Intradiaphragmatic extralobar pulmonary sequestration in adult

Journal of Cardiothoracic Surgery, Jun 2014

Extralobar pulmonary sequestrations may be located in intrathoracic or extrathoracic areas. Extrathoracic intradiaphragmatic extralobar pulmonary sequestrations are an extremely rare subset of bronchopulmonary sequestrations and there have been very few reported cases until now. We describe a 48-year-old Korean woman found to have left peridiaphragmatic lesion on computed tomography. We performed thoracoscopic surgery and successfully resected the tumor. Based on the histological findings, it was diagnosed as an intradiaphragmatic extralobar pulmonary sequestration. Postoperative course was uneventful. Intradiaphragmatic extralobar pulmonary sequestration in adult is extremely rare, so we report the case with a literature review.

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Intradiaphragmatic extralobar pulmonary sequestration in adult

Jang-Hoon Lee 0 Mi-Jin Kim 1 0 Department of Thoracic and Cardiovascular Surgery, College of Medicine, Yeungnam University , Daemyeong 5-dong, Nam-gu, Daegu Zip code 705-717 , Korea 1 Department of Pathology, College of Medicine, Yeungnam University , Daegu , Korea Extralobar pulmonary sequestrations may be located in intrathoracic or extrathoracic areas. Extrathoracic intradiaphragmatic extralobar pulmonary sequestrations are an extremely rare subset of bronchopulmonary sequestrations and there have been very few reported cases until now. We describe a 48-year-old Korean woman found to have left peridiaphragmatic lesion on computed tomography. We performed thoracoscopic surgery and successfully resected the tumor. Based on the histological findings, it was diagnosed as an intradiaphragmatic extralobar pulmonary sequestration. Postoperative course was uneventful. Intradiaphragmatic extralobar pulmonary sequestration in adult is extremely rare, so we report the case with a literature review. - Background Extralobar pulmonary sequestrations are most commonly found within the thoracic cavity, but have been described in extrathoracic areas as so called extrathoracic extralobar pulmonary sequestrations. Sequestrations within the diaphragm (intradiaphragmatic extralobar sequestration) are extremely rare. In this case, the tumor was resected using thoracoscopic surgery, and based on the histological findings, it was diagnosed as an intradiaphragmatic extralobar pulmonary sequestration. We herein report a case of an extrathoracic intradiaphragmatic extralobar pulmonary sequestration in a 48-year-old Korean woman. Case presentation A 48-year-old Korean female patient presented with an abnormal mass lesion that was detected by abdominal computed tomography in a visit to our hospital. She had experienced intermittent abdominal pain for several months. She had no other specific past medical history and no history of trauma. The patients vital signs were stable and laboratory tests were normal. Chest X-ray showed no abnormal findings and the computed tomography of her chest showed a 4-cm-sized round mass with areas of calcification in the left hemidiaphragmatic area (Figure 1). After a review of the diagnostic imaging, we were still unable to localize the mass, but we concluded that the lesion was most likely located in the left pleural space based on its proximity to the diaphragm. We decided to remove the mass. The patient was taken to the operating room for thoracoscopic surgery. General anesthesia with double lumen endotracheal tube intubation and one lung ventilation was done. Two 5-mm ports and one 10-mm port were placed in the left chest (fifth intercostal space in the midclavicular line, sixth intercostal space in the anterior axillary line, and eighth intercostal space in the posterior axillary line) for the thoracoscopic approach. No mass was visualized in the pleural space, but a bulge was visualized in the diaphragm consistent with the location of the lesion noted on chest computed tomography. The diaphragm was opened with electrocautery around the mass lesion. Then we identified the mass in the diaphragm (Figure 2). The mass was adhered to the crucial fibers of the diaphragm but was relatively well marginated. We dissected carefully, and a small feeding vessel was noted and clipped. The dissection was relatively easy and the mass was removed. The diaphragm defect was closed with interrupted polyester sutures and one chest tube was placed. The postoperative course was uneventful. The chest tube was removed on the third postoperative day and the patient Figure 1 Preoperative chest computed tomography findings. was discharged the following day. The specimen measured about 4 cm in diameter, 9.4 gram in weight, and was well-defined and reddish. Cut sections of the mass showed sponge-like appearance with cartilage and yellowcolored mucoid materials. Histologic evaluation of the specimen was consistent with the diagnosis of an extralobar sequestration (Figure 3). Discussion Pulmonary sequestration was first defined by Pryce in 1949 [1] as characterized by a non-functional lung without communication with the bronchial tree and the presence of an aberrant blood supply. On the basis of morphological patterns, they are divided into two types: intralobar and extralobar. An intralobar sequestration Figure 2 Intraoperative thoracoscopic images. (A) Incision of diaphragm (black arrow), Intradiaphragmatic mass (red arrow) was identified. (B) Diaphragmatic bulge (black arrow). Incision site of diaphragm (red arrow). (C) Small aberrant vessels were clipped (black arrow). (D) Yellowish mucoid materials were drained (black arrow). Figure 3 Pathologic findings of the resected specimen. (A) Gross findings. (B) Dilated mucin-filled airways and remnants of cartilaginous bronchi (x 100, hematoxylin and eosin stain). (C) Normal lung tissue is not observed (x 100, hematoxylin and eosin stain). (D) Dilated airways are lined by (...truncated)


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Jang-Hoon Lee, Mi-Jin Kim. Intradiaphragmatic extralobar pulmonary sequestration in adult, Journal of Cardiothoracic Surgery, 2014, pp. 112, 9, DOI: 10.1186/1749-8090-9-112