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Emergent airway management in a case of fibrodysplasia ossificans progressiva
Fibrodysplasia ossificans progressiva (FOP), or Stone man syndrome, is rare and one of the most disabling genetic conditions of the connective tissue due to progressive extraskeletal ossification. It usually presents in the first decade of life as painful inflammatory swellings, either spontaneously...
Autores principales: | , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
Medknow Publications & Media Pvt Ltd
2014
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Materias: | |
Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4234798/ https://www.ncbi.nlm.nih.gov/pubmed/25425787 http://dx.doi.org/10.4103/0970-9185.142865 |
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author | Parekh, Uma R. Read, Selina Desai, Vimal Budde, Arne O. |
author_facet | Parekh, Uma R. Read, Selina Desai, Vimal Budde, Arne O. |
author_sort | Parekh, Uma R. |
collection | PubMed |
description | Fibrodysplasia ossificans progressiva (FOP), or Stone man syndrome, is rare and one of the most disabling genetic conditions of the connective tissue due to progressive extraskeletal ossification. It usually presents in the first decade of life as painful inflammatory swellings, either spontaneously or in response to trauma, which later ossify and lead to severe disability. Progressive spinal deformity including thoracolumbar kyphoscoliosis leads to thoracic insufficiency syndrome, increasing the risk for pneumonia and right sided heart failure. We present the airway management in a 22-year-old male, diagnosed with FOP with severe disability, who required urgent airway intervention as a result of respiratory failure from pnuemonia. Tracheostomy triggers ossification and consequent airway obstruction at the tracheostomy site and laryngoscopy triggers temporomandibular joint ankylosis. Therefore, awake fiber-optic endotracheal intubation is recommended in these patients. Use of an airway endoscopy mask enabled us to simultaneously maintain non-invasive ventilation and intubate the patient in a situation where tracheostomy needed to be avoided. |
format | Online Article Text |
id | pubmed-4234798 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2014 |
publisher | Medknow Publications & Media Pvt Ltd |
record_format | MEDLINE/PubMed |
spelling | pubmed-42347982014-11-25 Emergent airway management in a case of fibrodysplasia ossificans progressiva Parekh, Uma R. Read, Selina Desai, Vimal Budde, Arne O. J Anaesthesiol Clin Pharmacol Case Report Fibrodysplasia ossificans progressiva (FOP), or Stone man syndrome, is rare and one of the most disabling genetic conditions of the connective tissue due to progressive extraskeletal ossification. It usually presents in the first decade of life as painful inflammatory swellings, either spontaneously or in response to trauma, which later ossify and lead to severe disability. Progressive spinal deformity including thoracolumbar kyphoscoliosis leads to thoracic insufficiency syndrome, increasing the risk for pneumonia and right sided heart failure. We present the airway management in a 22-year-old male, diagnosed with FOP with severe disability, who required urgent airway intervention as a result of respiratory failure from pnuemonia. Tracheostomy triggers ossification and consequent airway obstruction at the tracheostomy site and laryngoscopy triggers temporomandibular joint ankylosis. Therefore, awake fiber-optic endotracheal intubation is recommended in these patients. Use of an airway endoscopy mask enabled us to simultaneously maintain non-invasive ventilation and intubate the patient in a situation where tracheostomy needed to be avoided. Medknow Publications & Media Pvt Ltd 2014 /pmc/articles/PMC4234798/ /pubmed/25425787 http://dx.doi.org/10.4103/0970-9185.142865 Text en Copyright: © Journal of Anaesthesiology Clinical Pharmacology http://creativecommons.org/licenses/by-nc-sa/3.0 This is an open-access article distributed under the terms of the Creative Commons Attribution-Noncommercial-Share Alike 3.0 Unported, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited. |
spellingShingle | Case Report Parekh, Uma R. Read, Selina Desai, Vimal Budde, Arne O. Emergent airway management in a case of fibrodysplasia ossificans progressiva |
title | Emergent airway management in a case of fibrodysplasia ossificans progressiva |
title_full | Emergent airway management in a case of fibrodysplasia ossificans progressiva |
title_fullStr | Emergent airway management in a case of fibrodysplasia ossificans progressiva |
title_full_unstemmed | Emergent airway management in a case of fibrodysplasia ossificans progressiva |
title_short | Emergent airway management in a case of fibrodysplasia ossificans progressiva |
title_sort | emergent airway management in a case of fibrodysplasia ossificans progressiva |
topic | Case Report |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4234798/ https://www.ncbi.nlm.nih.gov/pubmed/25425787 http://dx.doi.org/10.4103/0970-9185.142865 |
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