Case Report
Medwave 2015 Dic;15(11):e6341 doi: 10.5867/medwave.2015.11.6341
Intradiaphragmatic bronchopulmonary sequestration: a case report and review
Camila González Parra, Marisol Chávez Gatica, Camila Cuevas Vergara, Jorge Morales Sepúlveda, David Schnettler Rodríguez
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Key Words: bronchopulmonary sequestration, intradiaphragmatic, congenital, pulmonary

Abstract

Bronchopulmonary sequestration is a rare congenital malformation, consisting in a mass of nonfunctioning lung tissue with no connection to the tracheobronchial tree. It can be classified into intra- and extra-lobar. Extra-lobar bronchopulmonary sequestration accounts for 25% of them; of these, only 1% are intra-diaphragmatic. There is little international literature about intra-diaphragmatic bronchopulmonary sequestration and its diagnosis and treatment remain a challenge for the surgeon. Our case is a newborn with antenatal diagnosis of an abdominal mass by ultrasound and magnetic resonance imaging (MRI) showing a left adrenal tumor that appeared to be a neuroblastoma. Postnatal computed tomography (CT) revealed the likelihood of an intra-diaphragmatic bronchopulmonary sequestration, although neuroblastoma could not be ruled out. Abdominal laparoscopy was performed in the bulging left hemidiaphragmatic area. A combined thoracoscopic approach was decided which showed that the defect was located in between both cavities. This is the first case of intra-diaphragmatic bronchopulmonary sequestration reported in Chile.


 

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El secuestro broncopulmonar es una malformación congénita rara, consistente en una masa de tejido pulmonar no funcionante sin conexión al árbol traqueobronquial. Esta puede clasificarse en intra y extralobar. El extralobar corresponde al 25% de los secuestros broncopulmonares y sólo el 1% es intradiafragmático. Existe escasa literatura internacional sobre secuestro broncopulmonar intradiafragmático, por lo que su diagnóstico y tratamiento es un desafío para el cirujano. Nuestro caso es un recién nacido con diagnóstico antenatal de una masa abdominal por ecografía y resonancia magnética nuclear, las cuales mostraron un tumor adrenal que impresiona ser un neuroblastoma. Se realizó tomografía axial computarizada postnatal donde el secuestro broncopulmonar intradiafragmático apareció como más probable. No se pudo excluir el neuroblastoma. Se realizó laparoscopía abdominal en la que se observó un bulto en el hemidiafragma izquierdo. Se decidió un abordaje toracoscópico combinado, el cual mostró que el defecto se encontraba entre ambas cavidades. Este es el primer caso reportado de secuestro broncopulmonar intradiafragmático en Chile.

Authors: Camila González Parra[1], Marisol Chávez Gatica[1], Camila Cuevas Vergara[1], Jorge Morales Sepúlveda[1], David Schnettler Rodríguez[2]

Affiliation:
[1] Facultad Medicina, Universidad Católica del Maule, Región del Maule, Chile
[2] Hospital Regional de Talca, Región del Maule, Chile

E-mail: caami.gonzalez@gmail.com

Author address:
[1] Calle 4 Sur 824
Villa Las Brisas de San Miguel
Talca
Región del Maule
Chile

Citation: González Parra C, Chávez Gatica M, Cuevas Vergara C, Morales Sepúlveda J, Schnettler Rodríguez D. Intradiaphragmatic bronchopulmonary sequestration: a case report and review . Medwave 2015 Dic;15(11):e6341 doi: 10.5867/medwave.2015.11.6341

Submission date: 26/10/2015

Acceptance date: 7/12/2015

Publication date: 18/12/2015

Origin: not requested

Type of review: reviewed by two external peer reviewers, double-blind

PubMed record

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  1. Weinbaum PJ, Bors-Koefoed R, Green KW, Prenatt L. Antenatal sonographic findings in a case of intra-abdominal pulmonary sequestration. Obstet Gynecol. 1989 May;73(5 Pt 2):860-2. | PubMed |
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  4. Caulet T. [Intradiaphragmatic estra-lobar pulmonary sequestration]. Arch Anat Pathol (Paris). 1962 Jun;10:150-1. | PubMed |
  5. Sadler TW, Langman J. Langman embriología médica: con orientación clínica. 12ª Ed. Buenos Aires: Médica Panamericana; 2009
  6. McAteer J, Stephenson J, Ricca R, Waldhausen JH, Gow KW. Intradiaphragmatic pulmonary sequestration: advantages of the thoracoscopic approach. J Pediatr Surg. 2012 Aug;47(8):1607-10. | CrossRef | PubMed |
  7. Nijagal A, Jelin E, Feldstein VA, Courtier J, Urisman A, Jones KD, et al. The diagnosis and management of intradiaphragmatic extralobar pulmonary sequestrations: a report of 4 cases. J Pediatr Surg. 2012 Aug;47(8):1501-5. | CrossRef | PubMed |
  8. Meier AH, Eggli KD, Cilley RE. Intradiaphragmatic extralobar sequestration-a rare pulmonary anomaly. J Pediatr Surg. 2009 Dec;44(12):e27-9. | CrossRef | PubMed |
  9. Yue SW, Guo H, Zhang YG, Gao JB, Ma XX, Ding PX. The clinical value of computer tomographic angiography for the diagnosis and therapeutic planning of patients with pulmonary sequestration. Eur J Cardiothorac Surg. 2013 May;43(5):946-51. | CrossRef | PubMed |
  10. Chun H, Gang Y, Xiaochun Z, Jin T, Bo X, Limin W, et al. Diagnosis and management of intradiaphragmatic extralobar pulmonary sequestration: a report of 11 cases. J Pediatr Surg. 2015 Aug;50(8):1269-72. | CrossRef | PubMed |
Weinbaum PJ, Bors-Koefoed R, Green KW, Prenatt L. Antenatal sonographic findings in a case of intra-abdominal pulmonary sequestration. Obstet Gynecol. 1989 May;73(5 Pt 2):860-2. | PubMed |

Rostión CG. Cirugía pediátrica. 2ª ed. Chile: Mediterráneo; 2014.

Alvarez García N, Escartín Villacampa R, Fernández Atuán R, Sainz Samitier A, Elías Pollina J. [Intradiaphragmatic extralobar pulmonary sequestration]. An Pediatr (Barc). 2014 Jun;80(6):410-1. | CrossRef | PubMed |

Caulet T. [Intradiaphragmatic estra-lobar pulmonary sequestration]. Arch Anat Pathol (Paris). 1962 Jun;10:150-1. | PubMed |

Sadler TW, Langman J. Langman embriología médica: con orientación clínica. 12ª Ed. Buenos Aires: Médica Panamericana; 2009

McAteer J, Stephenson J, Ricca R, Waldhausen JH, Gow KW. Intradiaphragmatic pulmonary sequestration: advantages of the thoracoscopic approach. J Pediatr Surg. 2012 Aug;47(8):1607-10. | CrossRef | PubMed |

Nijagal A, Jelin E, Feldstein VA, Courtier J, Urisman A, Jones KD, et al. The diagnosis and management of intradiaphragmatic extralobar pulmonary sequestrations: a report of 4 cases. J Pediatr Surg. 2012 Aug;47(8):1501-5. | CrossRef | PubMed |

Meier AH, Eggli KD, Cilley RE. Intradiaphragmatic extralobar sequestration-a rare pulmonary anomaly. J Pediatr Surg. 2009 Dec;44(12):e27-9. | CrossRef | PubMed |

Yue SW, Guo H, Zhang YG, Gao JB, Ma XX, Ding PX. The clinical value of computer tomographic angiography for the diagnosis and therapeutic planning of patients with pulmonary sequestration. Eur J Cardiothorac Surg. 2013 May;43(5):946-51. | CrossRef | PubMed |

Chun H, Gang Y, Xiaochun Z, Jin T, Bo X, Limin W, et al. Diagnosis and management of intradiaphragmatic extralobar pulmonary sequestration: a report of 11 cases. J Pediatr Surg. 2015 Aug;50(8):1269-72. | CrossRef | PubMed |