Intradiaphragmatic pulmonary sequestrations: a surgical challenge. Case series

Bronchopulmonary sequestrations (BPSs) are rare congenital anomalies characterized by non-functioning embryonic lung tissue receiving anomalous blood supply. They are most commonly located within the thorax (supradiaphragmatic) or into the abdominal cavity (infradiaphragmatic). Intradiaphragmatic ex...

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Main Authors: Chiara Oreglio, Francesca Tocchioni, Marco Ghionzoli, Annamaria Buccoliero, Antonino Morabito, Francesco Morini
Format: Article
Language:English
Published: Frontiers Media S.A. 2023-05-01
Series:Frontiers in Surgery
Subjects:
Online Access:https://www.frontiersin.org/articles/10.3389/fsurg.2023.1181007/full
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author Chiara Oreglio
Chiara Oreglio
Francesca Tocchioni
Marco Ghionzoli
Marco Ghionzoli
Annamaria Buccoliero
Antonino Morabito
Antonino Morabito
Francesco Morini
Francesco Morini
author_facet Chiara Oreglio
Chiara Oreglio
Francesca Tocchioni
Marco Ghionzoli
Marco Ghionzoli
Annamaria Buccoliero
Antonino Morabito
Antonino Morabito
Francesco Morini
Francesco Morini
author_sort Chiara Oreglio
collection DOAJ
description Bronchopulmonary sequestrations (BPSs) are rare congenital anomalies characterized by non-functioning embryonic lung tissue receiving anomalous blood supply. They are most commonly located within the thorax (supradiaphragmatic) or into the abdominal cavity (infradiaphragmatic). Intradiaphragmatic extralobar BPs (IDEPS) are an exceptionally rare finding, representing a diagnostic and operative challenge. We report three cases of IDEPS and their surgical management, describing our experience and approach to such rare clinical entities. From 2016 to 2022, we treated 3 cases of IDEPS. Surgical techniques, histopathological findings and clinical outcomes were retrospectively evaluated for each case and compared. Three different surgical techniques were used to approach each lesion, from open thoracotomy to a combined laparoscopic and thoracoscopic approach. Histopathological analysis of the specimens revealed hybrid pathological features, proper of both congenital pulmonary airway malformation (CPAM) and extralobar pulmonary sequestration. IDEPS represent a surgical challenge for pediatric surgeons, given their complex surgical planning. In our experience, the thoracoscopic approach is safe and feasible when performed by trained surgeons, even though a combined thoracoscopic-laparoscopic approach allows for optimal vessels control. The presence of CPAM elements within the lesions supports their surgical removal. Additional studies are required to better characterize IDEPS and their management.
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spelling doaj.art-f1b462cf409b4473911d44d6924eceb52023-05-25T04:27:55ZengFrontiers Media S.A.Frontiers in Surgery2296-875X2023-05-011010.3389/fsurg.2023.11810071181007Intradiaphragmatic pulmonary sequestrations: a surgical challenge. Case seriesChiara Oreglio0Chiara Oreglio1Francesca Tocchioni2Marco Ghionzoli3Marco Ghionzoli4Annamaria Buccoliero5Antonino Morabito6Antonino Morabito7Francesco Morini8Francesco Morini9Department of Neuroscience, Psychology, Drug Research and Child Health (NEUROFARBA), University of Florence, Florence, ItalyDepartment of Pediatric and Neonatal Surgery, Meyer Children’s Hospital IRCCS, Florence, ItalyDepartment of Pediatric and Neonatal Surgery, Meyer Children’s Hospital IRCCS, Florence, ItalyDepartment of Pediatric and Adolescent Surgery, University of Pisa, Pisa, ItalyDepartment of Surgical, Medical, Molecular Pathology and of the Critic Area, University of Pisa, Pisa, ItalyDepartment of Pathology, Meyer Children’s Hospital IRCCS, Florence, ItalyDepartment of Neuroscience, Psychology, Drug Research and Child Health (NEUROFARBA), University of Florence, Florence, ItalyDepartment of Pediatric and Neonatal Surgery, Meyer Children’s Hospital IRCCS, Florence, ItalyDepartment of Neuroscience, Psychology, Drug Research and Child Health (NEUROFARBA), University of Florence, Florence, ItalyDepartment of Pediatric and Neonatal Surgery, Meyer Children’s Hospital IRCCS, Florence, ItalyBronchopulmonary sequestrations (BPSs) are rare congenital anomalies characterized by non-functioning embryonic lung tissue receiving anomalous blood supply. They are most commonly located within the thorax (supradiaphragmatic) or into the abdominal cavity (infradiaphragmatic). Intradiaphragmatic extralobar BPs (IDEPS) are an exceptionally rare finding, representing a diagnostic and operative challenge. We report three cases of IDEPS and their surgical management, describing our experience and approach to such rare clinical entities. From 2016 to 2022, we treated 3 cases of IDEPS. Surgical techniques, histopathological findings and clinical outcomes were retrospectively evaluated for each case and compared. Three different surgical techniques were used to approach each lesion, from open thoracotomy to a combined laparoscopic and thoracoscopic approach. Histopathological analysis of the specimens revealed hybrid pathological features, proper of both congenital pulmonary airway malformation (CPAM) and extralobar pulmonary sequestration. IDEPS represent a surgical challenge for pediatric surgeons, given their complex surgical planning. In our experience, the thoracoscopic approach is safe and feasible when performed by trained surgeons, even though a combined thoracoscopic-laparoscopic approach allows for optimal vessels control. The presence of CPAM elements within the lesions supports their surgical removal. Additional studies are required to better characterize IDEPS and their management.https://www.frontiersin.org/articles/10.3389/fsurg.2023.1181007/fullchildrencongenital lung malformationCPAMbronchopulmonary sequestrationthoracoscopylaparoscopy
spellingShingle Chiara Oreglio
Chiara Oreglio
Francesca Tocchioni
Marco Ghionzoli
Marco Ghionzoli
Annamaria Buccoliero
Antonino Morabito
Antonino Morabito
Francesco Morini
Francesco Morini
Intradiaphragmatic pulmonary sequestrations: a surgical challenge. Case series
Frontiers in Surgery
children
congenital lung malformation
CPAM
bronchopulmonary sequestration
thoracoscopy
laparoscopy
title Intradiaphragmatic pulmonary sequestrations: a surgical challenge. Case series
title_full Intradiaphragmatic pulmonary sequestrations: a surgical challenge. Case series
title_fullStr Intradiaphragmatic pulmonary sequestrations: a surgical challenge. Case series
title_full_unstemmed Intradiaphragmatic pulmonary sequestrations: a surgical challenge. Case series
title_short Intradiaphragmatic pulmonary sequestrations: a surgical challenge. Case series
title_sort intradiaphragmatic pulmonary sequestrations a surgical challenge case series
topic children
congenital lung malformation
CPAM
bronchopulmonary sequestration
thoracoscopy
laparoscopy
url https://www.frontiersin.org/articles/10.3389/fsurg.2023.1181007/full
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