Abstract:Pulmonary sequestration is a congenital malformation characterized by cystic, non-functioning embryonic lung tissue supplied by an abnormal systemic artery. It's a rare disease widely variable in clinical presentation and severity, depending mostly on the degree of lung involvement and location in the thoracic cavity. Most patients have recurrent infections and inflammatory conditions in the affected lobe. Surgical resection continues to be the gold standard of therapy and in this context anatomical resection … Show more
“…11 New techniques have emerged in the treatment of ILS, such as endovascular embolization of the feeding artery before the actual removal of the mass, which decreases the perioperative risk of bleeding. [11][12][13][14] Also previously reported is the injection of methylene blue in the aberrant artery to make a clear and macroscopic distinction between healthy tissue and ILS. 15,16 This coloration allows for a complete resection without the need of a lobectomy, therefore sparing healthy lung parenchyma.…”
Bronchopulmonary sequestration is a rare congenital lung dysplasia. An intralobar sequestration (ILS) is a nonfunctional mass within the lung parenchyma without bronchial communication and with aberrant systemic arterial blood supply. Surgical resection or close observation can be proposed in the management of asymptomatic and low-risk ILS, but there is a lack of consensus. Endovascular embolization before thoracoscopic resection of ILS has been described to limit perioperative bleeding. Another technique previously reported is the injection of methylene blue in the feeding artery to macroscopically mark the sequestration from the healthy lung. In that way, a nonanatomical resection can be performed instead of a lobectomy without the risk of leaving abnormal lung tissue in place. We describe the first two cases of these two techniques combined: a 3-year-old girl with an ILS in the right lower lobe with an artery originating from the abdominal aorta, and a 14-month-old girl with an ILS in the right lower lobe with an artery coming from the celiac trunk.The combination of embolization and injection of methylene blue in the aberrant artery leads to a clear macroscopic demarcation of the blue-colored ILS from the healthy lung parenchyma and allowed safe nonanatomical resection of the ILS without risk of bleeding or compromising normal lung tissue.
“…11 New techniques have emerged in the treatment of ILS, such as endovascular embolization of the feeding artery before the actual removal of the mass, which decreases the perioperative risk of bleeding. [11][12][13][14] Also previously reported is the injection of methylene blue in the aberrant artery to make a clear and macroscopic distinction between healthy tissue and ILS. 15,16 This coloration allows for a complete resection without the need of a lobectomy, therefore sparing healthy lung parenchyma.…”
Bronchopulmonary sequestration is a rare congenital lung dysplasia. An intralobar sequestration (ILS) is a nonfunctional mass within the lung parenchyma without bronchial communication and with aberrant systemic arterial blood supply. Surgical resection or close observation can be proposed in the management of asymptomatic and low-risk ILS, but there is a lack of consensus. Endovascular embolization before thoracoscopic resection of ILS has been described to limit perioperative bleeding. Another technique previously reported is the injection of methylene blue in the feeding artery to macroscopically mark the sequestration from the healthy lung. In that way, a nonanatomical resection can be performed instead of a lobectomy without the risk of leaving abnormal lung tissue in place. We describe the first two cases of these two techniques combined: a 3-year-old girl with an ILS in the right lower lobe with an artery originating from the abdominal aorta, and a 14-month-old girl with an ILS in the right lower lobe with an artery coming from the celiac trunk.The combination of embolization and injection of methylene blue in the aberrant artery leads to a clear macroscopic demarcation of the blue-colored ILS from the healthy lung parenchyma and allowed safe nonanatomical resection of the ILS without risk of bleeding or compromising normal lung tissue.
“…Pulmonary sequestration is a rare congenital bronchopulmonary malformation of the foregut characterized by cystic, nonfunctioning lung tissue without normal communication with the bronchial tree. [2,3] Pulmonary sequestration has 2 types: intra- and extrapulmonary sequestration. Pulmonary sequestration accounts for approximately 0.15% to 6.40% of all congenital dysplasias of the lungs.…”
Rationale:
The presence of a mediastinal neurilemmoma accompanied by intrapulmonary sequestration is a rare occurrence. The clinical symptoms of a neurilemmoma depend on the site of the tumor. Diagnosis of pulmonary sequestration mainly depends on the presence of aberrant feeding arteries.
Patient concerns:
A 78-year-old woman was admitted to our hospital with a mediastinal space-occupying lesion of 50 years. Computed tomography and magnetic resonance imaging showed 2 roundish low-density shadows in the left posterior mediastinum.
Diagnosis:
The pathological findings of the upper cystic mass support the diagnosis of neurilemmoma. A branch of aorta was found supplying blood to the lower mass; it was considered a pulmonary sequestration.
Interventions:
Left-sided thoracotomy was planned to remove the chest space-occupying lesions. Two masses were completely removed. Severe adhesion between the left lower lobe and the diaphragm was successfully separated, the aberrant feeding vessel was properly managed, and the lower lobe was resected completely.
Outcomes:
The patient experienced remission of symptoms, had no significant postoperative complications, and was discharged from the hospital.
Lessons:
Special attention should be paid to neurological involvement of the neurilemmoma and the fragile feeding arteries of the intrapulmonary sequestration. Early diagnosis and treatment are important in such cases.
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