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Association of intestinal malrotation and Bochdalek hernia in an adult: a case report

BACKGROUND: Late presentations of congenital diaphragmatic hernia are rare and differ from the classic neonatal presentation. The association with other congenital malformations in children, mainly intestinal malrotation, is well documented. The diagnosis of this association in adults is very rare,...

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Autores principales: Salústio, Raquel, Nabais, Celso, Paredes, Bárbara, Sousa, Francisco V, Porto, Eusébio, Fradique, Caldeira
Formato: Online Artículo Texto
Lenguaje:English
Publicado: BioMed Central 2014
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4030037/
https://www.ncbi.nlm.nih.gov/pubmed/24886112
http://dx.doi.org/10.1186/1756-0500-7-296
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author Salústio, Raquel
Nabais, Celso
Paredes, Bárbara
Sousa, Francisco V
Porto, Eusébio
Fradique, Caldeira
author_facet Salústio, Raquel
Nabais, Celso
Paredes, Bárbara
Sousa, Francisco V
Porto, Eusébio
Fradique, Caldeira
author_sort Salústio, Raquel
collection PubMed
description BACKGROUND: Late presentations of congenital diaphragmatic hernia are rare and differ from the classic neonatal presentation. The association with other congenital malformations in children, mainly intestinal malrotation, is well documented. The diagnosis of this association in adults is very rare, and depends on a high degree of suspicion. CASE PRESENTATION: We report a case of a 50-year-old female Caucasian patient with a previous history of intestinal malrotation diagnosed in adolescence and treated conservatively. She was referred to the hospital with signs and symptoms of intestinal obstruction. The patient undertook computed tomography that confirmed small bowel obstruction with no obvious cause, and a right subphrenic abscess with right empyema was also present. An exploratory laparotomy was performed that revealed an intestinal malrotation associated with a right gangrenous and perforated Bochdalek hernia. Resection of the affected small bowel, closure of the Bochdalek foramen and the Ladd procedure were carried out. CONCLUSION: This case shows a rare association of two rare conditions in adults, and highlights the challenge in reaching the diagnosis and management options.
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spelling pubmed-40300372014-05-23 Association of intestinal malrotation and Bochdalek hernia in an adult: a case report Salústio, Raquel Nabais, Celso Paredes, Bárbara Sousa, Francisco V Porto, Eusébio Fradique, Caldeira BMC Res Notes Case Report BACKGROUND: Late presentations of congenital diaphragmatic hernia are rare and differ from the classic neonatal presentation. The association with other congenital malformations in children, mainly intestinal malrotation, is well documented. The diagnosis of this association in adults is very rare, and depends on a high degree of suspicion. CASE PRESENTATION: We report a case of a 50-year-old female Caucasian patient with a previous history of intestinal malrotation diagnosed in adolescence and treated conservatively. She was referred to the hospital with signs and symptoms of intestinal obstruction. The patient undertook computed tomography that confirmed small bowel obstruction with no obvious cause, and a right subphrenic abscess with right empyema was also present. An exploratory laparotomy was performed that revealed an intestinal malrotation associated with a right gangrenous and perforated Bochdalek hernia. Resection of the affected small bowel, closure of the Bochdalek foramen and the Ladd procedure were carried out. CONCLUSION: This case shows a rare association of two rare conditions in adults, and highlights the challenge in reaching the diagnosis and management options. BioMed Central 2014-05-13 /pmc/articles/PMC4030037/ /pubmed/24886112 http://dx.doi.org/10.1186/1756-0500-7-296 Text en Copyright © 2014 Salústio et al.; licensee BioMed Central Ltd. http://creativecommons.org/licenses/by/4.0 This is an Open Access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/4.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly credited. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated.
spellingShingle Case Report
Salústio, Raquel
Nabais, Celso
Paredes, Bárbara
Sousa, Francisco V
Porto, Eusébio
Fradique, Caldeira
Association of intestinal malrotation and Bochdalek hernia in an adult: a case report
title Association of intestinal malrotation and Bochdalek hernia in an adult: a case report
title_full Association of intestinal malrotation and Bochdalek hernia in an adult: a case report
title_fullStr Association of intestinal malrotation and Bochdalek hernia in an adult: a case report
title_full_unstemmed Association of intestinal malrotation and Bochdalek hernia in an adult: a case report
title_short Association of intestinal malrotation and Bochdalek hernia in an adult: a case report
title_sort association of intestinal malrotation and bochdalek hernia in an adult: a case report
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4030037/
https://www.ncbi.nlm.nih.gov/pubmed/24886112
http://dx.doi.org/10.1186/1756-0500-7-296
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