arteries. The second case of inoperable "pulmonary thrombo-embolic disease" was subsequently found at limited post mortem to have giant cell arteritis, which affected widespread small peripheral pulmonary arterial vessels. The other two cases were of aortitis occurring in the background of immune-mediated disease, which had been treated with aggressive immunosuppression regimens. The first of these was a case of Cogan's syndrome complicated by descending aortitis, a rarely reported phenomenon, with co-existent acute endocarditis of the aortic valve leaflets. Most cases of endocarditis in this context occur secondary to and in continuity with ascending aortitis. That this case, and a case of ascending aortitis occurring in the context of relapsing polychondritis occurred in the face of aggressive immunosuppression with an apparent clinical response, underscores the need to not accept a clinical picture at face value. This has implications for clinical management, particularly in the follow-up of surgical prosthetic devices such as grafts which may be used in these cases. All four cases emphasise the continued importance of histology and the post-mortem examination in elucidating previously undetected or unsuspected disease.
Item Type:
Article
Language:
en
Description:
We present four patients with vasculitis manifesting with unusual clinical or pathological features, generating surgical problems. Two cases presented with pulmonary hypertension, with investigations and radiological evidence prompting clinical suspicion of pulmonary thrombo-embolic disease. First case, with an antecedant history of Wegener's granulomatosis (WG), demonstrated following "embolectomy", WG involving the large pulmonary elastic arteries. The second case of inoperable "pulmonary thrombo-embolic disease" was subsequently found at limited post mortem to have giant cell arteritis, which affected widespread small peripheral pulmonary arterial vessels. The other two cases were of aortitis occurring in the background of immune-mediated disease, which had been treated with aggressive immunosuppression regimens. The first of these was a case of Cogan's syndrome complicated by descending aortitis, a rarely reported phenomenon, with co-existent acute endocarditis of the aortic valve leaflets. Most cases of endocarditis in this context occur secondary to and in continuity with ascending aortitis. That this case, and a case of ascending aortitis occurring in the context of relapsing polychondritis occurred in the face of aggressive immunosuppression with an apparent clinical response, underscores the need to not accept a clinical picture at face value. This has implications for clinical management, particularly in the follow-up of surgical prosthetic devices such as grafts which may be used in these cases. All four cases emphasise the continued importance of histology and the post-mortem examination in elucidating previously undetected or unsuspected disease.
MeSH:
Adult; Aorta, Thoracic; Aortitis; Cogan Syndrome; Female; Giant Cell Arteritis; Humans; Hypertension, Pulmonary; Middle Aged; Vasculitis; Wegener Granulomatosis
ISSN:
1432-2307

Full metadata record

DC FieldValue Language
dc.contributor.authorJansen, Michaelen
dc.contributor.authorSaleh, Sheikhen
dc.contributor.authorBolster, Margoten
dc.contributor.authorO'Donnell, Aonghusen
dc.contributor.authorAhern, Thomasen
dc.contributor.authorSpence, Liamen
dc.contributor.authorSheppard, Mary Nen
dc.contributor.authorBurke, Louiseen
dc.date.accessioned2012-01-05T15:43:56Z-
dc.date.available2012-01-05T15:43:56Z-
dc.date.issued2010-01-
dc.identifier.citationThoracic vasculitis presenting as surgical problems. 2010, 456 (1):91-6 Virchows Arch.en
dc.identifier.issn1432-2307-
dc.identifier.pmid20012090-
dc.identifier.doi10.1007/s00428-009-0865-0-
dc.identifier.urihttp://hdl.handle.net/10147/200282-
dc.descriptionWe present four patients with vasculitis manifesting with unusual clinical or pathological features, generating surgical problems. Two cases presented with pulmonary hypertension, with investigations and radiological evidence prompting clinical suspicion of pulmonary thrombo-embolic disease. First case, with an antecedant history of Wegener's granulomatosis (WG), demonstrated following "embolectomy", WG involving the large pulmonary elastic arteries. The second case of inoperable "pulmonary thrombo-embolic disease" was subsequently found at limited post mortem to have giant cell arteritis, which affected widespread small peripheral pulmonary arterial vessels. The other two cases were of aortitis occurring in the background of immune-mediated disease, which had been treated with aggressive immunosuppression regimens. The first of these was a case of Cogan's syndrome complicated by descending aortitis, a rarely reported phenomenon, with co-existent acute endocarditis of the aortic valve leaflets. Most cases of endocarditis in this context occur secondary to and in continuity with ascending aortitis. That this case, and a case of ascending aortitis occurring in the context of relapsing polychondritis occurred in the face of aggressive immunosuppression with an apparent clinical response, underscores the need to not accept a clinical picture at face value. This has implications for clinical management, particularly in the follow-up of surgical prosthetic devices such as grafts which may be used in these cases. All four cases emphasise the continued importance of histology and the post-mortem examination in elucidating previously undetected or unsuspected disease.en
dc.description.abstractWe present four patients with vasculitis manifesting with unusual clinical or pathological features, generating surgical problems. Two cases presented with pulmonary hypertension, with investigations and radiological evidence prompting clinical suspicion of pulmonary thrombo-embolic disease. First case, with an antecedant history of Wegener's granulomatosis (WG), demonstrated following "embolectomy", WG involving the large pulmonary elastic arteries. The second case of inoperable "pulmonary thrombo-embolic disease" was subsequently found at limited post mortem to have giant cell arteritis, which affected widespread small peripheral pulmonary arterial vessels. The other two cases were of aortitis occurring in the background of immune-mediated disease, which had been treated with aggressive immunosuppression regimens. The first of these was a case of Cogan's syndrome complicated by descending aortitis, a rarely reported phenomenon, with co-existent acute endocarditis of the aortic valve leaflets. Most cases of endocarditis in this context occur secondary to and in continuity with ascending aortitis. That this case, and a case of ascending aortitis occurring in the context of relapsing polychondritis occurred in the face of aggressive immunosuppression with an apparent clinical response, underscores the need to not accept a clinical picture at face value. This has implications for clinical management, particularly in the follow-up of surgical prosthetic devices such as grafts which may be used in these cases. All four cases emphasise the continued importance of histology and the post-mortem examination in elucidating previously undetected or unsuspected disease.-
dc.language.isoenen
dc.subject.meshAdult-
dc.subject.meshAorta, Thoracic-
dc.subject.meshAortitis-
dc.subject.meshCogan Syndrome-
dc.subject.meshFemale-
dc.subject.meshGiant Cell Arteritis-
dc.subject.meshHumans-
dc.subject.meshHypertension, Pulmonary-
dc.subject.meshMiddle Aged-
dc.subject.meshVasculitis-
dc.subject.meshWegener Granulomatosis-
dc.titleThoracic vasculitis presenting as surgical problems.en
dc.typeArticleen
dc.contributor.departmentDepartment of Histopathology, Cork University Hospital, Wilton, Cork, Eire, Ireland. jansmichael@gmail.comen
dc.identifier.journalVirchows Archiv : an international journal of pathologyen
dc.description.provinceMunster-

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