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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...

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Autores principales: Parekh, Uma R., Read, Selina, Desai, Vimal, Budde, Arne O.
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Medknow Publications & Media Pvt Ltd 2014
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.
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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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