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Bilateral Communicating Intralobar Sequestration and Microgastria
Bilateral Communicating Intralobar Sequestration and Microgastria Sonali Nagendran, MBBCh, Navroop Johal, MRCS, Pat Set, FRCR, Jeffrey Brain, FRCS, Adil Aslam, FRCS, and Madan Samuel, DM Department of Pediatric Surgery, Addenbrooke’s Hospital, Cambridge, United Kingdom
Fig 1.
Fig 2.
FEATURE ARTICLES
A
male infant was born at 39 weeks’ gestation. Antenatal scans demonstrated an absent stomach, but no thoracic abnormalities. On day 5, feeding difficulties and respiratory distress developed. A chest roentgenogram demonstrated aspiration pneumonia. A contrast study revealed microgastria and severe gastroesophageal reflux, which was treated with a fundoplication. The recurrent chest infections continued to develop postoperatively. A computed tomography scan demonstrated bilateral intralobar pulmonary sequestrations, which occupied the medial right lower lobe and the entire left lower lobe and communicated with each other (Fig 1). A large abnormal artery arising from the descending thoracic aorta bifurcated to supply both masses (Figs 2 and 3). At age 15 months, he underwent two lateral thoracotomies. This delineated the bilateral intralobar sequestrations, which communicated with each other within the pleural cavity to form a “saddle-shaped” appearance. A bronchoesophageal fistula communicated with the leftsided sequestration. The fistula was repaired, and a bilateral lower lobe segmentectomy (segments 7, 8 and 9) was performed to completely excise the sequestrations. The patient has since made a good recovery. This patient presented with a bilateral communicating intralobar sequestration. Although Cerruti and colleagues [1] described a bilateral intralobar sequestration in a horseshoe lung with a suspected bridging tunnel, this was not Address correspondence to Dr Samuel, Department of Pediatric Surgery, Addenbrooke’s Hospital, Hills Rd, Cambridge, CB2 0QQ, United Kingdom; e-mail: [email protected].
© 2009 by The Society of Thoracic Surgeons Published by Elsevier Inc
Fig 3.
a true intrapleural communication as was seen in this patient. The association between these two extremely rare anomalies of microgastria and pulmonary sequestration highlights the need for clinicians to have a high index of suspicion for multiple pathologies in children with recurrent respiratory symptoms.
Reference 1. Cerruti MM, Marmolejos F, Cacciarelli T. Bilateral intralobar pulmonary sequestration with horseshoe lung. Ann Thorac Surg 1993;55:509 –10. Ann Thorac Surg 2009;88:2040 • 0003-4975/09/$36.00 doi:10.1016/j.athoracsur.2009.02.070
Fig 1.
Fig 2.
FEATURE ARTICLES
A
male infant was born at 39 weeks’ gestation. Antenatal scans demonstrated an absent stomach, but no thoracic abnormalities. On day 5, feeding difficulties and respiratory distress developed. A chest roentgenogram demonstrated aspiration pneumonia. A contrast study revealed microgastria and severe gastroesophageal reflux, which was treated with a fundoplication. The recurrent chest infections continued to develop postoperatively. A computed tomography scan demonstrated bilateral intralobar pulmonary sequestrations, which occupied the medial right lower lobe and the entire left lower lobe and communicated with each other (Fig 1). A large abnormal artery arising from the descending thoracic aorta bifurcated to supply both masses (Figs 2 and 3). At age 15 months, he underwent two lateral thoracotomies. This delineated the bilateral intralobar sequestrations, which communicated with each other within the pleural cavity to form a “saddle-shaped” appearance. A bronchoesophageal fistula communicated with the leftsided sequestration. The fistula was repaired, and a bilateral lower lobe segmentectomy (segments 7, 8 and 9) was performed to completely excise the sequestrations. The patient has since made a good recovery. This patient presented with a bilateral communicating intralobar sequestration. Although Cerruti and colleagues [1] described a bilateral intralobar sequestration in a horseshoe lung with a suspected bridging tunnel, this was not Address correspondence to Dr Samuel, Department of Pediatric Surgery, Addenbrooke’s Hospital, Hills Rd, Cambridge, CB2 0QQ, United Kingdom; e-mail: [email protected].
© 2009 by The Society of Thoracic Surgeons Published by Elsevier Inc
Fig 3.
a true intrapleural communication as was seen in this patient. The association between these two extremely rare anomalies of microgastria and pulmonary sequestration highlights the need for clinicians to have a high index of suspicion for multiple pathologies in children with recurrent respiratory symptoms.
Reference 1. Cerruti MM, Marmolejos F, Cacciarelli T. Bilateral intralobar pulmonary sequestration with horseshoe lung. Ann Thorac Surg 1993;55:509 –10. Ann Thorac Surg 2009;88:2040 • 0003-4975/09/$36.00 doi:10.1016/j.athoracsur.2009.02.070