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A rare case of rapidly enlarging tracheal lobular capillary hemangioma presenting as difficult to ventilate acute asthma during pregnancy

BACKGROUND: Lobular Capillary Hemangioma (LCH) is a benign tumour that is known to be hormone responsive and have a relatively high incidence during pregnancy, the most common site being the gingival surfaces. A tracheal origin for this tumour is extremely rare, with no case reported so far in this...

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Autores principales: Prakash, Shivesh, Bihari, Shailesh, Wiersema, Ubbo
Formato: Online Artículo Texto
Lenguaje:English
Publicado: BioMed Central 2014
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4007586/
https://www.ncbi.nlm.nih.gov/pubmed/24612935
http://dx.doi.org/10.1186/1471-2466-14-41
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author Prakash, Shivesh
Bihari, Shailesh
Wiersema, Ubbo
author_facet Prakash, Shivesh
Bihari, Shailesh
Wiersema, Ubbo
author_sort Prakash, Shivesh
collection PubMed
description BACKGROUND: Lobular Capillary Hemangioma (LCH) is a benign tumour that is known to be hormone responsive and have a relatively high incidence during pregnancy, the most common site being the gingival surfaces. A tracheal origin for this tumour is extremely rare, with no case reported so far in this patient population, and the only reported clinical presentation of tracheal LCH in the literature is with haemoptysis. CASE PRESENTATION: We describe a case of a 23-year-old known asthmatic who presented at 32 weeks gestation with life-threatening respiratory failure resembling acute severe asthma, requiring invasive ventilation which was extremely difficult. This was subsequently found to be due to a large tracheal LCH producing a ball-valve phenomenon and predominantly expiratory airflow limitation similar to acute asthma. The endotracheal tube was advanced past the lesion under bronchoscopic guidance, and urgent Caesarean section performed due to foetal distress. The tumour was subsequently debulked and the trachea stented, facilitated by bi-femoral veno-venous extra-corporeal membrane oxygenation with relatively low dose of heparin. CONCLUSION: To our knowledge, this is the first report of a unique presentation and management of largest tracheal LCH so far occurring during pregnancy. Pulmonary and critical care physicians should be aware of this unique differential of refractory asthma, the aggressive nature of this benign tumour due to hormonal influences during pregnancy, and feasibility of using bi-femoral veno-venous extra-corporeal membrane oxygenation with low dose heparin as a rescue, given the high risk of bleeding.
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spelling pubmed-40075862014-05-03 A rare case of rapidly enlarging tracheal lobular capillary hemangioma presenting as difficult to ventilate acute asthma during pregnancy Prakash, Shivesh Bihari, Shailesh Wiersema, Ubbo BMC Pulm Med Case Report BACKGROUND: Lobular Capillary Hemangioma (LCH) is a benign tumour that is known to be hormone responsive and have a relatively high incidence during pregnancy, the most common site being the gingival surfaces. A tracheal origin for this tumour is extremely rare, with no case reported so far in this patient population, and the only reported clinical presentation of tracheal LCH in the literature is with haemoptysis. CASE PRESENTATION: We describe a case of a 23-year-old known asthmatic who presented at 32 weeks gestation with life-threatening respiratory failure resembling acute severe asthma, requiring invasive ventilation which was extremely difficult. This was subsequently found to be due to a large tracheal LCH producing a ball-valve phenomenon and predominantly expiratory airflow limitation similar to acute asthma. The endotracheal tube was advanced past the lesion under bronchoscopic guidance, and urgent Caesarean section performed due to foetal distress. The tumour was subsequently debulked and the trachea stented, facilitated by bi-femoral veno-venous extra-corporeal membrane oxygenation with relatively low dose of heparin. CONCLUSION: To our knowledge, this is the first report of a unique presentation and management of largest tracheal LCH so far occurring during pregnancy. Pulmonary and critical care physicians should be aware of this unique differential of refractory asthma, the aggressive nature of this benign tumour due to hormonal influences during pregnancy, and feasibility of using bi-femoral veno-venous extra-corporeal membrane oxygenation with low dose heparin as a rescue, given the high risk of bleeding. BioMed Central 2014-03-10 /pmc/articles/PMC4007586/ /pubmed/24612935 http://dx.doi.org/10.1186/1471-2466-14-41 Text en Copyright © 2014 Prakash et al.; licensee BioMed Central Ltd. http://creativecommons.org/licenses/by/2.0 This is an Open Access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/2.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly credited.
spellingShingle Case Report
Prakash, Shivesh
Bihari, Shailesh
Wiersema, Ubbo
A rare case of rapidly enlarging tracheal lobular capillary hemangioma presenting as difficult to ventilate acute asthma during pregnancy
title A rare case of rapidly enlarging tracheal lobular capillary hemangioma presenting as difficult to ventilate acute asthma during pregnancy
title_full A rare case of rapidly enlarging tracheal lobular capillary hemangioma presenting as difficult to ventilate acute asthma during pregnancy
title_fullStr A rare case of rapidly enlarging tracheal lobular capillary hemangioma presenting as difficult to ventilate acute asthma during pregnancy
title_full_unstemmed A rare case of rapidly enlarging tracheal lobular capillary hemangioma presenting as difficult to ventilate acute asthma during pregnancy
title_short A rare case of rapidly enlarging tracheal lobular capillary hemangioma presenting as difficult to ventilate acute asthma during pregnancy
title_sort rare case of rapidly enlarging tracheal lobular capillary hemangioma presenting as difficult to ventilate acute asthma during pregnancy
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4007586/
https://www.ncbi.nlm.nih.gov/pubmed/24612935
http://dx.doi.org/10.1186/1471-2466-14-41
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